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...
Autores principales: | , , , |
---|---|
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 |