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Jha R, Blitz SE, Chua MMJ, Warren AEL, Lee JW, Rolston JD. Surgical management of status epilepticus: A systematic review. Epilepsia Open 2024; 9:850-864. [PMID: 38456595 PMCID: PMC11145616 DOI: 10.1002/epi4.12924] [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: 08/11/2023] [Revised: 11/07/2023] [Accepted: 02/09/2024] [Indexed: 03/09/2024] Open
Abstract
Status Epilepticus (SE), unresponsive to medical management, is associated with high morbidity and mortality. Surgical management is typically considered in these refractory cases. The best surgical approach for affected patients remains unclear; however, given the lack of controlled trials exploring the role of surgery. We performed a systematic review according to PRIMSA guidelines, including case reports and series describing surgical interventions for patients in SE. Cases (157 patients, median age 12.9 years) were followed for a median of 12 months. Patients were in SE for a median of 21 days before undergoing procedures including: focal resection (36.9%), functional hemispherectomy (21%), lobar resection (12.7%), vagus nerve stimulation (VNS) (12.7%), deep brain stimulation (DBS) (6.4%), multiple subpial transection (MST) (3.8%), responsive neurostimulation (RNS) (1.9%), and cortical stimulator placement (1.27%), with 24 patients undergoing multiple procedures. Multiple SE semiologies were identified. 47.8% of patients had focal seizures, and 65% of patients had focal structural abnormalities on MRI. SE persisted for 36.8 ± 47.7 days prior to surgical intervention. SE terminated following surgery in 81.5%, terminated with additional adjuncts in 10.2%, continued in 1.9%, and was not specified in 6.4% of patients. Long-term seizure outcomes were favorable, with the majority improved and 51% seizure-free. Eight patients passed away in follow-up, of which three were in SE. Seizures emerging from one hemisphere were both more likely to immediately terminate (OR 4.7) and lead to long-term seizure-free status (OR 3.9) compared to nonunilateral seizures. No other predictors, including seizure focality, SE duration, or choice of surgical procedure, were predictors of SE termination. Surgical treatment of SE can be effective in terminating SE and leading to sustained seizure freedom, with many different procedures showing efficacy if matched appropriately with SE semiology and etiology. PLAIN LANGUAGE SUMMARY: Patients with persistent seizures (Status Epilepticus) that do not stop following medications can be treated effectively with surgery. Here, we systematically review the entirety of existing literature on surgery for treating status epilepticus to better identify how and when surgery is used and what patients do after surgery.
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Affiliation(s)
- Rohan Jha
- Harvard Medical SchoolBostonMassachusettsUSA
- Department of Neurosurgery, Harvard Medical SchoolBrigham and Women's HospitalBostonMassachusettsUSA
| | - Sarah E. Blitz
- Harvard Medical SchoolBostonMassachusettsUSA
- Department of Neurosurgery, Harvard Medical SchoolBrigham and Women's HospitalBostonMassachusettsUSA
| | - Melissa M. J. Chua
- Department of Neurosurgery, Harvard Medical SchoolBrigham and Women's HospitalBostonMassachusettsUSA
| | - Aaron E. L. Warren
- Department of Neurosurgery, Harvard Medical SchoolBrigham and Women's HospitalBostonMassachusettsUSA
| | - Jong Woo Lee
- Department of Neurology, Harvard Medical SchoolBrigham and Women's HospitalBostonMassachusettsUSA
| | - John D. Rolston
- Department of Neurosurgery, Harvard Medical SchoolBrigham and Women's HospitalBostonMassachusettsUSA
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Mantoan Ritter L, Selway R. Perspective: Vagal nerve stimulation in the treatment of new-onset refractory status epilepticus. Front Neurol 2023; 14:1172898. [PMID: 37153661 PMCID: PMC10157031 DOI: 10.3389/fneur.2023.1172898] [Citation(s) in RCA: 6] [Impact Index Per Article: 3.0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Journal Information] [Subscribe] [Scholar Register] [Received: 02/24/2023] [Accepted: 03/30/2023] [Indexed: 05/10/2023] Open
Abstract
Introduction Resistance to drug therapy is a major hurdle in new-onset refractory status epilepticus (NORSE) treatment and there is urgent need to develop new treatment approaches. Non-drug approaches such as neuromodulation offer significant benefits and should be investigated as new adjunct treatment modalities. An important unanswered question is whether desynchronizing networks by vagal nerve stimulation (VNS) may improve seizure control in NORSE patients. Main text We present a summary of published NORSE cases treated with VNS and our own data, discuss possible mechanisms of action, review VNS implantation timing, stimulation setting titration protocols and outcomes. Further, we propose avenues for future research. Discussion We advocate for consideration of VNS for NORSE both in early and late stages of the presentation and hypothesize a possible additional benefit from implantation in the acute phase of the disease. This should be pursued in the context of a clinical trial, harmonizing inclusion criteria, accuracy of documentation and treatment protocols. A study planned within our UK-wide NORSE-UK network will answer the question if VNS may confer benefits in aborting unremitting status epilepticus, modulate ictogenesis and reduce long-term chronic seizure burden.
