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Adrenocorticotropic Hormone Induced Status Dystonicus in a Child with West Syndrome

Dystonia is a movement disorder characterized by involuntary sustained or intermittent muscle contraction causing repetitive twisting movements and abnormal postures. Status dystonicus (SD) is an enigmatic disease of cryptic etiology. We hereby report a child with West syndrome (WS) who went on to d...

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Autores principales: Singh, Jasmine, Aulakh, Roosy
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Jaypee Brothers Medical Publishers 2022
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9363804/
https://www.ncbi.nlm.nih.gov/pubmed/36042765
http://dx.doi.org/10.5005/jp-journals-10071-24289
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author Singh, Jasmine
Aulakh, Roosy
author_facet Singh, Jasmine
Aulakh, Roosy
author_sort Singh, Jasmine
collection PubMed
description Dystonia is a movement disorder characterized by involuntary sustained or intermittent muscle contraction causing repetitive twisting movements and abnormal postures. Status dystonicus (SD) is an enigmatic disease of cryptic etiology. We hereby report a child with West syndrome (WS) who went on to develop SD following intramuscular adrenocorticotropic hormone (ACTH) injection. An 11-month-old male child presented with complaints of flexor spasms for 2 months. The diagnosis of WS was confirmed by electroencephalography (EEG), which showed hypsarrhythmia. Intramuscular ACTH was added, and oral trihexyphenidyl was started for dystonia. On day 7 of ACTH, the child developed frequent opisthotonic posturing. Management protocol for grade IV SD was initiated. Administration of N-terminal of ACTH in rat locus coeruleus has been shown to produce human dystonia-like movement and abnormal posturing. HOW TO CITE THIS ARTICLE: Singh J, Aulakh R. Adrenocorticotropic Hormone Induced Status Dystonicus in a Child with West Syndrome. Indian J Crit Care Med 2022;26(8):961–962.
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spelling pubmed-93638042022-08-29 Adrenocorticotropic Hormone Induced Status Dystonicus in a Child with West Syndrome Singh, Jasmine Aulakh, Roosy Indian J Crit Care Med Case Report Dystonia is a movement disorder characterized by involuntary sustained or intermittent muscle contraction causing repetitive twisting movements and abnormal postures. Status dystonicus (SD) is an enigmatic disease of cryptic etiology. We hereby report a child with West syndrome (WS) who went on to develop SD following intramuscular adrenocorticotropic hormone (ACTH) injection. An 11-month-old male child presented with complaints of flexor spasms for 2 months. The diagnosis of WS was confirmed by electroencephalography (EEG), which showed hypsarrhythmia. Intramuscular ACTH was added, and oral trihexyphenidyl was started for dystonia. On day 7 of ACTH, the child developed frequent opisthotonic posturing. Management protocol for grade IV SD was initiated. Administration of N-terminal of ACTH in rat locus coeruleus has been shown to produce human dystonia-like movement and abnormal posturing. HOW TO CITE THIS ARTICLE: Singh J, Aulakh R. Adrenocorticotropic Hormone Induced Status Dystonicus in a Child with West Syndrome. Indian J Crit Care Med 2022;26(8):961–962. Jaypee Brothers Medical Publishers 2022-08 /pmc/articles/PMC9363804/ /pubmed/36042765 http://dx.doi.org/10.5005/jp-journals-10071-24289 Text en Copyright © 2022; Jaypee Brothers Medical Publishers (P) Ltd. https://creativecommons.org/licenses/by-nc/4.0/© The Author(s). 2022 Open Access This article is distributed under the terms of the Creative Commons Attribution 4.0 International License (https://creativecommons.org/licenses/by-nc/4.0/), which permits unrestricted use, distribution, and non-commercial reproduction in any medium, provided you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons license, and indicate if changes were made. The Creative Commons Public Domain Dedication waiver (http://creativecommons.org/publicdomain/zero/1.0/ (https://creativecommons.org/publicdomain/zero/1.0/) ) applies to the data made available in this article, unless otherwise stated.
spellingShingle Case Report
Singh, Jasmine
Aulakh, Roosy
Adrenocorticotropic Hormone Induced Status Dystonicus in a Child with West Syndrome
title Adrenocorticotropic Hormone Induced Status Dystonicus in a Child with West Syndrome
title_full Adrenocorticotropic Hormone Induced Status Dystonicus in a Child with West Syndrome
title_fullStr Adrenocorticotropic Hormone Induced Status Dystonicus in a Child with West Syndrome
title_full_unstemmed Adrenocorticotropic Hormone Induced Status Dystonicus in a Child with West Syndrome
title_short Adrenocorticotropic Hormone Induced Status Dystonicus in a Child with West Syndrome
title_sort adrenocorticotropic hormone induced status dystonicus in a child with west syndrome
topic Case Report
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9363804/
https://www.ncbi.nlm.nih.gov/pubmed/36042765
http://dx.doi.org/10.5005/jp-journals-10071-24289
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