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1
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Liu AN, Yang JY, Chen XY, Wu SS, Ji Zhi SN, Zheng SM. Refractory Crohn's disease complicated with Guillain-Barré syndrome: A case report. World J Clin Cases 2025; 13:103618. [DOI: 10.12998/wjcc.v13.i18.103618] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 11/25/2024] [Revised: 01/24/2025] [Accepted: 02/17/2025] [Indexed: 02/27/2025] Open
Abstract
BACKGROUND Inflammatory bowel disease (IBD) comprises a group of chronic inflammatory gastrointestinal disorders, including Crohn's disease (CD) and ulcerative colitis, with uncertain etiologies. The natural course of IBD can be accompanied by extraintestinal manifestations involving the skin, mucous membranes, musculoskeletal structures, eyes, cardiovascular system and nervous system. Guillain-Barré syndrome (GBS) is a type of peripheral neuropathy. However, the etiology and pathogenesis of IBD combined with GBS are unclear, and only a few clinical cases have been reported. Here, we report a case of refractory CD complicated by GBS and review the previous literature to improve the understanding of these diseases.
CASE SUMMARY A 34-year-old man had a 9-year history of refractory CD. He became unresponsive to multiple drugs and experienced recurrent intestinal fistulas. After several abdominal surgeries and treatment with ustekinumab, he achieved clinical remission. Unfortunately, he developed GBS during maintenance treatment with ustekinumab. According to previous reports, in some patients with IBD combined with GBS, GBS may be a comorbidity, an extraintestinal manifestation of IBD, or an adverse reaction to IBD therapeutic drugs. After a comprehensive evaluation, we suspected that GBS might have been a comorbidity in this patient. To avoid fatal disease relapse after medication discontinuation, we concluded that ustekinumab should not be withdrawn. On the basis of a joint decision between doctors and the patient, we decided to continue maintenance treatment with ustekinumab along with intravenous immunoglobulin, dexamethasone and traditional Chinese medicine acupuncture, which resulted in a steady improvement in his GBS symptoms and sustained remission of CD.
CONCLUSION When IBD is complicated by a neurological disease, it is first necessary to analyze the patient's condition and then choose the corresponding treatment strategy. If the neurological disease is a specific comorbidity, treatment of both IBD and the comorbid disease should be considered. For IBD patients with extraintestinal manifestations involving the nervous system, neurological manifestations tend to resolve when the active IBD is controlled. When an adverse drug reaction is suspected, the medication should be discontinued, and symptomatic treatment should be administered.
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Affiliation(s)
- A-Niu Liu
- Department of Gastroenterology and Hepatology, Chengdu Medical College, Chengdu 610500, Sichuan Province, China
- Department of Gastroenterology and Hepatology, The General Hospital of Western Theater Command, Chengdu 610083, Sichuan Province, China
| | - Jia-Yi Yang
- Department of Radiology, Wuxi People’s Hospital, Nanjing Medical University, Nanjing 214023, Jiangsu Province, China
| | - Xing-Yu Chen
- Department of Gastroenterology and Hepatology, The General Hospital of Western Theater Command, Chengdu 610083, Sichuan Province, China
| | - Shan-Shan Wu
- Department of Gastroenterology and Hepatology, The General Hospital of Western Theater Command, Chengdu 610083, Sichuan Province, China
| | - Se-Niu Ji Zhi
- Department of Gastroenterology and Hepatology, The General Hospital of Western Theater Command, Chengdu 610083, Sichuan Province, China
| | - Shu-Mei Zheng
- Department of Gastroenterology and Hepatology, The General Hospital of Western Theater Command, Chengdu 610083, Sichuan Province, China
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Nasiri A, Alshammari T, Alsolaihim A, Alfattah H, Alahmri A. Tacrolimus associated Guillain-Barre syndrome. Arch Clin Cases 2025; 12:75-79. [PMID: 40416578 PMCID: PMC12096302 DOI: 10.22551/2025.47.1202.10317] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 05/27/2025] Open
Abstract
Guillain-Barré syndrome (GBS) is a rare but serious neuropathy in hematopoietic stem cell transplant recipients. Immunosuppressants, particularly tacrolimus, have been implicated as potential triggers. We present a 27-year-old man with BCR-ABL-positive acute myeloid leukemia who developed an acute demyelinating polyneuropathy possibly related to tacrolimus therapy post-transplantation, highlighting diagnostic challenges and management considerations. The patient developed progressive ascending weakness, areflexia, sensory loss, and bulbar symptoms 58 days after an allogeneic stem cell transplant from an HLA-matched sibling donor. Cerebrospinal fluid (CSF) analysis showed elevated protein (1,900 mg/L) with lymphocytic pleocytosis (51 cells/μL), an atypical finding for GBS. Magnetic resonance imaging revealed subtle nerve root enhancement, and nerve conduction studies demonstrated markedly slowed conduction velocities and prolonged distal latencies consistent with an acute inflammatory demyelinating polyneuropathy. Extensive infectious work-up (including viral PCR panels and cultures) was negative, and no leukemic cells were seen in CSF. Tacrolimus was discontinued (trough level 3.1 ng/mL, below therapeutic range) and intravenous immunoglobulin (2 g/kg total over five days) initiated. The patient's neurological deficits improved rapidly, with near-complete recovery within four weeks. Notably, withdrawal of tacrolimus immunosuppression did not precipitate graft-versus-host disease, and the patient's acute leukemia remained in remission on ponatinib monotherapy. This case illustrates an acute demyelinating polyneuropathy in a post-transplant patient, associated with tacrolimus. It underscores the importance of careful diagnostic assessment of GBS in transplant recipients, including consideration of atypical CSF findings and alternative diagnoses. Prompt recognition and management - including immunosuppressant adjustment and immunotherapy - can achieve full neurological recovery without compromising transplant outcomes.
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Affiliation(s)
- Abdulrahman Nasiri
- Department of Internal Medicine, College of Medicine, Imam Mohammad Ibn Saud Islamic University, Riyadh, Saudi Arabia
- Department of Hematology, King Faisal Specialist Hospital & Research Centre, Riyadh, Saudi Arabia
| | - Tamam Alshammari
- Department of Hematology, King Faisal Specialist Hospital & Research Centre, Riyadh, Saudi Arabia
| | - Alanood Alsolaihim
- Neuroscience Center, King Faisal Specialist Hospital and Research Center, Riyadh, Saudi Arabia
| | - Huda Alfattah
- Department of Hematology, King Faisal Specialist Hospital & Research Centre, Riyadh, Saudi Arabia
| | - Ali Alahmri
- Department of Hematology, King Faisal Specialist Hospital & Research Centre, Riyadh, Saudi Arabia
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3
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Bellanti R, Rinaldi S. Guillain-Barré syndrome: a comprehensive review. Eur J Neurol 2024; 31:e16365. [PMID: 38813755 PMCID: PMC11235944 DOI: 10.1111/ene.16365] [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: 03/26/2024] [Revised: 05/12/2024] [Accepted: 05/13/2024] [Indexed: 05/31/2024]
Abstract
Guillain-Barré syndrome (GBS) is a potentially devastating yet treatable disorder. A classically postinfectious, immune-mediated, monophasic polyradiculoneuropathy, it is the leading global cause of acquired neuromuscular paralysis. In most cases, the immunopathological process driving nerve injury is ill-defined. Diagnosis of GBS relies on clinical features, supported by laboratory findings and electrophysiology. Although previously divided into primary demyelinating or axonal variants, this dichotomy is increasingly challenged, and is not endorsed by the recent European Academy of Neurology (EAN)/Peripheral Nerve Society (PNS) guidelines. Intravenous immunoglobulin and plasma exchange remain the primary modalities of treatment, regardless of the electrophysiological subtype. Most patients recover, but approximately one-third require mechanical ventilation, and 5% die. Disease activity and treatment response are currently monitored through interval neurological examination and outcome measures, and the potential role of fluid biomarkers is under ongoing scrutiny. Novel potential therapies for GBS are being explored but none have yet modified clinical practice. This review provides a comprehensive update on the pathological and clinical aspects of GBS for clinicians and scientists.
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Affiliation(s)
- Roberto Bellanti
- Nuffield Department of Clinical NeurosciencesUniversity of OxfordOxfordUK
| | - Simon Rinaldi
- Nuffield Department of Clinical NeurosciencesUniversity of OxfordOxfordUK
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4
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van Doorn PA, Van den Bergh PYK, Hadden RDM, Avau B, Vankrunkelsven P, Attarian S, Blomkwist-Markens PH, Cornblath DR, Goedee HS, Harbo T, Jacobs BC, Kusunoki S, Lehmann HC, Lewis RA, Lunn MP, Nobile-Orazio E, Querol L, Rajabally YA, Umapathi T, Topaloglu HA, Willison HJ. European Academy of Neurology/Peripheral Nerve Society Guideline on diagnosis and treatment of Guillain-Barré syndrome. Eur J Neurol 2023; 30:3646-3674. [PMID: 37814552 DOI: 10.1111/ene.16073] [Citation(s) in RCA: 67] [Impact Index Per Article: 33.5] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 07/13/2023] [Revised: 08/25/2023] [Accepted: 08/28/2023] [Indexed: 10/11/2023]
Abstract
Guillain-Barré syndrome (GBS) is an acute polyradiculoneuropathy. Symptoms may vary greatly in presentation and severity. Besides weakness and sensory disturbances, patients may have cranial nerve involvement, respiratory insufficiency, autonomic dysfunction and pain. To develop an evidence-based guideline for the diagnosis and treatment of GBS, using Grading of Recommendations, Assessment, Development and Evaluation (GRADE) methodology a Task Force (TF) of the European Academy of Neurology (EAN) and the Peripheral Nerve Society (PNS) constructed 14 Population/Intervention/Comparison/Outcome questions (PICOs) covering diagnosis, treatment and prognosis of GBS, which guided the literature search. Data were extracted and summarised in GRADE Summaries of Findings (for treatment PICOs) or Evidence Tables (for diagnostic and prognostic PICOs). Statements were prepared according to GRADE Evidence-to-Decision (EtD) frameworks. For the six intervention PICOs, evidence-based recommendations are made. For other PICOs, good practice points (GPPs) are formulated. For diagnosis, the principal GPPs are: GBS is more likely if there is a history of recent diarrhoea or respiratory infection; CSF examination is valuable, particularly when the diagnosis is less certain; electrodiagnostic testing is advised to support the diagnosis; testing for anti-ganglioside antibodies is of limited clinical value in most patients with typical motor-sensory GBS, but anti-GQ1b antibody testing should be considered when Miller Fisher syndrome (MFS) is suspected; nodal-paranodal antibodies should be tested when autoimmune nodopathy is suspected; MRI or ultrasound imaging should be considered in atypical cases; and changing the diagnosis to acute-onset chronic inflammatory demyelinating polyradiculoneuropathy (A-CIDP) should be considered if progression continues after 8 weeks from onset, which occurs in around 5% of patients initially diagnosed with GBS. For treatment, the TF recommends intravenous immunoglobulin (IVIg) 0.4 g/kg for 5 days, in patients within 2 weeks (GPP also within 2-4 weeks) after onset of weakness if unable to walk unaided, or a course of plasma exchange (PE) 12-15 L in four to five exchanges over 1-2 weeks, in patients within 4 weeks after onset of weakness if unable to walk unaided. The TF recommends against a second IVIg course in GBS patients with a poor prognosis; recommends against using oral corticosteroids, and weakly recommends against using IV corticosteroids; does not recommend PE followed immediately by IVIg; weakly recommends gabapentinoids, tricyclic antidepressants or carbamazepine for treatment of pain; does not recommend a specific treatment for fatigue. To estimate the prognosis of individual patients, the TF advises using the modified Erasmus GBS outcome score (mEGOS) to assess outcome, and the modified Erasmus GBS Respiratory Insufficiency Score (mEGRIS) to assess the risk of requiring artificial ventilation. Based on the PICOs, available literature and additional discussions, we provide flow charts to assist making clinical decisions on diagnosis, treatment and the need for intensive care unit admission.