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Affiliation(s)
- Laura Mantoan Ritter
- Epilepsy Centre, Clinical Neurosciences Department, King's College NHS Foundation Trust, London, United Kingdom
- Maurice Wohl Clinical Neuroscience Institute, Institute of Psychiatry, Psychology and Neuroscience, King's College London, London, United Kingdom
- *Correspondence: Laura Mantoan Ritter
| | - Richard Selway
- Epilepsy Centre, Clinical Neurosciences Department, King's College NHS Foundation Trust, London, United Kingdom
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3
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Espino PH, Burneo JG, Moscol G, Gofton T, MacDougall K, Suller Marti A. Long-term outcomes after NORSE: Treatment with vagus nerve stimulation. Epilepsia Open 2022; 7:822-828. [PMID: 36177520 PMCID: PMC9712472 DOI: 10.1002/epi4.12654] [Citation(s) in RCA: 11] [Impact Index Per Article: 3.7] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 05/19/2022] [Accepted: 09/26/2022] [Indexed: 12/30/2022] Open
Abstract
New-onset refractory status epilepticus (NORSE) is associated with high mortality, therapy-resistant epilepsy (TRE), and poor cognitive and functional outcomes. Some patients develop multifocal TRE, for whom surgery with a curative intention, is not an option. In these patients, vagus nerve stimulation (VNS) is performed as a palliative treatment. We report the long-term outcomes regarding seizure frequency, functional and cognitive outcome, and effectiveness of VNS in two patients with TRE as a consequence of NORSE. In the first patient with cryptogenic NORSE, VNS implantation occurred during the acute stage, probably contributing to the cessation of her status epilepticus. However, in the long-term follow-up, the patient persisted with daily multifocal seizures. In the second patient, VNS implantation was delayed to manage his epilepsy when the NORSE, ultimately due to autoimmune encephalitis, had resolved. During long-term follow-up, no reduction in seizure frequency was achieved. This evidence supporting the use of VNS in patients with TRE after NORSE warrants further investigation.
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Affiliation(s)
- Poul H. Espino
- Epilepsy Program, Schulich School of Medicine and DentistryWestern UniversityLondonOntarioCanada
| | - Jorge G. Burneo
- Epilepsy Program, Schulich School of Medicine and DentistryWestern UniversityLondonOntarioCanada,Neuroepidemiology Unit, Schulich School of Medicine and DentistryWestern UniversityLondonOntarioCanada
| | - Gaby Moscol
- Epilepsy Program, Schulich School of Medicine and DentistryWestern UniversityLondonOntarioCanada
| | - Teneille Gofton
- Epilepsy Program, Schulich School of Medicine and DentistryWestern UniversityLondonOntarioCanada
| | - Keith MacDougall
- Epilepsy Program, Schulich School of Medicine and DentistryWestern UniversityLondonOntarioCanada
| | - Ana Suller Marti
- Epilepsy Program, Schulich School of Medicine and DentistryWestern UniversityLondonOntarioCanada,Paediatrics Department, Schulich School of Medicine and DentistryWestern UniversityLondonOntarioCanada
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4
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Zou Q, Chen P, Chen J, Chen D, Xia H, Chen L, Feng H, Feng L. Multisystem Involvement Induced by Human Parvovirus B19 Infection in a Non-immunosuppressed Adult: A Case Report. Front Med (Lausanne) 2022; 9:808205. [PMID: 35492345 PMCID: PMC9039281 DOI: 10.3389/fmed.2022.808205] [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: 11/03/2021] [Accepted: 03/16/2022] [Indexed: 11/13/2022] Open
Abstract
Background Human parvovirus B19 (B19V) infection is usually symptomless and occurs in the childhood. While in immunocompromised adults, B19V infection also presents various clinical symptoms due to the host's immune status. The classic symptoms include erythema, anemia, arthropathy, and edema, but neurological involvement is rare. Case Presentation In this report, we present a case of B19V infection caused multiple organ dysfunction in a non-immunosuppressed adult. Metagenomic next-generation sequencing (mNGS) was used and successfully detected the pathogen in multiple types of samples, including blood, cerebrospinal fluid (CSF), and bronchoalveolar lavage fluid (BALF). The diagnosis was subsequently confirmed by polymerase chain reaction (PCR). He was treated with intravenous gamma globulin, resulting in a significant resolution of symptoms after 1 month. Conclusion Multisystem involvement induced by B19V infection was found in this case report. mNGS performed great advantages in rapidly and accurately diagnosing B19V infection in multiple types of samples, which helps the timely adjustment of treatment and improves the prognosis.
