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Sjogren’s syndrome related autoimmune encephalopathy presenting as depression and involuntary agitation in a 15-year-old girl: a case report

INTRODUCTION: Sjogren’s syndrome is an autoimmune disease that commonly involves exocrinopathy. Although studies have reported psychiatric manifestations resulting from Sjogren’s syndrome, few studies have focused on such manifestations in pediatric patients. Herein, we present a case of an adolesce...

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Autores principales: Lee, Yu-Tung, Wu, Chao-Yi, Chou, I-Jun, Liu, Chun-Hao
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Frontiers Media S.A. 2023
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10345200/
https://www.ncbi.nlm.nih.gov/pubmed/37457704
http://dx.doi.org/10.3389/fimmu.2023.1145948
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author Lee, Yu-Tung
Wu, Chao-Yi
Chou, I-Jun
Liu, Chun-Hao
author_facet Lee, Yu-Tung
Wu, Chao-Yi
Chou, I-Jun
Liu, Chun-Hao
author_sort Lee, Yu-Tung
collection PubMed
description INTRODUCTION: Sjogren’s syndrome is an autoimmune disease that commonly involves exocrinopathy. Although studies have reported psychiatric manifestations resulting from Sjogren’s syndrome, few studies have focused on such manifestations in pediatric patients. Herein, we present a case of an adolescent girl with depression and involuntary self-harm behaviors related to Sjogren’s syndrome with central nervous system involvement. CASE PRESENTATION: A 15-year-old girl, with an underlying history of epilepsy, developed depressive symptoms of a year’s duration, accompanied by three seizure episodes and involuntary self-harm behaviors. The self-harm behaviors, which included head banging and arm scratching, were sudden onset, involuntary, and unable to be recalled afterwards. After admission to our ward, the patient was positive for serum antinuclear antibodies and Schirmer’s test. Moreover, 24-hour electroencephalography revealed epileptiform discharges during the mood swing episodes. Positive findings for antinuclear antibodies and anti-SSA antibodies in both serum and cerebrospinal fluid, suggested central nervous system involvement in Sjogren’s syndrome. After rituximab treatment, her mood became euthymic, and her involuntary self-harm behaviors ceased. CONCLUSION: Central nervous system involvement leading to psychiatric presentations has rarely been reported in adolescents with Sjogren’s syndrome. When treating adolescent patients with involuntary self-harm behaviors and neurological symptoms, it is crucial to consider autoimmune encephalitis related to Sjogren’s syndrome in the differential diagnosis.
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spelling pubmed-103452002023-07-15 Sjogren’s syndrome related autoimmune encephalopathy presenting as depression and involuntary agitation in a 15-year-old girl: a case report Lee, Yu-Tung Wu, Chao-Yi Chou, I-Jun Liu, Chun-Hao Front Immunol Immunology INTRODUCTION: Sjogren’s syndrome is an autoimmune disease that commonly involves exocrinopathy. Although studies have reported psychiatric manifestations resulting from Sjogren’s syndrome, few studies have focused on such manifestations in pediatric patients. Herein, we present a case of an adolescent girl with depression and involuntary self-harm behaviors related to Sjogren’s syndrome with central nervous system involvement. CASE PRESENTATION: A 15-year-old girl, with an underlying history of epilepsy, developed depressive symptoms of a year’s duration, accompanied by three seizure episodes and involuntary self-harm behaviors. The self-harm behaviors, which included head banging and arm scratching, were sudden onset, involuntary, and unable to be recalled afterwards. After admission to our ward, the patient was positive for serum antinuclear antibodies and Schirmer’s test. Moreover, 24-hour electroencephalography revealed epileptiform discharges during the mood swing episodes. Positive findings for antinuclear antibodies and anti-SSA antibodies in both serum and cerebrospinal fluid, suggested central nervous system involvement in Sjogren’s syndrome. After rituximab treatment, her mood became euthymic, and her involuntary self-harm behaviors ceased. CONCLUSION: Central nervous system involvement leading to psychiatric presentations has rarely been reported in adolescents with Sjogren’s syndrome. When treating adolescent patients with involuntary self-harm behaviors and neurological symptoms, it is crucial to consider autoimmune encephalitis related to Sjogren’s syndrome in the differential diagnosis. Frontiers Media S.A. 2023-06-29 /pmc/articles/PMC10345200/ /pubmed/37457704 http://dx.doi.org/10.3389/fimmu.2023.1145948 Text en Copyright © 2023 Lee, Wu, Chou and Liu 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
Lee, Yu-Tung
Wu, Chao-Yi
Chou, I-Jun
Liu, Chun-Hao
Sjogren’s syndrome related autoimmune encephalopathy presenting as depression and involuntary agitation in a 15-year-old girl: a case report
title Sjogren’s syndrome related autoimmune encephalopathy presenting as depression and involuntary agitation in a 15-year-old girl: a case report
title_full Sjogren’s syndrome related autoimmune encephalopathy presenting as depression and involuntary agitation in a 15-year-old girl: a case report
title_fullStr Sjogren’s syndrome related autoimmune encephalopathy presenting as depression and involuntary agitation in a 15-year-old girl: a case report
title_full_unstemmed Sjogren’s syndrome related autoimmune encephalopathy presenting as depression and involuntary agitation in a 15-year-old girl: a case report
title_short Sjogren’s syndrome related autoimmune encephalopathy presenting as depression and involuntary agitation in a 15-year-old girl: a case report
title_sort sjogren’s syndrome related autoimmune encephalopathy presenting as depression and involuntary agitation in a 15-year-old girl: a case report
topic Immunology
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10345200/
https://www.ncbi.nlm.nih.gov/pubmed/37457704
http://dx.doi.org/10.3389/fimmu.2023.1145948
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