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Li Z, Wu J, Wu S, Guo S, Cao M, Cheng W, Wang H, Li L, Yin Y. Towards an optimal diagnostic and prognostic model based on semi-quantitative assessment of 18F-FDG PET in children with autoimmune encephalitis. Front Immunol 2025; 16:1457758. [PMID: 40242763 PMCID: PMC12000777 DOI: 10.3389/fimmu.2025.1457758] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 07/01/2024] [Accepted: 02/26/2025] [Indexed: 04/18/2025] Open
Abstract
Purpose The metabolic pattern in autoimmune encephalitis (AE) has been frequently reported. Through this semi-quantitative analysis, we aim to explore a practical diagnostic model based on positron emission tomography (PET) for timely diagnosis of pediatric AE with high accuracy. Moreover, we aim to identify factors that affect the prognosis of pediatric AE and explore the utility of PET as a prognostic biomarker. Method Data were collected from 93 AE patients and 67 non-AE patients (age range: 1-18 years old). Semi-quantitative parameters of 18F-FDG PET imaging were evaluated, including the score of cortical lesion extent and the ratios of lesion-to-basal ganglia and thalamus. The Clinical Assessment Scale in Autoimmune Encephalitis (CASE) was used to rate the disease severity and long-term outcome. Multivariate statistical analysis was used to establish a diagnostic model and analyze predictors. Results The diagnostic model includes three PET parameters. The sensitivity, specificity, and accuracy of the model are 91.4%, 85.1%, and 88.8%, respectively. Participants were followed up for a median of 34 months. Logistic regression analysis indicated that male, initial CASE score >4.5,memory dysfunction, and the ratio of the maximum SUV of the lesion to thalamus (SUVRmaxL/T) < 0.577 are independent factors associated with poor prognosis in AE. We established a prognostic model through these predictors. Conclusion 18F-FDG PET plays a vital role in the diagnosis and prognosis of AE. The PET-based diagnostic model has higher specificity and accuracy than visual analysis. The prognostic model is a useful predictive tool for the long-term prognosis of children with AE.
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Affiliation(s)
- Ziyuan Li
- Department of Nuclear Medicine, Xinhua Hospital Affiliated To Shanghai Jiao Tong University School of Medicine, Shanghai, China
| | - Jing Wu
- Department of Pediatric Neurology, Xinhua Hospital Affiliated To Shanghai Jiao Tong University School of Medicine, Shanghai, China
| | - Shuqi Wu
- Department of Nuclear Medicine, Xinhua Hospital Affiliated To Shanghai Jiao Tong University School of Medicine, Shanghai, China
| | - Shenrui Guo
- Department of Nuclear Medicine, Xinhua Hospital Affiliated To Shanghai Jiao Tong University School of Medicine, Shanghai, China
| | - Mingming Cao
- Department of Nuclear Medicine, Xinhua Hospital Affiliated To Shanghai Jiao Tong University School of Medicine, Shanghai, China
| | - Weiwei Cheng
- Department of Nuclear Medicine, Xinhua Hospital Affiliated To Shanghai Jiao Tong University School of Medicine, Shanghai, China
| | - Hui Wang
- Department of Nuclear Medicine, Xinhua Hospital Affiliated To Shanghai Jiao Tong University School of Medicine, Shanghai, China
| | - Ling Li
- Department of Pediatric Neurology, Xinhua Hospital Affiliated To Shanghai Jiao Tong University School of Medicine, Shanghai, China
| | - Yafu Yin
- Department of Nuclear Medicine, Xinhua Hospital Affiliated To Shanghai Jiao Tong University School of Medicine, Shanghai, China
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Hung WC, Chen HH, Yeh HJ, Wang AG, Cheng HC. Ocular Electrophysiologic Studies in a Patient With Anti-NMDA Receptor Encephalitis and Visual Dysfunction: A Case Report. J Neuroophthalmol 2025; 45:e4-e6. [PMID: 38206753 DOI: 10.1097/wno.0000000000002084] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Grants] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 01/13/2024]
Affiliation(s)
- Wei-Che Hung
- Department of Ophthalmology (W-CH, A-GW, H-CC), Taipei Veterans General Hospital, Taipei, Taiwan; Division of Pediatric Neurosurgery (H-HC), The Neurological Institute, Taipei Veterans General Hospital, Taipei, Taiwan; Institute of Public Health (H-JY), National Yang Ming Chiao Tung University, Taipei, Taiwan; The Department of Physical Medicine and Rehabilitation (H-JY), Taipei Hospital, Ministry of Health and Welfare, New Taipei City, Taiwan; Department of Ophthalmology (A-GW, H-CC), School of Medicine, National Yang Ming Chiao Tung University, Taipei, Taiwan; Program in Molecular Medicine (H-CC), College of Life Sciences, National Yang Ming Chiao Tung University, Taipei, Taiwan; Department of Life Sciences and Institute of Genome Sciences (H-CC), College of Life Sciences, National Yang Ming Chiao Tung University, Taipei, Taiwan; and Brain Research Center (H-CC), National Yang Ming Chiao Tung University, Taipei, Taiwan
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Guasp M, Dalmau J. Autoimmune Encephalitis. Med Clin North Am 2025; 109:443-461. [PMID: 39893022 DOI: 10.1016/j.mcna.2024.09.001] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 02/04/2025]
Abstract
Autoimmune encephalitides (AE) constitute a broad group of inflammatory brain disorders characterized by prominent neuropsychiatric symptoms, frequently in association with autoantibodies against neural (neuronal or glial) antigens. The most frequent AE are anti-NMDA receptor encephalitis, acute disseminated encephalomyelitis (associated with MOG antibodies in 60% of patients), and limbic encephalitis (with several immunologic subtypes, anti-LGI1 encephalitis being the most frequent). The first 2 predominantly affect children and young adults, whereas limbic encephalitis usually affects patients older than 50 years. Despite the severity of symptoms, prompt diagnosis and treatment lead to substantial recovery in most patients.
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Affiliation(s)
- Mar Guasp
- Neuroimmunology Unit, Department of Neurology, Hospital Clínic de Barcelona, University of Barcelona, C/ Casanova, 143; Floor 3A, Barcelona 08036, Spain; Institut d'Investigacions Biomèdiques August Pi i Sunyer (IDIBAPS)-CaixaResearch Institute, Barcelona, Spain; Centro de Investigación Biomédica en red, enfermedades raras (CIBERER), Madrid, Spain
| | - Josep Dalmau
- Neuroimmunology Unit, Department of Neurology, Hospital Clínic de Barcelona, University of Barcelona, C/ Casanova, 143; Floor 3A, Barcelona 08036, Spain; Institut d'Investigacions Biomèdiques August Pi i Sunyer (IDIBAPS)-CaixaResearch Institute, Barcelona, Spain; Centro de Investigación Biomédica en red, enfermedades raras (CIBERER), Madrid, Spain; Department of Neurology, Perelman School of Medicine, University of Pennsylvania, Philadelphia, PA, USA.
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Tebartz van Elst L, Runge K, Meyer PT, Urbach H, Venhoff N, Prüss H. The Neuropsychiatric Checklist for Autoimmune Psychosis: A Narrative Review. Biol Psychiatry 2025:S0006-3223(25)00988-6. [PMID: 39987981 DOI: 10.1016/j.biopsych.2025.02.889] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 05/29/2024] [Revised: 02/11/2025] [Accepted: 02/15/2025] [Indexed: 02/25/2025]
Abstract
Autoimmune encephalitis (AE) is a rapidly evolving topic in both neurology and psychiatry. A recent international consensus article defined criteria for possible, probable, and definite autoimmune psychosis (AP) inspired by the principles established in neurology for the definition of AE. This has stimulated much clinical research on AP but also criticism of the validity of the criteria for possible AP, justifying additional clinical investigations such as lumbar puncture. In clinical practice, it is often difficult to decide how far diagnostic procedures such as lumbar punctures and immunotherapies should go in unclear cases. Against this background, we have 3 aims in this review. First, we summarize and compare the available concepts for the diagnosis of AP in a systematic literature review. Second, we present an overview of typical specific and nonspecific findings that can be obtained in laboratory, electroencephalography, magnetic resonance imaging, cerebrospinal fluid, and [18F]fluorodeoxyglucose positron emission tomography studies in the context of AP. Thirdly, we summarize these findings and present the Neuropsychiatric Checklist for AP as a tool for clinical assessment of the likelihood of AP, with reference to the typical red-flag symptoms and the specific and many unspecific findings that can be identified in additional investigations. We suggest that this instrument may be a useful tool for a comprehensive, possibly uniform, and standardized case assessment in the context of possible AP.
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Affiliation(s)
- Ludger Tebartz van Elst
- Department of Psychiatry and Psychotherapy, Section for Experimental Neuropsychiatry, Medical Center-University of Freiburg, Faculty of Medicine, University of Freiburg, Freiburg, Germany.
| | - Kimon Runge
- Department of Psychiatry and Psychotherapy, Section for Experimental Neuropsychiatry, Medical Center-University of Freiburg, Faculty of Medicine, University of Freiburg, Freiburg, Germany
| | - Philipp T Meyer
- Department of Nuclear Medicine, Medical Center-University of Freiburg, Faculty of Medicine, University of Freiburg, Freiburg, Germany
| | - Horst Urbach
- Department of Neuroradiology, Medical Center-University of Freiburg, Faculty of Medicine, University of Freiburg, Freiburg, Germany
| | - Nils Venhoff
- Department of Rheumatology and Clinical Immunology, Medical Center-University of Freiburg, Faculty of Medicine, University of Freiburg, Freiburg, Germany
| | - Harald Prüss
- Department of Neurology and Experimental Neurology, Charité Universitätsmedizin Berlin, Berlin, Germany; German Center for Neurodegenerative Diseases, Berlin, Berlin, Germany
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Jesse S, Riemann M, Schneider H, Ringelstein M, Melzer N, Vogel N, Pfeffer LK, Friese MA, Sühs KW, Hudasch D, Schwenkenbecher P, Günther A, Geis C, Wickel J, Lesser M, Kather A, Leypoldt F, Dargvainiene J, Markewitz R, Wandinger KP, Thaler FS, Kuchling J, Wurdack K, Sabater L, Finke C, Lewerenz J. Frequency, characteristics, and immunological accompaniments of ataxia in anti-NMDAR antibody-associated encephalitis. Front Immunol 2024; 15:1500904. [PMID: 39735552 PMCID: PMC11681429 DOI: 10.3389/fimmu.2024.1500904] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 09/24/2024] [Accepted: 11/18/2024] [Indexed: 12/31/2024] Open
Abstract
Introduction Very rarely, adult NMDAR antibody-associated encephalitis (NMDAR-E) leads to persistent cerebellar atrophy and ataxia. Transient cerebellar ataxia is common in pediatric NMDAR-E. Immune-mediated cerebellar ataxia may be associated with myelin oligodendrocyte glycoprotein (MOG), aquaporin-4 (AQP-4), kelch-like family member 11 (KLHL11), and glutamate kainate receptor subunit 2 (GluK2) antibodies, all of which may co-occur in NMDAR-E. Here, we aimed to investigate the frequency, long-term outcome, and immunological concomitants of ataxia in NMDAR-E. Methods In this observational study, patients with definite NMDAR-E with a follow-up of >12 months were recruited from the GENERATE registry. Cases with documented ataxia were analyzed in detail. Results In 12 of 62 patients (19%), ataxia was documented. Bilateral cerebellar ataxia without additional focal CNS findings was found in four (one child and three adults); one of these was previously reported as a case with persistent cerebellar atrophy and ataxia. Two patients with bilateral cerebellar ataxia had additional focal neurological symptoms, optic neuritis and facial palsy. Two patients developed hemiataxia: one with diplopia suggesting brainstem dysfunction and the other probably resulting from cerebellar diaschisis due to contralateral status epilepticus. In all but the one developing cerebellar atrophy, cerebellar ataxia was transient and not associated with a worse long-term outcome. In all five patients with cerebellar ataxia tested, MOG, AQP-4, GluK2, and KLHL11 antibodies were negative. In two additional patients negative for both MOG and AQP-4 antibodies, ataxia was sensory and explained by cervical myelitis as part of multiple sclerosis (MS) manifesting temporal relation to NMDAR-E. One of the patients with bilateral ataxia with focal neurological deficits was also diagnosed with MS upon follow-up. Finally, in two patients, ataxia was explained by cerebral hypoxic damage following circulatory failure during an ICU stay with severe NMDAR-E. Discussion Ataxia of different types is quite common in NMDAR-E. Cerebellar ataxia in NMDAR-E is mostly transient. NMDAR-E followed by persistent ataxia and cerebellar atrophy is very rare. Cerebellar ataxia in NMDAR-E may not be explained by concomitant KLHL11, MOG, AQP-4, or GluK2 autoimmunity. Of note, ataxia in NMDAR-E may result from treatment complications and, most interestingly, from MS manifesting in temporal association with NMDAR-E.
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Affiliation(s)
- Sarah Jesse
- Department of Neurology, University Hospital Ulm, Ulm, Germany
| | - Marie Riemann
- Department of Neurology, University Hospital Ulm, Ulm, Germany
| | - Hauke Schneider
- Department of Neurology, Augsburg University, Augsburg, Germany
| | - Marius Ringelstein
- Department of Neurology, Medical Faculty, Heinrich Heine University of Düsseldorf, Düsseldorf, Germany
- Department of Neurology, Centre for Neurology and Neuropsychiatry, LVR-Klinikum, Heinrich-Heine-University Düsseldorf, Düsseldorf, Germany
| | - Nico Melzer
- Department of Neurology, Medical Faculty, Heinrich Heine University of Düsseldorf, Düsseldorf, Germany
| | - Niklas Vogel
- Department of Neurology, Medical Faculty, Heinrich Heine University of Düsseldorf, Düsseldorf, Germany
| | - Lena Kristina Pfeffer
- Institute of Neuroimmunology and Multiple Sclerosis and Department of Neurology, University Medical Center Hamburg-Eppendorf, Hamburg, Germany
| | - Manuel A. Friese
- Institute of Neuroimmunology and Multiple Sclerosis and Department of Neurology, University Medical Center Hamburg-Eppendorf, Hamburg, Germany
| | | | - Dominica Hudasch
- Department of Neurology, Hannover Medical School, Hannover, Germany
| | | | - Albrecht Günther
- Section of Translational Neuroimmunology, Department of Neurology, Jena University Hospital, Jena, Germany
| | - Christian Geis
- Section of Translational Neuroimmunology, Department of Neurology, Jena University Hospital, Jena, Germany
| | - Jonathan Wickel
- Section of Translational Neuroimmunology, Department of Neurology, Jena University Hospital, Jena, Germany
| | - Martin Lesser
- Department of Neurology, Carl Gustav Carus University Dresden, Dresden, Germany
| | - Annika Kather
- Department of Neurology, Carl Gustav Carus University Dresden, Dresden, Germany
| | - Frank Leypoldt
- Institute of Clinical Chemistry, University Hospital Schleswig-Holstein, Kiel/Lubeck, Germany
- Department of Neurology, University Hospital Schleswig-Holstein, Kiel, Germany
| | - Justina Dargvainiene
- Institute of Clinical Chemistry, University Hospital Schleswig-Holstein, Kiel/Lubeck, Germany
| | - Robert Markewitz
- Institute of Clinical Chemistry, University Hospital Schleswig-Holstein, Kiel/Lubeck, Germany
| | - Klaus-Peter Wandinger
- Institute of Clinical Chemistry, University Hospital Schleswig-Holstein, Kiel/Lubeck, Germany
| | - Franziska S. Thaler
- Institute of Clinical Neuroimmunology, LMU University Hospital, LMU Munich, Munich, Germany
- Biomedical Center (BMC), Faculty of Medicine, LMU Munich, Munich, Germany
| | - Joseph Kuchling
- Department of Neurology and Experimental Neurology, Charité – Universitätsmedizin Berlin, Berlin, Germany
| | - Katharina Wurdack
- Department of Neurology and Experimental Neurology, Charité – Universitätsmedizin Berlin, Berlin, Germany
| | - Lidia Sabater
- Fundació de Recerca Biomèdica Clínic Barcelona-Institut d’Investigacions August Pi i Sunyer-Caixa Research Institute, Universitat de Barcelona, Barcelona, Spain
- Spanish National Network for Research on Rare Diseases (CIBERER), Madrid, Spain
| | - Carsten Finke
- Biomedical Center (BMC), Faculty of Medicine, LMU Munich, Munich, Germany
| | - Jan Lewerenz
- Department of Neurology, University Hospital Ulm, Ulm, Germany
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Fan B, Zhou X, Pang L, Long Q, Lv C, Zheng J. Aberrant functional hubs and related networks attributed to cognitive impairment in patients with anti‑N‑methyl‑D‑aspartate receptor encephalitis. Biomed Rep 2024; 21:104. [PMID: 38827495 PMCID: PMC11140295 DOI: 10.3892/br.2024.1792] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 10/10/2023] [Accepted: 03/06/2024] [Indexed: 06/04/2024] Open
Abstract
Anti-N-methyl-D-aspartate receptor (anti-NMDAR) encephalitis results in severe neuropsychiatric symptoms and persistent cognitive impairment; however, the underlying mechanism is still not fully understood. The present study utilized the degree centrality (DC), functional connectivity (FC) and multivariate pattern analysis (MVPA) to further explore neurofunctional symptoms in patients with anti-NMDAR encephalitis. A total of 29 patients with anti-NMDAR encephalitis and 26 healthy controls (HCs) were enrolled for neuropsychological assessment and resting-state functional MRI (rs-fMRI) scans. DC, FC and MVPA were examined to investigate cerebral functional activity and distinguish neuroimaging characteristics between the patient and HC groups based on the rs-fMRI data. Compared with the HCs, the patients exhibited cognitive deficits, anxiety and depression. In the DC analysis, the patients exhibited significantly decreased DC strength in the left rectus gyrus, left caudate nucleus (LCN) and bilateral superior medial frontal gyrus, as well as increased DC strength in the cerebellar anterior lobe, compared with the HCs. In the subsequent FC analysis, the LCN showed decreased FC strength in the bilateral middle frontal gyrus and right precuneus. Furthermore, correlation analysis indicated that disrupted cerebral functional activity was significantly correlated with the alerting effect and Hamilton Depression Scale score. Using DC maps and receiver operating characteristic curve analysis, the MVPA classifier exhibited an area under curve of 0.79, and the accuracy classification rate was 76.36%, with a sensitivity of 79.31% and a specificity of 78.18%. The present study revealed that the disrupted functional activity of hub and related networks in the cerebellum, including the default mode network and executive control network, contributed to deficits in cognition and emotion in patients with anti-NMDAR encephalitis. In conclusion, the present study provided imaging evidence and primary diagnostic markers for pathological and compensatory mechanisms of anti-NMDAR encephalitis, with the aim of improving the understanding of this disease.
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Affiliation(s)
- Binglin Fan
- Department of Neurology, The First Affiliated Hospital of Guangxi Medical University, Nanning, Guangxi Zhuang Autonomous Region 530021, P.R. China
| | - Xia Zhou
- Department of Neurology, The First Affiliated Hospital of Guangxi Medical University, Nanning, Guangxi Zhuang Autonomous Region 530021, P.R. China
| | - Linlin Pang
- Department of Neurology, The First Affiliated Hospital of Guangxi Medical University, Nanning, Guangxi Zhuang Autonomous Region 530021, P.R. China
| | - Qijia Long
- Department of Neurology, The First Affiliated Hospital of Guangxi Medical University, Nanning, Guangxi Zhuang Autonomous Region 530021, P.R. China
| | - Caitiao Lv
- Department of Neurology, The First Affiliated Hospital of Guangxi Medical University, Nanning, Guangxi Zhuang Autonomous Region 530021, P.R. China
| | - Jinou Zheng
- Department of Neurology, The First Affiliated Hospital of Guangxi Medical University, Nanning, Guangxi Zhuang Autonomous Region 530021, P.R. China
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Dai Y, Zhu Z, Tang Y, Xiao L, Liu X, Zhang M, Xiao B, Hu K, Long L, Xie Y, Hu S. The clinical and predictive value of 18F-FDG PET/CT metabolic patterns in a clinical Chinese cohort with autoimmune encephalitis. CNS Neurosci Ther 2024; 30:e14821. [PMID: 38948940 PMCID: PMC11215490 DOI: 10.1111/cns.14821] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Grants] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 02/26/2024] [Revised: 05/29/2024] [Accepted: 06/14/2024] [Indexed: 07/02/2024] Open
Abstract
AIMS To investigate the diagnostic and predictive role of 18F-FDG PET/CT in patients with autoimmune encephalitis (AE) as a whole group. METHODS Thrty-five patients (20 females and 15 males) with AE were recruited. A voxel-to-voxel semi-quantitative analysis based on SPM12 was used to analyze 18F-FDG PET/CT imaging data compared to healthy controls. Further comparison was made in different prognostic groups categorized by modified Rankin Scale (mRS). RESULTS In total, 24 patients (68.6%) were tested positive neuronal antibodies in serum and/or CSF. Psychiatric symptoms and seizure attacks were major clinical symptoms. In the acute stage, 13 patients (37.1%) demonstrated abnormal brain MRI results, while 33 (94.3%) presented abnormal metabolism patterns. 18F-FDG PET/CT was more sensitive than MRI (p < 0.05). Patients with AE mainly presented mixed metabolism patterns compared to the matched controls, demonstrating hypermetabolism mainly in the cerebellum, BG, MTL, brainstem, insula, middle frontal gyrus, and relatively hypometabolism in the frontal cortex, occipital cortex, temporal gyrus, right parietal gyrus, left cingulate gyrus (p < 0.05, FWE corrected). After a median follow-up of 26 months, the multivariable analysis identified a decreased level of consciousness as an independent risk factor associated with poor outcome of AE (HR = 3.591, p = 0.016). Meanwhile, decreased metabolism of right superior frontal gyrus along with increased metabolism of the middle and upper brainstem was more evident in patients with poor outcome (p < 0.001, uncorrected). CONCLUSION 18F-FDG PET/CT was more sensitive than MRI to detect neuroimaging abnormalities of AE. A mixed metabolic pattern, characterized by large areas of cortical hypometabolism with focal hypermetabolism was a general metabolic pattern. Decreased metabolism of right superior frontal gyrus with increased metabolism of the middle and upper brainstem may predict poor long-term prognosis of AE.
