PMID- 36389693
OWN - NLM
STAT- MEDLINE
DCOM- 20221121
LR  - 20221121
IS  - 1664-3224 (Electronic)
IS  - 1664-3224 (Linking)
VI  - 13
DP  - 2022
TI  - Anti-NMDAR encephalitis in Crohn's disease undergoing long-term infliximab 
      treatment: A case report.
PG  - 957575
LID - 10.3389/fimmu.2022.957575 [doi]
LID - 957575
AB  - Infliximab, a chimeric monoclonal antibody against anti-tumor necrosis factor-alpha 
      (TNF-alpha), has revolutionized the management of inflammatory bowel disease. 
      However, a recent nested case-control study showed that anti-TNF-alpha 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-alpha 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-alpha. Our case highlights the need for clinicians to recognize 
      the possibility of a paradoxical autoimmune response occurring with novel 
      biological therapies.
CI  - Copyright (c) 2022 Oh, Kwon, Lee and Lee.
FAU - Oh, Shin Ju
AU  - Oh SJ
AD  - Center for Crohn's and Colitis, Department of Gastroenterology, Kyung Hee 
      University Hospital, Kyung Hee University College of Medicine, Seoul, South 
      Korea.
FAU - Kwon, Young Nam
AU  - Kwon YN
AD  - Department of Neurology, Seoul National University Hospital, Seoul National 
      University College of Medicine, Seoul, South Korea.
FAU - Lee, Chang Kyun
AU  - Lee CK
AD  - Center for Crohn's and Colitis, Department of Gastroenterology, Kyung Hee 
      University Hospital, Kyung Hee University College of Medicine, Seoul, South 
      Korea.
FAU - Lee, Jin San
AU  - Lee JS
AD  - Department of Neurology, Kyung Hee University Hospital, Kyung Hee University 
      College of Medicine, Seoul, South Korea.
LA  - eng
PT  - Case Reports
PT  - Research Support, Non-U.S. Gov't
DEP - 20221031
PL  - Switzerland
TA  - Front Immunol
JT  - Frontiers in immunology
JID - 101560960
RN  - B72HH48FLU (Infliximab)
RN  - 0 (Tumor Necrosis Factor Inhibitors)
RN  - 0 (Tumor Necrosis Factor-alpha)
SB  - IM
MH  - Male
MH  - Humans
MH  - Infant, Newborn
MH  - Adult
MH  - *Anti-N-Methyl-D-Aspartate Receptor Encephalitis/diagnosis
MH  - Infliximab/adverse effects
MH  - *Crohn Disease/complications/drug therapy
MH  - Case-Control Studies
MH  - Tumor Necrosis Factor Inhibitors
MH  - Tumor Necrosis Factor-alpha/therapeutic use
PMC - PMC9659735
OTO - NOTNLM
OT  - Crohn 's disease
OT  - anti-N-methyl-D-aspartate
OT  - anti-tumor necrosis factor-alpha
OT  - autoimmune encephalitis
OT  - infliximab
COIS- The authors declare that the research was conducted in the absence of any 
      commercial or financial relationships that could be construed as a potential 
      conflict of interest.
EDAT- 2022/11/18 06:00
MHDA- 2022/11/22 06:00
PMCR- 2022/01/01
CRDT- 2022/11/17 12:43
PHST- 2022/05/31 00:00 [received]
PHST- 2022/10/19 00:00 [accepted]
PHST- 2022/11/17 12:43 [entrez]
PHST- 2022/11/18 06:00 [pubmed]
PHST- 2022/11/22 06:00 [medline]
PHST- 2022/01/01 00:00 [pmc-release]
AID - 10.3389/fimmu.2022.957575 [doi]
PST - epublish
SO  - Front Immunol. 2022 Oct 31;13:957575. doi: 10.3389/fimmu.2022.957575. eCollection 
      2022.