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Affiliation(s)
- Pieter A van Doorn
- Department of Neurology, Erasmus MC, University Medical Center, Rotterdam, The Netherlands
| | - Peter Y K Van den Bergh
- Neuromuscular Reference Centre, Department of Neurology, University Hospital Saint-Luc, Brussels, Belgium
| | | | - Bert Avau
- Cochrane Belgium, CEBAM, Leuven, Belgium
- CEBaP, Belgian Red Cross, Mechelen, Belgium
| | - Patrik Vankrunkelsven
- Department of Public Health and Primary Care KU Leuven, Cochrane Belgium, CEBAM, Leuven, Belgium
| | - Shahram Attarian
- Centre de Référence des Maladies Neuromusculaires et de la SLA, APHM, CHU Timone, Marseille, France
| | | | - David R Cornblath
- Department of Neurology, Johns Hopkins University School of Medicine, Baltimore, Maryland, USA
| | - H Stephan Goedee
- Department of Neurology, University Medical Center Utrecht, Brain Center UMC Utrecht, Utrecht, The Netherlands
| | - Thomas Harbo
- Department of Neurology, Aarhus University Hospital, Aarhus, Denmark
| | - Bart C Jacobs
- Department of Neurology and Immunology, Erasmus MC, University Medical Center, Rotterdam, The Netherlands
| | - Susumu Kusunoki
- Department of Neurology, Faculty of Medicine, Kindai University, Osaka, Japan
| | - Helmar C Lehmann
- Department of Neurology, Medical Faculty Köln, University Hospital Köln, Cologne, Germany
| | - Richard A Lewis
- Department of Neurology, Cedars-Sinai Medical Center, Los Angeles, California, USA
| | - Michael P Lunn
- Department of Neurology and MRC Centre for Neuromuscular Diseases, National Hospital for Neurology and Neurosurgery, London, UK
| | - Eduardo Nobile-Orazio
- Neuromuscular and Neuroimmunology Service, IRCCS Humanitas Research Institute, Department of Medical Biotechnology and Translational Medicine, University of Milan, Milan, Italy
| | - Luis Querol
- Neuromuscular Diseases Unit, Neurology Department, Hospital de la Santa Creu I Sant Pau, Universitat Autònoma de Barcelona, Barcelona, Spain
| | - Yusuf A Rajabally
- Neuromuscular Service, Neurology, Queen Elizabeth Hospital Birmingham, Birmingham, UK
| | | | | | - Hugh J Willison
- Glasgow Biomedical Research Centre, University of Glasgow, Glasgow, UK
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5
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van Doorn PA, Van den Bergh PYK, Hadden RDM, Avau B, Vankrunkelsven P, Attarian S, Blomkwist-Markens PH, Cornblath DR, Goedee HS, Harbo T, Jacobs BC, Kusunoki S, Lehmann HC, Lewis RA, Lunn MP, Nobile-Orazio E, Querol L, Rajabally YA, Umapathi T, Topaloglu HA, Willison HJ. European Academy of Neurology/Peripheral Nerve Society Guideline on diagnosis and treatment of Guillain-Barré syndrome. J Peripher Nerv Syst 2023; 28:535-563. [PMID: 37814551 DOI: 10.1111/jns.12594] [Citation(s) in RCA: 25] [Impact Index Per Article: 12.5] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Grants] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 07/13/2023] [Revised: 08/25/2023] [Accepted: 08/28/2023] [Indexed: 10/11/2023]
Abstract
Guillain-Barré syndrome (GBS) is an acute polyradiculoneuropathy. Symptoms may vary greatly in presentation and severity. Besides weakness and sensory disturbances, patients may have cranial nerve involvement, respiratory insufficiency, autonomic dysfunction and pain. To develop an evidence-based guideline for the diagnosis and treatment of GBS, using Grading of Recommendations, Assessment, Development and Evaluation (GRADE) methodology, a Task Force (TF) of the European Academy of Neurology (EAN) and the Peripheral Nerve Society (PNS) constructed 14 Population/Intervention/Comparison/Outcome questions (PICOs) covering diagnosis, treatment and prognosis of GBS, which guided the literature search. Data were extracted and summarised in GRADE Summaries of Findings (for treatment PICOs) or Evidence Tables (for diagnostic and prognostic PICOs). Statements were prepared according to GRADE Evidence-to-Decision (EtD) frameworks. For the six intervention PICOs, evidence-based recommendations are made. For other PICOs, good practice points (GPPs) are formulated. For diagnosis, the principal GPPs are: GBS is more likely if there is a history of recent diarrhoea or respiratory infection; CSF examination is valuable, particularly when the diagnosis is less certain; electrodiagnostic testing is advised to support the diagnosis; testing for anti-ganglioside antibodies is of limited clinical value in most patients with typical motor-sensory GBS, but anti-GQ1b antibody testing should be considered when Miller Fisher syndrome (MFS) is suspected; nodal-paranodal antibodies should be tested when autoimmune nodopathy is suspected; MRI or ultrasound imaging should be considered in atypical cases; and changing the diagnosis to acute-onset chronic inflammatory demyelinating polyradiculoneuropathy (A-CIDP) should be considered if progression continues after 8 weeks from onset, which occurs in around 5% of patients initially diagnosed with GBS. For treatment, the TF recommends intravenous immunoglobulin (IVIg) 0.4 g/kg for 5 days, in patients within 2 weeks (GPP also within 2-4 weeks) after onset of weakness if unable to walk unaided, or a course of plasma exchange (PE) 12-15 L in four to five exchanges over 1-2 weeks, in patients within 4 weeks after onset of weakness if unable to walk unaided. The TF recommends against a second IVIg course in GBS patients with a poor prognosis; recommends against using oral corticosteroids, and weakly recommends against using IV corticosteroids; does not recommend PE followed immediately by IVIg; weakly recommends gabapentinoids, tricyclic antidepressants or carbamazepine for treatment of pain; does not recommend a specific treatment for fatigue. To estimate the prognosis of individual patients, the TF advises using the modified Erasmus GBS outcome score (mEGOS) to assess outcome, and the modified Erasmus GBS Respiratory Insufficiency Score (mEGRIS) to assess the risk of requiring artificial ventilation. Based on the PICOs, available literature and additional discussions, we provide flow charts to assist making clinical decisions on diagnosis, treatment and the need for intensive care unit admission.
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Affiliation(s)
- Pieter A van Doorn
- Department of Neurology, Erasmus MC, University Medical Center, Rotterdam, The Netherlands
| | - Peter Y K Van den Bergh
- Neuromuscular Reference Centre, Department of Neurology, University Hospital Saint-Luc, Brussels, Belgium
| | | | - Bert Avau
- Cochrane Belgium, CEBAM, Leuven, Belgium
- CEBaP, Belgian Red Cross, Mechelen, Belgium
| | - Patrik Vankrunkelsven
- Department of Public Health and Primary Care KU Leuven, Cochrane Belgium, CEBAM, Leuven, Belgium
| | - Shahram Attarian
- Centre de Référence des Maladies Neuromusculaires et de la SLA, APHM, CHU Timone, Marseille, France
| | | | - David R Cornblath
- Department of Neurology, Johns Hopkins University School of Medicine, Baltimore, Maryland, USA
| | - H Stephan Goedee
- Department of Neurology, University Medical Center Utrecht, Brain Center UMC Utrecht, Utrecht, The Netherlands
| | - Thomas Harbo
- Department of Neurology, Aarhus University Hospital, Aarhus, Denmark
| | - Bart C Jacobs
- Department of Neurology and Immunology, Erasmus MC, University Medical Center, Rotterdam, The Netherlands
| | - Susumu Kusunoki
- Department of Neurology, Faculty of Medicine, Kindai University, Osaka, Japan
| | - Helmar C Lehmann
- Department of Neurology, Medical Faculty Köln, University Hospital Köln, Cologne, Germany
| | - Richard A Lewis
- Department of Neurology, Cedars-Sinai Medical Center, Los Angeles, California, USA
| | - Michael P Lunn
- Department of Neurology and MRC Centre for Neuromuscular Diseases, National Hospital for Neurology and Neurosurgery, London, UK
| | - Eduardo Nobile-Orazio
- Neuromuscular and Neuroimmunology Service, IRCCS Humanitas Research Institute, Department of Medical Biotechnology and Translational Medicine, University of Milan, Milan, Italy
| | - Luis Querol
- Neuromuscular Diseases Unit, Neurology Department, Hospital de la Santa Creu I Sant Pau, Universitat Autònoma de Barcelona, Barcelona, Spain
| | - Yusuf A Rajabally
- Neuromuscular Service, Neurology, Queen Elizabeth Hospital Birmingham, Birmingham, UK
| | | | | | - Hugh J Willison
- Glasgow Biomedical Research Centre, University of Glasgow, Glasgow, UK
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Kubota T, Shijo T, Ikeda K, Mitobe Y, Umezawa S, Misu T, Hasegawa T, Aoki M. Distal Chronic Inflammatory Demyelinating Polyneuropathy Following COVID-19 Vaccination in a Patient with Solitary Plasmacytoma: A Case Report and Literature Review. Intern Med 2023; 62:2419-2425. [PMID: 37587059 PMCID: PMC10484767 DOI: 10.2169/internalmedicine.1365-22] [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: 11/28/2022] [Accepted: 04/24/2023] [Indexed: 08/18/2023] Open
Abstract
We herein report a rare case of distal chronic inflammatory demyelinating polyneuropathy (CIDP) following coronavirus disease 2019 (COVID-19) vaccination. A 39-year-old woman with a solitary plasmacytoma developed general weakness 7 days after receiving the second dose of the Pfizer-BioNTech COVID-19 vaccine, which had progressed for 3 months. A neurological examination revealed limb weakness with areflexia. Serological tests identified the presence of IgG antibodies against anti-GM1 and anti-GM2 gangliosides. Comprehensive evaluations met the criteria of distal CIDP. Intravenous immunoglobulin, intravenous methylprednisolone, oral prednisolone, and plasma exchange were administered, and she gradually improved. Physicians should be aware of CIDP as a rare complication of COVID-19 vaccination.