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Affiliation(s)
- Yuwei Dai
- Department of Neurology, Xiangya HospitalCentral South UniversityChangshaHunanP.R. China
- National Clinical Research Center for Geriatric Diseases, Xiangya HospitalCentral South UniversityChangshaHunanP.R. China
- Clinical Research Center for Epileptic disease of Hunan ProvinceCentral South UniversityChangshaHunanP.R. China
| | - Zehua Zhu
- National Clinical Research Center for Geriatric Diseases, Xiangya HospitalCentral South UniversityChangshaHunanP.R. China
- Department of Nuclear Medicine, Xiangya HospitalCentral South UniversityChangshaHunanP.R. China
- Key Laboratory of Biological Nanotechnology of National Health Commission, Xiangya HospitalCentral South UniversityChangshaHunanP.R. China
- Division of Life Sciences and Medicine, Department of Nuclear Medicine, The First Affiliated Hospital of USTCUniversity of Science and Technology of ChinaHefeiAnhuiP.R. China
| | - Yongxiang Tang
- National Clinical Research Center for Geriatric Diseases, Xiangya HospitalCentral South UniversityChangshaHunanP.R. China
- Department of Nuclear Medicine, Xiangya HospitalCentral South UniversityChangshaHunanP.R. China
- Key Laboratory of Biological Nanotechnology of National Health Commission, Xiangya HospitalCentral South UniversityChangshaHunanP.R. China
| | - Ling Xiao
- National Clinical Research Center for Geriatric Diseases, Xiangya HospitalCentral South UniversityChangshaHunanP.R. China
- Department of Nuclear Medicine, Xiangya HospitalCentral South UniversityChangshaHunanP.R. China
- Key Laboratory of Biological Nanotechnology of National Health Commission, Xiangya HospitalCentral South UniversityChangshaHunanP.R. China
| | - Xianghe Liu
- Department of Neurology, Xiangya HospitalCentral South UniversityChangshaHunanP.R. China
- National Clinical Research Center for Geriatric Diseases, Xiangya HospitalCentral South UniversityChangshaHunanP.R. China
- Clinical Research Center for Epileptic disease of Hunan ProvinceCentral South UniversityChangshaHunanP.R. China
| | - Min Zhang
- Department of Neurology, Xiangya HospitalCentral South UniversityChangshaHunanP.R. China
- National Clinical Research Center for Geriatric Diseases, Xiangya HospitalCentral South UniversityChangshaHunanP.R. China
- Clinical Research Center for Epileptic disease of Hunan ProvinceCentral South UniversityChangshaHunanP.R. China
| | - Bo Xiao
- Department of Neurology, Xiangya HospitalCentral South UniversityChangshaHunanP.R. China
- National Clinical Research Center for Geriatric Diseases, Xiangya HospitalCentral South UniversityChangshaHunanP.R. China
- Clinical Research Center for Epileptic disease of Hunan ProvinceCentral South UniversityChangshaHunanP.R. China
| | - Kai Hu
- Department of Neurology, Xiangya HospitalCentral South UniversityChangshaHunanP.R. China
- National Clinical Research Center for Geriatric Diseases, Xiangya HospitalCentral South UniversityChangshaHunanP.R. China
- Clinical Research Center for Epileptic disease of Hunan ProvinceCentral South UniversityChangshaHunanP.R. China
| | - Lili Long
- Department of Neurology, Xiangya HospitalCentral South UniversityChangshaHunanP.R. China
- National Clinical Research Center for Geriatric Diseases, Xiangya HospitalCentral South UniversityChangshaHunanP.R. China
- Clinical Research Center for Epileptic disease of Hunan ProvinceCentral South UniversityChangshaHunanP.R. China
| | - Yuanyuan Xie
- Department of Neurology, Xiangya HospitalCentral South UniversityChangshaHunanP.R. China
- National Clinical Research Center for Geriatric Diseases, Xiangya HospitalCentral South UniversityChangshaHunanP.R. China
- Clinical Research Center for Epileptic disease of Hunan ProvinceCentral South UniversityChangshaHunanP.R. China
| | - Shuo Hu
- National Clinical Research Center for Geriatric Diseases, Xiangya HospitalCentral South UniversityChangshaHunanP.R. China
- Department of Nuclear Medicine, Xiangya HospitalCentral South UniversityChangshaHunanP.R. China
- Key Laboratory of Biological Nanotechnology of National Health Commission, Xiangya HospitalCentral South UniversityChangshaHunanP.R. China
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Gagnon MH, Derenoncourt PR, Rayamahi S, Taylor S, Parikh AK, Ponisio MR, Khanna G. Unusual imaging findings associated with abdominal pediatric germ cell tumors. Pediatr Radiol 2024; 54:1093-1104. [PMID: 38462578 DOI: 10.1007/s00247-024-05894-9] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 11/15/2023] [Revised: 02/19/2024] [Accepted: 02/20/2024] [Indexed: 03/12/2024]
Abstract
Germ cell tumors of childhood are tumors arising from germline cells in gonadal or extragonadal locations. Extragonadal germ cell tumors are characteristically located in the midline, arising intracranially or in the mediastinum, retroperitoneum, or pelvis. These tumors are generally easily diagnosed due to typical sites of origin, characteristic imaging findings, and laboratory markers. However, germ cell tumors can be associated with unusual clinical syndromes or imaging features that can perplex the radiologist. This review will illustrate atypical imaging/clinical manifestations and complications of abdominal germ cell tumors in childhood. These features include unusual primary tumors such as multifocal primaries; local complications such as ovarian torsion or ruptured dermoid; atypical presentations of metastatic disease associated with burned-out primary tumor, growing teratoma syndrome, and gliomatosis peritonei; endocrine manifestations such as precocious puberty and hyperthyroidism; and antibody mediated paraneoplastic syndrome such as anti-N-methyl-D-aspartate-receptor antibody-mediated encephalitis. This review aims to illustrate unusual imaging features associated with the primary tumor, metastatic disease, or distant complications of abdominal germ cell tumors of childhood.
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Affiliation(s)
- Marie-Helene Gagnon
- Department of Radiology & Imaging Sciences, Emory University and Children's Healthcare of Atlanta, 1405 Clifton Road NE, Atlanta, GA, 30322, USA.
| | - Paul-Robert Derenoncourt
- Mallinckrodt Institute of Radiology, Washington University in Saint Louis, 510 S Kingshighway Blvd, St. Louis, MO, 63110, USA
| | - Sampanna Rayamahi
- Mallinckrodt Institute of Radiology, Washington University in Saint Louis, 510 S Kingshighway Blvd, St. Louis, MO, 63110, USA
| | - Susan Taylor
- Department of Radiology & Imaging Sciences, Emory University and Children's Healthcare of Atlanta, 1405 Clifton Road NE, Atlanta, GA, 30322, USA
| | - Ashishkumar K Parikh
- Department of Radiology & Imaging Sciences, Emory University and Children's Healthcare of Atlanta, 1405 Clifton Road NE, Atlanta, GA, 30322, USA
| | - Maria R Ponisio
- Mallinckrodt Institute of Radiology, Washington University in Saint Louis, 510 S Kingshighway Blvd, St. Louis, MO, 63110, USA
| | - Geetika Khanna
- Department of Radiology & Imaging Sciences, Emory University and Children's Healthcare of Atlanta, 1405 Clifton Road NE, Atlanta, GA, 30322, USA
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Hartung TJ, Cooper G, Jünger V, Komnenić D, Ryan L, Heine J, Chien C, Paul F, Prüss H, Finke C. The T1-weighted/T2-weighted ratio as a biomarker of anti-NMDA receptor encephalitis. J Neurol Neurosurg Psychiatry 2024; 95:366-373. [PMID: 37798094 PMCID: PMC10958321 DOI: 10.1136/jnnp-2023-332069] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Grants] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 06/21/2023] [Accepted: 09/21/2023] [Indexed: 10/07/2023]
Abstract
BACKGROUND Anti-N-methyl-D-aspartate receptor (NMDAR) encephalitis rarely causes visible lesions in conventional MRI, yet advanced imaging detects extensive white matter damage. To improve prognostic capabilities, we evaluate the T1-weighted/T2-weighted (T1w/T2w) ratio, a measure of white matter integrity computable from clinical MRI sequences, in NMDAR encephalitis and examine its associations with cognitive impairment. METHODS T1-weighted and T2-weighted MRI were acquired cross-sectionally at 3 Tesla in 53 patients with NMDAR encephalitis (81% women, mean age 29 years) and 53 matched healthy controls. Quantitative and voxel-wise group differences in T1w/T2w ratios and associations with clinical and neuropsychological outcomes were assessed. P-values were false discovery rate (FDR) adjusted where multiple tests were conducted. RESULTS Patients with NMDAR encephalitis had significantly lower T1w/T2w ratios across normal appearing white matter (p=0.009, Hedges' g=-0.51), which was associated with worse verbal episodic memory performance (r=0.39, p=0.005, p(FDR)=0.026). White matter integrity loss was observed in the corticospinal tract, superior longitudinal fascicle, optic radiation and callosal body with medium to large effects (Cohen's d=[0.42-1.17]). In addition, patients showed decreased T1w/T2w ratios in the hippocampus (p=0.002, p(FDR)=0.005, Hedges' g=-0.62), amygdala (p=0.002, p(FDR)=0.005, Hedges' g=-0.63) and thalamus (p=0.010, p(FDR)=0.019, Hedges' g=-0.51). CONCLUSIONS The T1w/T2w ratio detects microstructural changes in grey and white matter of patients with NMDAR encephalitis that correlate with cognitive performance. Computable from conventional clinical MRI sequences, this measure shows promise in bridging the clinico-radiological dissociation in NMDAR encephalitis and could serve as an imaging outcome measure in clinical trials.
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Affiliation(s)
- Tim Julian Hartung
- Department of Neurology and Experimental Neurology, Charité - Universitätsmedizin Berlin, Berlin, Germany
| | - Graham Cooper
- Department of Neurology and Experimental Neurology, Charité - Universitätsmedizin Berlin, Berlin, Germany
| | - Valentin Jünger
- Experimental and Clinical Research Center, Max Delbrueck Center for Molecular Medicine and Charité - Universitätsmedizin Berlin, Berlin, Germany
- Neuroscience Clinical Research Center (NCRC), Charité - Universitätsmedizin Berlin, Berlin, Germany
- Department of Neuroradiology, Charité - Universitätsmedizin Berlin, Berlin, Germany
| | - Darko Komnenić
- Department of Neurology and Experimental Neurology, Charité - Universitätsmedizin Berlin, Berlin, Germany
- Berlin School of Mind and Brain, Humboldt-Universität zu Berlin, Berlin, Germany
| | - Lara Ryan
- Berlin School of Mind and Brain, Humboldt-Universität zu Berlin, Berlin, Germany
- Einstein Center for Neurosciences Berlin, Charité - Universitätsmedizin Berlin, Berlin, Germany
| | - Josephine Heine
- Department of Neurology and Experimental Neurology, Charité - Universitätsmedizin Berlin, Berlin, Germany
| | - Claudia Chien
- Experimental and Clinical Research Center, Max Delbrueck Center for Molecular Medicine and Charité - Universitätsmedizin Berlin, Berlin, Germany
- Neuroscience Clinical Research Center (NCRC), Charité - Universitätsmedizin Berlin, Berlin, Germany
- Medizinische Klinik m.S. Psychosomatik, Charité - Universitätsmedizin Berlin, Berlin, Germany
| | - Friedemann Paul
- Experimental and Clinical Research Center, Max Delbrueck Center for Molecular Medicine and Charité - Universitätsmedizin Berlin, Berlin, Germany
| | - Harald Prüss
- Department of Neurology and Experimental Neurology, Charité - Universitätsmedizin Berlin, Berlin, Germany
- German Center for Neurodegenerative Diseases (DZNE) Berlin, Berlin, Germany
| | - Carsten Finke
- Department of Neurology and Experimental Neurology, Charité - Universitätsmedizin Berlin, Berlin, Germany
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10
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Kalra S, Tripathi M, Tripathi M, Sonar RS, Pandey AK, Jaleel J, Singh RK, Kumar P, Damle NA, Bal C. Role of FDG PET/CT in definitive and presumed autoimmune encephalitis. Nucl Med Commun 2024; 45:121-127. [PMID: 37982572 DOI: 10.1097/mnm.0000000000001790] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/21/2023]
Abstract
OBJECTIVE F-18 Fluorodeoxyglucose PET/CT (FDG-PET) is emerging as a useful imaging adjunct to MRI in the initial diagnostic evaluation of autoimmune encephalitis (AIE)-though presently it is not included in the diagnostic criteria. MATERIALS AND METHODS In this prospective study we enrolled a total of 52 patients with clinically diagnosed and treated AIE. MRI evaluation was done in each case along with CSF and EEG where feasible. FDG-PET was done for all and images were interpreted visually and using SPM. RESULTS The mean age group of patients included was 38.5 ± 22.6 years with 31 females and 21 males. 23 antibody-positive cases underwent PET, the most common antibody detected was anti-NMDAR type followed by anti-LGI 1. Most common metabolic pattern in NMDARE was hypermetabolism in basal ganglia and hypometabolism in parieto-occipital cortices and ovarian teratoma was detected in two of these patients on whole-body PET. A metabolic pattern consistent with AIE was demonstrated in 22/29 (75.8%) antibody-negative patients with hypermetabolism in basal ganglia and mesial temporal cortices. The overall sensitivity of FDG PET was 86% (45/52). MRI abnormalities were detected in 22/52 (42%) cases, 10/23 antibody positive and 12/29 antibody negative cases. PET was positive in 23/30 (76%) MRI negative cases. CONCLUSION Sensitivity of FDG PET for supporting a diagnosis of AIE was higher compared to MRI in both antibody-positive (definitive) and antibody-negative (presumed) AIE. Specific metabolic patterns can be demonstrated on FDG PET in AIE, prompting an early diagnosis so that timely treatment can be instituted.
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11
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Beutler BD, Moody AE, Thomas JM, Sugar BP, Ulanja MB, Antwi-Amoabeng D, Tsikitas LA. Anti-N-methyl-D-aspartate receptor-associated encephalitis: A review of clinicopathologic hallmarks and multimodal imaging manifestations. World J Radiol 2024; 16:1-8. [PMID: 38312349 PMCID: PMC10835429 DOI: 10.4329/wjr.v16.i1.1] [Citation(s) in RCA: 2] [Impact Index Per Article: 2.0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 09/30/2023] [Revised: 12/04/2023] [Accepted: 12/25/2023] [Indexed: 01/23/2024] Open
Abstract
Anti-N-methyl-D-aspartate receptor-associated encephalitis (NMDARE) is a rare immune-mediated neuroinflammatory condition characterized by the rapid onset of neuropsychiatric symptoms and autonomic dysfunction. The mechanism of pathogenesis remains incompletely understood, but is thought to be related to antibodies targeting the GluN1 subunit of the NMDA receptor with resultant downstream dysregulation of dopaminergic pathways. Young adults are most frequently affected; the median age at diagnosis is 21 years. There is a strong female predilection with a female sex predominance of 4:1. NMDARE often develops as a paraneoplastic process and is most commonly associated with ovarian teratoma. However, NMDARE has also been described in patients with small cell lung cancer, clear cell renal carcinoma, and other benign and malignant neoplasms. Diagnosis is based on correlation of the clinical presentation, electroencephalography, laboratory studies, and imaging. Computed tomography, positron emission tomography, and magnetic resonance imaging are essential to identify an underlying tumor, exclude clinicopathologic mimics, and predict the likelihood of long-term functional impairment. Nuclear imaging may be of value for prognostication and to assess the response to therapy. Treatment may involve high-dose corticosteroids, intravenous immunoglobulin, and plasma exchange. Herein, we review the hallmark clinicopathologic features and imaging findings of this rare but potentially devastating condition and summarize diagnostic criteria, treatment regimens, and proposed pathogenetic mechanisms.
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Affiliation(s)
- Bryce David Beutler
- Department of Radiology, University of Southern California, Keck School of Medicine, Los Angeles, CA 90033, United States
| | - Alastair E Moody
- Department of Anesthesiology, University of Utah, Salt Lake City, UT 84132, United States
| | - Jerry Mathew Thomas
- Department of Radiology, University of Southern California, Keck School of Medicine, Los Angeles, CA 90033, United States
| | - Benjamin Phillip Sugar
- Department of Radiology, University of Southern California, Keck School of Medicine, Los Angeles, CA 90033, United States
| | - Mark B Ulanja
- Department of Internal Medicine, Christus Ochsner St. Patrick Hospital, Lake Charles, LA 70601, United States
| | - Daniel Antwi-Amoabeng
- Department of Internal Medicine, Christus Ochsner St. Patrick Hospital, Lake Charles, LA 70601, United States
| | - Lucas Anthony Tsikitas
- Department of Radiology, University of Southern California, Keck School of Medicine, Los Angeles, CA 90033, United States
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12
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Kuchling J, Jurek B, Kents M, Kreye J, Geis C, Wickel J, Mueller S, Koch SP, Boehm-Sturm P, Prüss H, Finke C. Impaired functional connectivity of the hippocampus in translational murine models of NMDA-receptor antibody associated neuropsychiatric pathology. Mol Psychiatry 2024; 29:85-96. [PMID: 37875549 PMCID: PMC11078734 DOI: 10.1038/s41380-023-02303-9] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Grants] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 10/10/2022] [Revised: 09/28/2023] [Accepted: 10/11/2023] [Indexed: 10/26/2023]
Abstract
Decreased hippocampal connectivity and disruption of functional networks are established resting-state functional MRI (rs-fMRI) features that are associated with neuropsychiatric symptom severity in human anti-N-methyl-D-aspartate receptor (NMDAR) encephalitis. However, the underlying pathophysiology of NMDAR encephalitis remains poorly understood. Application of patient-derived monoclonal antibodies against the NR1 (GluN1) subunit of the NMDAR now allows for the translational investigation of functional connectivity in experimental murine NMDAR antibody disease models with neurodevelopmental disorders. Using rs-fMRI, we studied functional connectivity alterations in (1) adult C57BL/6 J mice that were intrathecally injected with a recombinant human NR1 antibody over 14 days (n = 10) and in (2) a newly established mouse model with in utero exposure to a human recombinant NR1 antibody (NR1-offspring) at the age of (2a) 8 weeks (n = 15) and (2b) 10 months (n = 14). Adult NR1-antibody injected mice showed impaired functional connectivity within the left hippocampus compared to controls, resembling impaired connectivity patterns observed in human NMDAR encephalitis patients. Similarly, NR1-offspring showed significantly reduced functional connectivity in the hippocampus after 8 weeks, and impaired connectivity in the hippocampus was likewise observed in NR1-offspring at the age of 10 months. We successfully reproduced functional connectivity changes within the hippocampus in different experimental murine systems that were previously observed in human NMDAR encephalitis patients. Translational application of this method within a combined imaging and histopathological framework will allow future experimental studies to identify the underlying biological mechanisms and may eventually facilitate non-invasive monitoring of disease activity and treatment responses in autoimmune encephalitis.
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Grants
- J.Ku is participant in the BIH-Charité Junior Clinician Scientist Program
- J.Kr is participant in the BIH-Charité Clinician Scientist Program funded by the Charité – Universitätsmedizin Berlin and the Berlin Institute of Health.
