Precision Assessment of Anti-NMDA Receptor Encephalitis: A Case Report on Integrating Clinical Course, Immunophenotyping, and Comprehensive Symptomatology in a Pediatric Patient With Adjunctive Hydrogen Therapy

Shang-Chiang Lin; Jeng-Wei Lu; Ting-Chun Lin; Yueh-Feng Sung; Yi-Jung Ho; Fu-Min Wang; Shan-Wen Lui; Ting-Yu Hsieh; Kuang-Yih Wang; Feng-Cheng Liu

doi:10.21873/invivo.13858

Precision Assessment of Anti-NMDA Receptor Encephalitis: A Case Report on Integrating Clinical Course, Immunophenotyping, and Comprehensive Symptomatology in a Pediatric Patient With Adjunctive Hydrogen Therapy

In Vivo. 2025 Jan-Feb;39(1):539-547. doi: 10.21873/invivo.13858.

Authors

Shang-Chiang Lin¹, Jeng-Wei Lu^{2

3}, Ting-Chun Lin⁴, Yueh-Feng Sung⁵, Yi-Jung Ho^{4

6}, Fu-Min Wang⁷, Shan-Wen Lui⁸, Ting-Yu Hsieh⁸, Kuang-Yih Wang⁹, Feng-Cheng Liu¹⁰

Affiliations

¹ Department of Internal Medicine, Tri-Service General Hospital, National Defense Medical Center, Taipei, Taiwan, R.O.C.
² Biotech Research and Innovation Centre, University of Copenhagen, Copenhagen, Denmark.
³ The Finsen Laboratory, Rigshospitalet/National University Hospital, Faculty of Health and Medical Sciences, University of Copenhagen, Copenhagen, Denmark.
⁴ Graduate Institute of Life Science, National Defense Medical Center, Taipei, Taiwan, R.O.C.
⁵ Department of Neurology, Tri-Service General Hospital, National Defense Medical Center, Taipei, Taiwan, R.O.C.
⁶ School of Pharmacy, National Defense Medical Center, Taipei, Taiwan, R.O.C.
⁷ Department of Pediatric Hematology and Oncology, Tri-Service General Hospital, National Defense Medical Center, Taipei, Taiwan, R.O.C.
⁸ School of Medicine, National Defense Medical Center, Taipei, Taiwan, R.O.C.
⁹ Rheumatology/Immunology and Allergy, Department of Internal Medicine, Tri-Service General Hospital, National Defense Medical Center, Taipei, Taiwan, R.O.C.
¹⁰ Rheumatology/Immunology and Allergy, Department of Internal Medicine, Tri-Service General Hospital, National Defense Medical Center, Taipei, Taiwan, R.O.C.; [email protected].

Abstract

Background/aim: Anti-N-methyl-D-aspartate receptor (NMDAR) encephalitis, though rare, is the most common form of autoimmune encephalitis, predominantly affecting young individuals, particularly females. Standard treatments include corticosteroids, intravenous immunoglobulins (IVIG), and plasmapheresis, with rituximab recommended for those unresponsive to first-line therapies. However, reliable biomarkers for clinical assessment remain elusive. This study investigated the efficacy of adjunctive hydrogen therapy in a patient with anti-NMDAR encephalitis.

Case report: This case report describes a 14-year-old boy with anti-NMDAR encephalitis who exhibited poor response to initial treatment, but showed significant improvement with rituximab and adjunctive hydrogen therapy. Immunophenotyping revealed correlations between treatment outcomes and shifts in B cell subsets, PD-1+ cytotoxic T cells, and regulatory T cell subtypes.

Conclusion: This case underscores the importance of integration traditional clinical assessments with advanced diagnostics such as flow cytometry-based immunophenotyping, and suggests a potential role for hydrogen therapy in modulating immune response in this complex autoimmune condition.

Keywords: B cells; Case report; PD-1+ cytotoxic T cells; anti-NMDA receptor encephalitis; hydrogen therapy; regulatory T cells.

Publication types

Case Reports

MeSH terms

Adolescent
Anti-N-Methyl-D-Aspartate Receptor Encephalitis* / diagnosis
Anti-N-Methyl-D-Aspartate Receptor Encephalitis* / drug therapy
Anti-N-Methyl-D-Aspartate Receptor Encephalitis* / therapy
Humans
Hydrogen*
Immunophenotyping*
Male
Rituximab / therapeutic use
Treatment Outcome

Substances

Hydrogen
Rituximab