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Affiliation(s)
- Takafumi Kubota
- Department of Neurology, Tohoku University Graduate School of Medicine, Japan
| | - Tomomi Shijo
- Department of Neurology, Tohoku University Graduate School of Medicine, Japan
| | - Kensho Ikeda
- Department of Neurology, Tohoku University Graduate School of Medicine, Japan
| | - Yoshihiko Mitobe
- Department of Neurology, Tohoku University Graduate School of Medicine, Japan
| | - Shu Umezawa
- Department of Neurology, Tohoku University Graduate School of Medicine, Japan
| | - Tatsuro Misu
- Department of Neurology, Tohoku University Graduate School of Medicine, Japan
| | - Takafumi Hasegawa
- Department of Neurology, Tohoku University Graduate School of Medicine, Japan
| | - Masashi Aoki
- Department of Neurology, Tohoku University Graduate School of Medicine, Japan
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7
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Malekpour M, Khanmohammadi S, Meybodi MJE, Shekouh D, Rahmanian MR, Kardeh S, Azarpira N. COVID-19 as a trigger of Guillain-Barré syndrome: A review of the molecular mechanism. Immun Inflamm Dis 2023; 11:e875. [PMID: 37249286 DOI: 10.1002/iid3.875] [Citation(s) in RCA: 8] [Impact Index Per Article: 4.0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 03/20/2023] [Revised: 04/30/2023] [Accepted: 05/03/2023] [Indexed: 05/31/2023] Open
Abstract
Severe acute respiratory syndrome coronavirus 2 (SARS-CoV-2) caused a pandemic with serious complications. After coronavirus disease 2019 (COVID-19), several post-acute COVID-19 syndromes (PACSs) and long-COVID sequels were reported. PACSs involve many organs, including the nervous, gustatory, and immune systems. One of the PACSs after SARS-CoV-2 infection and vaccination is Guillain-Barré syndrome (GBS). The incidence rate of GBS after SARS-CoV-2 infection or vaccination is low. However, the high prevalence of COVID-19 and severe complications of GBS, for example, autonomic dysfunction and respiratory failure, highlight the importance of post-COVID-19 GBS. It is while patients with simultaneous COVID-19 and GBS seem to have higher admission rates to the intensive care unit, and demyelination is more aggressive in post-COVID-19 GBS patients. SARS-CoV-2 can trigger GBS via several pathways like direct neurotropism and neurovirulence, microvascular dysfunction and oxidative stress, immune system disruption, molecular mimicry, and autoantibody production. Although there are few molecular studies on the molecular and cellular mechanisms of GBS occurrence after SARS-CoV-2 infection and vaccination, we aimed to discuss the possible pathomechanism of post-COVID-19 GBS by gathering the most recent molecular evidence.
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Affiliation(s)
- Mahdi Malekpour
- Student Research Committee, Shiraz University of Medical Sciences, Shiraz, Iran
- Transplant Research Center, Shiraz University of Medical Sciences, Shiraz, Iran
| | - Shaghayegh Khanmohammadi
- Network of Immunity in Infection, Malignancy and Autoimmunity (NIIMA), Universal Scientific Education and Research Network (USERN), Tehran, Iran
- Research Center for Immunodeficiencies, Children's Medical Center, Tehran University of Medical Sciences, Tehran, Iran
| | - Mohammad Javad Entezari Meybodi
- Student Research Committee, Shiraz University of Medical Sciences, Shiraz, Iran
- Transplant Research Center, Shiraz University of Medical Sciences, Shiraz, Iran
| | - Dorsa Shekouh
- Student Research Committee, Shiraz University of Medical Sciences, Shiraz, Iran
| | - Mohammad Reza Rahmanian
- Student Research Committee, Shiraz University of Medical Sciences, Shiraz, Iran
- Transplant Research Center, Shiraz University of Medical Sciences, Shiraz, Iran
| | - Sina Kardeh
- Central Clinical School, Monash University, Melbourne, Australia
| | - Negar Azarpira
- Transplant Research Center, Shiraz University of Medical Sciences, Shiraz, Iran
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8
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Huang C, Zhang Y, Deng S, Ren Y, Lu W. Trauma-Related Guillain-Barré Syndrome: Systematic Review of an Emerging Concept. Front Neurol 2020; 11:588290. [PMID: 33240210 PMCID: PMC7681248 DOI: 10.3389/fneur.2020.588290] [Citation(s) in RCA: 14] [Impact Index Per Article: 2.8] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 07/28/2020] [Accepted: 10/02/2020] [Indexed: 02/05/2023] Open
Abstract
Guillain-Barré syndrome (GBS) is mainly associated with preceding exposure to an infectious agent, although the precise pathogenic mechanisms and causes remain unknown. Increasing evidence indicates an association between trauma-related factors and GBS. Here, we performed a systematic review, summarized the current scientific literature related to the onset of GBS associated with trauma, and explored the possible pathogenesis. A literature search of various electronic databases was performed up to May 2020 to identify studies reporting diverse trauma-related triggers of GBS. Data were extracted, summarized descriptively, and evaluated with respect to possible mechanisms. In total, 100 publications, including 136 cases and 6 case series involving GBS triggered by injury, surgery, intracranial hemorrhage, and heatstroke, met our eligibility criteria. The median age of the patients was 53 [interquartile range (IQR) 45-63] years, and 72.1% of the patients were male. The median number of days between the trigger to onset of GBS symptoms was 9 (IQR 6.5-13). Overall, 121 patients (89.0%) developed post-injury/surgical GBS, whereas 13 (9.6%) and 2 (1.5%) patients had preexisting spontaneous intracranial hemorrhage and heatstroke, respectively. The main locations of injury or surgeries preceding GBS were the spine and brain. Based on available evidence, we highlight possible mechanisms of GBS induced by these triggers. Moreover, we propose the concept of "trauma-related GBS" as a new research direction, which may help uncover more pathogenic mechanisms than previously considered for typical GBS triggered by infection or vaccination.
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Affiliation(s)
- Chuxin Huang
- Department of Neurology, The Second Xiangya Hospital, Central South University, Changsha, China
- Department of Radiology, The Second Xiangya Hospital, Central South University, Changsha, China
| | - Yiliu Zhang
- Department of Neurology, The Second Xiangya Hospital, Central South University, Changsha, China
| | - Shuwen Deng
- Department of Neurology, The Second Xiangya Hospital, Central South University, Changsha, China
| | - Yijun Ren
- Department of Neurology, The Second Xiangya Hospital, Central South University, Changsha, China
| | - Wei Lu
- Department of Neurology, The Second Xiangya Hospital, Central South University, Changsha, China
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9
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Bao L, Chen X, Li Q, Zhang R, Shi H, Cui G. Surgery and Guillain-Barré Syndrome: A Single-Center Retrospective Study Focused on Clinical and Electrophysiological Subtypes. Neuropsychiatr Dis Treat 2020; 16:969-974. [PMID: 32346291 PMCID: PMC7167305 DOI: 10.2147/ndt.s241128] [Citation(s) in RCA: 15] [Impact Index Per Article: 3.0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 12/04/2019] [Accepted: 03/22/2020] [Indexed: 12/15/2022] Open
Abstract
BACKGROUND Surgery-related Guillain-Barré syndrome (GBS) is often underestimated and sometimes difficult to diagnose. This study aimed to elucidate the clinical features and electrophysiological subtypes of post-surgical GBS. METHODS We retrospectively reviewed 17 patients who developed post-surgical GBS after a recent surgery between 2015 and 2019. Clinical characteristics, electrophysiological examinations, lumbar puncture results and prognosis were assessed. As controls, we selected 66 patients hospitalized with non-surgical GBS. RESULTS The median duration from the surgery to the onset of GBS symptoms was 16.0 days. The main types of surgeries preceding GBS were orthopedic, gastrointestinal and neurosurgery. Symmetrical distal limbs weakness was present in all 17 post-surgical GBS patients. The incidence of respiratory failure, autonomic dysfunction and muscle atrophy in post-surgical GBS patients was significantly higher than that in non-surgical GBS patients. Hughes Functional Grading Scale (HFGS) scores were also higher in the post-surgical GBS group both at the time of peak disease and 6 months after discharge. Electrophysiological studies revealed significant motor amplitudes reduction with relative preserved nerve conduction velocities and distal latencies, suggesting axonal subtypes of GBS. CONCLUSION GBS should be considered in patients with rapidly progressive muscle weakness after surgery. Such patients often exhibit axonal subtypes of GBS with severe motor dysfunction, high risk of respiratory failure, and poor prognosis.