- C.G. is funded by the Deutsche Forschungsgemeinschaft (DFG, German Research Foundation: grant numbers GE2519/8-1, GE2519/9-1, FOR3004 and GE2519/11-1), by the German Ministry of Education and Research (BMBF: grant numbers 01EW1901, 01GM1908B), and receives funding from Hermann und Lilly Schilling Foundation.
- H.P. is funded by the Deutsche Forschungsgemeinschaft (DFG, German Research Foundation: grant numbers PR 1274/2-1, PR 1274/3-1, FOR3004 and PR 1274/5-1), by the German Ministry of Education and Research (BMBF: grant numbers 01GM1908D, CONNECT-GENERATE), and by the Helmholtz Association (HIL-A03).
- C.F. is funded by the Deutsche Forschungsgemeinschaft (DFG, German Research Foundation: grant numbers FI 2309/1-1 and FI 2309/2-1), and by the German Ministry of Education and Research (BMBF; grant numbers 01GM1908D, CONNECT-GENERATE)
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Affiliation(s)
- Joseph Kuchling
- Department of Neurology and Experimental Neurology, Charité - Universitätsmedizin Berlin, Berlin, Germany
- Neurocure Cluster of Excellence, NeuroCure Clinical Research Center, Charité - Universitätsmedizin Berlin, Berlin, Germany
| | - Betty Jurek
- Department of Neurology and Experimental Neurology, Charité - Universitätsmedizin Berlin, Berlin, Germany
- German Center for Neurodegenerative Diseases (DZNE) Berlin, Berlin, Germany
| | - Mariya Kents
- Department of Neurology and Experimental Neurology, Charité - Universitätsmedizin Berlin, Berlin, Germany
- German Center for Neurodegenerative Diseases (DZNE) Berlin, Berlin, Germany
| | - Jakob Kreye
- Department of Neurology and Experimental Neurology, Charité - Universitätsmedizin Berlin, Berlin, Germany
- German Center for Neurodegenerative Diseases (DZNE) Berlin, Berlin, Germany
- Berlin Institute of Health at Charité - Universitätsmedizin Berlin, Charitéplatz 1, 10117, Berlin, Germany
| | - Christian Geis
- Section of Translational Neuroimmunology, Hans Berger Department of Neurology, Jena University Hospital, Jena, Germany
| | - Jonathan Wickel
- Section of Translational Neuroimmunology, Hans Berger Department of Neurology, Jena University Hospital, Jena, Germany
| | - Susanne Mueller
- Department of Neurology and Experimental Neurology, Charité - Universitätsmedizin Berlin, Berlin, Germany
- Neurocure Cluster of Excellence, Core Facility 7 T Experimental MRIs, Charité - Universitätsmedizin Berlin, Berlin, Germany
- Berlin Center for Stroke Research, Charité - Universitätsmedizin Berlin, Berlin, Germany
| | - Stefan Paul Koch
- Department of Neurology and Experimental Neurology, Charité - Universitätsmedizin Berlin, Berlin, Germany
- Neurocure Cluster of Excellence, Core Facility 7 T Experimental MRIs, Charité - Universitätsmedizin Berlin, Berlin, Germany
- Berlin Center for Stroke Research, Charité - Universitätsmedizin Berlin, Berlin, Germany
| | - Philipp Boehm-Sturm
- Department of Neurology and Experimental Neurology, Charité - Universitätsmedizin Berlin, Berlin, Germany
- Neurocure Cluster of Excellence, Core Facility 7 T Experimental MRIs, Charité - Universitätsmedizin Berlin, Berlin, Germany
- Berlin Center for Stroke Research, Charité - Universitätsmedizin Berlin, Berlin, Germany
| | - Harald Prüss
- Department of Neurology and Experimental Neurology, Charité - Universitätsmedizin Berlin, Berlin, Germany.
- German Center for Neurodegenerative Diseases (DZNE) Berlin, Berlin, Germany.
| | - Carsten Finke
- Department of Neurology and Experimental Neurology, Charité - Universitätsmedizin Berlin, Berlin, Germany.
- Humboldt-Universität zu Berlin, Berlin School of Mind and Brain, Berlin, Germany.
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13
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Baishya J, Basher RK. Antibody-Specific PET Finding in Autoimmune Encephalitis: How Accurate? Ann Indian Acad Neurol 2024; 27:5-6. [PMID: 38495249 PMCID: PMC10941909 DOI: 10.4103/aian.aian_338_23] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Track Full Text] [Download PDF] [Journal Information] [Subscribe] [Scholar Register] [Received: 04/18/2023] [Revised: 04/20/2023] [Accepted: 04/22/2023] [Indexed: 03/19/2024] Open
Affiliation(s)
- Jitupam Baishya
- Department of Neurology, Postgraduate Institute of Medical Education and Research, Chandigarh, India
| | - Rajinder Kumar Basher
- Department of Nuclear Medicine, Postgraduate Institute of Medical Education and Research, Chandigarh, India
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14
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Wu D, Guo Y, Li C, Pang X, Xu S, Zhang J, Wei L, Li Q, Du J, Tian Y, Wang K. Dynamic and static changes of amplitude of low-frequency fluctuations in anti‑N‑methyl‑D‑aspartate receptor encephalitis. Brain Imaging Behav 2023; 17:652-663. [PMID: 37673808 DOI: 10.1007/s11682-023-00790-6] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Accepted: 08/21/2023] [Indexed: 09/08/2023]
Abstract
BACKGROUND Previous neuroimaging research has examined static local brain activity changes in patients with anti-N-methyl-d-aspartate receptor (anti-NMDAR) encephalitis. However, the dynamic properties of local brain activity in anti-NMDAR encephalitis remain unknown. METHODS This study used a combination of the amplitude of low-frequency fluctuation (ALFF) method and a sliding-window dynamic analysis approach to examine the time-varying local brain activity changes in anti-NMDAR encephalitis. RESULTS Results showed that patients with anti-NMDAR encephalitis exhibited increased dynamic ALFF (dALFF) variability in the left inferior occipital gyrus compared to healthy controls (HCs), while the patients exhibited decreased sALFF in widespread regions, including the left inferior frontal gyrus, left medial frontal gyrus, bilateral putamen, left medial superior frontal gyrus. dALFF had superior classification performance in distinguishing anti-NMDAR encephalitis patients from HCs over sALFF, but sALFF was correlated with multiple clinical and neuropsychological measures. CONCLUSIONS These findings may shed light on anti-NMDAR encephalitis brain dysfunction from the perspective of dynamic local brain activity. sALFF and dALFF analyses provide complementary information, emphasizing the potential usefulness of combining sALFF and dALFF in elucidating the neuropathological mechanisms of autoimmune encephalitis and may ultimately inform future disease diagnosis.
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Affiliation(s)
- Dongpeng Wu
- Department of Neurology, The First Affiliated Hospital of Anhui Medical University, 218 Jixi Road, Hefei, 230022, Anhui Province, China
| | - Yuanyuan Guo
- Department of Neurology, The First Affiliated Hospital of Anhui Medical University, 218 Jixi Road, Hefei, 230022, Anhui Province, China
| | - Chenglong Li
- Department of Neurology, The First Affiliated Hospital of Anhui Medical University, 218 Jixi Road, Hefei, 230022, Anhui Province, China
| | - Xiaonan Pang
- Department of Neurology, The Second Affiliated Hospital of Anhui Medical University, Hefei, 230601, China
| | - Si Xu
- Department of Neurology, The Second Affiliated Hospital of Anhui Medical University, Hefei, 230601, China
| | - Juanjuan Zhang
- Department of Neurology, The First Affiliated Hospital of Anhui Medical University, 218 Jixi Road, Hefei, 230022, Anhui Province, China
| | - Ling Wei
- Department of Neurology, The First Affiliated Hospital of Anhui Medical University, 218 Jixi Road, Hefei, 230022, Anhui Province, China
| | - Qianqian Li
- Department of Psychology and Sleep Medicine, The Second Affiliated Hospital of Anhui Medical University, Hefei, 230601, China
| | - Jing Du
- Department of Neurology, The Second Affiliated Hospital of Anhui Medical University, Hefei, 230601, China.
| | - Yanghua Tian
- Department of Neurology, The First Affiliated Hospital of Anhui Medical University, 218 Jixi Road, Hefei, 230022, Anhui Province, China.
- Department of Neurology, The Second Affiliated Hospital of Anhui Medical University, Hefei, 230601, China.
- Department of Psychology and Sleep Medicine, The Second Affiliated Hospital of Anhui Medical University, Hefei, 230601, China.
| | - Kai Wang
- Department of Neurology, The First Affiliated Hospital of Anhui Medical University, 218 Jixi Road, Hefei, 230022, Anhui Province, China
- Anhui Province Key Laboratory of Cognition and Neuropsychiatric Disorders, Hefei, 230022, China
- School of Mental Health and Psychological Sciences, Anhui Medical University, Hefei, 230022, China
- Collaborative Innovation Center of Neuropsychiatric Disorders and Mental Health, Hefei, 230022, China
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15
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Liu L, Lyu Z, Li H, Bai L, Wan Y, Li P. Enhancing the clinical diagnosis of the acute and subacute phases of autoimmune encephalitis and predicting the risk factors: the potential advantages of 18F-FDG PET/CT. BMC Med Imaging 2023; 23:193. [PMID: 37986052 PMCID: PMC10662540 DOI: 10.1186/s12880-023-01148-6] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 02/15/2023] [Accepted: 10/31/2023] [Indexed: 11/22/2023] Open
Abstract
BACKGROUND 2-deoxy-2-[18F]fluoro-D-glucose positron emission tomography (18F-FDG PET) could help evaluate metabolic abnormalities by semi-quantitative measurement to identify autoimmune encephalitis (AE). Few studies have been conducted to analyze the prognostic factors of AE. The study aimed to explore the values of diagnosis and treatment evaluation by 18F-FDG PET and preliminarily discussed the potential value in predicting the prognosis of AE patients. METHODS AE patients underwent 18F-FDG PET/CT and magnetic resonance imaging (MRI). There were two steps to analyse 18F-FDG PET imaging data. The first step was visual assessment. The second step was to analyse 18F-FDG PET parameters using Scenium software (Siemens Molecular Imaging Ltd). The mean standardized uptake value (SUVmean) and maximum standardized uptake value (SUVmax) of brain relative regional metabolism (BRRM) were quantified in the case and control groups according to the anatomical automatic labeling (AAL) partition. The main statistical method was the Kruskal-Wallis test. Finally, the simple linear regression method was used to analyse the relationships between 18F-FDG PET parameters and the modified Rankin Scale (mRS) scores before and after treatment. RESULTS The results on 18F-FDG PET showed that visual assessment abnormalities were in the mesial temporal lobe (MTL) (70.8%), (mainly infringing on the hippocampus and amygdala), basal ganglia (62.5%), frontal lobes (37.5%), occipital lobes (29.2%), and parietal lobes (12.5%). The positive rate of abnormalities on 18F-FDG PET was more sensitive than that on MRI (95.5% vs 32.2%, p = 0.001). The number of lesions on PET was positively correlated with the mRS scores before and after treatment, and the correlation before treatment was more significant. Before treatment, the SUVmean of the left occipital lobe was the most remarkable (SUVmean, R2 = 0.082, p > 0.05) factor associated with the mRS score, and the correlation was negative. With regard to prognosis, the SUVmax of the MTL was the most notable (R2 = 0.1471, p > 0.05) factor associated with the mRS score after treatment, and the correlation was positive. CONCLUSIONS 18F-FDG PET could be more sensitive and informative than MRI in the early phases of AE. The common pattern of AE was high MTL metabolism on 18F-FDG PET, which was associated with hypometabolism of the occipital lobe, and the number of lesions on PET before treatment may be significant factors in assessing disease severity. The SUVmax of MTL hypermetabolism may serve as a prognostic biomarker in AE.
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Affiliation(s)
- Lili Liu
- Department of PET/CT, The Second Affiliated Hospital of Harbin Medical University, No.246 Xuefu Road, Harbin, 150001, Heilongjiang, People's Republic of China
| | - Zhehao Lyu
- Department of Nuclear Medicine, The First Affiliated Hospital of Harbin Medical University, Postal Street No.23, Harbin, 150001, Heilongjiang, People's Republic of China
| | - Huimin Li
- Department of Nuclear Medicine, Inner Mongolia Autonomous Region People's Hospital, No.20 Zhaowuda Road, Hohhot, 010017, People's Republic of China
| | - Lin Bai
- Department of PET/CT, The Second Affiliated Hospital of Harbin Medical University, No.246 Xuefu Road, Harbin, 150001, Heilongjiang, People's Republic of China
| | - Yong Wan
- Department of PET/CT, The Second Affiliated Hospital of Harbin Medical University, No.246 Xuefu Road, Harbin, 150001, Heilongjiang, People's Republic of China
| | - Ping Li
- Department of PET/CT, The Second Affiliated Hospital of Harbin Medical University, No.246 Xuefu Road, Harbin, 150001, Heilongjiang, People's Republic of China.
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Yuan L, Mao G, Zhang Y, Xu Y, Chen Q, Shan B, Cui T, Ai L. Typical metabolic pattern of 18F-FDG PET in Anti-NMDAR encephalitis in the acute and subacute phases and its correlation with T2 FLAIR-MRI features. BMC Neurosci 2023; 24:51. [PMID: 37749547 PMCID: PMC10521454 DOI: 10.1186/s12868-023-00823-2] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Grants] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 12/28/2022] [Accepted: 09/19/2023] [Indexed: 09/27/2023] Open
Abstract
BACKGROUND/AIMS Early diagnosis of Anti-N-methyl-D-aspartate receptor (anti-NMDAR) encephalitis with non-invasive imaging modalities benefiting is crucial to guarantee prompt treatments decision-making and good prognosis for patients. The present study aimed to explore the correlation of MRI features with brain metabolism characteristics of 18F-fluorodeoxyglucose positron emission tomography (18F-FDG PET) and to describe the metabolic patterns in Anti-N-methyl-D-aspartate receptor (anti-NMDAR) encephalitis at acute and subacute phases. Twenty-four patients with anti-NMDAR encephalitis confirmed by serum and/or CSF tests at acute and subacute phases, 9 females and 15 males, with an age range of 6-80 years, were enrolled in this retrospective study as encephalitis group. 18F-FDG PET and MRI findings of all patients were investigated and interpreted with visual analysis. Chi-square test was performed to compare the diagnostic sensitivity between MRI and PET. Independent sample t-test was used to compare the standardized uptake value ratio (SUVR) of each ROI between the encephalitis group and control group, which consisted of 24 healthy volunteers of the same age and gender. RESULTS There was no statistical difference in the diagnostic sensitivity between FDG PET (23/24, 95.83%) and MRI (18/24, 75.00%) in anti-NMDAR encephalitis patients (P > 0.05). Three categories of abnormalities shown on T2 FLAIR, including shallow of sulci and swelling of brain tissue, increased signal in the sulci, increased signal on brain gray matter or adjacent white matter presented hypermetabolism on PET, excepting increased signal in brain linear structure with hypometabolism of the basal ganglia on PET. We identified 19 brain regions with hypermetabolism and 16 brain regions with hypometabolism that exhibited statistically significant changes in SUVRs between anti-NMDAR encephalitis group and control group (FDR P < 0.05). CONCLUSION Anteroposterior glucose metabolism gradient (frontal-temporal/parietal-occipital) is proved to be a typical pattern of anti-NMDAR encephalitis at the acute and subacute phases in both visual and statistical testing. Interestingly, the pattern is also commonly found in the anterior and posterior portions of the parietal lobe and cingular cortex, which may be a potential indicator for the diagnosis of this disorder. In addition, MRI is an important and reliable neuroimaging modality to assist in the correct evaluation of activity changes on individual 18F-FDG PET.
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Affiliation(s)
- Leilei Yuan
- Department of Nuclear Medicine, Beijing Tiantan Hospital, Capital Medical University, Beijing, 100070, China
| | - Guangjuan Mao
- Beijing Engineering Research Center of Radiographic Techniques and Equipment, Institute of High Energy Physics, Chinese Academy of Sciences, Beijing, 100049, China
- School of Nuclear Science and Technology, University of Chinese Academy of Sciences, Beijing, 100049, China
| | - Yudi Zhang
- Department of Neurology, The Second Hospital of Hebei Medical University, Shijiazhuang, 050000, China
| | - Yang Xu
- Department of Nuclear Medicine, Beijing Tiantan Hospital, Capital Medical University, Beijing, 100070, China
| | - Qian Chen
- Department of Nuclear Medicine, Beijing Tiantan Hospital, Capital Medical University, Beijing, 100070, China
| | - Baoci Shan
- Beijing Engineering Research Center of Radiographic Techniques and Equipment, Institute of High Energy Physics, Chinese Academy of Sciences, Beijing, 100049, China
- School of Nuclear Science and Technology, University of Chinese Academy of Sciences, Beijing, 100049, China
| | - Tao Cui
- Department of Neurology, Beijing Tiantan Hospital, Capital Medical University, Beijing, 100070, China.
- China National Clinical Research Center for Neurological Diseases, Beijing, 100070, China.
| | - Lin Ai
- Department of Nuclear Medicine, Beijing Tiantan Hospital, Capital Medical University, Beijing, 100070, China.
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Bai S, Zhang C, Yao X, Shao H, Huang G, Liu J, Hao Y, Guan Y. A novel classification model based on cerebral 18F-FDG uptake pattern facilitates the diagnosis of acute/subacute seropositive autoimmune encephalitis. J Neuroradiol 2023; 50:492-501. [PMID: 37142216 DOI: 10.1016/j.neurad.2023.05.001] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 11/14/2022] [Revised: 05/01/2023] [Accepted: 05/01/2023] [Indexed: 05/06/2023]
Abstract
PURPOSE To explore the intrinsic alteration of cerebral 18F-FDG metabolism in acute/subacute seropositive autoimmune encephalitis (AE) and to propose a universal classification model based on 18F-FDG metabolic patterns to predict AE. METHODS Cerebral 18F-FDG PET images of 42 acute/subacute seropositive AE patients and 45 healthy controls (HCs) were compared using voxelwise and region of interest (ROI)-based schemes. The mean standardized uptake value ratios (SUVRs) of 59 subregions according to a modified Automated Anatomical Labeling (AAL) atlas were compared using a t-test. Subjects were randomly divided into a training set (70%) and a testing set (30%). Logistic regression models were built based on the SUVRs and the models were evaluated by determining their predictive value in the training and testing sets. RESULTS The 18F-FDG uptake pattern in the AE group was characterized by increased SUVRs in the brainstem, cerebellum, basal ganglia, and temporal lobe, and decreased SUVRs in the occipital, and frontal regions with voxelwise analysis (false discovery rate [FDR] p<0.05). Utilizing ROI-based analysis, we identified 15 subareas that exhibited statistically significant changes in SUVRs among AE patients compared to HC (FDR p<0.05). Further, a logistic regression model incorporating SUVRs from the calcarine cortex, putamen, supramarginal gyrus, cerebelum_10, and hippocampus successfully enhanced the positive predictive value from 0.76 to 0.86 when compared to visual assessments. This model also demonstrated potent predictive ability, with AUC values of 0.94 and 0.91 observed for the training and testing sets, respectively. CONCLUSIONS During the acute/subacute stages of seropositive AE, alterations in SUVRs appear to be concentrated within physiologically significant regions, ultimately defining the general cerebral metabolic pattern. By incorporating these key regions into a new classification model, we have improved the overall diagnostic efficiency of AE.
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Affiliation(s)
- Shuwei Bai
- Department of Neurology, The Second Affiliated Hospital of Xinjiang Medical University, Urumqi 830063, China; Department of Neurology, Ren Ji Hospital, Shanghai Jiao Tong University School of Medicine, Shanghai, China
| | - Chenpeng Zhang
- Department of Nuclear Medicine, Ren Ji Hospital, Shanghai Jiao Tong University School of Medicine, Shanghai, China
| | - Xiaoying Yao
- Department of Neurology, Ren Ji Hospital, Shanghai Jiao Tong University School of Medicine, Shanghai, China
| | - Hongda Shao
- Department of Nuclear Medicine, Ren Ji Hospital, Shanghai Jiao Tong University School of Medicine, Shanghai, China
| | - Gan Huang
- Department of Nuclear Medicine, Ren Ji Hospital, Shanghai Jiao Tong University School of Medicine, Shanghai, China
| | - Jianjun Liu
- Department of Nuclear Medicine, Ren Ji Hospital, Shanghai Jiao Tong University School of Medicine, Shanghai, China.
| | - Yong Hao
- Department of Neurology, Ren Ji Hospital, Shanghai Jiao Tong University School of Medicine, Shanghai, China.
| | - Yangtai Guan
- Department of Neurology, Ren Ji Hospital, Shanghai Jiao Tong University School of Medicine, Shanghai, China.