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Affiliation(s)
- Qihang Zou
- Department of Neurology, The First Affiliated Hospital, Sun Yat-sen University, Guangzhou, China.,Guangdong Provincial Key Laboratory of Diagnosis and Treatment of Major Neurological Diseases, National Key Clinical Department and Key Discipline of Neurology, Guangzhou, China
| | - Peisong Chen
- Department of Laboratory Medicine, The First Affiliated Hospital, Sun Yat-sen University, Guangzhou, China
| | - Jiaxin Chen
- Department of Neurology, The First Affiliated Hospital, Sun Yat-sen University, Guangzhou, China.,Guangdong Provincial Key Laboratory of Diagnosis and Treatment of Major Neurological Diseases, National Key Clinical Department and Key Discipline of Neurology, Guangzhou, China
| | - Dingbang Chen
- Department of Neurology, The First Affiliated Hospital, Sun Yat-sen University, Guangzhou, China.,Guangdong Provincial Key Laboratory of Diagnosis and Treatment of Major Neurological Diseases, National Key Clinical Department and Key Discipline of Neurology, Guangzhou, China
| | - Han Xia
- Department of Scientific Affairs, Hugobiotech Co., Ltd., Beijing, China
| | - Ling Chen
- Department of Neurology, The First Affiliated Hospital, Sun Yat-sen University, Guangzhou, China.,Guangdong Provincial Key Laboratory of Diagnosis and Treatment of Major Neurological Diseases, National Key Clinical Department and Key Discipline of Neurology, Guangzhou, China
| | - Huiyu Feng
- Department of Neurology, The First Affiliated Hospital, Sun Yat-sen University, Guangzhou, China.,Guangdong Provincial Key Laboratory of Diagnosis and Treatment of Major Neurological Diseases, National Key Clinical Department and Key Discipline of Neurology, Guangzhou, China
| | - Li Feng
- Department of Neurology, The First Affiliated Hospital, Sun Yat-sen University, Guangzhou, China.,Guangdong Provincial Key Laboratory of Diagnosis and Treatment of Major Neurological Diseases, National Key Clinical Department and Key Discipline of Neurology, Guangzhou, China
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5
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Dibué-Adjei M, Brigo F, Yamamoto T, Vonck K, Trinka E. Vagus nerve stimulation in refractory and super-refractory status epilepticus - A systematic review. Brain Stimul 2019; 12:1101-1110. [PMID: 31126871 DOI: 10.1016/j.brs.2019.05.011] [Citation(s) in RCA: 58] [Impact Index Per Article: 9.7] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 01/21/2019] [Revised: 05/06/2019] [Accepted: 05/08/2019] [Indexed: 11/16/2022] Open
Abstract
RATIONALE Refractory status epilepticus (RSE) is the persistence of status epilepticus despite second-line treatment. Super-refractory SE (SRSE) is characterized by ongoing status despite 48 h of anaesthetic treatment. Due to the high case fatality in RSE of 16-39%, off label treatments without strong evidence of efficacy in RSE are often administered. In single case-reports and small case series totalling 28 patients, acute implantation of VNS in RSE was associated with 76% and 26% success rate in generalized and focal RSE respectively. We performed an updated systematic review of the literature on efficacy of VNS in RSE/SRSE by including all reported patients. METHODS We systematically searched EMBASE, CENTRAL, Opengre.eu, and ClinicalTrials.gov, and PubMed databases