Successful Management of Anti‐mGluR1 Encephalitis with Immunosuppressive Treatment: Dengue Virus as a Trigger?

Chaumont, Hugo; Petit, Aimée; Mameri, Tanicia; Schollhammer, Romain; Honnorat, Jérôme; Lannuzel, Annie

doi:10.1002/mdc3.12841

Cited by 4 publications

(4 citation statements)

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“…However, one case was preceded by herpes zoster infection in the trigeminal nerve a month before the disease onset, another was preceded by streptococcal pharyngitis 2 months prior, and yet another case was found to have evidence of dengue virus infection. These findings suggest a post-infectious element in the occurrence of anti-mGluR1 encephalitis or that infection may trigger its onset ( 4 , 14 , 15 ). Unlike most autoimmune disorders which favor women, coincident autoimmunity in anti-mGluR1 encephalitis patients affected both genders equally ( 26 ).…”

Section: Discussionmentioning

confidence: 85%

Anti-mGluR1 encephalitis: Case illustration and systematic review

2023

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BackgroundThe literature for immune-mediated neurological disorders is evolving like no other field of neurological illnesses. Many new antibodies or disorders have been described in the last decade. The cerebellum is a brain structure susceptible to these immune-mediated pathologies, and anti-metabotropic glutamate receptor 1 (mGluR1) antibody has a predilection to the cerebellar tissue. Anti-mGluR1 encephalitis is a rare autoimmune disease affecting the central and peripheral nervous systems, triggering an acute or subacute cerebellar syndrome with varying degrees of severity. Anti-mGluR1 encephalitis is a rare autoimmune disease affecting the central nervous system. We aimed to systematically review reported cases of anti-mGluR1 encephalitis and summarize their clinical presentation, management, outcomes, and case reports.MethodsA search of the PubMed and Google Scholar databases was conducted and included all cases of anti-mGluR1 encephalitis published in English before October 1, 2022. A comprehensive systematic review was conducted using “metabotropic glutamate receptor type 1,” “mGluR1,” autoantibodies,” “autoantibodies,” “autoimmunity,” and “antibody” as keywords. The risk of bias assessment of the evidence was performed using appropriate tools. The qualitative variables were presented as frequency and percentage.ResultsIncluding our case, 36 cases of anti-mGluR1 encephalitis (19 males, median age 52.5 years, 11.1% pediatric cases) have been reported. The most common clinical manifestations are ataxia, dysarthria, and nystagmus. Initial imaging was normal in 44.4% of patients; however, 75% of patients showed abnormality later in the disease course. The first-line therapy options include glucocorticoids, intravenous immunoglobulin, and plasma exchange. Rituximab is the most commonly used second-line treatment. Complete remission was achieved in only 22.2% of patients, and 61.8% were disabled by the end of their course.ConclusionAnti-mGluR1 encephalitis manifests as symptoms of cerebellar pathology. Although the natural history has not been completely elucidated, early diagnosis with prompt initiation of immunotherapy could be imperative. Any patient suspected to have autoimmune cerebellitis should be tested for the presence of anti-mGluR1 antibody in the serum and cerebrospinal fluid. Escalation to an aggressive therapy approach should be applied in cases that do not respond to first-line therapies, and extended follow-up durations are required in all cases.

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Section: Discussionmentioning

confidence: 85%

Anti-mGluR1 encephalitis: Case illustration and systematic review

2023

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“…This patient concomitantly had Hodgkin lymphoma (Ophelia syndrome) ( 15 ). In addition, in Guadaloupe, a case of anti-mGluR1 encephalitis that presented with acute cerebellar ataxia was associated with a possible trigger by dengue virus infection ( 16 ).…”

Section: Resultsmentioning

confidence: 99%

Autoimmune Encephalitis in Latin America: A Critical Review

et al. 2021

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Autoimmune encephalitis is an increasingly recognized cause of encephalitis. The majority of case series report patients residing in developed countries in the northern hemisphere. The epidemiologic features of autoimmune encephalitis in Latin America are still unclear. The aim of the study was to perform a review of the clinical presentation of autoimmune encephalitis in Latin America and compare to world literature. References were identified by an in-depth literature search and selected on the basis of relevance to the topic and authors' judgment. We selected clinical studies and case reports published from 2007 to July, 2020 including patients from Latin American countries. Of the 379 patients included, the majority were cases of anti-NMDA receptor encephalitis (93.14%), followed by anti-VGKC-complex encephalitis (N = 17; 4.48%), anti-GAD encephalitis (N = 9; 2.37%), anti-AMPA receptor encephalitis (N = 1; 0.26%), anti-GABA receptor encephalitis (N = 1; 0. 26%), anti-mGluR5 encephalitis (N = 1; 0. 26%), and anti-mGluR1 encephalitis (N = 1; 0. 26%). Reported cases of Anti-NMDA encephalitis in Latin-America had a very slight female predominance, lower prevalence of associated tumors and a lower incidence of extreme delta brush on electroencephalogram. Autoimmune encephalitis is possibly underdiagnosed in underdeveloped countries. Its outcome after treatment, however, appears to be similarly favorable in Latin American patients as has been reported in developed countries based on available case reports and case series. Regional specificities in the manifestation of autoimmune encephalitis could be related to epidemiologic factors, such as the presence of different triggers and different genetic and immunologic background, that need to be studied by future research.

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“…Research by Khojah et al ( 4 ) highlighted that 25% of patients experienced one or more prodromal symptoms—ranging from fever, headache, and fatigue to weight loss, nausea, vomiting, night sweats, and flu-like manifestations—with a median interval of 30 days leading up to neurological symptom onset. In a literature review, three patients reported prior infections, including a trigeminal herpes zoster infection a month before onset ( 5 ), a streptococcal pharyngitis 2 months prior ( 6 ), and a dengue virus infection ( 7 ). The link between such infections and anti-mGluR1 encephalitis remains ambiguous.…”

Section: Discussionmentioning

confidence: 99%

Cerebellar encephalitis associated with anti-mGluR1 antibodies: a case report and comprehensive literature review

Chen,

et al. 2024

Front. Neurol.

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Anti-metabotropic glutamate receptor 1 encephalitis is an uncommon autoimmune condition characterized by a subacute onset of cerebellar syndrome. Frequently, it also manifests as sleep disorders and cognitive or behavioral changes. While immunotherapy is the primary treatment approach, the disease remains poorly understood. Herein, we present a case of anti-metabotropic glutamate receptor 1 encephalitis, highlighting its primary cerebellar syndrome manifestation. The first magnetic resonance imaging scan showed no obvious abnormality. Lumbar puncture showed increased cerebrospinal fluid pressure, increased white blood cell count and protein level. The next-generation sequencing of cerebrospinal fluid showed Epstein–Barr virus infection, and the patient was diagnosed with viral cerebellar encephalitis. However, antiviral therapy was ineffective. Finally, anti-metabotropic glutamate receptor 1 was measured at 1:1,000, and the patient was definitely diagnosed with anti-metabotropic glutamate receptor 1 encephalitis. Therefore, clinicians should pay attention to such diseases to avoid misdiagnosis.

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Successful Management of Anti‐mGluR1 Encephalitis with Immunosuppressive Treatment: Dengue Virus as a Trigger?

Abstract: View Supplementary Video 1

Cited by 4 publications

References 3 publications

Anti-mGluR1 encephalitis: Case illustration and systematic review

Anti-mGluR1 encephalitis: Case illustration and systematic review

Autoimmune Encephalitis in Latin America: A Critical Review

Cerebellar encephalitis associated with anti-mGluR1 antibodies: a case report and comprehensive literature review

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