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Zhang C, Hao Y, Huang G, Xin M, Bai S, Guan Y, Liu J. Hypometabolism of the left middle/medial frontal lobe on FDG-PET in anti-NMDA receptor encephalitis: Comparison with MRI and EEG findings. CNS Neurosci Ther 2023; 29:1624-1635. [PMID: 36815303 PMCID: PMC10173717 DOI: 10.1111/cns.14125] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 11/13/2022] [Revised: 02/02/2023] [Accepted: 02/05/2023] [Indexed: 02/24/2023] Open
Abstract
OBJECTIVES To investigate changes in brain-glucose metabolism in anti-N-methyl-D-aspartate receptor (NMDAR) encephalitis, and compare results with MRI and electroencephalography (EEG) findings at different disease stages. METHODS The clinical data of 18 patients (median age, 35 years; 11 men) were retrospectively collected. Patients were divided into groups based on the time of symptom onset to examination, (≤1 month, >1 but ≤3 months, >3 months). Two-sample t-test results were compared with age and sex-paired healthy controls using statistical parametric mapping and verified using a NeuroQ software normal database with a discriminating z-score of 2. RESULTS Abnormal patterns on FDG-PET differed over time (T = 3.21-8.74, Z = 2.68-4.23, p < 0.005). Regional analysis showed hypometabolic left middle or medial frontal cortex in 4/5, 5/7, and 5/6 patients, respectively. Time-subgroup analysis revealed hypermetabolic supertemporal cortex in 4/5, 5/7, and 2/6, patients, respectively. MRI and EEG abnormalities in any region and stage occurred in 10/18 and 10/16 patients, respectively. MRI and EEG time-subgroup analysis showed abnormalities in 5/9, 4/5, and 1/4, and 1/3, 6/7, and 3/6 patients, respectively. Abnormal temporal lobes were detected most frequently in MRI analyses and occurred in 3/10 patients. CONCLUSIONS Decreased left middle/medial frontal metabolism could be common to all stages. Metabolism in other regions, MRI, and EEG results were associated with the progression of anti-NMDAR encephalitis. The sensitivity rate of FDG-PET was superior to that of MRI and EEG.
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Affiliation(s)
- Chenpeng Zhang
- Department of Nuclear Medicine, Ren Ji Hospital, Shanghai Jiao Tong University School of Medicine, Shanghai, China
| | - Yong Hao
- Department of Neurology, Ren Ji Hospital, Shanghai Jiao Tong University School of Medicine, Shanghai, China
| | - Gan Huang
- Department of Nuclear Medicine, Ren Ji Hospital, Shanghai Jiao Tong University School of Medicine, Shanghai, China
| | - Mei Xin
- Department of Nuclear Medicine, Ren Ji Hospital, Shanghai Jiao Tong University School of Medicine, Shanghai, China
| | - Shuwei Bai
- Department of Neurology, Ren Ji Hospital, Shanghai Jiao Tong University School of Medicine, Shanghai, China
| | - Yangtai Guan
- Department of Neurology, Ren Ji Hospital, Shanghai Jiao Tong University School of Medicine, Shanghai, China
| | - Jianjun Liu
- Department of Nuclear Medicine, Ren Ji Hospital, Shanghai Jiao Tong University School of Medicine, Shanghai, China
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Wada T, Mori H, Shindo K. Serial assessment of multimodality imaging in anti-leucine-rich glioma-inactivated 1 antibody encephalitis: A case report. eNeurologicalSci 2022; 29:100426. [PMID: 36161067 PMCID: PMC9494171 DOI: 10.1016/j.ensci.2022.100426] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 06/13/2022] [Revised: 07/29/2022] [Accepted: 09/10/2022] [Indexed: 11/29/2022] Open
Abstract
In autoimmune encephalitis, abnormalities of diffusion-weighted imaging (DWI), fluid-attenuated inversion recovery (FLAIR), arterial spin labeling (ASL) in magnetic resonance imaging (MRI), single-photon emission computed tomography (SPECT) and 18F-fluorodeoxyglucose-positron emission tomography (18F-FDG-PET) have been reported. However, there are few studies of long-term follow-up of imaging. We report a case of anti-leucine-rich glioma-inactivated 1 antibody encephalitis whose MRI (DWI, FLAIR and ASL), 99mTcHM-PAO SPECT (PAO-SPECT) and 18F-FDG-PET were evaluated through the clinical course. ASL, PAO-SPECT and 18F-FDG-PET consistently showed abnormalities in almost the same area. Serial assessment of these imaging modalities is useful in evaluating disease activity and efficacy of treatment.
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Affiliation(s)
- Takafumi Wada
- Department of Neurology, Kurashiki Central Hospital, 1-1-1 Miwa, Kurashiki/Okayama, 710-8602, Japan
| | - Hitoshi Mori
- Department of Neurology, Kurashiki Central Hospital, 1-1-1 Miwa, Kurashiki/Okayama, 710-8602, Japan
| | - Katsuro Shindo
- Department of Neurology, Kurashiki Central Hospital, 1-1-1 Miwa, Kurashiki/Okayama, 710-8602, Japan
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20
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Jha S, Nagaraj C, Mundlamuri RC, Alladi S, Nashi S, Kenchaiah R, Mahadevan A, Bhat M, Saini J, Netravathi M. FDG-PET in Autoimmune Encephalitis: Utility, Pattern of Abnormalities, and Correlation with Autoantibodies. Ann Indian Acad Neurol 2022; 25:1122-1129. [PMID: 36911487 PMCID: PMC9996532 DOI: 10.4103/aian.aian_645_22] [Citation(s) in RCA: 4] [Impact Index Per Article: 1.3] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 07/27/2022] [Revised: 10/04/2022] [Accepted: 10/14/2022] [Indexed: 03/14/2023] Open
Abstract
Background Fluorodeoxyglucose-positron emission tomography (FDG-PET) in autoimmune encephalitis (AE) as an adjunctive investigation helps in characterizing the type of AE based on characteristic metabolic patterns. Objectives We aimed to study the following: (i) the sensitivity of FDG-PET in the diagnosis of AE, (ii) describe abnormal patterns of metabolism of various subtypes of AE, and (iii) correlate serum serology with FDG-PET abnormalities. Materials and Methods This study was conducted at a tertiary university hospital in South India. The demographic profile, clinical features, and investigations (FDG-PET, magnetic resonance imaging (MRI) brain, electroencephalography (EEG), cerebrospinal fluid (CSF)) were reviewed. The nuclear medicine physician performed blinded qualitative visual and semi-quantitative analysis of the 18-FDG-PET (fluorine 18-FDG-PET) findings of these patients. Results Twenty-nine (M:F: 11:18) patients were recruited; among them, 22 (75.8%) patients had autoimmune antibodies; the rest seven (24.1%) patients were seronegative. Among the 22 seropositive patients, 9 (31%) patients were positive for anti-N-methyl-D-aspartate receptor (NMDAR), 8 (28%) for anti-leucine-rich glioma inactivated 1 (LGI-1), 4 (14%) for anti-contactin-associated protein 2 (CASPR2), 1 (3%) for anti-glutamic acid decarboxylase (GAD)-65, and rest 7 (24%) patients were seronegative. The patterns most commonly observed were isolated hypermetabolism (41%), isolated hypometabolism (41%), and combined hypermetabolism with hypometabolism (18%). The fraction of abnormalities was lower for MRI (17/22; 73.9%) than for FDG-PET (27/29; 93.1%). FDG-PET correlated with serology in 10 (34%) cases [NMDAR: 6 (60%) and LGI-1: 4 (40%)]. The sensitivity of FDG-PET was 94.1% when compared with MRI. Discussion and Conclusion FDG-PET correlated with serology in only one-third of patients. The most consistent pattern in both seropositive and seronegative AE is characterized by parieto-occipital hypometabolism and fronto-temporal with basal ganglia hypermetabolism.
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Affiliation(s)
- Shreyashi Jha
- Department of Neurology, National Institute of Mental Health and Neurosciences (NIMHANS), Bengaluru, Karnataka, India
| | - Chandana Nagaraj
- Department of Neuroimaging and Interventional Neuroradiology (NIIR), National Institute of Mental Health and Neurosciences (NIMHANS), Bengaluru, Karnataka, India
| | - R. C. Mundlamuri
- Department of Neurology, National Institute of Mental Health and Neurosciences (NIMHANS), Bengaluru, Karnataka, India
| | - Suvarna Alladi
- Department of Neurology, National Institute of Mental Health and Neurosciences (NIMHANS), Bengaluru, Karnataka, India
| | - Saraswati Nashi
- Department of Neurology, National Institute of Mental Health and Neurosciences (NIMHANS), Bengaluru, Karnataka, India
| | - Raghavendra Kenchaiah
- Department of Neurology, National Institute of Mental Health and Neurosciences (NIMHANS), Bengaluru, Karnataka, India
| | - Anita Mahadevan
- Department of Neuropathology, National Institute of Mental Health and Neurosciences (NIMHANS), Bengaluru, Karnataka, India
| | - Maya Bhat
- Department of Neuroimaging and Interventional Neuroradiology (NIIR), National Institute of Mental Health and Neurosciences (NIMHANS), Bengaluru, Karnataka, India
| | - Jitender Saini
- Department of Neuroimaging and Interventional Neuroradiology (NIIR), National Institute of Mental Health and Neurosciences (NIMHANS), Bengaluru, Karnataka, India
| | - M. Netravathi
- Department of Neurology, National Institute of Mental Health and Neurosciences (NIMHANS), Bengaluru, Karnataka, India
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21
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Li G, Liu X, Yu T, Ren J, Wang Q. Positron emission tomography in autoimmune encephalitis: Clinical implications and future directions. Acta Neurol Scand 2022; 146:708-715. [PMID: 36259555 DOI: 10.1111/ane.13717] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 07/26/2022] [Revised: 10/04/2022] [Accepted: 10/08/2022] [Indexed: 11/28/2022]
Abstract
18 F-fluoro-deoxyglucose position emission tomography (18 F-FDG-PET) has been proven as a sensitive and reliable tool for diagnosis of autoimmune encephalitis (AE). More attention was paid to this kind of imaging because of the shortage of MRI, EEG, and CSF findings. FDG-PET has been assessed in a few small studies and case reports showing apparent abnormalities in cases where MRI does not. Here, we summarized the patterns (specific or not) in AE with different antibodies detected and the clinical outlook for the wide application of FDG-PET considering some limitations. Specific patterns based on antibody subtypes and clinical symptoms were critical for identifying suspicious AE, the most common of which was the anteroposterior gradient in anti- N -methyl- d -aspartate receptor (NMDAR) encephalitis and the medial temporal lobe hypermetabolism in limbic encephalitis. And the dynamic changes of metabolic presentations in different phases provided us the potential to inspect the evolution of AE and predict the functional outcomes. Except for the visual assessment, quantitative analysis was recently reported in some voxel-based studies of regions of interest, which suggested some clues of the future evaluation of metabolic abnormalities. Large prospective studies need to be conducted controlling the time from symptom onset to examination with the same standard of FDG-PET scanning.
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Affiliation(s)
- Gongfei Li
- Department of Neurology, Beijing Tiantan Hospital, Capital Medical University, Beijing, China.,China National Clinical Research Center for Neurological Diseases, Beijing, China
| | - Xiao Liu
- Department of Neurology, Beijing Tiantan Hospital, Capital Medical University, Beijing, China.,China National Clinical Research Center for Neurological Diseases, Beijing, China
| | - Tingting Yu
- Department of Neurology, Beijing Tiantan Hospital, Capital Medical University, Beijing, China.,China National Clinical Research Center for Neurological Diseases, Beijing, China
| | - Jiechuan Ren
- Department of Neurology, Beijing Tiantan Hospital, Capital Medical University, Beijing, China.,China National Clinical Research Center for Neurological Diseases, Beijing, China
| | - Qun Wang
- Department of Neurology, Beijing Tiantan Hospital, Capital Medical University, Beijing, China.,China National Clinical Research Center for Neurological Diseases, Beijing, China.,Beijing Institute for Brain Disorders, Collaborative Innovation Center for Brain Disorders, Capital Medical University, Beijing, China
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22
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Hu CC, Pan XL, Zhang MX, Chen HF. Paroxysmal speech disorder as the initial symptom in a young adult with anti-N-methyl-D-aspartate receptor encephalitis: A case report. World J Clin Cases 2022; 10:8648-8655. [PMID: 36157799 PMCID: PMC9453376 DOI: 10.12998/wjcc.v10.i24.8648] [Citation(s) in RCA: 2] [Impact Index Per Article: 0.7] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 01/19/2022] [Revised: 04/22/2022] [Accepted: 07/22/2022] [Indexed: 02/05/2023] Open
Abstract
BACKGROUND Anti-N-methyl-D-aspartate receptor (anti-NMDAR) encephalitis is a treatable but frequently misdiagnosed autoimmune disease. Speech dysfunction, as one of the common manifestations of anti-NMDAR encephalitis, is usually reported as a symptom secondary to psychiatric symptoms or seizures rather than the initial symptom in a paroxysmal form. We report a case of anti-NMDAR encephalitis with paroxysmal speech disorder as a rare initial manifestation, and hope that it will contribute to the literature.
CASE SUMMARY A 39-year-old man with anti-NMDAR encephalitis initially presented with paroxysmal nonfluent aphasia and was misdiagnosed with a transient ischemic attack and cerebral infarction successively. The patient subsequently presented with seizures, but no abnormalities were found on brain magnetic resonance imaging or electroencephalogram. Cerebrospinal fluid (CSF) analysis revealed mild pleocytosis and increased protein levels. Anti-NMDAR antibodies in serum and CSF were detected for a conclusive diagnosis. After immunotherapy, the patient made a full recovery.
CONCLUSION This case suggests that paroxysmal speech disorder may be the presenting symptom of anti-NMDAR encephalitis in a young patient.
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Affiliation(s)
- Chuan-Chen Hu
- Department of Neurology, The Affiliated Jinhua Hospital, School of Medicine, Zhejiang University, Jinhua 321000, Zhejiang Province, China
| | - Xiao-Ling Pan
- Department of Neurology, The Affiliated Jinhua Hospital, School of Medicine, Zhejiang University, Jinhua 321000, Zhejiang Province, China
| | - Mei-Xia Zhang
- Department of Neurology, The Affiliated Jinhua Hospital, School of Medicine, Zhejiang University, Jinhua 321000, Zhejiang Province, China
| | - Hong-Fang Chen
- Department of Neurology, The Affiliated Jinhua Hospital, School of Medicine, Zhejiang University, Jinhua 321000, Zhejiang Province, China
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23
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Massa F, Franciotta D, Grisanti S, Roccatagliata L, Morbelli S, Beltramini S, Uccelli A, Schenone A, Benedetti L. Intravenous immunoglobulin bridging to rituximab in NMDAR encephalitis patients non-responders to first-line treatments. Neurol Sci 2022; 43:6441-6447. [PMID: 35953578 PMCID: PMC9616745 DOI: 10.1007/s10072-022-06313-3] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 01/31/2022] [Accepted: 08/02/2022] [Indexed: 11/25/2022]
Abstract
Background The immunotherapy strategy for autoimmune encephalitis is based on several types and schedules of both first- and second-line drugs. Failing to respond to the latter prompts the use of non-conventional rescue therapies, with higher risks of severe adverse effects. We report on a protocol that entails the use of intravenous immunoglobulin cycles to bridge the 4-month period that the second-line drug rituximab needs to exert its full therapeutic effects. Methods Three patients with NMDAR encephalitis who were non-responders to first-line treatments entered the study. The protocol consisted of six monthly cycles of intravenous immunoglobulins (IVIG, 0.4 mg/kg/die for 5 days), starting 1 month after the last rituximab infusion (1000 mg at days 0 and 15). Brain MRI and [18F]-FDG-PET were performed at onset and at six and 18 months after onset. Results In the three patients, substantial improvements of disability or complete recovery were achieved, without modifications over the 30-to-50-month follow-up. No adverse events nor laboratory test abnormalities were recorded. Imaging findings paralleled the favorable disease courses. Brain [18F]-FDG-PET was more sensitive than MRI in detecting abnormalities. Discussion Our observations suggest that the herein-described protocol might be used in patients with NMDAR encephalitis at risk for poor prognosis in the mid-term when they need to shift to rituximab. [18F]-FDG-PET confirmed to be a sensitive tool to detect the minimal brain lesions that can underlie isolated cognitive and psychiatric symptoms.
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24
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Long Q, Lv Z, Zhao J, Shi K, Li C, Fan B, Zheng J. Cerebral gray matter volume changes in patients with anti-N-methyl-D-aspartate receptor encephalitis: A voxel-based morphometry study. Front Neurol 2022; 13:892242. [PMID: 35959389 PMCID: PMC9358280 DOI: 10.3389/fneur.2022.892242] [Citation(s) in RCA: 6] [Impact Index Per Article: 2.0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 03/08/2022] [Accepted: 07/04/2022] [Indexed: 01/19/2023] Open
Abstract
Background Anti-N-methyl-D-aspartate receptor (NMDAR) encephalitis is an autoimmune disease with typical clinical features. Whether and how cerebral gray matter structural damage inherent to the disorder affects cognitive function in patients is still unclear. Therefore, this study aimed to explore the changes in cerebral gray matter volume and whether these alterations contribute to cognitive impairment and mood disorders. Methods Forty patients with anti-NMDAR encephalitis and forty healthy controls (HCs) matched for gender, age, and education were recruited. All participants underwent attention network tests (ANT), neuropsychological tests and magnetic resonance imaging (MRI). Voxel-based morphological analysis (VBM) and correlation analysis was performed on all participants. Finally, according to the course of disease, patients were divided into two groups: NMDARE_SD (short duration; course ≤ 2 years since diagnosis) and NMDARE_LD (long duration; course >2 years since diagnosis), to evaluate gray matter volume changes that differ as a function of disease course. Results Compared to HCs, patients with anti-NMDAR encephalitis showed decreased executive control ability and lower MoCA score, while increased anxiety and depression as reflected by HAMA and HAMD24 scores (all P < 0.05). In VBM analysis, patients showed decreased gray matter volume in bilateral thalamus, left medial prefrontal cortex (mPFC_L), left superior temporal gyrus (STG_L), and left rectus gyrus. In the analysis stratified by disease course, the NMDARE_LD group exhibited decreased gray matter volume in the left precuneus and right posterior cerebellar lobe compared to the NMDARE_SD group. Conclusions Patients with anti-NMDAR encephalitis have cognitive, executive, and emotional dysfunction, and the sites of gray matter atrophy are concentrated in the thalamus, frontal lobe, and temporal lobe. These abnormalities may be involved in the process of cognitive and affective dysfunction.Patients with different courses of anti-NMDAR encephalitis have different brain atrophy sites. These results may help to clarify the contradiction between clinical and imaging manifestations of anti NMDAR encephalitis, which is worthy of further longitudinal studies.
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Affiliation(s)
| | | | | | | | | | | | - Jinou Zheng
- Department of Neurology, The First Affiliated Hospital of Guangxi Medical University, Nanning, China
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25
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Arbizu J, Gállego Pérez-Larraya J, Hilario A, Gómez Grande A, Rubí S, Camacho V. Actualización en el diagnóstico de la encefalitis. Rev Esp Med Nucl Imagen Mol 2022. [PMID: 35701317 DOI: 10.1016/j.remn.2022.05.001] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/30/2022]
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26
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Arbizu J, Gállego Pérez-Larraya J, Hilario A, Gómez Grande A, Rubí S, Camacho V. Update on the diagnosis of encephalitis. Rev Esp Med Nucl Imagen Mol 2022; 41:247-257. [DOI: 10.1016/j.remnie.2022.05.003] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 05/09/2022] [Accepted: 05/10/2022] [Indexed: 11/25/2022]
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Liang CW, Chen TC. Anti-NMDA receptor encephalitis presenting as fever with undetermined cause. Int J Infect Dis 2022; 122:365-367. [PMID: 35718296 DOI: 10.1016/j.ijid.2022.06.019] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 02/25/2022] [Revised: 06/10/2022] [Accepted: 06/13/2022] [Indexed: 10/18/2022] Open
Abstract
Autoimmune encephalitis is a rare disease which should be differentiated with aseptic encephalitis. Possessing anti-N-methyl-D-aspartate (NMDA) receptor autoantibody is the leading cause of autoimmune encephalitis. However, it may pose a diagnostic challenge to clinicians, especially non-psychiatric or non-neurologic specialist, resulting in a delayed initiation of treatment. Hence, we shared a case of anti-NMDA receptor encephalitis that got hospitalized at infectious diseases ward with the presentation of acute febrile illness that preceded characteristic neuropsychiatric symptoms.