to identify studies reporting the use of VNS for RSE and/or SRSE. We also searched conference abstracts from AES and ILAE meetings. RESULTS 45 patients were identified in total of which 38 were acute implantations of VNS in RSE/SRSE. Five cases had VNS implantation for epilepsia partialis continua, one for refractory electrical status epilepticus in sleep and one for acute encephalitis with refractory repetitive focal seizures. Acute VNS implantation was associated with cessation of RSE/SRSE in 74% (28/38) of acute cases. Cessation did not occur in 18% (7/38) of cases and four deaths were reported (11%); all of them due to the underlying disease and unlikely related to VNS implantation. Median duration of the RSE/SRSE episode pre and post VNS implantation was 18 days (range: 3-1680 days) and 8 days (range: 3-84 days) respectively. Positive outcomes occurred in 82% (31/38) of cases. CONCLUSION VNS can interrupt RSE and SRSE in 74% of patients; data originate from reported studies classified as level IV and the risk for reporting bias is high. Further prospective studies are warranted to investigate acute VNS in RSE and SRSE.
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Affiliation(s)
- Maxine Dibué-Adjei
- LivaNova Deutschland GmbH, LivaNova PLC-owned Subsidiary, Lindberghstraße 25, 80939, Munich, Germany; Department of Neurosurgery, Medical Faculty, Heinrich-Heine-University, Moorenstraße 5, D-40225, Düsseldorf, Germany.
| | - Francesco Brigo
- Department of Neuroscience, Biomedicine and Movement Sciences, University of Verona, Verona, Italy; Department of Neurology, Franz Tappeiner Hospital, Merano, Italy
| | - Takamichi Yamamoto
- Comprehensive Epilepsy Center, Seirei Hamamatsu General Hospital, Shizuoka, Japan
| | - Kristl Vonck
- Brain Research Team, Department of Neurology, Ghent University, Ghent, Belgium
| | - Eugen Trinka
- Department of Neurology, Christian-Doppler University Hospital, Paracelsus Medical University, Centre for Cognitive Neuroscience, Salzburg, Austria; Institute of Public Health, Medical Decision Making and HTA, UMIT, Private University for Health Sciences, Medical Informatics and Technology, Hall in Tyrol, Austria
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6
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Clinical manifestations and treatment outcomes of parvovirus B19 encephalitis in immunocompetent adults. J Neurovirol 2017; 23:903-907. [PMID: 28895082 DOI: 10.1007/s13365-017-0568-2] [Citation(s) in RCA: 3] [Impact Index Per Article: 0.4] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 03/28/2017] [Revised: 08/02/2017] [Accepted: 08/21/2017] [Indexed: 10/18/2022]
Abstract
Parvovirus B19 (PVB19) has rarely been identified as a cause of encephalitis in immunocompetent adults, in whom clinical information regarding PVB19 encephalitis has remained unclear. Herein, we report the clinical presentations, laboratory and imaging findings, and treatment outcomes of five immunocompetent adults with PVB19 encephalitis. Although none of the patients showed any distinctive features of PVB19 infection, they showed various clinical manifestations, including one instance of brainstem involvement. Additionally, immunotherapy can be considered an effective approach, especially in immunocompetent adults with PVB19 encephalitis who are resistant to the initial management.