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Affiliation(s)
- Lei Bao
- Department of Neurology, The Affiliated Hospital of Xuzhou Medical University, Xuzhou, Jiangsu 221004, People's Republic of China
| | - Xueting Chen
- Department of Neurology, The Affiliated Hospital of Xuzhou Medical University, Xuzhou, Jiangsu 221004, People's Republic of China
| | - Qingjie Li
- Department of Neurology, The Affiliated Hospital of Xuzhou Medical University, Xuzhou, Jiangsu 221004, People's Republic of China
| | - Ruixue Zhang
- Department of Neurology, The Affiliated Hospital of Xuzhou Medical University, Xuzhou, Jiangsu 221004, People's Republic of China
| | - Hongjuan Shi
- Department of Neurology, The Affiliated Hospital of Xuzhou Medical University, Xuzhou, Jiangsu 221004, People's Republic of China
| | - Guiyun Cui
- Department of Neurology, The Affiliated Hospital of Xuzhou Medical University, Xuzhou, Jiangsu 221004, People's Republic of China
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10
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Chowdhary P, Kale S, Saha T, Banerjee S. Guillain–Barre syndrome-like presentation and thrombotic microangiopathy with calcineurin inhibitor - A case report. INDIAN JOURNAL OF TRANSPLANTATION 2020. [DOI: 10.4103/ijot.ijot_70_19] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/04/2022] Open
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11
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Xu YL, Zhao WH, Tang ZY, Li ZQ, Long Y, Cheng P, Luo J. Guillain-Barré syndrome in a patient with multiple myeloma after bortezomib therapy: A case report. World J Clin Cases 2019; 7:2905-2909. [PMID: 31616710 PMCID: PMC6789385 DOI: 10.12998/wjcc.v7.i18.2905] [Citation(s) in RCA: 4] [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: 04/10/2019] [Revised: 08/13/2019] [Accepted: 08/26/2019] [Indexed: 02/05/2023] Open
Abstract
BACKGROUND Bortezomib is a first-line drug approved for patients with multiple myeloma (MM) and has significantly increased their overall survival. However, bortezomib-induced peripheral neuropathy (PN) remains a significant side effect that has led to its discontinuation in some patients. Guillain-Barré syndrome (GBS) is recognized as an immune-mediated PN characterized by the involvement of multiple nerve roots and peripheral nerves and albuminocytologic dissociation in cerebrospinal fluid (CSF) tests. Intravenous immunoglobulin (IVIG) and plasmapheresis are effective.
CASE SUMMARY A 45-year-old man diagnosed with stage III MM (λ type) was treated with bortezomib and dexamethasone. Fourteen days after the second course, he complained of intense burning sensation in the lower limbs and hands, loss of tactile sensation, and pain in the distal area of both thighs and in the distal part of both wrist joints. Neurological examination revealed absence of knee and ankle reflexes. CSF examination revealed albuminocytologic dissociation. Nerve conduction studies indicated sensory nerve action potential amplitudes, conduction velocity decrease, and F wave latency prolongation. He was diagnosed as MM complicated with GBS. Subsequently, he was treated with high-dose IVIG (400 mg/kg/d for five days). His symptoms fully resolved without relapse at the 6-month follow-up.
CONCLUSION Our case highlights the differential diagnosis and management of complications after bortezomib treatment in MM.
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Affiliation(s)
- Yu-Ling Xu
- Department of Hematology, the First Affiliated Hospital of Guangxi Medical University, Nanning 530021, Guangxi Zhuang Autonomous Region, China
| | - Wei-Hua Zhao
- Department of Hematology, the First Affiliated Hospital of Guangxi Medical University, Nanning 530021, Guangxi Zhuang Autonomous Region, China
| | - Zhong-Yuan Tang
- Department of Hematology, the First Affiliated Hospital of Guangxi Medical University, Nanning 530021, Guangxi Zhuang Autonomous Region, China
| | - Zhong-Qing Li
- Department of Hematology, the First Affiliated Hospital of Guangxi Medical University, Nanning 530021, Guangxi Zhuang Autonomous Region, China
| | - Yuan Long
- Department of Hematology, the First Affiliated Hospital of Guangxi Medical University, Nanning 530021, Guangxi Zhuang Autonomous Region, China
| | - Peng Cheng
- Department of Hematology, the First Affiliated Hospital of Guangxi Medical University, Nanning 530021, Guangxi Zhuang Autonomous Region, China
| | - Jun Luo
- Department of Hematology, the First Affiliated Hospital of Guangxi Medical University, Nanning 530021, Guangxi Zhuang Autonomous Region, China
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12
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Balaguer-Rosello A, Bataller L, Piñana JL, Montoro J, Lorenzo I, Villalba A, Freiria C, Santiago M, Sevilla T, Muelas N, Guerreiro M, Carretero C, Gómez I, Solves P, Sanz MÁ, Sanz G, Sanz J. Noninfectious Neurologic Complications after Allogeneic Hematopoietic Stem Cell Transplantation. Biol Blood Marrow Transplant 2019; 25:1818-1824. [PMID: 31132454 DOI: 10.1016/j.bbmt.2019.05.024] [Citation(s) in RCA: 25] [Impact Index Per Article: 4.2] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 04/01/2019] [Revised: 05/18/2019] [Accepted: 05/21/2019] [Indexed: 12/19/2022]
Abstract
Although allogeneic hematopoietic stem cell transplantation (allo-HSCT) can be associated with neurologic complications, data on noninfectious etiologies are scanty. Therefore, we analyzed the incidence, clinical characteristics, risk factors, and influence on outcomes of noninfectious neurologic complications (NCs) in 971 consecutive patients with hematologic malignancies undergoing allo-HSCT at our center between January 2000 and December 2016. We evaluated NCs affecting the central nervous system (CNS) and peripheral nervous system (PNS). The median duration of follow-up of survivors was 71 months (range, 11 to 213 months). A total of 467 patients received a matched sibling donor (MSD) transplant, 381 received umbilical cord blood (UCB), 74 received a haploidentical transplant, and 49 received a matched unrelated donor (MUD) transplant. One hundred forty-nine (15.3%) NCs were documented at a median of 78 days after transplantation (range, 5 days before to 3722 days after). The cumulative incidence risk of developing NC was 7.5% (95% confidence interval, 6% to 8.2%) at day +90 and 13% at 5 years. The 5-year cumulative incidence of NCs was 10.8% after MSD allo-HSCT and 15.3% after alternative donor (UCB, MUD, haploidentical) allo-HSCT (P = .004). There were 101 (68%) CNS complications, including encephalopathy, n = 46 (31%); headache, n = 20 (13%); stroke, n = 15 (10%); seizures, n = 9 (6%), posterior reversible encephalopathy syndrome, n = 6 (4%), and myelopathy, n = 5 (3%). PNS complications (32%) included neuropathies, n = 25 (17%), and myopathies and neuromuscular junction disorders, n = 23 (17%), with 17% of the total PNS complications being immune-related. In multivariable analysis, donor type other than MSD, age ≥40 years, development of acute graft-versus-host disease (GVHD) grade II-IV (hazard ratio [HR], 3.3; P < .00001), and extensive chronic GVHD (HR, 3.2; P = .0002) were independently associated with increased risk of NCs. The 5-year overall survival (OS) was 21% in patients who developed NCs and 41% for those who did not (P < .0001). This difference in OS was observed in patients developing CNS NCs, but not in those developing PNS complications. In conclusion, our study reveals NCs as a frequent and heterogeneous complication that, when affecting CNS, is associated with poor prognosis following allo-HSCT.
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Affiliation(s)
| | - Luis Bataller
- Neurology Department, Hospital Universitari i Politècnic La Fe, Valencia, Spain
| | - José Luis Piñana
- Hematology Department, Hospital Universitari i Politècnic La Fe, Valencia, Spain; CIBERONC, Instituto Carlos III, Madrid, Spain
| | - Juan Montoro
- Hematology Department, Hospital Universitari i Politècnic La Fe, Valencia, Spain
| | - Ignacio Lorenzo
- Hematology Department, Hospital Universitari i Politècnic La Fe, Valencia, Spain
| | - Ana Villalba
- Hematology Department, Hospital Universitari i Politècnic La Fe, Valencia, Spain
| | - Carmen Freiria
- Hematology Department, Hospital Universitari i Politècnic La Fe, Valencia, Spain
| | - Marta Santiago
- Hematology Department, Hospital Universitari i Politècnic La Fe, Valencia, Spain
| | - Teresa Sevilla
- Neurology Department, Hospital Universitari i Politècnic La Fe, Valencia, Spain
| | - Nuria Muelas
- Neurology Department, Hospital Universitari i Politècnic La Fe, Valencia, Spain
| | - Manuel Guerreiro
- Hematology Department, Hospital Universitari i Politècnic La Fe, Valencia, Spain
| | - Carlos Carretero
- Hematology Department, Hospital Universitari i Politècnic La Fe, Valencia, Spain
| | - Inés Gómez
- Hematology Department, Hospital Universitari i Politècnic La Fe, Valencia, Spain
| | - Pilar Solves
- Hematology Department, Hospital Universitari i Politècnic La Fe, Valencia, Spain
| | - Miguel Ángel Sanz
- Hematology Department, Hospital Universitari i Politècnic La Fe, Valencia, Spain; CIBERONC, Instituto Carlos III, Madrid, Spain
| | - Guillermo Sanz
- Hematology Department, Hospital Universitari i Politècnic La Fe, Valencia, Spain; CIBERONC, Instituto Carlos III, Madrid, Spain
| | - Jaime Sanz
- Hematology Department, Hospital Universitari i Politècnic La Fe, Valencia, Spain; CIBERONC, Instituto Carlos III, Madrid, Spain
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13
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Ren XY, Liu X, Huang QS, Wang QM, He Y, Zhu XL, Han W, Chen H, Chen YH, Wang FR, Wang JZ, Zhang YY, Mo XD, Chen Y, Wang Y, Fu HX, Chang YJ, Xu LP, Liu KY, Huang XJ, Zhang XH. Incidence, Risk Factors, and Outcome of Immune-Mediated Neuropathies (IMNs) following Haploidentical Hematopoietic Stem Cell Transplantation. Biol Blood Marrow Transplant 2019; 25:1629-1636. [PMID: 31048087 DOI: 10.1016/j.bbmt.2019.04.021] [Citation(s) in RCA: 4] [Impact Index Per Article: 0.7] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 01/02/2019] [Revised: 03/14/2019] [Accepted: 04/24/2019] [Indexed: 12/18/2022]
Abstract
Immune-mediated neuropathies (IMNs) following hematopoietic stem cell transplantation have been described recently, which, excluding Guillain-Barré syndrome and chronic inflammatory demyelinating polyneuropathy, may present with atypical patterns. This retrospective, nested, case-control study reviewed data from 3858 patients who received haploidentical hematopoietic stem cell transplantation (haplo-HSCT) during the past 10 years at a single center, and 40 patients (1.04%) with IMN following haplo-HSCT were identified. Chronic graft-versus-host disease (cGVHD) (P = .043) and cytomegalovirus (CMV) viremia (P = .035) were recognized as independent risk factors for the development of IMN after haplo-HSCT. There were no significant differences in overall survival (P = .619), disease-free survival (P = .609), nonrelapse mortality (P = .87), or the incidence of relapse (P = .583) between patients with and without IMN after haplo-HSCT. However, patients with post-transplant IMN were at higher risk of developing cGVHD (P = .012) than patients who did not develop IMN. Twenty-four of the 40 patients with IMN (60%) attained neurologic improvement after treatments including vitamins B1 and B12 and/or immunomodulatory agents. However, 19 (47.5%) patients still had persistent motor/sensory deficits despite receiving timely treatment. More studies are needed to help develop standardized diagnostic and therapeutic strategies for patients with post-transplant IMN.