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Affiliation(s)
- Chih-Wei Liang
- Department of Internal Medicine, Kaohsiung Medical University Hospital, Kaohsiung Medical University, Kaohsiung, Taiwan
| | - Tun-Chieh Chen
- Department of Internal Medicine and Infection Control Office, Kaohsiung Municipal Ta-Tung Hospital, Kaohsiung Medical University, Kaohsiung, Taiwan; Division of Infectious Diseases, Department of Internal Medicine, Kaohsiung Medical University Hospital, Kaohsiung Medical University, Kaohsiung, Taiwan; School of Medicine, College of Medicine, Kaohsiung Medical University, Kaohsiung, Taiwan.
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28
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Guo Y, Lv X, Wei Q, Wu Y, Chen Y, Ji Y, Hou Q, Lv H, Zhou N, Wang K, Tian Y. Impaired neurovascular coupling and cognitive deficits in anti-N-methyl-D-aspartate receptor encephalitis. Brain Imaging Behav 2022; 16:1065-1076. [PMID: 34735667 DOI: 10.1007/s11682-021-00588-4] [Citation(s) in RCA: 12] [Impact Index Per Article: 4.0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Grants] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 01/05/2021] [Accepted: 10/17/2021] [Indexed: 10/19/2022]
Abstract
Anti-N-methyl-D-aspartate receptor (anti-NMDAR) encephalitis is a recently identified autoimmune disorder with heterogeneous neurological, psychiatric, and cognitive manifestations. The NMDAR is a key signaling node for neurovascular coupling, the mechanism by which cerebral blood perfusion is enhanced to meet local metabolic requirements from increased neuronal activity. Therefore, anti-NMDAR encephalitis may disrupt neurovascular coupling and induce cognitive deficits. This study examined neurovascular coupling and cognitive function in anti-NMDAR encephalitis patients to identify prognostic biomarkers, reveal potential pathogenic mechanisms, and provide clues to possible therapeutic strategies. In this study, twenty-three anti-NMDAR encephalitis patients and thirty healthy controls received neuropsychological testing and multimodal magnetic resonance imaging (MRI). Cerebral blood flow (CBF) was calculated from arterial spin labeling, and regional homogeneity (ReHo) was computed from functional MRI. Pearson's correlation coefficients between CBF and ReHo were calculated to obtain neurovascular coupling. At the whole gray matter level, CBF‒ReHo coupling was reduced in patients compared to healthy controls. At the regional level, CBF‒ReHo was significantly lower among patients in the precentral gyrus, frontal gyrus, insula, cuneus, inferior parietal lobe, supramarginal gyrus, angular gyrus, precuneus, temporal gyrus, and temporal pole. Reduced CBF‒ReHo in the left superior medial frontal gyrus of patients was significantly correlated with a deficit in verbal inhibition control, and the reduced CBF‒ReHo in the left insula was significantly correlated with impaired executive function. In conclusion, anti-NMDAR encephalitis is associated with both global and regional disruptions in neurovascular coupling that may in turn lead to deficits in specific cognitive domains.
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Affiliation(s)
- Yuanyuan Guo
- Department of Neurology, The First Affiliated Hospital of Anhui Medical University, 218 Jixi Road, Anhui Province, Hefei, 230022, China
| | - Xinyi Lv
- Department of Neurology, The First Affiliated Hospital of USTC, Division of Life Sciences and Medicine, University of Science and Technology of China, Hefei, Anhui, 230001, China
| | - Qiang Wei
- Department of Neurology, The First Affiliated Hospital of Anhui Medical University, 218 Jixi Road, Anhui Province, Hefei, 230022, China
- Anhui Province Key Laboratory of Cognition and Neuropsychiatric Disorders, 230022, Hefei, China
| | - Yue Wu
- Department of Neurology, The First Affiliated Hospital of Anhui Medical University, 218 Jixi Road, Anhui Province, Hefei, 230022, China
| | - Yue Chen
- Department of Neurology, The First Affiliated Hospital of Anhui Medical University, 218 Jixi Road, Anhui Province, Hefei, 230022, China
| | - Yang Ji
- Department of Neurology, The First Affiliated Hospital of Anhui Medical University, 218 Jixi Road, Anhui Province, Hefei, 230022, China
| | - Qiangqiang Hou
- Department of Neurology, The First Affiliated Hospital of Anhui Medical University, 218 Jixi Road, Anhui Province, Hefei, 230022, China
| | - Huaming Lv
- Department of Neurology, The First Affiliated Hospital of Anhui Medical University, 218 Jixi Road, Anhui Province, Hefei, 230022, China
| | - Nong Zhou
- Department of Neurology, The First Affiliated Hospital of Anhui Medical University, 218 Jixi Road, Anhui Province, Hefei, 230022, China
| | - Kai Wang
- Department of Neurology, The First Affiliated Hospital of Anhui Medical University, 218 Jixi Road, Anhui Province, Hefei, 230022, China.
- Anhui Province Key Laboratory of Cognition and Neuropsychiatric Disorders, 230022, Hefei, China.
- Institute of Artificial Intelligence, Hefei Comprehensive National Science Center, 230022, Hefei, China.
- School of Mental Health and Psychological Sciences, Anhui Medical University, 230022, Hefei, China.
- Collaborative Innovation Center of Neuropsychiatric Disorders and Mental Health, 230022, Hefei, China.
| | - Yanghua Tian
- Department of Neurology, The First Affiliated Hospital of Anhui Medical University, 218 Jixi Road, Anhui Province, Hefei, 230022, China.
- Anhui Province Key Laboratory of Cognition and Neuropsychiatric Disorders, 230022, Hefei, China.
- Institute of Artificial Intelligence, Hefei Comprehensive National Science Center, 230022, Hefei, China.
- School of Mental Health and Psychological Sciences, Anhui Medical University, 230022, Hefei, China.
- Collaborative Innovation Center of Neuropsychiatric Disorders and Mental Health, 230022, Hefei, China.
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Simabukuro MM, da Silva GD, Castro LHM, Lucato LT. A critical review and update on autoimmune encephalitis: understanding the alphabet soup. ARQUIVOS DE NEURO-PSIQUIATRIA 2022; 80:143-158. [PMID: 35976312 PMCID: PMC9491421 DOI: 10.1590/0004-282x-anp-2022-s122] [Citation(s) in RCA: 4] [Impact Index Per Article: 1.3] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Subscribe] [Scholar Register] [Received: 03/30/2022] [Accepted: 04/29/2022] [Indexed: 05/24/2023]
Abstract
Autoimmune encephalitis (AE) comprises a group of diseases mediated by antibodies against neuronal cell surface or synaptic antigens, such as ion channels or neurotransmitter receptors. New clinical syndromes and their associated antibodies were and are still being characterized over the last two decades. The fact that their main clinical features are interdisciplinary, - encompassing neuropsychiatric symptoms, cognitive dysfunction, epileptic seizures, movement and sleep disorders - has led to a surge of interest in this field. Some of these diseases present with a well-defined syndrome, being recognizable on clinical grounds. Correct diagnosis is important since AE are potentially treatable diseases, despite their severity. On the other hand, an increasing number of neuronal antibodies being described casts doubt upon the way we should utilize antibody testing and interpret results. In this article we review, summarize and update the current knowledge on antibody mediated encephalitis.
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Affiliation(s)
| | | | | | - Leandro Tavares Lucato
- Universidade de São Paulo, Faculdade de Medicina, Instituto de Radiologia São Paulo, SP, Brazil
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Alves IS, Coutinho AMN, Vieira APF, Rocha BP, Passos UL, Gonçalves VT, Silva PDS, Zhan MX, Pinho PC, Delgado DS, Docema MFL, Lee HW, Policeni BA, Leite CC, Martin MGM, Amancio CT. Imaging Aspects of the Hippocampus. Radiographics 2022; 42:822-840. [PMID: 35213261 DOI: 10.1148/rg.210153] [Citation(s) in RCA: 6] [Impact Index Per Article: 2.0] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/11/2022]
Abstract
The hippocampus is one of the most sophisticated structures in the brain, owing to its complex anatomy, intriguing functions, relationship with other structures, and relevant associated symptoms. Despite being a structure analyzed for centuries, its anatomy and physiology in the human body are still being extensively studied, as well as associated pathologic conditions and potential biomarkers. It can be affected by a broad group of diseases that can be classified as congenital, degenerative, infectious or inflammatory, neoplastic, vascular, or toxic-metabolic disease. The authors present the anatomy and close structures, function, and development of the hippocampus, as well as an original algorithm for imaging diagnosis. The algorithm includes pathologic conditions that typically affect the hippocampus and groups them into nodular (space occupying) and nonnodular pathologic conditions, serving as a guide to narrow the differential diagnosis. MRI is the imaging modality of choice for evaluation of the hippocampus, and CT and nuclear medicine also improve the analysis. The MRI differential diagnosis depends on anatomic recognition and careful characterization of associated imaging findings such as volumetric changes, diffusion restriction, cystic appearance, hyperintensity at T1-weighted imaging, enhancement, or calcification, which play a central role in diagnosis along with clinical findings. Some pathologic conditions arising from surrounding structures such as the amygdala are also important to recognize. Pathologic conditions of the hippocampus can be a challenge to diagnose because they usually manifest as similar clinical syndromes, so the imaging findings play a potential role in guiding the final diagnosis. Online supplemental material is available for this article. ©RSNA, 2022.
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Affiliation(s)
- Isabela S Alves
- From the Neuroradiology Section, Department of Radiology, Hospital Sírio-Libanês, Adma Jafet 91, Bela Vista, São Paulo SP 01308-050, Brazil (I.S.A., A.M.N.C., A.P.F.V., B.P.R., U.L.P., V.T.G., P.C.P., D.S.D., M.F.L.D., H.W.L., M.G.M.M., C.T.A.); Neuroradiology Section, Department of Radiology, University of São Paulo, Brazil (A.M.N.C., P.C.P., C.C.L., M.G.M.M.); Department of Neurology, Prevent Senior, São Paulo, Brazil (P.D.S.S.); and Neuroradiology Section, Department of Radiology, University of Iowa, Iowa City, Iowa (M.X.Z., B.A.P.)
| | - Artur M N Coutinho
- From the Neuroradiology Section, Department of Radiology, Hospital Sírio-Libanês, Adma Jafet 91, Bela Vista, São Paulo SP 01308-050, Brazil (I.S.A., A.M.N.C., A.P.F.V., B.P.R., U.L.P., V.T.G., P.C.P., D.S.D., M.F.L.D., H.W.L., M.G.M.M., C.T.A.); Neuroradiology Section, Department of Radiology, University of São Paulo, Brazil (A.M.N.C., P.C.P., C.C.L., M.G.M.M.); Department of Neurology, Prevent Senior, São Paulo, Brazil (P.D.S.S.); and Neuroradiology Section, Department of Radiology, University of Iowa, Iowa City, Iowa (M.X.Z., B.A.P.)
| | - Ana P F Vieira
- From the Neuroradiology Section, Department of Radiology, Hospital Sírio-Libanês, Adma Jafet 91, Bela Vista, São Paulo SP 01308-050, Brazil (I.S.A., A.M.N.C., A.P.F.V., B.P.R., U.L.P., V.T.G., P.C.P., D.S.D., M.F.L.D., H.W.L., M.G.M.M., C.T.A.); Neuroradiology Section, Department of Radiology, University of São Paulo, Brazil (A.M.N.C., P.C.P., C.C.L., M.G.M.M.); Department of Neurology, Prevent Senior, São Paulo, Brazil (P.D.S.S.); and Neuroradiology Section, Department of Radiology, University of Iowa, Iowa City, Iowa (M.X.Z., B.A.P.)
| | - Bruno P Rocha
- From the Neuroradiology Section, Department of Radiology, Hospital Sírio-Libanês, Adma Jafet 91, Bela Vista, São Paulo SP 01308-050, Brazil (I.S.A., A.M.N.C., A.P.F.V., B.P.R., U.L.P., V.T.G., P.C.P., D.S.D., M.F.L.D., H.W.L., M.G.M.M., C.T.A.); Neuroradiology Section, Department of Radiology, University of São Paulo, Brazil (A.M.N.C., P.C.P., C.C.L., M.G.M.M.); Department of Neurology, Prevent Senior, São Paulo, Brazil (P.D.S.S.); and Neuroradiology Section, Department of Radiology, University of Iowa, Iowa City, Iowa (M.X.Z., B.A.P.)
| | - Ula L Passos
- From the Neuroradiology Section, Department of Radiology, Hospital Sírio-Libanês, Adma Jafet 91, Bela Vista, São Paulo SP 01308-050, Brazil (I.S.A., A.M.N.C., A.P.F.V., B.P.R., U.L.P., V.T.G., P.C.P., D.S.D., M.F.L.D., H.W.L., M.G.M.M., C.T.A.); Neuroradiology Section, Department of Radiology, University of São Paulo, Brazil (A.M.N.C., P.C.P., C.C.L., M.G.M.M.); Department of Neurology, Prevent Senior, São Paulo, Brazil (P.D.S.S.); and Neuroradiology Section, Department of Radiology, University of Iowa, Iowa City, Iowa (M.X.Z., B.A.P.)
| | - Vinicius T Gonçalves
- From the Neuroradiology Section, Department of Radiology, Hospital Sírio-Libanês, Adma Jafet 91, Bela Vista, São Paulo SP 01308-050, Brazil (I.S.A., A.M.N.C., A.P.F.V., B.P.R., U.L.P., V.T.G., P.C.P., D.S.D., M.F.L.D., H.W.L., M.G.M.M., C.T.A.); Neuroradiology Section, Department of Radiology, University of São Paulo, Brazil (A.M.N.C., P.C.P., C.C.L., M.G.M.M.); Department of Neurology, Prevent Senior, São Paulo, Brazil (P.D.S.S.); and Neuroradiology Section, Department of Radiology, University of Iowa, Iowa City, Iowa (M.X.Z., B.A.P.)
| | - Paulo D S Silva
- From the Neuroradiology Section, Department of Radiology, Hospital Sírio-Libanês, Adma Jafet 91, Bela Vista, São Paulo SP 01308-050, Brazil (I.S.A., A.M.N.C., A.P.F.V., B.P.R., U.L.P., V.T.G., P.C.P., D.S.D., M.F.L.D., H.W.L., M.G.M.M., C.T.A.); Neuroradiology Section, Department of Radiology, University of São Paulo, Brazil (A.M.N.C., P.C.P., C.C.L., M.G.M.M.); Department of Neurology, Prevent Senior, São Paulo, Brazil (P.D.S.S.); and Neuroradiology Section, Department of Radiology, University of Iowa, Iowa City, Iowa (M.X.Z., B.A.P.)
| | - Malia X Zhan
- From the Neuroradiology Section, Department of Radiology, Hospital Sírio-Libanês, Adma Jafet 91, Bela Vista, São Paulo SP 01308-050, Brazil (I.S.A., A.M.N.C., A.P.F.V., B.P.R., U.L.P., V.T.G., P.C.P., D.S.D., M.F.L.D., H.W.L., M.G.M.M., C.T.A.); Neuroradiology Section, Department of Radiology, University of São Paulo, Brazil (A.M.N.C., P.C.P., C.C.L., M.G.M.M.); Department of Neurology, Prevent Senior, São Paulo, Brazil (P.D.S.S.); and Neuroradiology Section, Department of Radiology, University of Iowa, Iowa City, Iowa (M.X.Z., B.A.P.)
| | - Paula C Pinho
- From the Neuroradiology Section, Department of Radiology, Hospital Sírio-Libanês, Adma Jafet 91, Bela Vista, São Paulo SP 01308-050, Brazil (I.S.A., A.M.N.C., A.P.F.V., B.P.R., U.L.P., V.T.G., P.C.P., D.S.D., M.F.L.D., H.W.L., M.G.M.M., C.T.A.); Neuroradiology Section, Department of Radiology, University of São Paulo, Brazil (A.M.N.C., P.C.P., C.C.L., M.G.M.M.); Department of Neurology, Prevent Senior, São Paulo, Brazil (P.D.S.S.); and Neuroradiology Section, Department of Radiology, University of Iowa, Iowa City, Iowa (M.X.Z., B.A.P.)
| | - Daniel S Delgado
- From the Neuroradiology Section, Department of Radiology, Hospital Sírio-Libanês, Adma Jafet 91, Bela Vista, São Paulo SP 01308-050, Brazil (I.S.A., A.M.N.C., A.P.F.V., B.P.R., U.L.P., V.T.G., P.C.P., D.S.D., M.F.L.D., H.W.L., M.G.M.M., C.T.A.); Neuroradiology Section, Department of Radiology, University of São Paulo, Brazil (A.M.N.C., P.C.P., C.C.L., M.G.M.M.); Department of Neurology, Prevent Senior, São Paulo, Brazil (P.D.S.S.); and Neuroradiology Section, Department of Radiology, University of Iowa, Iowa City, Iowa (M.X.Z., B.A.P.)
| | - Marcos F L Docema
- From the Neuroradiology Section, Department of Radiology, Hospital Sírio-Libanês, Adma Jafet 91, Bela Vista, São Paulo SP 01308-050, Brazil (I.S.A., A.M.N.C., A.P.F.V., B.P.R., U.L.P., V.T.G., P.C.P., D.S.D., M.F.L.D., H.W.L., M.G.M.M., C.T.A.); Neuroradiology Section, Department of Radiology, University of São Paulo, Brazil (A.M.N.C., P.C.P., C.C.L., M.G.M.M.); Department of Neurology, Prevent Senior, São Paulo, Brazil (P.D.S.S.); and Neuroradiology Section, Department of Radiology, University of Iowa, Iowa City, Iowa (M.X.Z., B.A.P.)
| | - Hae W Lee
- From the Neuroradiology Section, Department of Radiology, Hospital Sírio-Libanês, Adma Jafet 91, Bela Vista, São Paulo SP 01308-050, Brazil (I.S.A., A.M.N.C., A.P.F.V., B.P.R., U.L.P., V.T.G., P.C.P., D.S.D., M.F.L.D., H.W.L., M.G.M.M., C.T.A.); Neuroradiology Section, Department of Radiology, University of São Paulo, Brazil (A.M.N.C., P.C.P., C.C.L., M.G.M.M.); Department of Neurology, Prevent Senior, São Paulo, Brazil (P.D.S.S.); and Neuroradiology Section, Department of Radiology, University of Iowa, Iowa City, Iowa (M.X.Z., B.A.P.)
| | - Bruno A Policeni
- From the Neuroradiology Section, Department of Radiology, Hospital Sírio-Libanês, Adma Jafet 91, Bela Vista, São Paulo SP 01308-050, Brazil (I.S.A., A.M.N.C., A.P.F.V., B.P.R., U.L.P., V.T.G., P.C.P., D.S.D., M.F.L.D., H.W.L., M.G.M.M., C.T.A.); Neuroradiology Section, Department of Radiology, University of São Paulo, Brazil (A.M.N.C., P.C.P., C.C.L., M.G.M.M.); Department of Neurology, Prevent Senior, São Paulo, Brazil (P.D.S.S.); and Neuroradiology Section, Department of Radiology, University of Iowa, Iowa City, Iowa (M.X.Z., B.A.P.)
| | - Claudia C Leite
- From the Neuroradiology Section, Department of Radiology, Hospital Sírio-Libanês, Adma Jafet 91, Bela Vista, São Paulo SP 01308-050, Brazil (I.S.A., A.M.N.C., A.P.F.V., B.P.R., U.L.P., V.T.G., P.C.P., D.S.D., M.F.L.D., H.W.L., M.G.M.M., C.T.A.); Neuroradiology Section, Department of Radiology, University of São Paulo, Brazil (A.M.N.C., P.C.P., C.C.L., M.G.M.M.); Department of Neurology, Prevent Senior, São Paulo, Brazil (P.D.S.S.); and Neuroradiology Section, Department of Radiology, University of Iowa, Iowa City, Iowa (M.X.Z., B.A.P.)
| | - Maria G M Martin
- From the Neuroradiology Section, Department of Radiology, Hospital Sírio-Libanês, Adma Jafet 91, Bela Vista, São Paulo SP 01308-050, Brazil (I.S.A., A.M.N.C., A.P.F.V., B.P.R., U.L.P., V.T.G., P.C.P., D.S.D., M.F.L.D., H.W.L., M.G.M.M., C.T.A.); Neuroradiology Section, Department of Radiology, University of São Paulo, Brazil (A.M.N.C., P.C.P., C.C.L., M.G.M.M.); Department of Neurology, Prevent Senior, São Paulo, Brazil (P.D.S.S.); and Neuroradiology Section, Department of Radiology, University of Iowa, Iowa City, Iowa (M.X.Z., B.A.P.)
| | - Camila T Amancio
- From the Neuroradiology Section, Department of Radiology, Hospital Sírio-Libanês, Adma Jafet 91, Bela Vista, São Paulo SP 01308-050, Brazil (I.S.A., A.M.N.C., A.P.F.V., B.P.R., U.L.P., V.T.G., P.C.P., D.S.D., M.F.L.D., H.W.L., M.G.M.M., C.T.A.); Neuroradiology Section, Department of Radiology, University of São Paulo, Brazil (A.M.N.C., P.C.P., C.C.L., M.G.M.M.); Department of Neurology, Prevent Senior, São Paulo, Brazil (P.D.S.S.); and Neuroradiology Section, Department of Radiology, University of Iowa, Iowa City, Iowa (M.X.Z., B.A.P.)