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7
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Palermo CI, Costanzo CM, Franchina C, Castiglione G, Giuliano L, Russo R, Conti A, Sofia V, Scalia G. Focal epilepsy as a long term sequela of Parvovirus B19 encephalitis. J Clin Virol 2016; 80:20-3. [PMID: 27130981 DOI: 10.1016/j.jcv.2016.04.007] [Citation(s) in RCA: 4] [Impact Index Per Article: 0.4] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 02/01/2016] [Revised: 04/08/2016] [Accepted: 04/11/2016] [Indexed: 11/19/2022]
Abstract
Human Parvovirus B19 (PVB19), the etiological agent of the fifth disease, is associated with a large spectrum of pathologies, among which is encephalitis. Since it has been detected from the central nervous system in children or in immunocompromised patients, its causative role in serious neurological manifestations is still unclear. Here we report the case of an 18-year-old healthy boy who developed encephalitis complicated by prolonged status epilepticus. The detection of PVB19 DNA in his serum and, subsequently, in his cerebrospinal fluid supports the hypothesis that this virus could potentially play a role in the pathogenesis of neurological complications. In addition, the detection of viral DNA and the presence of specific IgM and IgG antibodies in serum, together with clinical findings such as skin rash, support the presence of a disseminated viral infection. In the presence of neurological disorders, especially when there are no specific signs, but seizures and rash are present, it is important to search for PVB19 both in immunocompromised and immunocompetent patients. Moreover, the introduction of the PVB19 DNA test into diagnostic protocols of neuropathies, especially those undiagnosed, could clarify the etiological agent that otherwise could remain unrecognized.
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Affiliation(s)
- Concetta Ilenia Palermo
- Department of Biomedical and Biotechnological Sciences, University of Catania, Italy; O.U. Central Laboratory, Clinical Virology Unit, A.O.U Policlinico-Vittorio Emanuele, O.P "Gaspare Rodolico", Catania, Italy.
| | - Carmela Maria Costanzo
- Department of Biomedical and Biotechnological Sciences, University of Catania, Italy; O.U. Central Laboratory, Clinical Virology Unit, A.O.U Policlinico-Vittorio Emanuele, O.P "Gaspare Rodolico", Catania, Italy
| | - Concetta Franchina
- Department of Biomedical and Biotechnological Sciences, University of Catania, Italy; O.U. Central Laboratory, Clinical Virology Unit, A.O.U Policlinico-Vittorio Emanuele, O.P "Gaspare Rodolico", Catania, Italy
| | - Giacomo Castiglione
- O.U. Intensive Care, A.O.U Policlinico-Vittorio Emanuele, O.P "Vittorio Emanuele II", Catania, Italy
| | - Loretta Giuliano
- Department "G. F. Ingrassia", Section of Neurosciences, University of Catania, Italy
| | - Raffaela Russo
- Department of Biomedical and Biotechnological Sciences, University of Catania, Italy; O.U. Central Laboratory, Clinical Virology Unit, A.O.U Policlinico-Vittorio Emanuele, O.P "Gaspare Rodolico", Catania, Italy
| | - Alessandro Conti
- O.U. Intensive Care, A.O.U Policlinico-Vittorio Emanuele, O.P "Vittorio Emanuele II", Catania, Italy
| | - Vito Sofia
- Department "G. F. Ingrassia", Section of Neurosciences, University of Catania, Italy
| | - Guido Scalia
- Department of Biomedical and Biotechnological Sciences, University of Catania, Italy; O.U. Central Laboratory, Clinical Virology Unit, A.O.U Policlinico-Vittorio Emanuele, O.P "Gaspare Rodolico", Catania, Italy
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8
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Watanabe T, Kawashima H. Acute encephalitis and encephalopathy associated with human parvovirus B19 infection in children. World J Clin Pediatr 2015; 4:126-134. [PMID: 26566485 PMCID: PMC4637803 DOI: 10.5409/wjcp.v4.i4.126] [Citation(s) in RCA: 19] [Impact Index Per Article: 1.9] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 06/24/2015] [Revised: 08/11/2015] [Accepted: 09/10/2015] [Indexed: 02/06/2023] Open
Abstract
Reports of neurologic manifestations of human parvovirus B19 (B19) infection have been on the rise. Acute encephalitis and encephalopathy is the most common, accounting for 38.8% of total B19-associated neurological manifestations. To date, 34 children with B19 encephalitis and encephalopathy have been reported, which includes 21 encephalitis and 13 encephalopathy cases. Ten (29%) were immunocompromised and 17 (39%) had underlying diseases. Fever at the onset of disease and rash presented in 44.1% and 20.6% of patients, respectively. Neurological manifestations include alteration of consciousness occurred in all patients, seizures in 15 (44.1%) patients, and focal neurologic signs in 12 (35.3%) patients. Anemia and pleocytosis in cerebrospinal fluid (CSF) occurred in 56.3% and 48.1% of patients, respectively. Serum Anti-B19 IgM (82.6%) and CSF B19 DNA (90%) were positive in the majority of cases. Some patients were treated with intravenous immunoglobulins and/or steroids, although an accurate evaluation of the efficacy of these treatment modalities cannot be determined. Nineteen (57.6%) patients recovered completely, 11 (33.3%) patients had some neurological sequelae and 3 (8.8%) patients died. Although the precise pathogenesis underlying the development of B19 encephalitis and encephalopathy is unclear, direct B19 infection or NS1protein of B19 toxicity in the brain, and immune-mediated brain injuries have been proposed.