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Affiliation(s)
- Xi-Ying Ren
- Peking University People's Hospital, Peking University Institute of Hematology, Beijing, China; Beijing Key Laboratory of Hematopoietic Stem Cell Transplantation, Beijing, China; Collaborative Innovation Center of Hematology, Peking University, Beijing, China
| | - Xiao Liu
- Peking University People's Hospital, Peking University Institute of Hematology, Beijing, China; Beijing Key Laboratory of Hematopoietic Stem Cell Transplantation, Beijing, China; Collaborative Innovation Center of Hematology, Peking University, Beijing, China
| | - Qiu-Sha Huang
- Peking University People's Hospital, Peking University Institute of Hematology, Beijing, China; Beijing Key Laboratory of Hematopoietic Stem Cell Transplantation, Beijing, China; Collaborative Innovation Center of Hematology, Peking University, Beijing, China
| | - Qian-Ming Wang
- Peking University People's Hospital, Peking University Institute of Hematology, Beijing, China; Beijing Key Laboratory of Hematopoietic Stem Cell Transplantation, Beijing, China; Collaborative Innovation Center of Hematology, Peking University, Beijing, China
| | - Yun He
- Peking University People's Hospital, Peking University Institute of Hematology, Beijing, China; Beijing Key Laboratory of Hematopoietic Stem Cell Transplantation, Beijing, China; Collaborative Innovation Center of Hematology, Peking University, Beijing, China
| | - Xiao-Lu Zhu
- Peking University People's Hospital, Peking University Institute of Hematology, Beijing, China; Beijing Key Laboratory of Hematopoietic Stem Cell Transplantation, Beijing, China; Collaborative Innovation Center of Hematology, Peking University, Beijing, China
| | - Wei Han
- Peking University People's Hospital, Peking University Institute of Hematology, Beijing, China; Beijing Key Laboratory of Hematopoietic Stem Cell Transplantation, Beijing, China; Collaborative Innovation Center of Hematology, Peking University, Beijing, China
| | - Huan Chen
- Peking University People's Hospital, Peking University Institute of Hematology, Beijing, China; Beijing Key Laboratory of Hematopoietic Stem Cell Transplantation, Beijing, China; Collaborative Innovation Center of Hematology, Peking University, Beijing, China
| | - Yu-Hong Chen
- Peking University People's Hospital, Peking University Institute of Hematology, Beijing, China; Beijing Key Laboratory of Hematopoietic Stem Cell Transplantation, Beijing, China; Collaborative Innovation Center of Hematology, Peking University, Beijing, China
| | - Feng-Rong Wang
- Peking University People's Hospital, Peking University Institute of Hematology, Beijing, China; Beijing Key Laboratory of Hematopoietic Stem Cell Transplantation, Beijing, China; Collaborative Innovation Center of Hematology, Peking University, Beijing, China
| | - Jing-Zhi Wang
- Peking University People's Hospital, Peking University Institute of Hematology, Beijing, China; Beijing Key Laboratory of Hematopoietic Stem Cell Transplantation, Beijing, China; Collaborative Innovation Center of Hematology, Peking University, Beijing, China
| | - Yuan-Yuan Zhang
- Peking University People's Hospital, Peking University Institute of Hematology, Beijing, China; Beijing Key Laboratory of Hematopoietic Stem Cell Transplantation, Beijing, China; Collaborative Innovation Center of Hematology, Peking University, Beijing, China
| | - Xiao-Dong Mo
- Peking University People's Hospital, Peking University Institute of Hematology, Beijing, China; Beijing Key Laboratory of Hematopoietic Stem Cell Transplantation, Beijing, China; Collaborative Innovation Center of Hematology, Peking University, Beijing, China
| | - Yao Chen
- Peking University People's Hospital, Peking University Institute of Hematology, Beijing, China; Beijing Key Laboratory of Hematopoietic Stem Cell Transplantation, Beijing, China; Collaborative Innovation Center of Hematology, Peking University, Beijing, China
| | - Yu Wang
- Peking University People's Hospital, Peking University Institute of Hematology, Beijing, China; Beijing Key Laboratory of Hematopoietic Stem Cell Transplantation, Beijing, China; Collaborative Innovation Center of Hematology, Peking University, Beijing, China
| | - Hai-Xia Fu
- Peking University People's Hospital, Peking University Institute of Hematology, Beijing, China; Beijing Key Laboratory of Hematopoietic Stem Cell Transplantation, Beijing, China; Collaborative Innovation Center of Hematology, Peking University, Beijing, China
| | - Ying-Jun Chang
- Peking University People's Hospital, Peking University Institute of Hematology, Beijing, China; Beijing Key Laboratory of Hematopoietic Stem Cell Transplantation, Beijing, China; Collaborative Innovation Center of Hematology, Peking University, Beijing, China
| | - Lan-Ping Xu
- Peking University People's Hospital, Peking University Institute of Hematology, Beijing, China; Beijing Key Laboratory of Hematopoietic Stem Cell Transplantation, Beijing, China; Collaborative Innovation Center of Hematology, Peking University, Beijing, China
| | - Kai-Yan Liu
- Peking University People's Hospital, Peking University Institute of Hematology, Beijing, China; Beijing Key Laboratory of Hematopoietic Stem Cell Transplantation, Beijing, China; Collaborative Innovation Center of Hematology, Peking University, Beijing, China
| | - Xiao-Jun Huang
- Peking University People's Hospital, Peking University Institute of Hematology, Beijing, China; Beijing Key Laboratory of Hematopoietic Stem Cell Transplantation, Beijing, China; Collaborative Innovation Center of Hematology, Peking University, Beijing, China
| | - Xiao-Hui Zhang
- Peking University People's Hospital, Peking University Institute of Hematology, Beijing, China; Beijing Key Laboratory of Hematopoietic Stem Cell Transplantation, Beijing, China; Collaborative Innovation Center of Hematology, Peking University, Beijing, China.
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Rudant J, Dupont A, Mikaeloff Y, Bolgert F, Coste J, Weill A. Surgery and risk of Guillain-Barré syndrome: A French nationwide epidemiologic study. Neurology 2018; 91:e1220-e1227. [PMID: 30143563 DOI: 10.1212/wnl.0000000000006246] [Citation(s) in RCA: 48] [Impact Index Per Article: 6.9] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 02/19/2018] [Accepted: 06/28/2018] [Indexed: 12/11/2022] Open
Abstract
OBJECTIVE To assess the association between Guillain-Barré syndrome (GBS) and recent surgery based on French nationwide data. METHODS Data were extracted from the French health administrative databases (SNIIRAM/PMSI). All patients hospitalized for GBS between 2009 and 2014 were identified by ICD-10 code G61.0 as main diagnosis. Patients previously hospitalized for GBS in 2006, 2007, and 2008 were excluded. Surgical procedures were identified from the hospital database. Hospitalizations for surgery with no infection diagnosis code entered during the hospital stay were also identified. The association between GBS and a recent surgical procedure was estimated using a case-crossover design. Case and referent windows were defined as 1-60 days and 366-425 days before GBS hospitalization, respectively. Analyses were adjusted for previous episodes of gastroenteritis and respiratory tract infection, identified by drug dispensing data. RESULTS Of the 8,364 GBS cases included, 175 and 257 patients had undergone a surgical procedure in the referent and case windows, respectively (adjusted odds ratio [OR] = 1.53, 95% confidence interval [CI]: 1.25-1.88). A slightly weaker association was observed for surgical procedures with no identified infection during the hospitalization (OR = 1.40, 95% CI: 1.12-1.73). Regarding the type of surgery, only surgical procedures on bones and digestive organs were significantly associated with GBS (OR and 95% CI = 2.78 [1.68-4.60] and 2.36 [1.32-4.21], respectively). CONCLUSION In this large nationwide epidemiologic study, GBS was moderately associated with any type of recent surgery and was more strongly associated with bone and digestive organ surgery.
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Affiliation(s)
- Jérémie Rudant
- From Caisse Nationale de l'Assurance Maladie (J.R., A.D., J.C., A.W.), Direction de la Stratégie des Études et des Statistiques, Département Études en Santé Publique, Paris; Assistance Publique-Hôpitaux de Paris (Y.M.), Hôpital Bicêtre, Unité de Rééducation Neurologique Infantile, Bicêtre; Université Paris-Saclay (Y.M.), Université Paris-Sud, CESP, Inserm, Villejuif; and Assistance Publique-Hôpitaux de Paris (F.B.), Hôpital Pitié-Salpêtrière, Réanimation Neurologique, Neurologie 1, Paris, France.