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[18F]FDG brain PET and clinical symptoms in different autoantibodies of autoimmune encephalitis: a systematic review. Neurol Sci 2022; 43:4701-4718. [PMID: 35486333 DOI: 10.1007/s10072-022-06094-9] [Citation(s) in RCA: 8] [Impact Index Per Article: 2.7] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 01/01/2022] [Accepted: 04/21/2022] [Indexed: 12/17/2022]
Abstract
INTRODUCTION Autoimmune encephalitis (AE) is caused by the antibodies that target receptors and intracellular or surface proteins. To achieve the appropriate therapeutic results, early and proper diagnosis is still the most important issue. In this review, we provide an overview of FDG-PET imaging findings in AE patients and possible relation to different subtypes and clinical features. METHODS PubMed, Web of Science, and Scopus were searched in August 2021 using a predefined search strategy. RESULTS After two-step reviewing, 22 studies with a total of 332 participants were entered into our qualitative synthesis. In anti-NMDAR encephalitis, decreased activity in the occipital lobe was present, in addition, to an increase in frontal, parietal, and specifically medial temporal activity. Anti-VGKC patients showed altered metabolism in cortical and subcortical regions such as striata and cerebellum. Abnormal metabolism in patients with anti-LGI1 has been reported in diverse areas of the brain including medial temporal, hippocampus, cerebellum, and basal ganglia all of which had hypermetabolism. Hypometabolism in parietal, frontal, occipital lobes, temporal, frontal, and hippocampus was observed in AE patients with anti-GAD antibodies. CONCLUSION Our results indicate huge diversity in metabolic patterns among different AE subtypes and it is hard to draw a firm conclusion. Moreover, the timing of imaging, seizures, and acute treatments can alter the PET patterns strongly. Further prospective investigations with specific inclusion and exclusion criteria should be carried out to identify the metabolic defect in different AE subtypes.
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Doroudinia A, Safarpour Lima B, Bakhshayesh Karam M, Ghadimi N, Yousefi F. Interesting Manifestation of Autoimmune Encephalitis on FDG PET Scan. Clin Nucl Med 2022; 47:e190-e191. [PMID: 34392290 DOI: 10.1097/rlu.0000000000003866] [Citation(s) in RCA: 2] [Impact Index Per Article: 0.7] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/25/2022]
Abstract
ABSTRACT We are presenting a 22-year-old man with intractable seizures. Autoimmune epilepsy, vasculitis, and paraneoplastic disorder were among initial differential diagnoses. His initial laboratory tests and toxicology screen were unremarkable. His initial brain MRI demonstrated generalized cortical atrophy. Features such as progressive encephalopathy, neuropsychiatric symptoms, personality change, and autonomic dysfunction were in favor of autoimmune encephalitis. Autoantibody evaluations including anti-NMDA receptor (NR1) IgG were negative in both serum and CSF samples. FDG PET scan demonstrated intense FDG uptake in the basal ganglia, more prominent in the caudate nuclei and putamina, which is one of the known autoimmune encephalitis imaging features.
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Affiliation(s)
- Abtin Doroudinia
- From the Chronic Respiratory Diseases Research Center, National Research Institute of Tuberculosis and Lung Diseases
| | - Behnam Safarpour Lima
- Department of Neurology, Imam Hossein Medical and Educational Center, Shahid Beheshti University of Medical Sciences, Tehran-Iran
| | - Mehrdad Bakhshayesh Karam
- From the Chronic Respiratory Diseases Research Center, National Research Institute of Tuberculosis and Lung Diseases
| | - Niloufar Ghadimi
- From the Chronic Respiratory Diseases Research Center, National Research Institute of Tuberculosis and Lung Diseases
| | - Farhad Yousefi
- From the Chronic Respiratory Diseases Research Center, National Research Institute of Tuberculosis and Lung Diseases
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Seery N, Butzkueven H, O'Brien TJ, Monif M. Contemporary advances in anti-NMDAR antibody (Ab)-mediated encephalitis. Autoimmun Rev 2022; 21:103057. [PMID: 35092831 DOI: 10.1016/j.autrev.2022.103057] [Citation(s) in RCA: 17] [Impact Index Per Article: 5.7] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 01/09/2022] [Accepted: 01/23/2022] [Indexed: 12/18/2022]
Abstract
The study of antibody (Ab)-mediated encephalitis has advanced dramatically since the discovery of antibodies directed against the N-methyl-D-aspartate receptor (NMDAR) in association with a unique neuro-psychiatric syndrome, over a decade-and-a-half ago. Anti-NMDAR Ab-mediated encephalitis now represents the most well characterised form of autoimmune encephalitis. The disease most commonly manifests in young women, but all ages and both sexes can be affected. Autoantibodies may arise in the context of two well-recognised disease triggers in a proportion of patients, and ultimately facilitate NMDAR displacement from synapses. Various CSF cytokines, chemokines, and other molecules have been explored as candidate biomarkers but are limited in sensitivity and specificity. The clinical spectrum is diverse, with evolution and a combination of neuro-psychiatric abnormalities at disease nadir common. Anti-NMDAR Ab-mediated encephalitis is immunotherapy responsive, and a near-majority ultimately acquire a broadly favourable clinical outcome. The diagnosis, and more particularly, the management of the disease can still hold considerable challenges. Moreover, well-defined biomarkers remain elusive. The present review will therefore delineate pathogenic and clinical advances to date in anti-NMDAR antibody-mediated encephalitis.
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Affiliation(s)
- Nabil Seery
- Department of Neuroscience, Central Clinical School, Faculty of Medicine, Nursing and Health Science, Monash University, Melbourne, Victoria, Australia; Department of Neurology, Alfred Hospital, Melbourne, Victoria, Australia
| | - Helmut Butzkueven
- Department of Neuroscience, Central Clinical School, Faculty of Medicine, Nursing and Health Science, Monash University, Melbourne, Victoria, Australia; Department of Neurology, Alfred Hospital, Melbourne, Victoria, Australia
| | - Terence J O'Brien
- Department of Neuroscience, Central Clinical School, Faculty of Medicine, Nursing and Health Science, Monash University, Melbourne, Victoria, Australia; Department of Neurology, Alfred Hospital, Melbourne, Victoria, Australia
| | - Mastura Monif
- Department of Neuroscience, Central Clinical School, Faculty of Medicine, Nursing and Health Science, Monash University, Melbourne, Victoria, Australia; Department of Neurology, Alfred Hospital, Melbourne, Victoria, Australia; Department of Neurology, Royal Melbourne Hospital, Melbourne, Victoria, Australia.
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Endres D, Pollak TA, Bechter K, Denzel D, Pitsch K, Nickel K, Runge K, Pankratz B, Klatzmann D, Tamouza R, Mallet L, Leboyer M, Prüss H, Voderholzer U, Cunningham JL, Domschke K, Tebartz van Elst L, Schiele MA. Immunological causes of obsessive-compulsive disorder: is it time for the concept of an "autoimmune OCD" subtype? Transl Psychiatry 2022; 12:5. [PMID: 35013105 PMCID: PMC8744027 DOI: 10.1038/s41398-021-01700-4] [Citation(s) in RCA: 51] [Impact Index Per Article: 17.0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Grants] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 05/23/2021] [Revised: 10/09/2021] [Accepted: 10/19/2021] [Indexed: 12/13/2022] Open
Abstract
Obsessive-compulsive disorder (OCD) is a highly disabling mental illness that can be divided into frequent primary and rarer organic secondary forms. Its association with secondary autoimmune triggers was introduced through the discovery of Pediatric Autoimmune Neuropsychiatric Disorder Associated with Streptococcal infection (PANDAS) and Pediatric Acute onset Neuropsychiatric Syndrome (PANS). Autoimmune encephalitis and systemic autoimmune diseases or other autoimmune brain diseases, such as multiple sclerosis, have also been reported to sometimes present with obsessive-compulsive symptoms (OCS). Subgroups of patients with OCD show elevated proinflammatory cytokines and autoantibodies against targets that include the basal ganglia. In this conceptual review paper, the clinical manifestations, pathophysiological considerations, diagnostic investigations, and treatment approaches of immune-related secondary OCD are summarized. The novel concept of "autoimmune OCD" is proposed for a small subgroup of OCD patients, and clinical signs based on the PANDAS/PANS criteria and from recent experience with autoimmune encephalitis and autoimmune psychosis are suggested. Red flag signs for "autoimmune OCD" could include (sub)acute onset, unusual age of onset, atypical presentation of OCS with neuropsychiatric features (e.g., disproportionate cognitive deficits) or accompanying neurological symptoms (e.g., movement disorders), autonomic dysfunction, treatment resistance, associations of symptom onset with infections such as group A streptococcus, comorbid autoimmune diseases or malignancies. Clinical investigations may also reveal alterations such as increased levels of anti-basal ganglia or dopamine receptor antibodies or inflammatory changes in the basal ganglia in neuroimaging. Based on these red flag signs, the criteria for a possible, probable, and definite autoimmune OCD subtype are proposed.
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Affiliation(s)
- Dominique Endres
- Section for Experimental Neuropsychiatry, Department of Psychiatry and Psychotherapy, Medical Center - University of Freiburg, Faculty of Medicine, University of Freiburg, Freiburg, Germany.
- Department of Psychiatry and Psychotherapy, Medical Center - University of Freiburg, Faculty of Medicine, University of Freiburg, Freiburg, Germany.
| | - Thomas A Pollak
- Department of Psychosis Studies, Institute of Psychiatry, Psychology and Neuroscience, King's College London, London, UK
| | - Karl Bechter
- Department for Psychiatry and Psychotherapy II, Ulm University, Bezirkskrankenhaus Günzburg, Günzburg, Germany
| | - Dominik Denzel
- Department of Psychiatry and Psychotherapy, Medical Center - University of Freiburg, Faculty of Medicine, University of Freiburg, Freiburg, Germany
| | - Karoline Pitsch
- Department of Psychiatry and Psychotherapy, Medical Center - University of Freiburg, Faculty of Medicine, University of Freiburg, Freiburg, Germany
| | - Kathrin Nickel
- Section for Experimental Neuropsychiatry, Department of Psychiatry and Psychotherapy, Medical Center - University of Freiburg, Faculty of Medicine, University of Freiburg, Freiburg, Germany
- Department of Psychiatry and Psychotherapy, Medical Center - University of Freiburg, Faculty of Medicine, University of Freiburg, Freiburg, Germany
| | - Kimon Runge
- Section for Experimental Neuropsychiatry, Department of Psychiatry and Psychotherapy, Medical Center - University of Freiburg, Faculty of Medicine, University of Freiburg, Freiburg, Germany
- Department of Psychiatry and Psychotherapy, Medical Center - University of Freiburg, Faculty of Medicine, University of Freiburg, Freiburg, Germany
| | - Benjamin Pankratz
- Department of Psychiatry and Psychotherapy, Medical Center - University of Freiburg, Faculty of Medicine, University of Freiburg, Freiburg, Germany
| | - David Klatzmann
- AP-HP, Hôpital Pitié-Salpêtrière, Biotherapy (CIC-BTi) and Inflammation-Immunopathology-Biotherapy Department (i2B), Paris, France
- Sorbonne Université, INSERM, Immunology-Immunopathology-Immunotherapy (i3), Paris, France
| | - Ryad Tamouza
- Univ Paris Est Créteil, INSERM, IMRB, Translational Neuropsychiatry, AP-HP, DMU IMPACT, FHU ADAPT, Fondation FondaMental, Créteil, France
| | - Luc Mallet
- Univ Paris Est Créteil, INSERM, IMRB, Translational Neuropsychiatry, AP-HP, DMU IMPACT, FHU ADAPT, Fondation FondaMental, Créteil, France
| | - Marion Leboyer
- Univ Paris Est Créteil, INSERM, IMRB, Translational Neuropsychiatry, AP-HP, DMU IMPACT, FHU ADAPT, Fondation FondaMental, Créteil, France
| | - Harald Prüss
- Department of Neurology and Experimental Neurology, Charité - Universitätsmedizin Berlin, Berlin, Germany
- German Center for Neurodegenerative Diseases (DZNE) Berlin, Berlin, Germany
| | - Ulrich Voderholzer
- Schoen Clinic Roseneck, Prien am Chiemsee, Germany
- Department of Psychiatry and Psychotherapy, University Hospital Munich, Munich, Germany
| | - Janet L Cunningham
- Department of Neuroscience, Psychiatry, Uppsala University, Uppsala, Sweden
| | - Katharina Domschke
- Department of Psychiatry and Psychotherapy, Medical Center - University of Freiburg, Faculty of Medicine, University of Freiburg, Freiburg, Germany
- Centre for Basics in Neuromodulation, Faculty of Medicine, University of Freiburg, Freiburg, Germany
| | - Ludger Tebartz van Elst
- Section for Experimental Neuropsychiatry, Department of Psychiatry and Psychotherapy, Medical Center - University of Freiburg, Faculty of Medicine, University of Freiburg, Freiburg, Germany
- Department of Psychiatry and Psychotherapy, Medical Center - University of Freiburg, Faculty of Medicine, University of Freiburg, Freiburg, Germany
| | - Miriam A Schiele
- Department of Psychiatry and Psychotherapy, Medical Center - University of Freiburg, Faculty of Medicine, University of Freiburg, Freiburg, Germany
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Abdullah W, Hussein S, Ibrahim B. Anti-N-Methyl-D-Aspartate receptor encephalitis in pediatrics: A review of clinical manifestations, treatment, and prognosis. MUSTANSIRIYA MEDICAL JOURNAL 2022. [DOI: 10.4103/mj.mj_6_22] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Track Full Text] [Subscribe] [Scholar Register] [Indexed: 01/04/2023]
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36
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Endres D, Lüngen E, Hasan A, Kluge M, Fröhlich S, Lewerenz J, Bschor T, Haußleiter IS, Juckel G, Then Bergh F, Ettrich B, Kertzscher L, Oviedo-Salcedo T, Handreka R, Lauer M, Winter K, Zumdick N, Drews A, Obrocki J, Yalachkov Y, Bubl A, von Podewils F, Schneider U, Szabo K, Mattern M, Philipsen A, Domschke K, Wandinger KP, Neyazi A, Stich O, Prüss H, Leypoldt F, Tebartz van Elst L. Clinical manifestations and immunomodulatory treatment experiences in psychiatric patients with suspected autoimmune encephalitis: a case series of 91 patients from Germany. Mol Psychiatry 2022; 27:1479-1489. [PMID: 35046526 PMCID: PMC9095476 DOI: 10.1038/s41380-021-01396-4] [Citation(s) in RCA: 25] [Impact Index Per Article: 8.3] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 02/12/2021] [Revised: 09/26/2021] [Accepted: 11/23/2021] [Indexed: 12/14/2022]
Abstract
Autoimmune encephalitis (AE) can rarely manifest as a predominantly psychiatric syndrome without overt neurological symptoms. This study's aim was to characterize psychiatric patients with AE; therefore, anonymized data on patients with suspected AE with predominantly or isolated psychiatric syndromes were retrospectively collected. Patients with readily detectable neurological symptoms suggestive of AE (e.g., epileptic seizures) were excluded. Patients were classified as "probable psychiatric AE (pAE)," if well-characterized neuronal IgG autoantibodies were detected or "possible pAE" (e.g., with detection of nonclassical neuronal autoantibodies or compatible cerebrospinal fluid (CSF) changes). Of the 91 patients included, 21 (23%) fulfilled our criteria for probable (autoantibody-defined) pAE and 70 (77%) those for possible pAE. Among patients with probable pAE, 90% had anti-NMDA receptor (NMDA-R) autoantibodies. Overall, most patients suffered from paranoid-hallucinatory syndromes (53%). Patients with probable pAE suffered more often from disorientation (p < 0.001) and impaired memory (p = 0.001) than patients with possible pAE. Immunotherapies were performed in 69% of all cases, mostly with high-dose corticosteroids. Altogether, 93% of the patients with probable pAE and 80% of patients with possible pAE reportedly benefited from immunotherapies (p = 0.251). In summary, this explorative, cross-sectional evaluation confirms that autoantibody-associated AE syndromes can predominantly manifest as psychiatric syndromes, especially in anti-NMDA-R encephalitis. However, in three out of four patients, diagnosis of possible pAE was based on nonspecific findings (e.g., slight CSF pleocytosis), and well-characterized neuronal autoantibodies were absent. As such, the spectrum of psychiatric syndromes potentially responding to immunotherapies seems not to be limited to currently known autoantibody-associated AE. Further trials are needed.
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Affiliation(s)
- Dominique Endres
- grid.7708.80000 0000 9428 7911Section for Experimental Neuropsychiatry, Department of Psychiatry and Psychotherapy, Medical Center - University of Freiburg, Faculty of Medicine, University of Freiburg, Freiburg, Germany ,grid.7708.80000 0000 9428 7911Department of Psychiatry and Psychotherapy, Medical Center - University of Freiburg, Faculty of Medicine, University of Freiburg, Freiburg, Germany
| | - Eva Lüngen
- grid.7708.80000 0000 9428 7911Section for Experimental Neuropsychiatry, Department of Psychiatry and Psychotherapy, Medical Center - University of Freiburg, Faculty of Medicine, University of Freiburg, Freiburg, Germany ,grid.7708.80000 0000 9428 7911Department of Psychiatry and Psychotherapy, Medical Center - University of Freiburg, Faculty of Medicine, University of Freiburg, Freiburg, Germany
| | - Alkomiet Hasan
- grid.7307.30000 0001 2108 9006Department of Psychiatry, Psychotherapy and Psychosomatics, Medical Faculty, University of Augsburg, BKH Augsburg, Augsburg, Germany ,grid.411095.80000 0004 0477 2585Department of Psychiatry and Psychotherapy, University Hospital, Munich, Germany
| | - Michael Kluge
- grid.9647.c0000 0004 7669 9786Department of Psychiatry and Psychotherapy, University of Leipzig, Leipzig, Germany
| | - Sabrina Fröhlich
- Department of Psychiatry and Psychotherapy, Ludwig-Noll-Krankenhaus, Kassel, Germany ,Department of Neurology and Clinical Neurophysiology, DRK Hospital Nordhessen, Kassel, Germany
| | - Jan Lewerenz
- grid.6582.90000 0004 1936 9748Department of Neurology, University of Ulm, Ulm, Germany
| | - Tom Bschor
- grid.412282.f0000 0001 1091 2917Department of Psychiatry and Psychotherapy, University Hospital Dresden, Dresden, Germany
| | - Ida Sibylle Haußleiter
- grid.5570.70000 0004 0490 981XDepartment of Psychiatry, LWL-University Hospital, Ruhr University Bochum, Bochum, Germany
| | - Georg Juckel
- grid.5570.70000 0004 0490 981XDepartment of Psychiatry, LWL-University Hospital, Ruhr University Bochum, Bochum, Germany
| | - Florian Then Bergh
- grid.9647.c0000 0004 7669 9786Department of Neurology, University of Leipzig, Leipzig, Germany
| | - Barbara Ettrich
- grid.9647.c0000 0004 7669 9786Department of Neurology, University of Leipzig, Leipzig, Germany
| | - Lisa Kertzscher
- grid.9647.c0000 0004 7669 9786Department of Psychiatry and Psychotherapy, University of Leipzig, Leipzig, Germany
| | - Tatiana Oviedo-Salcedo
- grid.411095.80000 0004 0477 2585Department of Psychiatry and Psychotherapy, University Hospital, Munich, Germany
| | - Robert Handreka
- grid.460801.b0000 0004 0558 2150Department of Neurology, Carl-Thiem-Klinikum Cottbus, Cottbus, Germany
| | - Martin Lauer
- grid.411760.50000 0001 1378 7891Department of Psychiatry, Psychosomatics and Psychotherapy, Center of Mental Health, University Hospital Würzburg, Würzburg, Germany
| | - Klaas Winter
- grid.491868.a0000 0000 9601 2399Department of Psychiatry and Psychotherapy, Carl-Friedrich-Flemming-Klinik, Helios Kliniken Schwerin, Schwerin, Germany
| | - Norbert Zumdick
- Department of Psychiatry and Psychotherapy Medicine, St. Marien-Hospital Hamm, Hamm, Germany
| | - Anna Drews
- Department of Psychiatry and Psychotherapy, Vinzenz von Paul Hospital Rottenmünster, Rottweil, Germany
| | - Jost Obrocki
- Department of Psychiatry, Psychotherapy and Psychosomatic Medicine, Regio Klinikum Elmshorn, Elmshorn, Germany
| | - Yavor Yalachkov
- grid.411088.40000 0004 0578 8220Department of Neurology, University Hospital/Goethe University, Frankfurt/Main, Germany
| | - Anna Bubl
- grid.11749.3a0000 0001 2167 7588Department of Psychiatry and Psychotherapy, University of Saarland, Homburg/Saar, Germany
| | - Felix von Podewils
- grid.5603.0Department of Neurology, University Medicine Greifswald, Greifswald, Germany
| | - Udo Schneider
- grid.5570.70000 0004 0490 981XDepartment of Psychiatry and Psychotherapy, Ruhr-University Bochum Campus-OWL Lübbecke, Lübbecke, Germany
| | - Kristina Szabo
- grid.7700.00000 0001 2190 4373Department of Neurology and Mannheim Center for Translational Neuroscience, Medical Faculty Mannheim, Heidelberg University, Mannheim, Germany
| | - Margarete Mattern
- grid.5253.10000 0001 0328 4908Department of General Psychiatry, Center for Psychosocial Medicine, University Hospital Heidelberg, Heidelberg, Germany
| | - Alexandra Philipsen
- grid.10388.320000 0001 2240 3300Department of Psychiatry and Psychotherapy, University of Bonn, Bonn, Germany
| | - Katharina Domschke
- grid.7708.80000 0000 9428 7911Department of Psychiatry and Psychotherapy, Medical Center - University of Freiburg, Faculty of Medicine, University of Freiburg, Freiburg, Germany ,grid.5963.9Center for Basics in NeuroModulation, Faculty of Medicine, University of Freiburg, Freiburg, Germany
| | - Klaus-Peter Wandinger
- grid.412468.d0000 0004 0646 2097Neuroimmunology Section, Institute of Clinical Chemistry, University Hospital Schleswig-Holstein Kiel/Lübeck, Lübeck, Germany
| | - Alexandra Neyazi
- grid.10423.340000 0000 9529 9877Department of Psychiatry, Social Psychiatry and Psychotherapy, Hannover Medical School, Hannover, Germany
| | - Oliver Stich
- Neurology, Medical Care Center, Konstanz, Germany ,grid.5963.9Department of Neurology, Medical Center, Faculty of Medicine, University of Freiburg, Freiburg, Germany
| | - Harald Prüss
- grid.6363.00000 0001 2218 4662Department of Neurology and Experimental Neurology, Charité - Universitätsmedizin Berlin, Berlin, Germany ,grid.424247.30000 0004 0438 0426German Center for Neurodegenerative Diseases (DZNE), Berlin, Germany
| | - Frank Leypoldt
- grid.412468.d0000 0004 0646 2097Neuroimmunology Section, Institute of Clinical Chemistry, University Hospital Schleswig-Holstein Kiel/Lübeck, Lübeck, Germany ,grid.9764.c0000 0001 2153 9986Department of Neurology, Christian-Albrechts-University Kiel, Kiel, Germany
| | - Ludger Tebartz van Elst
- Section for Experimental Neuropsychiatry, Department of Psychiatry and Psychotherapy, Medical Center - University of Freiburg, Faculty of Medicine, University of Freiburg, Freiburg, Germany. .,Department of Psychiatry and Psychotherapy, Medical Center - University of Freiburg, Faculty of Medicine, University of Freiburg, Freiburg, Germany.