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Barah F, Whiteside S, Batista S, Morris J. Neurological aspects of human parvovirus B19 infection: a systematic review. Rev Med Virol 2014; 24:154-68. [PMID: 24459081 PMCID: PMC4238837 DOI: 10.1002/rmv.1782] [Citation(s) in RCA: 66] [Impact Index Per Article: 6.0] [Reference Citation Analysis] [Abstract] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 10/22/2013] [Revised: 11/28/2013] [Accepted: 11/29/2013] [Indexed: 02/06/2023]
Abstract
Parvovirus B19 has been linked with various clinical syndromes including neurological manifestations. However, its role in the latter remains not completely understood. Although the last 10 years witnessed a surge of case reports on B19-associated neurological aspects, the literature data remains scattered and heterogeneous, and epidemiological information on the incidence of B19-associated neurological aspects cannot be accurately extrapolated. The aim of this review is to identify the characteristics of cases of B19-associated neurological manifestations. A computerized systematic review of existing literature concerning cases of B19-related neurological aspects revealed 89 articles describing 129 patients; 79 (61.2%) were associated with CNS manifestations, 41 (31.8%) were associated with peripheral nervous system manifestations, and 9 (7.0%) were linked with myalgic encephalomyelitis. The majority of the cases (50/129) had encephalitis. Clinical characteristic features of these cases were analyzed, and possible pathological mechanisms were also described. In conclusion, B19 should be included in differential diagnosis of encephalitic syndromes of unknown etiology in all age groups. Diagnosis should rely on investigation of anti-B19 IgM antibodies and detection of B19 DNA in serum or CSF. Treatment of severe cases might benefit from a combined regime of intravenous immunoglobulins and steroids. To confirm these outcomes, goal-targeted studies are recommended to exactly identify epidemiological scenarios and explore potential pathogenic mechanisms of these complications. Performing retrospective and prospective and multicenter studies concerning B19 and neurological aspects in general, and B19 and encephalitic syndromes in particular, are required. © 2014 The Authors. Reviews in Medical Virology published by John Wiley & Sons, Ltd.
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Affiliation(s)
- Faraj Barah
- Center for Neuroscience and Cell Biology, Faculty of Medicine, University of Coimbra, Coimbra, Portugal
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10
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Grant JK, Yin NC, Zaytoun AM, Waseem H, Hobbs JA. Persistent adeno-associated virus 2 and parvovirus B19 sequences in post-mortem human cerebellum. THE CEREBELLUM 2009; 8:490-8. [PMID: 19585179 DOI: 10.1007/s12311-009-0126-4] [Citation(s) in RCA: 11] [Impact Index Per Article: 0.7] [Reference Citation Analysis] [Abstract] [Track Full Text] [Subscribe] [Scholar Register] [Received: 03/19/2009] [Accepted: 06/24/2009] [Indexed: 12/18/2022]
Abstract
We previously reported in a large cohort (N = 104) of post-mortem tissues the detection of both the non-pathogenic adeno-associated virus (AAV2) in approximately 13% and the pathogenic human parvovirus B19 (B19) in approximately 42% of human brains, particularly the dorsolateral prefrontal cortex. Multiple animal parvoviruses target the developing cerebellum (CBLM) resulting in hypoplasia and ataxia, but very little is known about the human parvoviruses and their ability to infect or cause disease in the CBLM. We have now confirmed in the above cohort the presence of AAV2 and B19 sequences in the CBLM. Our results show that approximately 27% and approximately 70% of human CBLM are positive by nested polymerase chain reaction for AAV2 and B19 sequences, respectively. We also document in a second cohort (N = 10) the presence of AAV2 (50%) and B19 (100%) sequences in the CBLM and correlate our results for B19 with studies from matched sera. Eighty percent (80%) of this cohort was positive for anti-B19 IgG, while none were IgM+, suggesting that most individuals had been previously infected with B19 but none acutely. To our knowledge, this study is the first to demonstrate that both AAV2 and B19 sequences are present at relatively high frequencies in the CBLM and are likely due to persistent rather than acute infection. Further studies will lead to insights into AAV2- and/or B19-CBLM interactions including mechanisms of infection, persistence, and possibly neuropathology, including cerebellar hypoplasia and ataxia.