| | - Axelle Dupont
- From Caisse Nationale de l'Assurance Maladie (J.R., A.D., J.C., A.W.), Direction de la Stratégie des Études et des Statistiques, Département Études en Santé Publique, Paris; Assistance Publique-Hôpitaux de Paris (Y.M.), Hôpital Bicêtre, Unité de Rééducation Neurologique Infantile, Bicêtre; Université Paris-Saclay (Y.M.), Université Paris-Sud, CESP, Inserm, Villejuif; and Assistance Publique-Hôpitaux de Paris (F.B.), Hôpital Pitié-Salpêtrière, Réanimation Neurologique, Neurologie 1, Paris, France
| | - Yann Mikaeloff
- From Caisse Nationale de l'Assurance Maladie (J.R., A.D., J.C., A.W.), Direction de la Stratégie des Études et des Statistiques, Département Études en Santé Publique, Paris; Assistance Publique-Hôpitaux de Paris (Y.M.), Hôpital Bicêtre, Unité de Rééducation Neurologique Infantile, Bicêtre; Université Paris-Saclay (Y.M.), Université Paris-Sud, CESP, Inserm, Villejuif; and Assistance Publique-Hôpitaux de Paris (F.B.), Hôpital Pitié-Salpêtrière, Réanimation Neurologique, Neurologie 1, Paris, France
| | - Francis Bolgert
- From Caisse Nationale de l'Assurance Maladie (J.R., A.D., J.C., A.W.), Direction de la Stratégie des Études et des Statistiques, Département Études en Santé Publique, Paris; Assistance Publique-Hôpitaux de Paris (Y.M.), Hôpital Bicêtre, Unité de Rééducation Neurologique Infantile, Bicêtre; Université Paris-Saclay (Y.M.), Université Paris-Sud, CESP, Inserm, Villejuif; and Assistance Publique-Hôpitaux de Paris (F.B.), Hôpital Pitié-Salpêtrière, Réanimation Neurologique, Neurologie 1, Paris, France
| | - Joël Coste
- From Caisse Nationale de l'Assurance Maladie (J.R., A.D., J.C., A.W.), Direction de la Stratégie des Études et des Statistiques, Département Études en Santé Publique, Paris; Assistance Publique-Hôpitaux de Paris (Y.M.), Hôpital Bicêtre, Unité de Rééducation Neurologique Infantile, Bicêtre; Université Paris-Saclay (Y.M.), Université Paris-Sud, CESP, Inserm, Villejuif; and Assistance Publique-Hôpitaux de Paris (F.B.), Hôpital Pitié-Salpêtrière, Réanimation Neurologique, Neurologie 1, Paris, France
| | - Alain Weill
- From Caisse Nationale de l'Assurance Maladie (J.R., A.D., J.C., A.W.), Direction de la Stratégie des Études et des Statistiques, Département Études en Santé Publique, Paris; Assistance Publique-Hôpitaux de Paris (Y.M.), Hôpital Bicêtre, Unité de Rééducation Neurologique Infantile, Bicêtre; Université Paris-Saclay (Y.M.), Université Paris-Sud, CESP, Inserm, Villejuif; and Assistance Publique-Hôpitaux de Paris (F.B.), Hôpital Pitié-Salpêtrière, Réanimation Neurologique, Neurologie 1, Paris, France
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Abstract
Guillain-Barré syndrome (GBS) is a term that is used to describe a group of immune-mediated peripheral neuropathies, with the most common feature being rapid polyradiculoneuropathy. The exact etiology of this syndrome is unknown. In the field of orthopedics, GBS has been reported to occur after total hip arthroplasty, orthopedic trauma, and spine surgery. We report a unique case of GBS after elective revision lumbar spine surgery. A 62-year-old female presented with persistent low back pain and radiculopathy and elected to have revision lumbar spine surgery. Approximately 24 to 36 hours after hospital discharge, she returned to the hospital with weakness in her legs. After an electromyography (EMG), the patient was diagnosed with GBS and placed on intravenous immunoglobulin (IVIG). She developed respiratory failure, which required intubation and eventually converted to a tracheostomy and was finally decannulated. Over the course of 12 months, she improved to her pre-surgical baseline, gaining 5/5 strength in her upper and lower extremities and was able to ambulate independently without any aids. This was a case of GBS that occurred in a patient approximately two weeks after revision lumbar surgery. GBS is a poorly understood and rare complication of lumbar spine surgery that needs to be recognized quickly to be effectively treated.
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Affiliation(s)
- Aymen Rashid
- Department of Orthopedic Surgery, SUNY Upstate Medical University
| | - Swamy Kurra
- Department of Orthopedic Surgery, SUNY Upstate Medical University
| | - William Lavelle
- Department of Orthopedic Surgery, SUNY Upstate Medical University
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16
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Pritchard J, Hughes RAC, Hadden RDM, Brassington R. Pharmacological treatment other than corticosteroids, intravenous immunoglobulin and plasma exchange for Guillain-Barré syndrome. Cochrane Database Syst Rev 2016; 11:CD008630. [PMID: 27846348 PMCID: PMC6464710 DOI: 10.1002/14651858.cd008630.pub4] [Citation(s) in RCA: 7] [Impact Index Per Article: 0.8] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 12/25/2022]
Abstract
BACKGROUND Plasma exchange and intravenous immunoglobulin, but not corticosteroids, are beneficial in Guillain-Barré syndrome (GBS). The efficacy of other pharmacological agents is unknown. This review was first published in 2011 and updated in 2013 and 2016. OBJECTIVES To assess the effects of pharmacological agents other than plasma exchange, intravenous immunoglobulin and corticosteroids for GBS. SEARCH METHODS On 18 January 2016, we searched the Cochrane Neuromuscular Specialised Register, Cochrane Central Register of Controlled Trials, MEDLINE, and Embase for treatments for GBS. We also searched clinical trials registries. SELECTION CRITERIA We included all randomised controlled trials (RCTs) or quasi-RCTs of acute GBS (within four weeks from onset) of all types and degrees of severity, and in individuals of all ages. We discarded trials that investigated only corticosteroids, intravenous immunoglobulin or plasma exchange. We included other pharmacological treatments or combinations of treatments compared with no treatment, placebo or another treatment. We also identified a number of non-randomised studies during the search, the results of which we considered in the Discussion. DATA COLLECTION AND ANALYSIS We followed standard Cochrane methodology. MAIN RESULTS We identified no new trials during this update of the review. In previous versions of this review we identified only very low quality evidence for four different interventions published in four studies. Each study had a high risk of bias in at least one respect. One RCT with 19 participants comparing interferon beta-1a and placebo showed no clinically important difference in any outcome between groups. Another with 10 participants comparing brain-derived neurotrophic factor and placebo showed no clinically important difference in any outcome between groups. A third with 37 participants comparing cerebrospinal fluid filtration and plasma exchange also showed no clinically important difference in any outcome between groups. In a fourth with 43 participants, the risk ratio for an improvement by one or more disability grade after eight weeks was greater with the Chinese herbal medicine tripterygium polyglycoside than with corticosteroids (risk ratio 1.47; 95% confidence interval 1.02 to 2.11); other outcomes in this trial showed no difference. Serious adverse events were uncommon with each of these treatments and in the control groups. AUTHORS' CONCLUSIONS The quality of the evidence was very low. Three small RCTs, comparing interferon beta-1a or brain-derived neurotrophic factor with placebo, and cerebrospinal fluid filtration with plasma exchange, showed no significant benefit or harm for any of the interventions. A fourth small trial showed that the Chinese herbal medicine, tripterygium polyglycoside, hastened recovery in people with GBS to a greater extent than corticosteroids, but this result needs confirmation. We were unable to draw any useful conclusions from the few observational studies we identified.
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Affiliation(s)
- Jane Pritchard
- Charing Cross HospitalNeuromuscular Unit 3 NorthFulham Palace RoadLondonUKW6 8RF
| | - Richard AC Hughes
- National Hospital for Neurology and NeurosurgeryMRC Centre for Neuromuscular DiseasesPO Box 114Queen SquareLondonUKWC1N 3BG
| | - Robert DM Hadden
- King's College HospitalDepartment of NeurologyDenmark HillLondonUKSE5 9RS
| | - Ruth Brassington
- National Hospital for Neurology and NeurosurgeryMRC Centre for Neuromuscular DiseasesPO Box 114Queen SquareLondonUKWC1N 3BG
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17
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Alhefzi M, Aycart M, Bueno E, Kueckelhaus M, Fischer S, Snook R, Sharfuddin A, Hadad I, Malla P, Amato A, Pomahac B, Marty F. Guillain-Barré syndrome associated with resistant cytomegalovirus infection after face transplantation. Transpl Infect Dis 2016; 18:288-92. [DOI: 10.1111/tid.12516] [Citation(s) in RCA: 12] [Impact Index Per Article: 1.3] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 08/27/2015] [Revised: 11/06/2015] [Accepted: 12/30/2015] [Indexed: 11/29/2022]
Affiliation(s)
- M. Alhefzi
- Division of Plastic Surgery; Department of Surgery; Brigham and Women's Hospital; Harvard Medical School; Boston Massachusetts USA
| | - M.A. Aycart
- Division of Plastic Surgery; Department of Surgery; Brigham and Women's Hospital; Harvard Medical School; Boston Massachusetts USA
| | - E.M. Bueno
- Division of Plastic Surgery; Department of Surgery; Brigham and Women's Hospital; Harvard Medical School; Boston Massachusetts USA
| | - M. Kueckelhaus
- Division of Plastic Surgery; Department of Surgery; Brigham and Women's Hospital; Harvard Medical School; Boston Massachusetts USA
| | - S. Fischer
- Division of Plastic Surgery; Department of Surgery; Brigham and Women's Hospital; Harvard Medical School; Boston Massachusetts USA
| | - R.J. Snook
- Department of Neurology; Indiana University School of Medicine; Indianapolis Indiana USA
| | - A.A. Sharfuddin
- Division of Nephrology; Department of Medicine; Indiana University School of Medicine; Indianapolis Indiana USA
| | - I. Hadad
- Division of Plastic Surgery; Indiana University School of Medicine; Indianapolis Indiana USA
| | - P. Malla
- Division of Neuromuscular Disease; Department of Neurology; Brigham and Women's Hospital; Harvard Medical School; Boston Massachusetts USA
| | - A.A. Amato
- Division of Neuromuscular Disease; Department of Neurology; Brigham and Women's Hospital; Harvard Medical School; Boston Massachusetts USA
| | - B. Pomahac
- Division of Plastic Surgery; Department of Surgery; Brigham and Women's Hospital; Harvard Medical School; Boston Massachusetts USA
| | - F.M. Marty
- Division of Infectious Diseases; Department of Medicine; Brigham and Women's Hospital; Harvard Medical School; Boston Massachusetts USA
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Acute demyelinating polyneuropathy after lung transplantation: guillain-barré syndrome or tacrolimus toxicity? Case Rep Transplant 2014; 2014:685010. [PMID: 25184071 PMCID: PMC4144080 DOI: 10.1155/2014/685010] [Citation(s) in RCA: 2] [Impact Index Per Article: 0.2] [Reference Citation Analysis] [Abstract] [Track Full Text] [Download PDF] [Journal Information] [Subscribe] [Scholar Register] [Received: 07/03/2014] [Accepted: 07/30/2014] [Indexed: 12/03/2022] Open
Abstract
Guillain-Barré syndrome (GBS) has been described after solid organ and bone marrow transplantation mostly due to viral infections and possibly calcineurin inhibitors. Incidence after bone marrow transplant is 0.3–0.7%, though incidence in other transplants is not well known. We present the first description of tacrolimus associated GBS in lung transplant recipients in the English language literature. The pathophysiology of tacrolimus-induced polyneuropathy is not known, but some have hypothesized that tacrolimus induces an inflammatory phenomenon by differential effects on T cell subsets. Diagnosis of association may be challenging and requires high index of suspicion. The optimal treatment of GBS-associated with tacrolimus after lung transplantation is unknown, although drug discontinuation may result in improvement in some patients, while some reports suggest that the use of IVIG and/or plasmapheresis may be helpful and safe in organ transplant recipients with severe symptoms.