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Ciano-Petersen NL, Cabezudo-García P, Muñiz-Castrillo S, Honnorat J, Serrano-Castro PJ, Oliver-Martos B. Current Status of Biomarkers in Anti-N-Methyl-D-Aspartate Receptor Encephalitis. Int J Mol Sci 2021; 22:13127. [PMID: 34884930 PMCID: PMC8658717 DOI: 10.3390/ijms222313127] [Citation(s) in RCA: 19] [Impact Index Per Article: 4.8] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Grants] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 11/08/2021] [Revised: 11/29/2021] [Accepted: 12/01/2021] [Indexed: 02/05/2023] Open
Abstract
The discovery of biomarkers in rare diseases is of paramount importance to allow a better diagnosis, improve predictions of outcomes, and prompt the development of new treatments. Anti-N-methyl-D-aspartate receptor (NMDAR) encephalitis is a rare autoimmune disorder associated with the presence of antibodies targeting the GluN1 subunit of the NMDAR. Since it was discovered in 2007, large efforts have been made towards the identification of clinical, paraclinical, and molecular biomarkers to better understand the immune mechanisms that govern the course of the disease as well as to define predictors of treatment response and long-term outcomes. However, most of these biomarkers are still in an exploratory phase, with only a few candidates reaching the final phases of the always-complex process of biomarker development, mainly due to the low incidence of the disease and its recent description. Clinical and paraclinical markers are probably the most widely explored in anti-NMDAR encephalitis, five of them combined in a clinical score to predict 1 year outcome. On the contrary, soluble molecules, such as persistent antibody positivity, antibody titers, cytokines, and other inflammatory mediators, have been proposed as biomarkers of clinical activity, inflammation, prognosis, and treatment response, but further studies are required for their clinical validation including larger and more homogenous cohorts of patients. Similarly, genetic susceptibility biomarkers are still in the exploratory phase and, therefore, weak conclusions can for now only be achieved. Thus, further studies are warranted to define biomarkers and unravel the underlying mechanisms driving rare diseases such as anti-NMDAR encephalitis. Future international collaborative studies with prospective designs that enable the enrollment of large cohorts will allow for the identification and validation of novel biomarkers for clinical decision-making.
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Affiliation(s)
- Nicolás Lundahl Ciano-Petersen
- Neuroimmunology and Neuroinflammation Group, Biomedical Research Institute of Málaga (IBIMA), 29007 Málaga, Spain; (N.L.C.-P.); (P.C.-G.)
- Red Andaluza de Investigación Clínica y Traslacional en Neurología (Neuro-RECA), 29010 Málaga, Spain
- French Reference Center on Paraneoplastic Neurological Syndromes and Autoimmune Encephalitis, Hospices Civils de Lyon, Hôpital Neurologique, 69677 Bron, France; (S.M.-C.); (J.H.)
- SynatAc Team, Institut NeuroMyoGène, INSERM U1217/CNRS UMR 5310, Université de Lyon, Université Claude Bernard Lyon 1, 69372 Lyon, France
| | - Pablo Cabezudo-García
- Neuroimmunology and Neuroinflammation Group, Biomedical Research Institute of Málaga (IBIMA), 29007 Málaga, Spain; (N.L.C.-P.); (P.C.-G.)
- Red Andaluza de Investigación Clínica y Traslacional en Neurología (Neuro-RECA), 29010 Málaga, Spain
| | - Sergio Muñiz-Castrillo
- French Reference Center on Paraneoplastic Neurological Syndromes and Autoimmune Encephalitis, Hospices Civils de Lyon, Hôpital Neurologique, 69677 Bron, France; (S.M.-C.); (J.H.)
- SynatAc Team, Institut NeuroMyoGène, INSERM U1217/CNRS UMR 5310, Université de Lyon, Université Claude Bernard Lyon 1, 69372 Lyon, France
| | - Jérôme Honnorat
- French Reference Center on Paraneoplastic Neurological Syndromes and Autoimmune Encephalitis, Hospices Civils de Lyon, Hôpital Neurologique, 69677 Bron, France; (S.M.-C.); (J.H.)
- SynatAc Team, Institut NeuroMyoGène, INSERM U1217/CNRS UMR 5310, Université de Lyon, Université Claude Bernard Lyon 1, 69372 Lyon, France
| | - Pedro Jesús Serrano-Castro
- Neuroimmunology and Neuroinflammation Group, Biomedical Research Institute of Málaga (IBIMA), 29007 Málaga, Spain; (N.L.C.-P.); (P.C.-G.)
- Red Andaluza de Investigación Clínica y Traslacional en Neurología (Neuro-RECA), 29010 Málaga, Spain
| | - Begoña Oliver-Martos
- Neuroimmunology and Neuroinflammation Group, Biomedical Research Institute of Málaga (IBIMA), 29007 Málaga, Spain; (N.L.C.-P.); (P.C.-G.)
- Red Andaluza de Investigación Clínica y Traslacional en Neurología (Neuro-RECA), 29010 Málaga, Spain
- Department of Cell Biology, Genetics and Physiology, Physiology Area, University of Malaga, 29010 Málaga, Spain
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Liu X, Yu T, Zhao X, Li G, Lv R, Ai L, Wang Q. 18 F-fluorodeoxy-glucose positron emission tomography pattern and prognostic predictors in patients with anti-GABAB receptor encephalitis. CNS Neurosci Ther 2021; 28:269-278. [PMID: 34837479 PMCID: PMC8739043 DOI: 10.1111/cns.13767] [Citation(s) in RCA: 2] [Impact Index Per Article: 0.5] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 07/24/2021] [Revised: 10/21/2021] [Accepted: 11/10/2021] [Indexed: 01/03/2023] Open
Abstract
Aims To identify the metabolic pattern and prognostic predictors in anti‐gamma‐aminobutyric‐acid B (GABAB) receptor encephalitis using 18F‐fluorodeoxy‐glucose positron emission tomography (18F‐FDG‐PET). Methods Twenty‐one patients diagnosed anti‐GABAB receptor encephalitis who underwent 18F‐FDG‐PET at first hospitalization were retrospectively reviewed. 18F‐FDG‐PET images were analyzed in comparison with controls. Further group comparisons of 18F‐FDG‐PET data were carried out between prognostic subgroups. Results 18F‐FDG‐PET was abnormal in 81% patients with anti‐GABAB receptor encephalitis and was more sensitive than MRI (81% vs. 42.9%, p = 0.025). Alter limbic lobe glucose metabolism (mostly hypermetabolism) was observed in 14 patients (66.7%), of whom 10 (10/14, 71.4%) demonstrated hypermetabolism in the medial temporal lobe (MTL). Group analysis also confirmed MTL hypermetabolism in association with relative frontal and parietal hypometabolism was a general metabolic pattern. After a median follow‐up of 33 months, the group comparisons revealed that patients with poor outcome demonstrated increased metabolism in the MTL compared to those with good outcome. Conclusion 18F‐FDG‐PET may be more sensitive than MRI in the early diagnosis of anti‐GABAB receptor encephalitis. MTL hypermetabolism was associated with relative frontal or parietal hypometabolism and may serve as a prognostic biomarker in anti‐GABAB receptor encephalitis.
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Affiliation(s)
- Xiao Liu
- Department of Neurology, Beijing Tiantan Hospital, Capital Medical University, Beijing, China.,China National Clinical Research Center for Neurological Diseases, Beijing, China
| | - Tingting Yu
- Department of Neurology, Beijing Tiantan Hospital, Capital Medical University, Beijing, China.,China National Clinical Research Center for Neurological Diseases, Beijing, China
| | - Xiaobin Zhao
- Department of Nuclear Medicine, Beijing Tiantan Hospital, Capital Medical University, Beijing, China
| | - Gongfei Li
- Department of Neurology, Beijing Tiantan Hospital, Capital Medical University, Beijing, China.,China National Clinical Research Center for Neurological Diseases, Beijing, China
| | - Ruijuan Lv
- Department of Neurology, Beijing Tiantan Hospital, Capital Medical University, Beijing, China.,China National Clinical Research Center for Neurological Diseases, Beijing, China
| | - Lin Ai
- Department of Nuclear Medicine, Beijing Tiantan Hospital, Capital Medical University, Beijing, China
| | - Qun Wang
- Department of Neurology, Beijing Tiantan Hospital, Capital Medical University, Beijing, China.,China National Clinical Research Center for Neurological Diseases, Beijing, China.,Collaborative Innovation Center for Brain Disorders, Beijing Institute of Brain Disorders, Capital Medical University, Beijing, China
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Desai K, Aneja A, Luthra M. Lung cancer masquerading as a paraneoplastic neurologic syndrome without a primary lung mass: Case report and review of literature. Lung India 2021; 38:577-580. [PMID: 34747743 PMCID: PMC8614603 DOI: 10.4103/lungindia.lungindia_893_20] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/04/2022] Open
Abstract
Paraneoplastic and autoimmune encephalitis (AIE) syndromes describe a range of inflammatory disorders of the brain. "Classic" paraneoplastic encephalitis syndromes occur due to a remote neoplasm and are associated with antibodies that target intracellular neuronal proteins while the more recently described AIE syndromes are not always paraneoplastic and occur in association with antibodies that target cell-surface neuronal receptors (e.g., anti-NMDA receptor, anti-LGI1, anti-GABAB receptor).[1] Diagnosis can be difficult and delayed due to nonspecific clinical, imaging, and laboratory findings, and in those syndromes associated with a neoplasm, the neurologic syndromes often precede the cancer diagnosis. We present a case of a 64-year-old patient diagnosed with anti-GABAB receptor encephalitis that subsequently revealed an underlying small cell lung cancer without a primary lung tumor. This case highlights the clinical challenge in diagnosing immune-mediated encephalitis, its methodical work up, and subsequent management.
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Affiliation(s)
- Krisha Desai
- Department of Medicine, School of Medicine, J. Willis Hurst Internal Medicine Residency Program, Emory University, Atlanta, Georgia
| | - Ankur Aneja
- Department of Medicine, School of Medicine, J. Willis Hurst Internal Medicine Residency Program, Emory University, Atlanta, Georgia
| | - Munish Luthra
- Department of Medicine, Division of Pulmonary, Allergy, Critical Care and Sleep Medicine, School of Medicine, Emory University, Atlanta, Georgia
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Bordonne M, Chawki MB, Doyen M, Kas A, Guedj E, Tyvaert L, Verger A. Brain 18F-FDG PET for the diagnosis of autoimmune encephalitis: a systematic review and a meta-analysis. Eur J Nucl Med Mol Imaging 2021; 48:3847-3858. [PMID: 33677643 DOI: 10.1007/s00259-021-05299-y] [Citation(s) in RCA: 60] [Impact Index Per Article: 15.0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 12/02/2020] [Accepted: 03/02/2021] [Indexed: 12/14/2022]
Abstract
OBJECTIVE To consolidate current understanding of detection sensitivity of brain 18F-FDG PET scans in the diagnosis of autoimmune encephalitis and to define specific metabolic imaging patterns for the most frequently occurring autoantibodies. METHODS A systematic and exhaustive search of data available in the literature was performed by querying the PubMed/MEDLINE and Cochrane databases for the search terms: ((PET) OR (positron emission tomography)) AND ((FDG) OR (fluorodeoxyglucose)) AND ((encephalitis) OR (brain inflammation)). Studies had to satisfy the following criteria: (i) include at least ten pediatric or adult patients suspected or diagnosed with autoimmune encephalitis according to the current recommendations, (ii) specifically present 18F-FDG PET and/or morphologic imaging findings. The diagnostic 18F-FDG PET detection sensitivity in autoimmune encephalitis was determined for all cases reported in this systematic review, according to a meta-analysis following the PRISMA method, and selected publication quality was assessed with the QUADAS-2 tool. RESULTS The search strategy identified 626 articles including references from publications. The detection sensitivity of 18F-FDG PET was 87% (80-92%) based on 21 publications and 444 patients included in the meta-analysis. We also report specific brain 18F-FDG PET imaging patterns for the main encephalitis autoantibody subtypes. CONCLUSION AND RELEVANCE Brain 18F-FDG PET has a high detection sensitivity and should be included in future diagnostic autoimmune encephalitis recommendations. Specific metabolic 18F-FDG PET patterns corresponding to the main autoimmune encephalitis autoantibody subtypes further enhance the value of this diagnostic.
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Affiliation(s)
- Manon Bordonne
- Department of Nuclear Medicine and Nancyclotep Imaging Platform, Université de Lorraine, CHRU Nancy, Rue du Morvan, 54500 Vandoeuvre-les-Nancy, F-54000, Nancy, France
| | - Mohammad B Chawki
- Department of Nuclear Medicine and Nancyclotep Imaging Platform, Université de Lorraine, CHRU Nancy, Rue du Morvan, 54500 Vandoeuvre-les-Nancy, F-54000, Nancy, France
| | - Matthieu Doyen
- Department of Nuclear Medicine and Nancyclotep Imaging Platform, Université de Lorraine, CHRU Nancy, Rue du Morvan, 54500 Vandoeuvre-les-Nancy, F-54000, Nancy, France
- Université de Lorraine, IADI, INSERM U1254, F-54000, Nancy, France
| | - Aurelie Kas
- Nuclear Medicine Department, Pitié-Salpêtrière Hospital, APHP Sorbonne-Université, Laboratoire d'Imagerie Biomédicale, Sorbonne Université, F-75000, Paris, France
| | - Eric Guedj
- Nuclear Medicine Department, Aix Marseille Univ, APHM, CNRS, Centrale Marseille, Institut Fresnel, Timone Hospital, CERIMED, F-13000, Marseille, France
| | - Louise Tyvaert
- Department of Neurology, Université de Lorraine, CRAN UMR 7039, CHRU, F-54000, Nancy, France
| | - Antoine Verger
- Department of Nuclear Medicine and Nancyclotep Imaging Platform, Université de Lorraine, CHRU Nancy, Rue du Morvan, 54500 Vandoeuvre-les-Nancy, F-54000, Nancy, France.
- Université de Lorraine, IADI, INSERM U1254, F-54000, Nancy, France.
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Abou Khaled KJ, Azar CE, Haidar MB. Ictal and interictal FDG-PET in anti-NMDAR encephalitis with mutism. Radiol Case Rep 2021; 16:3892-3897. [PMID: 34703513 PMCID: PMC8523870 DOI: 10.1016/j.radcr.2021.09.036] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.3] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 08/22/2021] [Revised: 09/13/2021] [Accepted: 09/15/2021] [Indexed: 11/24/2022] Open
Abstract
We report a case of a 27-year-old right-handed gentleman with mutism and seizures diagnosed with Anti–N-methyl-D-aspartate receptor (NMDAR) encephalitis without evidence of underlying malignancy. Brain MRI was unremarkable. Clinical seizures were controlled but long-term video-EEG monitoring was needed for better characterization of his clinical manifestations especially that language partially improved. It was crucial to identify whether this mutism was ictal in origin or not. Ictal brain Positron Emission Tomography with 18 F‐fluorodeoxyglucose (FDGPET) scan combined with EEG was done. It revealed left fronto-temporal, parietal, and crossed cerebellar hypermetabolism (or diaschisis) concomitant to the underlying rhythmic focal delta activity on EEG. Beside anti–epileptic drugs he was treated with escalating immunotherapy (intravenous solumedrol then immunoglobulins then full rituximab course). Six months later, EEG combined to FDG-PET scan were repeated, and were normal. At 3 years follow up the patient remains neurologically stable and seizure-free, off anti–epileptics drugs. Performing the FDGPET scan combined to EEG was useful to identify non–convulsive status epilepticus and should be performed early in anti–NMDAR encephalitis to guide treatment.
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Affiliation(s)
| | - Carine E Azar
- Department of Neurology, Saint-Joseph University, Beirut, Lebanon
| | - Mohamad B Haidar
- Department of Nuclear Medicine, American University of Beirut, Beirut, Lebanon
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Herrera-Mora P, Munive-Baez L, Ruiz García M, Galindo-Martínez A, Maldonado-Diaz DE, Delgado RD, Cárdenas G. Anti-N-methyl-D-aspartate receptor encephalitis: An observational and comparative study in Mexican children and adults. Clin Neurol Neurosurg 2021; 210:106986. [PMID: 34688092 DOI: 10.1016/j.clineuro.2021.106986] [Citation(s) in RCA: 2] [Impact Index Per Article: 0.5] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 11/04/2020] [Revised: 09/15/2021] [Accepted: 10/06/2021] [Indexed: 11/17/2022]
Abstract
OBJECTIVE To present a case series of encephalitis patients with anti-N-methyl-D-aspartate receptor antibodies, attending two neurological referral centers in a three-year period. METHODS A retrospective, descriptive, comparative study included child and adult patients in two neurological populations, positive for antibodies against the NR1 and NR2 subunits of the glutamate (NMDA) receptor in serum and CSF, as determined during a three-year period. RESULTS Sixty-six patients were included (40 children and 26 adults). Male patients were more affected (M: F ratio was 1:0.6). No differences in progression or hospitalization time were observed between groups. In children, 35% of patients showed herpetic infection before autoimmune encephalitis (P = 0.01). Among viral prodromal symptoms, upper respiratory tract infection (P = 0.02) and fever (P = 0.001) predominated in children, while infectious gastroenteritis was more frequent in adults (P = 0.03). Among neuropsychiatric signs, mental confusion (P = 0.0001) and orofacial dyskinesia/oromandibular dystonia (P = 0.0001) were frequent in children, while emotional lability (P = 0.03), catatonia (P = 0.0001), and headache (P = 0.005) predominated in adults. The score in the modified Rankin scale on admission was higher in children (4.3 ± 0.8 vs. 2.2 ± 1.3, P = 0.0001), but at one-year of clinical follow up no significant differences were found. CONCLUSIONS Male patients were predominantly affected in our population. One-third of all patients developed prodromal infection. Neuropsychiatric clinical complaints were different in children and adults. However, post-hospitalization recovery was similar between groups.