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Affiliation(s)
- James K Grant
- Department of Psychiatry, University of Florida College of Medicine, Gainesville, FL 32610, USA
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11
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Hobbs JA. Detection of adeno-associated virus 2 and parvovirus B19 in the human dorsolateral prefrontal cortex. J Neurovirol 2006; 12:190-9. [PMID: 16877300 DOI: 10.1080/13550280600827351] [Citation(s) in RCA: 27] [Impact Index Per Article: 1.4] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 02/07/2023]
Abstract
Although animal parvoviruses have long been recognized as causes of brain pathology in multiple animal models, especially during early development, human parvoviruses are rarely thought of as neurotropic or causes of neuropathology in humans. However, several recent case reports have suggested possible associations of parvovirus B19 (B19) infection with various neurological and neuropsychiatric symptoms. Adeno-associated virus 2 (AAV2) is related to B19 but has thus far not been shown to be associated with any human disease but is of clinical interest because of the recent use of recombinant AAV vectors in human gene therapy, including gene delivery to the brain. To date, there have been no large-scale studies of the propensity of wild-type human parvoviruses to infect the brain. The Stanley Medical Research Institute Brain Collection offered a unique opportunity to study a large sample (n = 104) of dorsolateral prefrontal cortex (DLPC) DNAs isolated from unaffected control, schizophrenic, and bipolar disorder brains for the presence of parvoviral sequences. This is the first investigator-blinded study to document the presence of parvoviral sequences in the DLPC by utilizing highly sensitive nested polymerase chain reaction (nPCR) and DNA sequencing. Of the overall sample, 6.7% to 12.5% were positive for AAV2, and 14.4% to 42.3% were positive for B19 sequences, with no statistical differences among subgroups. This is the first report to demonstrate the presence of human parvoviruses in a large cohort of adult DLPC, which underscores the need to gain a better insight into the basic biology of parvovirus-brain interactions, including mechanisms of infection and persistence.
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Affiliation(s)
- Jacqueline A Hobbs
- Department of Psychiatry and the Evelyn F. and William L. McKnight Brain Institute, University of Florida College of Medicine, Gainesville, Florida 32610, USA.
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12
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Abstract
Human parvovirus B19 is the agent that causes erythema infectiosum (fifth disease), a disease that is generally benign and self-limiting. This virus also is associated with severe disease in hemolytic or immunocompromised patients. It rarely causes meningoencephalitis in healthy children. Herein we present the case of a 10-year-old healthy girl with refractory status epilepticus possibly owing to human parvovirus B19 encephalitis who was successfully treated with high-dose corticosteroids. To our knowledge, this is the first report of human parvovirus B19 encephalitis complicated by refractory status epilepticus in a child.
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Affiliation(s)
- Ilknur Erol
- Department of Pediatrics, Neurology Division, Baskent University School of Medicine, 6.Cadde 72/3 Bahcelievler, 06490 Ankara, Turkey.
