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Successful Treatment of Carbamazepine-Induced Toxic Epidermal Necrolysis With Clinical Gastrointestinal Involvement: A Case Report

BACKGROUND: Stevens-Johnson syndrome/toxic epidermal necrolysis (SJS/TEN) is a rare and life-threatening disease of the skin and mucosal surfaces. Although gastrointestinal manifestations in adults are potential prognostic factors for disease severity, there are limited data on such cases and their...

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Autores principales: Chi, Le Quynh, Anh, Nguyen Thi Van, Le, Nguyen Ngoc Quynh, Ha, Nguyen Thi Thu, Tien, Hoang Minh, Dien, Tran Minh
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Frontiers Media S.A. 2022
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9043682/
https://www.ncbi.nlm.nih.gov/pubmed/35498784
http://dx.doi.org/10.3389/fped.2022.834037
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author Chi, Le Quynh
Anh, Nguyen Thi Van
Le, Nguyen Ngoc Quynh
Ha, Nguyen Thi Thu
Tien, Hoang Minh
Dien, Tran Minh
author_facet Chi, Le Quynh
Anh, Nguyen Thi Van
Le, Nguyen Ngoc Quynh
Ha, Nguyen Thi Thu
Tien, Hoang Minh
Dien, Tran Minh
author_sort Chi, Le Quynh
collection PubMed
description BACKGROUND: Stevens-Johnson syndrome/toxic epidermal necrolysis (SJS/TEN) is a rare and life-threatening disease of the skin and mucosal surfaces. Although gastrointestinal manifestations in adults are potential prognostic factors for disease severity, there are limited data on such cases and their standard management in the pediatric population. CASE PRESENTATION: We herein report the case of an 8-year-old girl with a 1-year history of epilepsy, who presented with bilateral conjunctivitis and progressively widespread bullous, and pruritic eruption based on erythematous skin after administration of carbamazepine. A diagnosis of carbamazepine-induced TEN was made, and the drug was immediately discontinued. The result of genetic screening showed that the patient was positive for the HLA-B(*)15:02 allele. Then, her condition got worse by developing gastrointestinal involvement, including hematemesis and severe watery bloody diarrhea. A combination of the intravenous immunoglobulin and the appropriate dose of systemic steroids have contributed to a favorable outcome in this case. Multidisciplinary care of mucocutaneous involvement, supplemental nutrition, and fluid replacement was also critically warranted. This report aims to contribute to the current literature on TEN-related gastrointestinal manifestations in pediatrics and highlights the need for further investigations in determining the optimal treatment in such cases. CONCLUSION: In conclusion, we reported the successful treatment of TEN-related gastrointestinal manifestations in a pediatric patient, which should be critically considered in patients with SJS/TEN. Since it may significantly contribute to the poor prognosis of the illness, further investigations in determining standard management in such cases are necessary.
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spelling pubmed-90436822022-04-28 Successful Treatment of Carbamazepine-Induced Toxic Epidermal Necrolysis With Clinical Gastrointestinal Involvement: A Case Report Chi, Le Quynh Anh, Nguyen Thi Van Le, Nguyen Ngoc Quynh Ha, Nguyen Thi Thu Tien, Hoang Minh Dien, Tran Minh Front Pediatr Pediatrics BACKGROUND: Stevens-Johnson syndrome/toxic epidermal necrolysis (SJS/TEN) is a rare and life-threatening disease of the skin and mucosal surfaces. Although gastrointestinal manifestations in adults are potential prognostic factors for disease severity, there are limited data on such cases and their standard management in the pediatric population. CASE PRESENTATION: We herein report the case of an 8-year-old girl with a 1-year history of epilepsy, who presented with bilateral conjunctivitis and progressively widespread bullous, and pruritic eruption based on erythematous skin after administration of carbamazepine. A diagnosis of carbamazepine-induced TEN was made, and the drug was immediately discontinued. The result of genetic screening showed that the patient was positive for the HLA-B(*)15:02 allele. Then, her condition got worse by developing gastrointestinal involvement, including hematemesis and severe watery bloody diarrhea. A combination of the intravenous immunoglobulin and the appropriate dose of systemic steroids have contributed to a favorable outcome in this case. Multidisciplinary care of mucocutaneous involvement, supplemental nutrition, and fluid replacement was also critically warranted. This report aims to contribute to the current literature on TEN-related gastrointestinal manifestations in pediatrics and highlights the need for further investigations in determining the optimal treatment in such cases. CONCLUSION: In conclusion, we reported the successful treatment of TEN-related gastrointestinal manifestations in a pediatric patient, which should be critically considered in patients with SJS/TEN. Since it may significantly contribute to the poor prognosis of the illness, further investigations in determining standard management in such cases are necessary. Frontiers Media S.A. 2022-04-13 /pmc/articles/PMC9043682/ /pubmed/35498784 http://dx.doi.org/10.3389/fped.2022.834037 Text en Copyright © 2022 Chi, Anh, Le, Ha, Tien and Dien. 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 Pediatrics
Chi, Le Quynh
Anh, Nguyen Thi Van
Le, Nguyen Ngoc Quynh
Ha, Nguyen Thi Thu
Tien, Hoang Minh
Dien, Tran Minh
Successful Treatment of Carbamazepine-Induced Toxic Epidermal Necrolysis With Clinical Gastrointestinal Involvement: A Case Report
title Successful Treatment of Carbamazepine-Induced Toxic Epidermal Necrolysis With Clinical Gastrointestinal Involvement: A Case Report
title_full Successful Treatment of Carbamazepine-Induced Toxic Epidermal Necrolysis With Clinical Gastrointestinal Involvement: A Case Report
title_fullStr Successful Treatment of Carbamazepine-Induced Toxic Epidermal Necrolysis With Clinical Gastrointestinal Involvement: A Case Report
title_full_unstemmed Successful Treatment of Carbamazepine-Induced Toxic Epidermal Necrolysis With Clinical Gastrointestinal Involvement: A Case Report
title_short Successful Treatment of Carbamazepine-Induced Toxic Epidermal Necrolysis With Clinical Gastrointestinal Involvement: A Case Report
title_sort successful treatment of carbamazepine-induced toxic epidermal necrolysis with clinical gastrointestinal involvement: a case report
topic Pediatrics
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9043682/
https://www.ncbi.nlm.nih.gov/pubmed/35498784
http://dx.doi.org/10.3389/fped.2022.834037
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