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19
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Wakerley BR, Yuki N. Infectious and noninfectious triggers in Guillain-Barré syndrome. Expert Rev Clin Immunol 2014; 9:627-39. [PMID: 23899233 DOI: 10.1586/1744666x.2013.811119] [Citation(s) in RCA: 98] [Impact Index Per Article: 8.9] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 12/24/2022]
Abstract
Guillain-Barré syndrome (GBS) is the commonest cause of acquired flaccid paralysis in the world and regarded by many as the prototype for postinfectious autoimmunity. Here the authors consider both infectious and noninfectious triggers of GBS and determine where possible what immunological mechanisms may account for this association. In approximately two-thirds of cases, an infectious trigger is reported in the weeks that lead up to disease onset, indicating that the host's response to infection must play an important role in disease pathogenesis. The most frequently identified bacteria, Campylobacter jejuni, through a process known as molecular mimicry, has been shown to induce cross-reactive anti-ganglioside antibodies, which can lead to the development of axonal-type GBS in some patients. Whether this paradigm can be extended to other infectious organisms or vaccines remains an important area of research and has public health implications. GBS has also been reported rarely in patients with underlying systemic diseases and immunocompromised states and although the exact mechanism is yet to be established, increased susceptibility to known infectious triggers should be considered most likely.
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Affiliation(s)
- Benjamin R Wakerley
- Department of Medicine, National University Hospital, 1E Kent Ridge Road, Singapore.
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20
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Piras E, Caocci G, Pisano V, Orrù F, Murgia F, Sanna M, Vacca A, La Nasa G. Guillain–Barré syndrome after human herpesvirus-6 reactivation in unrelated hematopoietic stem cell transplantation. Leuk Lymphoma 2013; 54:1332-3. [DOI: 10.3109/10428194.2012.740560] [Citation(s) in RCA: 4] [Impact Index Per Article: 0.3] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/13/2022]
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21
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Hughes RAC, Pritchard J, Hadden RDM. Pharmacological treatment other than corticosteroids, intravenous immunoglobulin and plasma exchange for Guillain-Barré syndrome. Cochrane Database Syst Rev 2013:CD008630. [PMID: 23450584 DOI: 10.1002/14651858.cd008630.pub3] [Citation(s) in RCA: 14] [Impact Index Per Article: 1.2] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 12/28/2022]
Abstract
BACKGROUND Plasma exchange and intravenous immunoglobulin, but not corticosteroids, are beneficial in Guillain-Barré syndrome (GBS). The efficacy of other pharmacological agents is unknown. This review was first published in 2011 and this update in 2013. OBJECTIVES To review systematically the evidence from randomised controlled trials (RCTs) for pharmacological agents other than plasma exchange, intravenous immunoglobulin and corticosteroids. SEARCH METHODS On 28 August 2012, we searched the Cochrane Neuromuscular Disease Group Specialized Register CENTRAL (2012, Issue 8 in The Cochrane Library), MEDLINE (January 1966 to August 2012) and EMBASE (January 1980 to August 2012) for treatments for GBS. We considered evidence from non-randomised studies in the Discussion. SELECTION CRITERIA We included all randomised or quasi-RCTs of acute (within four weeks from onset) GBS of all types, ages and degrees of severity. We discarded trials which only tested corticosteroids, intravenous immunoglobulin or plasma exchange. We included other pharmacological treatments or combinations of treatments compared with no treatment, placebo treatment or another treatment. DATA COLLECTION AND ANALYSIS Change in disability after four weeks was the primary outcome. Two authors checked references and extracted data independently. One author entered and another checked data in Review Manager (RevMan). We assessed risk of bias according to the Cochrane Handbook for Systematic Reviews of Interventions. We calculated mean differences and risk ratios with their 95% confidence intervals. We assessed strength of evidence with GradePro software. MAIN RESULTS Only very low quality evidence was found for four different interventions. This update of the review found no new trials. One RCT with 13 participants showed no significant difference in any outcome between interferon beta-1a and placebo. Another with 10 participants showed no significant difference in any outcome between brain-derived neurotrophic factor and placebo. A third with 37 participants showed no significant difference in any outcome between cerebrospinal fluid filtration and plasma exchange. In a fourth with 20 participants, the risk ratio of improving by one or more disability grades after eight weeks was significantly greater with the Chinese herbal medicine tripterygium polyglycoside than with corticosteroids (risk ratio 1.47; 95% confidence interval 1.02 to 2.11). Serious adverse events were uncommon with each of these treatments and not significantly commoner in the treated than the control groups. AUTHORS' CONCLUSIONS The quality of the evidence was very low. Three small RCTs, of interferon beta-1a, brain-derived neurotrophic factor and cerebrospinal fluid filtration, showed no significant benefit or harm. A fourth small trial showed that the Chinese herbal medicine tripterygium polyglycoside hastened recovery significantly more than corticosteroids but this result needs confirmation. It was not possible to draw useful conclusions from the few observational studies.
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Affiliation(s)
- Richard A C Hughes
- MRC Centre for Neuromuscular Diseases, National Hospital for Neurology and Neurosurgery, London, UK.
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22
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Baker MG, Kvalsvig A, Zhang J, Lake R, Sears A, Wilson N. Declining Guillain-Barré syndrome after campylobacteriosis control, New Zealand, 1988-2010. Emerg Infect Dis 2012; 18:226-33. [PMID: 22304786 PMCID: PMC3310455 DOI: 10.3201/eid1802.111126] [Citation(s) in RCA: 36] [Impact Index Per Article: 2.8] [Reference Citation Analysis] [Abstract] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/19/2022] Open
Abstract
Food safety measures that lower incidence of campylobacteriosis might also prevent Guillain-Barré syndrome.
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23
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Gensicke H, Datta AN, Dill P, Schindler C, Fischer D. Increased incidence of Guillain-Barré syndrome after surgery. Eur J Neurol 2012; 19:1239-44. [PMID: 22519650 DOI: 10.1111/j.1468-1331.2012.03730.x] [Citation(s) in RCA: 78] [Impact Index Per Article: 6.0] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 10/25/2011] [Accepted: 03/13/2012] [Indexed: 11/28/2022]
Abstract
BACKGROUND AND PURPOSE Antecedent surgery has been described to trigger Guillain-Barré syndrome (GBS), but its evidence is poor and based on case reports only. METHODS We performed a retrospective analysis of 63 patients with GBS admitted to the University Hospital Basel and University Children's Hospital Basel from January 2005 to December 2010. We calculated and compared the incidences of post-surgical and non-exposed patients with GBS in the study population and those reported previously in literature. RESULTS Six of 63 (9.5%) GBS cases had had a surgery within 6 weeks prior to GBS. The relative risk of developing GBS during the 6-week period after surgery is 13.1 times higher than the normal incidence in the study population (95% confidence interval: 5.68, 30.3; P ≤ 0.0001), suggesting an attributable risk of 4.1 cases per 100, 000 surgeries. In addition, the incidence of post-surgical GBS is significantly higher than influenza vaccine-associated GBS in the study population (P = 0.01) as well as in comparison with previous reported vaccine-associated GBS (P ≤ 0.0001) and background incidences (P ≤ 0.0001). CONCLUSION Surgery must be considered to be a potential risk factor for developing GBS.
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Affiliation(s)
- H Gensicke
- Department of Neurology, University Hospital Basel, Basel, Switzerland
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24
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Pustavoitau A, Bhardwaj A, Stevens R. Analytic Review: Neurological Complications of Transplantation. J Intensive Care Med 2011; 26:209-22. [DOI: 10.1177/0885066610389549] [Citation(s) in RCA: 30] [Impact Index Per Article: 2.1] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 01/05/2023]
Abstract
Recipients of solid organ or hematopoietic cell transplants are at risk of life-threatening neurological disorders including encephalopathy, seizures, infections and tumors of the central nervous system, stroke, central pontine myelinolysis, and neuromuscular disorders—often requiring admission to, or occurring in, the intensive care unit (ICU). Many of these complications are linked directly or indirectly to immunosuppressive therapy. However, neurological disorders may also result from graft versus host disease, or be an expression of the underlying disease which prompted transplantation, as well as injury induced during radiation, chemotherapy, surgery, and ICU stay. In rare cases, neuroinfectious pathogens may be transmitted with the transplanted tissue or organ. Diagnosis may be a challenge because clinical symptoms and findings on neuroimaging lack specificity, and a biological specimen or tissue diagnosis is often needed for definitive diagnosis. Management is centered on preventing further neurological injury, etiology-targeted therapy, and balancing the benefits and toxicities of specific immunosuppressive agents.
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Affiliation(s)
- Aliaksei Pustavoitau
- Departments of Anesthesiology Critical Care Medicine, Johns Hopkins University School of Medicine, Baltimore, MD, USA
| | - Anish Bhardwaj
- Departments of Neurology and Neurological Surgery, Tufts University School of Medicine, Boston, MA, USA,
| | - Robert Stevens
- Departments of Anesthesiology Critical Care Medicine, Neurology, Neurosurgery, and Radiology, Johns Hopkins University School of Medicine, Baltimore, MD, USA
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25
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Annaloro C, Onida F, Lambertenghi Deliliers G. Autologous hematopoietic stem cell transplantation in autoimmune diseases. Expert Rev Hematol 2011; 2:699-715. [PMID: 21082959 DOI: 10.1586/ehm.09.60] [Citation(s) in RCA: 14] [Impact Index Per Article: 1.0] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 12/13/2022]
Abstract
The term 'autoimmune diseases' encompasses a spectrum of diseases whose clinical manifestations and, possibly, biological features vary widely. The results of conventional treatment are considered unsatisfactory in aggressive forms, with subsets of patients having short life expectancies. Relying on wide experimental evidence and more feeble clinical data, some research groups have used autologous hematopoietic stem cell transplantation (HSCT) in the most disabling autoimmune diseases with the aim of resetting the patient's immune system. Immunoablative conditioning regimens are preferred over their myeloablative counterparts, and some form of in vivo and/or ex vivo T-cell depletion is generally adopted. Despite 15 years' experience, published controlled clinical trials are still lacking, with the evidence so far available coming from pilot studies and registry surveys. In multiple sclerosis, clinical improvement, or at least lasting disease stabilization, can be achieved in the majority of the patients; nevertheless, the worst results are observed in patients with progressive disease, where no benefit can be expected from conventional therapy. Concerning rheumatologic diseases, wide experience has been acquired in systemic sclerosis, with long-term improvements in cutaneous disease being frequently reported, although visceral involvement remains unchanged at best. Autografting has proved to be barely effective in rheumatoid arthritis and quite toxic in juvenile idiopathic arthritis, whereas it leads to clinical remission and the reversal of visceral impairment in the majority of patients with systemic lupus erythematosus. A promising indication is Crohn's disease, in which long-term endoscopic remission is frequently observed. Growing experience with autologous HCST in autoimmune diseases has progressively reduced concerns about transplant-related mortality and secondary myelodysplasia/leukemia. Therefore, a sustained complete remission seems to be within the reach of autografting in some autoimmune diseases; in others, the indications, risks and benefits of autografting need to be better defined. Consequently, the search for new drugs should also be encouraged.