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13
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Peets AD, Berthiaume LR, Bagshaw SM, Federico P, Doig CJ, Zygun DA. Prolonged refractory status epilepticus following acute traumatic brain injury: a case report of excellent neurological recovery. CRITICAL CARE : THE OFFICIAL JOURNAL OF THE CRITICAL CARE FORUM 2005; 9:R725-8. [PMID: 16280070 PMCID: PMC1414004 DOI: 10.1186/cc3884] [Citation(s) in RCA: 6] [Impact Index Per Article: 0.3] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Download PDF] [Figures] [Subscribe] [Scholar Register] [Received: 08/31/2005] [Accepted: 10/03/2005] [Indexed: 11/10/2022]
Abstract
Introduction Refractory status epilepticus (RSE) secondary to traumatic brain injury (TBI) may be under-recognized and is associated with significant morbidity and mortality. Methods This case report describes a 20 year old previously healthy woman who suffered a severe TBI as a result of a motor vehicle collision and subsequently developed RSE. Pharmacological coma, physiological support and continuous electroencephalography (cEEG) were undertaken. Results Following 25 days of pharmacological coma, electrographic and clinical seizures subsided and the patient has made an excellent cognitive recovery. Conclusion With early identification, aggressive physiological support, appropriate monitoring, including cEEG, and an adequate length of treatment, young trauma patients with no previous seizure history and limited structural damage to the brain can have excellent neurological recovery from prolonged RSE.
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Affiliation(s)
- Adam D Peets
- Research Fellow, Department of Critical Care, University of Calgary, Calgary, Alberta, Canada
| | - Luc R Berthiaume
- Research Fellow, Departments of Critical Care Medicine and Community Health Sciences, University of Calgary, Calgary, Alberta, Canada
| | - Sean M Bagshaw
- Research Fellow, Departments of Critical Care Medicine and Community Health Sciences, University of Calgary, Calgary, Alberta, Canada
| | - Paolo Federico
- Assistant Professor, Department of Neurosciences, University of Calgary, Alberta, Calgary, Canada
| | - Christopher J Doig
- Associate Professor, Departments of Critical Care Medicine and Community Health Sciences, University of Calgary, Calgary, Alberta, Canada
| | - David A Zygun
- Assistant Professor, Departments of Critical Care Medicine, Clinical Neurosciences and Community Health Sciences, University of Calgary, Calgary, Alberta, Canada
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14
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Nolan RC, Chidlow G, French MA. Parvovirus B19 encephalitis presenting as immune restoration disease after highly active antiretroviral therapy for human immunodeficiency virus infection. Clin Infect Dis 2003; 36:1191-4. [PMID: 12715316 DOI: 10.1086/374603] [Citation(s) in RCA: 39] [Impact Index Per Article: 1.8] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 12/02/2002] [Accepted: 01/10/2003] [Indexed: 12/19/2022] Open
Abstract
Illness occurring during the initial months of highly active antiretroviral therapy (HAART) for human immunodeficiency virus infection may be a consequence of the restoration of an immune response against opportunistic pathogens (i.e., immune restoration disease [IRD]). We describe a young man who had AIDS complicated by parvovirus B19 infection and RBC aplasia and who developed a painless, progressive dyspraxia of the left arm and an expressive dysphasia 4 weeks after commencing effective HAART. Neuroimaging demonstrated multiple right fronto-parietal lesions, and, following extensive investigations, including a brain biopsy, it was concluded that the brain lesions represented IRD associated with parvovirus B19 infection.
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Affiliation(s)
- Richard C Nolan
- Department of Clinical Immunology and Biochemical Genetics, Royal Perth Hospital, Perth WA-6001, Australia.
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15
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Yazawa S, Kawasaki S, Fujimoto C, Ohi T. Case report of meningoencephalitis during a concomitant mumps and parvovirus B19 infection. Clin Neurol Neurosurg 2002; 104:380-2. [PMID: 12140110 DOI: 10.1016/s0303-8467(02)00030-6] [Citation(s) in RCA: 6] [Impact Index Per Article: 0.3] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/16/2022]
Abstract
A 19-year-old, immunologically healthy man suffered from prolonged and intermittent high fever, left parotitis, systemic lymph node swelling, progressive liver dysfunction and leukocytopenia. 11 days after the fever onset, consciousness disturbance and generalized convulsion occurred. By the administration of gamma-globulin and steroid, the patient recovered completely. Serum titers of IgG and IgM specific for both human parvovirus B19 and mumps were elevated, and parvovirus B19 DNA was identified in the serum. It was speculated that overlap infection of mumps and parvovirus B19 made the disease more severe in this patient.
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Affiliation(s)
- Shogo Yazawa
- Department of Neurology, Miyazaki Prefectural Hospital of Nobeoka, Miyazaki, Japan.
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