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Affiliation(s)
- Patricia Herrera-Mora
- Department of Pediatric Neurology, Instituto Nacional de Pediatría, Mexico City, México
| | - Leticia Munive-Baez
- Department of Pediatric Neurology, Instituto Nacional de Pediatría, Mexico City, México
| | - Matilde Ruiz García
- Department of Pediatric Neurology, Instituto Nacional de Pediatría, Mexico City, México
| | | | - Daniela Ellis Maldonado-Diaz
- Department of Neuroinfectology, Instituto Nacional de Neurología y Neurocirugía Manuel Velasco Suárez, Mexico City, México
| | - Rosa Delia Delgado
- Department of Neuroimaging, Instituto Nacional de Neurología y Neurocirugía Manuel Velasco Suárez, Mexico City, México
| | - Graciela Cárdenas
- Department of Neuroinfectology, Instituto Nacional de Neurología y Neurocirugía Manuel Velasco Suárez, Mexico City, México.
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43
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Autoimmune cerebellar hypermetabolism: Report of three cases and literature overview. Rev Neurol (Paris) 2021; 178:337-346. [PMID: 34657731 DOI: 10.1016/j.neurol.2021.07.018] [Citation(s) in RCA: 2] [Impact Index Per Article: 0.5] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 04/21/2021] [Revised: 07/19/2021] [Accepted: 07/23/2021] [Indexed: 11/23/2022]
Abstract
We report three cases of vermian cerebellar hypermetabolism in patients with autoimmune encephalitis. One of our patients was positive for anti-Ma2 antibodies and one for anti-Zic4 antibodies while the remaining patient did not present any known antibodies. The seronegative patient deteriorated after immune checkpoint inhibitor treatment for a pulmonary adenocarcinoma and improved with immunosuppressive drugs, which is in favour of an underlying autoimmune mechanism. They all presented with subacute neurological symptoms. Brain magnetic resonance imaging was normal except in one patient, where hyperintensities were present on FLAIR sequence around the third ventricle and the cerebral aqueduct. 18F-FDG brain positron emission tomography with computed tomography (18F-FDG PET-CT) demonstrated an unusual vermian cerebellar hypermetabolism in the three cases. While cerebellar hypermetabolism on 18F-FDG PET-CT has been described in various neurological diseases, such vermian - and more broadly cerebellar - hypermetabolism was seldom described in previous studies on autoimmune encephalitis. When differential diagnoses have been ruled out, this pattern may be of interest for the positive diagnosis of autoimmune encephalitis in difficult diagnostic cases.
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Zhao X, Zhao S, Chen Y, Zhang Z, Li X, Liu X, Lv R, Wang Q, Ai L. Subcortical Hypermetabolism Associated With Cortical Hypometabolism Is a Common Metabolic Pattern in Patients With Anti-Leucine-Rich Glioma-Inactivated 1 Antibody Encephalitis. Front Immunol 2021; 12:672846. [PMID: 34616389 PMCID: PMC8488294 DOI: 10.3389/fimmu.2021.672846] [Citation(s) in RCA: 11] [Impact Index Per Article: 2.8] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 02/26/2021] [Accepted: 09/06/2021] [Indexed: 12/23/2022] Open
Abstract
Purpose Brain 18F-fluorodeoxyglucose positron emission tomography (FDG PET) is a sensitive technique for assisting in the diagnosis of patients with anti-leucine-rich glioma-inactivated 1 (LGI1) antibody encephalitis. However, the common pattern of this disorder assessed by FDG PET remains unknown. The present study aimed to explore the glucose metabolic patterns of this disorder based on PET voxel analysis. Methods This retrospective study enrolled 25 patients with anti-LGI1 encephalitis, who were admitted in Beijing Tiantan Hospital between September 2014 and July 2019. The glucose metabolic pattern was compared between the included patients and 44 age- and gender-matched healthy controls using Statistical Parametric Mapping. Then, the correlation between the metabolic pattern and scaled activities of daily living (ADLs) of the patients was assessed. Results The median time from symptom onset to PET scans was 9 w (range:2-53w). The groupwise analysis revealed that patients with anti-LGI1 encephalitis had left hippocampal hypermetabolism and hypometabolism in almost all neocortical regions. The individual-level results showed most patients presented a decreased metabolism in neocortical regions, as well as an increase in metabolism in the hippocampus and basal ganglia. Furthermore, the metabolic gradient between hippocampus and neocortical regions was positively associated with the ADLs (frontal lobe, r=0.529, P=0.008; parietal lobe, r=0.474, P=0.019; occipital lobe, r=0.413, P=0.045; temporal lobe, r=0.490, P=0.015), respectively. In addition, the patients with facio-brachial dystonic seizures (FBDS) presented bilateral putamen hypermetabolism, when compared to patients without FBDS and healthy controls. Conclusion Subcortical hypermetabolism associated with cortical hypometabolism presented with a common metabolic pattern in patients with anti-LGI1 encephalitis in the present study. The resolution of the metabolic gradient of the hippocampal hypermetabolism and neocortical hypometabolism may bring about improved clinical neurologic disability.
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Affiliation(s)
- Xiaobin Zhao
- Department of Nuclear Medicine, Beijing Tiantan Hospital, Capital Medical University, Beijing, China
| | - Shaokun Zhao
- State Key Laboratory of Cognitive Neuroscience and Learning & International Data Group/McGovern Institute for Brain Research, Beijing Normal University, Beijing, China.,Beijing Aging Brain Rejuvenation Initiative Centre, Beijing Normal University, Beijing, China
| | - Yaojing Chen
- State Key Laboratory of Cognitive Neuroscience and Learning & International Data Group/McGovern Institute for Brain Research, Beijing Normal University, Beijing, China.,Beijing Aging Brain Rejuvenation Initiative Centre, Beijing Normal University, Beijing, China
| | - Zhanjun Zhang
- State Key Laboratory of Cognitive Neuroscience and Learning & International Data Group/McGovern Institute for Brain Research, Beijing Normal University, Beijing, China.,Beijing Aging Brain Rejuvenation Initiative Centre, Beijing Normal University, Beijing, China
| | - Xiaotong Li
- Department of Nuclear Medicine, Beijing Tiantan Hospital, Capital Medical University, Beijing, China
| | - Xiao Liu
- Department of Neurology, Beijing Tiantan Hospital, Capital Medical University, Beijing, China
| | - Ruijuan Lv
- Department of Neurology, Beijing Tiantan Hospital, Capital Medical University, Beijing, China
| | - Qun Wang
- Department of Neurology, Beijing Tiantan Hospital, Capital Medical University, Beijing, China
| | - Lin Ai
- Department of Nuclear Medicine, Beijing Tiantan Hospital, Capital Medical University, Beijing, China
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45
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Decrease in the cortex/striatum metabolic ratio on [ 18F]-FDG PET: a biomarker of autoimmune encephalitis. Eur J Nucl Med Mol Imaging 2021; 49:921-931. [PMID: 34462791 DOI: 10.1007/s00259-021-05507-9] [Citation(s) in RCA: 9] [Impact Index Per Article: 2.3] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 03/15/2021] [Accepted: 07/27/2021] [Indexed: 10/20/2022]
Abstract
PURPOSE The aim of this [18F]-FDG PET study was to determine the diagnostic value of the cortex/striatum metabolic ratio in a large cohort of patients suffering from autoimmune encephalitis (AE) and to search for correlations with the course of the disease. METHODS We retrospectively collected clinical and paraclinical data of patients with AE, including brain 18F-FDG PET/CT. Whole-brain statistical analysis was performed using SPM8 software after activity parametrization to the striatum in comparison to healthy subjects. The discriminative performance of this metabolic ratio was evaluated in patients with AE using receiver operating characteristic curves against 44 healthy subjects and a control group of 688 patients with MCI. Relationship between cortex/striatum metabolic ratios and clinical/paraclinical data was assessed using univariate and multivariate analysis in patients with AE. RESULTS Fifty-six patients with AE were included. In comparison to healthy subjects, voxel-based statistical analysis identified one large cluster (p-cluster < 0.05, FWE corrected) of widespread decreased cortex/striatum ratio in patients with AE. The mean metabolic ratio was significantly lower for AE patients (1.16 ± 0.13) than that for healthy subjects (1.39 ± 0.08; p < 0.001) and than that for MCI patients (1.32 ± 0.11; p < 0.001). A ratio threshold of 1.23 allowed to detect AE patients with a sensitivity of 71% and a specificity of 82% against MCI patients, and 98% against healthy subjects. A lower cortex/striatum metabolic ratio had a trend towards shorter delay before 18F-FDG PET/CT (p = 0.07) in multivariate analysis. CONCLUSION The decrease in the cortex/striatal metabolic ratio has a good early diagnostic performance for the differentiation of AE patients from controls.
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46
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Li C, Pang X, Shi K, Long Q, Liu J, Zheng J. The Insula Is a Hub for Functional Brain Network in Patients With Anti- N-Methyl-D-Aspartate Receptor Encephalitis. Front Neurosci 2021; 15:642390. [PMID: 33790737 PMCID: PMC8005702 DOI: 10.3389/fnins.2021.642390] [Citation(s) in RCA: 2] [Impact Index Per Article: 0.5] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 12/16/2020] [Accepted: 01/29/2021] [Indexed: 01/17/2023] Open
Abstract
BACKGROUND In recent years, imaging technologies have been rapidly evolving, with an emphasis on the characterization of brain structure changes and functional imaging in patients with autoimmune encephalitis. However, the neural basis of anti-N-methyl-D-aspartate receptor (NMDAR) encephalitis and its linked cognitive decline is unclear. Our research aimed to assess changes in the functional brain network in patients with anti-NMDAR encephalitis and whether these changes lead to cognitive impairment. METHODS Twenty-one anti-NMDAR encephalitis patients and 22 age-, gender-, and education status-matched healthy controls were assessed using resting functional magnetic resonance imaging (fMRI) scanning and neuropsychological tests, including the Hamilton Depression Scale (HAMD24), the Montreal Cognitive Assessment (MoCA), and the Hamilton Anxiety Scale (HAMA). A functional brain network was constructed using fMRI, and the topology of the network parameters was analyzed using graph theory. Next, we extracted the aberrant topological parameters of the functional network as seeds and compared causal connectivity with the whole brain. Lastly, we explored the correlation of aberrant topological structures with deficits in cognitive performance. RESULTS Relative to healthy controls, anti-NMDAR encephalitis patients exhibited decreased MoCA scores and increased HAMA and HAMD24 scores (p < 0.05). The nodal clustering coefficient and nodal local efficiency of the left insula (Insula_L) were significantly decreased in anti-NMDAR encephalitis patients (p < 0.05 following Bonferroni correction). Moreover, anti-NMDAR encephalitis patients showed a weakened causal connectivity from the left insula to the left inferior parietal lobe (Parietal_Inf_L) compared to healthy controls. Conversely, the left superior parietal lobe (Parietal_sup_L) exhibited an enhanced causal connectivity to the left insula in anti-NMDAR encephalitis patients compared to controls. Unexpectedly, these alterations were not correlated with any neuropsychological test scores. CONCLUSION This research describes topological abnormalities in the functional brain network in anti-NMDAR encephalitis. These results will be conducive to understand the structure and function of the brain network of patients with anti-NMDAR encephalitis and further explore the neuropathophysiological mechanisms.
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Affiliation(s)
- Chunyan Li
- Department of Neurology, The First Affiliated Hospital of Guangxi Medical University, Nanning, China
| | - Xiaomin Pang
- Department of Radiology, The First Affiliated Hospital of Guangxi Medical University, Nanning, China
| | - Ke Shi
- Department of Neurology, The First Affiliated Hospital of Guangxi Medical University, Nanning, China
| | - Qijia Long
- Department of Neurology, The First Affiliated Hospital of Guangxi Medical University, Nanning, China
| | - Jinping Liu
- Department of Neurology, The First Affiliated Hospital of Guangxi Medical University, Nanning, China
| | - Jinou Zheng
- Department of Neurology, The First Affiliated Hospital of Guangxi Medical University, Nanning, China
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47
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Wang J, Duan Y, Zhang T, Huang J, Ren Z, Ye J, Wang N, Li Y, Chen X, Gao P, Li K, Liu Y. Aberrant multimodal brain networks in patients with anti-NMDA receptor encephalitis. CNS Neurosci Ther 2021; 27:652-663. [PMID: 33713553 PMCID: PMC8111502 DOI: 10.1111/cns.13632] [Citation(s) in RCA: 10] [Impact Index Per Article: 2.5] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 06/04/2020] [Revised: 01/26/2021] [Accepted: 02/17/2021] [Indexed: 11/30/2022] Open
Abstract
Aims To explore large‐scale brain network alterations and examine their clinical and neuropsychological relevance in patients with anti‐N‐methyl‐D‐aspartate receptor (NMDAR) encephalitis. Methods Twenty‐four patients with anti‐NMDAR encephalitis and 26 matched healthy controls (HCs) were enrolled in our study. Based on the multimodal MRI dataset, individual morphological, structural, and functional brain networks were constructed and compared between the two groups at multiple levels. The associations with clinical/neuropsychological variables and the discriminant ability of significant alterations were further studied. Results Multimodal network analysis revealed that anti‐NMDAR encephalitis mainly affected morphological and structural networks, but subtle alterations were observed in functional networks. Intriguingly, decreased network local efficiency was observed for both morphological and structural networks and increased nodal centrality in the lateral orbital gyrus was convergently observed among the three types of networks in the patients. Moreover, the alterations, particularly those from structural networks, accounted largely for cognitive deficits of the patients and could distinguish the diseased individuals from the HCs with excellent performance (area under the curve =0.933). Conclusions The current study provides a comprehensive view of characteristic multimodal network dysfunction in anti‐NMDAR encephalitis, which is crucial to establish new diagnostic biomarkers and promising therapeutic targets for the disease.
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Affiliation(s)
- Jinhui Wang
- Institute for Brain Research and Rehabilitation, Guangdong Key Laboratory of Mental Health and Cognitive Science, Center for Studies of Psychological Application, South China Normal University, Guangzhou, China.,Key Laboratory of Brain, Cognition and Education Sciences (South China Normal University), Ministry of Education, Guangzhou, China
| | - Yunyun Duan
- Department of Radiology, Beijing Tiantan Hospital, Capital Medical University, Beijing, China
| | - Tian Zhang
- Department of Radiology, Xuanwu Hospital, Capital Medical University, Beijing, China
| | - Jing Huang
- Department of Radiology, Xuanwu Hospital, Capital Medical University, Beijing, China
| | - Zhuoqiong Ren
- Department of Radiology, Xuanwu Hospital, Capital Medical University, Beijing, China
| | - Jing Ye
- Department of Radiology, Xuanwu Hospital, Capital Medical University, Beijing, China
| | - Ningkai Wang
- Institute for Brain Research and Rehabilitation, Guangdong Key Laboratory of Mental Health and Cognitive Science, Center for Studies of Psychological Application, South China Normal University, Guangzhou, China
| | - Yinzhi Li
- Institute for Brain Research and Rehabilitation, Guangdong Key Laboratory of Mental Health and Cognitive Science, Center for Studies of Psychological Application, South China Normal University, Guangzhou, China
| | - Xiaoya Chen
- Department of Radiology, The First Affiliated Hospital of Chongqing Medical University, Chongqing, China
| | - Peiyi Gao
- Department of Radiology, Beijing Tiantan Hospital, Capital Medical University, Beijing, China
| | - Kuncheng Li
- Department of Radiology, Xuanwu Hospital, Capital Medical University, Beijing, China
| | - Yaou Liu
- Department of Radiology, Beijing Tiantan Hospital, Capital Medical University, Beijing, China.,Tiantan Image Research Center, China National Clinical Research Center for Neurological Diseases, Beijing, China
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Caquot PA, Zizi G, Lelièvre M, Dejust S, Morland D. 18F-FDG PET/CT in Anti-Leucine-Rich Glioma-Inactivated 1 Antibody Encephalitis: Typical Pattern and Follow-up. Clin Nucl Med 2021; 46:250-251. [PMID: 33323741 DOI: 10.1097/rlu.0000000000003469] [Citation(s) in RCA: 2] [Impact Index Per Article: 0.5] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/27/2022]
Abstract
ABSTRACT We report the case of a 55-year-old man presenting pseudopsychiatric behavior disorders of subacute-onset. MRI showed a FLAIR (fluid-attenuated inversion recovery) hyperintensity in the left hippocampus. The diagnosis of limbic encephalitis was raised, and the patient was referred for an 18F-FDG PET/CT. PET/CT depicted an increased uptake of the left mesiotemporal structures and also an increased uptake of both cerebellum and striatal areas. This pattern was compatible with an anti-leucine-rich glioma-inactivated 1 antibody encephalitis that was later confirmed.
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Affiliation(s)
| | - Ghali Zizi
- From the Médecine Nucléaire, Institut Godinot
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49
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Ge J, Deng B, Guan Y, Bao W, Wu P, Chen X, Zuo C. Distinct cerebral 18F-FDG PET metabolic patterns in anti-N-methyl-D-aspartate receptor encephalitis patients with different trigger factors. Ther Adv Neurol Disord 2021; 14:1756286421995635. [PMID: 33717212 PMCID: PMC7919218 DOI: 10.1177/1756286421995635] [Citation(s) in RCA: 5] [Impact Index Per Article: 1.3] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 07/14/2020] [Accepted: 01/23/2021] [Indexed: 11/23/2022] Open
Abstract
Aim: Anti-N-methyl-D-aspartate receptor (anti-NMDAR) encephalitis is a subgroup of
treatable autoimmune encephalitis, characterized by rapid development of
psychosis, cognitive impairments and seizures. Etiologically, anti-NMDAR
encephalitis could be divided into three subgroups, which are paraneoplastic
(especially associated with ovarian teratoma), viral encephalitis-related
and cryptogenic. Each type is different in clinical course, treatment
strategies and prognosis. In this study, we aim to investigate whether
anti-NMDAR encephalitis patients with different trigger factors exhibit
distinct cerebral metabolic patterns detected by
18F-fluorodeoxyglucose positron emission tomography imaging. Methods: 24 patients with anti-NMDAR encephalitis in acute phase from Huashan
Hospital, Fudan University (Shanghai, China) were recruited in this study.
Each patient was classified into one of etiological subgroups. Positron
emission tomography images of individual patients were analyzed with both
routine visual reading and computer-supported reading by comparison with
those of the same 10 healthy controls using a voxel-wise statistical
parametric mapping analysis. Results: Patients in both the cryptogenic (13 patients) and paraneoplastic (five
patients) subgroups showed hypermetabolism in the frontal-temporal lobes and
basal ganglia, covarying with hypometabolism in the occipital regions.
Notably, the abnormal metabolism was usually asymmetric in the cryptogenic
subgroup, but relatively symmetric in the paraneoplastic subgroup. Moreover,
the other six patients secondary to viral encephalitis presented with
significant hypometabolism in the bilateral occipital regions, as well as in
the unilateral temporal lobes and part of basal ganglia (also is virus
infection side), but hypermetabolism in the contralateral temporal
areas. Conclusion: This study revealed that patients with anti-NMDAR encephalitis triggered by
different factors presented distinct cerebral metabolic patterns. Awareness
of these patterns may help to better understand the varying occurrence and
development of anti-NMDAR encephalitis in each subgroup, and could offer
valuable information to the early diagnosis, treatment and prognosis of this
disorder. Trial registration number ChiCTR2000029115 (Chinese clinical trial registry site, http://www.chictr.org)
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Affiliation(s)
- Jingjie Ge
- PET Center, Huashan Hospital, Fudan University, Shanghai, China
| | - Bo Deng
- Department of Neurology, Huashan Hospital, Fudan University, Shanghai, China
| | - Yihui Guan
- PET Center, Huashan Hospital, Fudan University, Shanghai, China
| | - Weiqi Bao
- PET Center, Huashan Hospital, Fudan University, Shanghai, China
| | - Ping Wu
- PET Center, Huashan Hospital, Fudan University, Shanghai, China
| | - Xiangjun Chen
- Department of Neurology, Huashan Hospital and Institute of Neurology, Fudan University, 12 Middle Wulumuqi Road, Jing'an District, Shanghai 200040, China
| | - Chuantao Zuo
- PET Center, Huashan Hospital, Fudan University, 518 East Wuzhong Road, Shanghai 200235, China
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50
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Reversible mixed perfusion on 123I-IMP SPECT in anti-AMPA receptor encephalitis: A case report. J Neurol Sci 2021; 421:117306. [PMID: 33450618 DOI: 10.1016/j.jns.2020.117306] [Citation(s) in RCA: 2] [Impact Index Per Article: 0.5] [Reference Citation Analysis] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 10/21/2020] [Revised: 12/10/2020] [Accepted: 12/28/2020] [Indexed: 12/31/2022]
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