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Affiliation(s)
- Claudio Annaloro
- Bone Marrow Transplantation Center-Hematology I, Fondazione Ospedale Maggiore Policlinico, Mangiagalli e Regina Elena, University of Milan, Via Francesco Sforza 35, Milan, Italy
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26
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Hughes RA, Pritchard J, Hadden RD. Pharmacological treatment other than corticosteroids, intravenous immunoglobulin and plasma exchange for Guillain Barré syndrome. Cochrane Database Syst Rev 2011:CD008630. [PMID: 21412923 DOI: 10.1002/14651858.cd008630.pub2] [Citation(s) in RCA: 15] [Impact Index Per Article: 1.1] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 01/17/2023]
Abstract
BACKGROUND Plasma exchange and intravenous immunoglobulin, but not corticosteroids, are beneficial in Guillain-Barré syndrome (GBS). The efficacy of other pharmacological agents is unknown. OBJECTIVES To review systematically the evidence from randomised controlled trials for pharmacological agents other than plasma exchange, intravenous immunoglobulin and corticosteroids. SEARCH STRATEGY We searched the Cochrane Neuromuscular Disease Group Specialized Register (5 July 2010), the Cochrane Central Register of Controlled Trials (CENTRAL) (The Cochrane Library 2010, Issue 2), MEDLINE (January 1966 to June 2010) and EMBASE (January 1980 to June 2010) for treatments for GBS. We considered evidence from non-randomised studies in the Discussion. SELECTION CRITERIA We included all randomised or quasi-randomised controlled trials of acute (within four weeks from onset) GBS of all types, ages and degrees of severity. We discarded trials which only tested corticosteroids, intravenous immunoglobulin or plasma exchange. We included other pharmacological treatments or combinations of treatments compared with no treatment, placebo treatment or another treatment. DATA COLLECTION AND ANALYSIS Change in disability after four weeks was the primary outcome. Two authors checked references and extracted data independently. One author entered and another checked data in Review Manager (RevMan). We assessed risk of bias according to the Cochrane Handbook for Systematic Reviews of Interventions. We calculated mean differences and risk ratios with their 95% confidence intervals. We assessed strength of evidence with GradePro software. MAIN RESULTS Only very low quality evidence was found for four different interventions. One randomised controlled trial with 13 participants showed no significant difference in any outcome between interferon beta-1a and placebo. Another with 10 participants showed no significant difference in any outcome between brain-derived neurotrophic factor and placebo. A third with 37 participants showed no significant difference in any outcome between cerebrospinal fluid filtration and plasma exchange. In a fourth with 20 participants, the risk ratio of improving by one or more disability grades after eight weeks was significantly greater with the Chinese herbal medicine tripterygium polyglycoside than with corticosteroids (risk ratio 1.47; 95% confidence interval 1.02 to 2.11). AUTHORS' CONCLUSIONS The quality of the evidence was very low. Three small randomised controlled trials, of interferon beta-1a, brain-derived neurotrophic factor and cerebrospinal fluid filtration, showed no significant benefit or harm. A fourth small trial showed that the Chinese herbal medicine tripterygium polyglycoside hastened recovery significantly more than corticosteroids but this result needs confirmation. It was not possible to draw useful conclusions from the few observational studies.
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Affiliation(s)
- Richard Ac Hughes
- MRC Centre for Neuromuscular Disease, National Hospital for Neurology and Neurosurgery, PO Box 114, Queen Square, London, UK, WC1N 3BG
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27
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Wang HX, Yan HM, Liu J, Duan LN, Wang ZD, Zhu L, Xue M, Guo ZK. Haploidentical hematopoietic stem-cell transplantation for non-Hodgkin lymphoma with bone marrow involvement. Leuk Lymphoma 2011; 50:1488-93. [PMID: 19811326 DOI: 10.1080/10428190903156745] [Citation(s) in RCA: 12] [Impact Index Per Article: 0.9] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 10/20/2022]
Abstract
Here, we report the preliminary results of haploidentical hematopoietic stem cell transplantation (Haplo-HSCT) with granulocyte-colony-stimulating factor (G-CSF) mobilized bone marrow grafts without T-cell depletion for 10 patients with refractory non-Hodgkin lymphoma accompanied by bone marrow involvement. Eight patients received a conditioning regimen consisting of high-doses of cytarabine and cyclophosphamide with total body irradiation, whereas two cases were preconditioned with busulfan, thiotepa, and cyclophosphamide. All patients had rapid hematopoietic engraftment with the mean time for neutrophil and platelet recovery being 16.6 days and 19.2 days, respectively. Three cases died within 6 months after transplantation from severe acute graft-versus-host disease, fungal infection, or relapse. The others are currently alive in complete remission at a median follow-up of 60.71 months (range: 44-81 months). The results here suggest that haplo-HSCT might provide an opportunity of myeloablative therapy for refractory lymphoma with marrow infiltration.
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Affiliation(s)
- Heng-Xiang Wang
- Department of Hematology, The General Hospital of Air Force, Beijing, China
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28
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Melguizo I, Gilbert M, Tummala S. Guillain-Barré syndrome and glioblastoma. J Neurooncol 2010; 104:371-3. [PMID: 21104295 DOI: 10.1007/s11060-010-0471-7] [Citation(s) in RCA: 3] [Impact Index Per Article: 0.2] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 06/11/2010] [Accepted: 11/12/2010] [Indexed: 12/01/2022]
Abstract
Guillain-Barré syndrome (GBS) is defined as an acute demyelinating peripheral neuropathy. We describe a case of GBS in a patient with glioblastoma undergoing chemotherapy treatment. A 57 year old woman diagnosed with glioblastoma developed a subacute progressive history of bilateral symmetric numbness of her fingers and toes, belt-type neuropathic pain, a left facial droop and upper and lower extremity muscle weakness. There was no evidence of a tumor mass or leptomeningeal disease in the spine. Electrophysiological studies confirmed the diagnosis. Although rare, GBS should be considered in primary brain tumor patients with generalized acute-subacute progressive weakness that is inconsistent with the location of their tumor, particularly if they are also on chemotherapy contributing to their immunosuppressive state.
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Affiliation(s)
- Isaac Melguizo
- Department of Neuro-oncology, The University of Texas M.D. Anderson Cancer Center, 1515 Holcombe Blvd, Unit Number: 431, Room Number: FC7.3057, Houston, TX 77030, USA.
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29
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Guillain-Barré syndrome mimicking acute methotrexate-associated encephalopathy in an adolescent patient with lymphoblastic lymphoma. J Pediatr Hematol Oncol 2010; 32:615-6. [PMID: 20463610 DOI: 10.1097/mph.0b013e3181d204b1] [Citation(s) in RCA: 3] [Impact Index Per Article: 0.2] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 11/25/2022]
Abstract
We describe an adolescent case of Guillain-Barré syndrome (GBS) mimicking acute methotrexate-associated encephalopathy during chemotherapy for lymphoblastic lymphoma. Although initial presentations of hemiparesis and irritability were suggestive of acute encephalopathy, the diminished deep tendon reflexes and subsequent rapid progression to flaccid triparesis with bulbar palsy were consistent with GBS. After the initiation of intravenous immunoglobulin therapy her symptoms improved rapidly, and the diagnosis of GBS was confirmed by nerve conduction studies and cerebrospinal fluid examination in recovery phase. GBS should be considered in the differential diagnosis of acute methotrexate-associated encephalopathy, although GBS is a rare neurologic complication.
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30
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Kaida K, Ariga T, Yu RK. Antiganglioside antibodies and their pathophysiological effects on Guillain-Barré syndrome and related disorders--a review. Glycobiology 2009; 19:676-92. [PMID: 19240270 DOI: 10.1093/glycob/cwp027] [Citation(s) in RCA: 115] [Impact Index Per Article: 7.2] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/13/2022] Open
Abstract
Guillain-Barré syndrome (GBS) is an acute immune-mediated polyradiculoneuropathy which can cause acute quadriplegia. Infection with micro-organisms, including Campylobacter jejuni (C. jejuni), Haemophilus influenzae, and Cytomegalovirus (CMV), is recognized as a main triggering event for the disease. Lipooligosaccharide (LOS) genes are responsible for the formation of human ganglioside-like LOS structures in infectious micro-organisms that can induce GBS. Molecular mimicry of LOSs on the surface of infectious agents and of ganglioside antigens on neural cells is thought to induce cross-reactive humoral and cellular immune responses. Patients with GBS develop antibodies against those gangliosides, resulting in autoimmune targeting of peripheral nerve sites, leading to neural damage. Heterogeneity of ganglioside expression in the peripheral nervous system (PNS) may underlie the differential clinical manifestation of the GBS variants. Recent studies demonstrate that some GBS sera react with ganglioside complexes consisting of two different gangliosides, such as GD1a and GD1b, or GM1 and GD1a, but not with each constituent ganglioside alone. The discovery of antiganglioside complex antibodies not only improves the detection rate of autoantibodies in GBS, but also provides a new concept in the antibody-antigen interaction through clustered carbohydrate epitopes. Although ganglioside mimicry is one of the possible etiological causes of GBS, unidentified factors may also contribute to the pathogenesis of GBS. While GBS is not considered a genetic disease, host factors, particularly human lymphocyte antigen type, appear to have a role in the pathogenesis of GBS following C. jejuni infection.
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Affiliation(s)
- Kenichi Kaida
- Institute of Molecular Medicine and Genetics, Medical College of Georgia, Augusta, GA 30912, USA
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