Cargando…

Anti-NMDAR encephalitis in Crohn’s disease undergoing long-term infliximab treatment: A case report

Infliximab, a chimeric monoclonal antibody against anti-tumor necrosis factor-α (TNF-α), has revolutionized the management of inflammatory bowel disease. However, a recent nested case-control study showed that anti-TNF-α therapy exposure in patients with autoimmune diseases is associated with an inc...

Descripción completa

Detalles Bibliográficos
Autores principales: Oh, Shin Ju, Kwon, Young Nam, Lee, Chang Kyun, Lee, Jin San
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Frontiers Media S.A. 2022
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9659735/
https://www.ncbi.nlm.nih.gov/pubmed/36389693
http://dx.doi.org/10.3389/fimmu.2022.957575
_version_ 1784830262725574656
author Oh, Shin Ju
Kwon, Young Nam
Lee, Chang Kyun
Lee, Jin San
author_facet Oh, Shin Ju
Kwon, Young Nam
Lee, Chang Kyun
Lee, Jin San
author_sort Oh, Shin Ju
collection PubMed
description Infliximab, a chimeric monoclonal antibody against anti-tumor necrosis factor-α (TNF-α), has revolutionized the management of inflammatory bowel disease. However, a recent nested case-control study showed that anti-TNF-α therapy exposure in patients with autoimmune diseases is associated with an increased risk of inflammatory central nervous system (CNS) events. A 27-year-old man diagnosed with Crohn’s disease at 17 years of age was referred to our clinic for suffering with Wernicke’s aphasia and the right-hand weakness over two weeks. Nine years of treatment for Crohn’s disease with infliximab anti-TNF-α therapy was well tolerated. An initial MRI revealed diffuse leptomeningeal enhancement along the bilateral cerebral sulci without any parenchymal abnormalities. Cerebrospinal fluid (CSF) and serum N-methyl-D-aspartate receptor (NMDAR) antibody testing yielded positive results. Anti-NMDAR encephalitis was diagnosed, and the patient was treated with rituximab. A follow-up brain MRI showed new multiple cerebral lesions in the left insular cortex and subcortical white matter of the left frontal and temporal gyri. Approximately 8 months after symptom onset, the CSF and serum NMDAR antibody converted to negative. Twelve months later, the patient fully recovered from anti-NMDAR encephalitis without any neurological deficits and is currently being treated with the anti-interleukin 12/23 agent ustekinumab for Crohn’s disease. This is the first report of not only a patient with infliximab-associated anti-NMDAR encephalitis in Crohn’s disease but also of an inflammatory non-demyelinating CNS event during long-term suppression of TNF-α. Our case highlights the need for clinicians to recognize the possibility of a paradoxical autoimmune response occurring with novel biological therapies.
format Online
Article
Text
id pubmed-9659735
institution National Center for Biotechnology Information
language English
publishDate 2022
publisher Frontiers Media S.A.
record_format MEDLINE/PubMed
spelling pubmed-96597352022-11-15 Anti-NMDAR encephalitis in Crohn’s disease undergoing long-term infliximab treatment: A case report Oh, Shin Ju Kwon, Young Nam Lee, Chang Kyun Lee, Jin San Front Immunol Immunology Infliximab, a chimeric monoclonal antibody against anti-tumor necrosis factor-α (TNF-α), has revolutionized the management of inflammatory bowel disease. However, a recent nested case-control study showed that anti-TNF-α therapy exposure in patients with autoimmune diseases is associated with an increased risk of inflammatory central nervous system (CNS) events. A 27-year-old man diagnosed with Crohn’s disease at 17 years of age was referred to our clinic for suffering with Wernicke’s aphasia and the right-hand weakness over two weeks. Nine years of treatment for Crohn’s disease with infliximab anti-TNF-α therapy was well tolerated. An initial MRI revealed diffuse leptomeningeal enhancement along the bilateral cerebral sulci without any parenchymal abnormalities. Cerebrospinal fluid (CSF) and serum N-methyl-D-aspartate receptor (NMDAR) antibody testing yielded positive results. Anti-NMDAR encephalitis was diagnosed, and the patient was treated with rituximab. A follow-up brain MRI showed new multiple cerebral lesions in the left insular cortex and subcortical white matter of the left frontal and temporal gyri. Approximately 8 months after symptom onset, the CSF and serum NMDAR antibody converted to negative. Twelve months later, the patient fully recovered from anti-NMDAR encephalitis without any neurological deficits and is currently being treated with the anti-interleukin 12/23 agent ustekinumab for Crohn’s disease. This is the first report of not only a patient with infliximab-associated anti-NMDAR encephalitis in Crohn’s disease but also of an inflammatory non-demyelinating CNS event during long-term suppression of TNF-α. Our case highlights the need for clinicians to recognize the possibility of a paradoxical autoimmune response occurring with novel biological therapies. Frontiers Media S.A. 2022-10-31 /pmc/articles/PMC9659735/ /pubmed/36389693 http://dx.doi.org/10.3389/fimmu.2022.957575 Text en Copyright © 2022 Oh, Kwon, Lee and Lee https://creativecommons.org/licenses/by/4.0/This is an open-access article distributed under the terms of the Creative Commons Attribution License (CC BY). The use, distribution or reproduction in other forums is permitted, provided the original author(s) and the copyright owner(s) are credited and that the original publication in this journal is cited, in accordance with accepted academic practice. No use, distribution or reproduction is permitted which does not comply with these terms.
spellingShingle Immunology
Oh, Shin Ju
Kwon, Young Nam
Lee, Chang Kyun
Lee, Jin San
Anti-NMDAR encephalitis in Crohn’s disease undergoing long-term infliximab treatment: A case report
title Anti-NMDAR encephalitis in Crohn’s disease undergoing long-term infliximab treatment: A case report
title_full Anti-NMDAR encephalitis in Crohn’s disease undergoing long-term infliximab treatment: A case report
title_fullStr Anti-NMDAR encephalitis in Crohn’s disease undergoing long-term infliximab treatment: A case report
title_full_unstemmed Anti-NMDAR encephalitis in Crohn’s disease undergoing long-term infliximab treatment: A case report
title_short Anti-NMDAR encephalitis in Crohn’s disease undergoing long-term infliximab treatment: A case report
title_sort anti-nmdar encephalitis in crohn’s disease undergoing long-term infliximab treatment: a case report
topic Immunology
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9659735/
https://www.ncbi.nlm.nih.gov/pubmed/36389693
http://dx.doi.org/10.3389/fimmu.2022.957575
work_keys_str_mv AT ohshinju antinmdarencephalitisincrohnsdiseaseundergoinglongterminfliximabtreatmentacasereport
AT kwonyoungnam antinmdarencephalitisincrohnsdiseaseundergoinglongterminfliximabtreatmentacasereport
AT leechangkyun antinmdarencephalitisincrohnsdiseaseundergoinglongterminfliximabtreatmentacasereport
AT leejinsan antinmdarencephalitisincrohnsdiseaseundergoinglongterminfliximabtreatmentacasereport