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Idiopathic Harlequin Syndrome in a Patient from Ethiopia: A Case Report

BACKGROUND: Harlequin syndrome is a rare disorder caused by autonomic nervous system dysfunction. It manifests as asymmetric facial flashing and sweating with contralateral anhidrosis. It may be primary (idiopathic) with a benign course or can occur secondary to structural abnormalities or iatrogeni...

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Autores principales: Melka, Dereje, Zebenigus, Mehila
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Research and Publications Office of Jimma University 2023
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10358395/
https://www.ncbi.nlm.nih.gov/pubmed/37484168
http://dx.doi.org/10.4314/ejhs.v33i2.24
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author Melka, Dereje
Zebenigus, Mehila
author_facet Melka, Dereje
Zebenigus, Mehila
author_sort Melka, Dereje
collection PubMed
description BACKGROUND: Harlequin syndrome is a rare disorder caused by autonomic nervous system dysfunction. It manifests as asymmetric facial flashing and sweating with contralateral anhidrosis. It may be primary (idiopathic) with a benign course or can occur secondary to structural abnormalities or iatrogenic factors. To our knowledge, there has been no report of idiopathic Harlequin syndrome published from Ethiopia. We are reporting this case since it signifies the existence of idiopathic Harlequin syndrome in our setting and the need to properly diagnose this condition. CASE PRESENTATION: We are reporting a 29-year-old female from Addis Ababa, Ethiopia, who presented with a complaint of left hemifacial hyperhidrosis of 8 years which worsen after routine household activities and exercise. Physical examination revealed left hemifacial hyperhidrosis with right-side anhidrosis. She was diagnosed with idiopathic Harlequin syndrome after an appropriate investigation revealed a nonremarkable finding. Symptomatic treatment showed no significant improvement and the patient was also counseled on the disease entity. CONCLUSIONS: The patient described here signifies an idiopathic Harlequin syndrome in an Ethiopian woman. This case highlights the existence of idiopathic Harlequin syndrome within our setting and the need to properly diagnose this condition.
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spelling pubmed-103583952023-07-21 Idiopathic Harlequin Syndrome in a Patient from Ethiopia: A Case Report Melka, Dereje Zebenigus, Mehila Ethiop J Health Sci Case Report BACKGROUND: Harlequin syndrome is a rare disorder caused by autonomic nervous system dysfunction. It manifests as asymmetric facial flashing and sweating with contralateral anhidrosis. It may be primary (idiopathic) with a benign course or can occur secondary to structural abnormalities or iatrogenic factors. To our knowledge, there has been no report of idiopathic Harlequin syndrome published from Ethiopia. We are reporting this case since it signifies the existence of idiopathic Harlequin syndrome in our setting and the need to properly diagnose this condition. CASE PRESENTATION: We are reporting a 29-year-old female from Addis Ababa, Ethiopia, who presented with a complaint of left hemifacial hyperhidrosis of 8 years which worsen after routine household activities and exercise. Physical examination revealed left hemifacial hyperhidrosis with right-side anhidrosis. She was diagnosed with idiopathic Harlequin syndrome after an appropriate investigation revealed a nonremarkable finding. Symptomatic treatment showed no significant improvement and the patient was also counseled on the disease entity. CONCLUSIONS: The patient described here signifies an idiopathic Harlequin syndrome in an Ethiopian woman. This case highlights the existence of idiopathic Harlequin syndrome within our setting and the need to properly diagnose this condition. Research and Publications Office of Jimma University 2023-03 /pmc/articles/PMC10358395/ /pubmed/37484168 http://dx.doi.org/10.4314/ejhs.v33i2.24 Text en © 2023 Dereje Melka, et al. https://creativecommons.org/licenses/by/4.0/This is an open access article distributed under the terms of the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original author and source are credited.
spellingShingle Case Report
Melka, Dereje
Zebenigus, Mehila
Idiopathic Harlequin Syndrome in a Patient from Ethiopia: A Case Report
title Idiopathic Harlequin Syndrome in a Patient from Ethiopia: A Case Report
title_full Idiopathic Harlequin Syndrome in a Patient from Ethiopia: A Case Report
title_fullStr Idiopathic Harlequin Syndrome in a Patient from Ethiopia: A Case Report
title_full_unstemmed Idiopathic Harlequin Syndrome in a Patient from Ethiopia: A Case Report
title_short Idiopathic Harlequin Syndrome in a Patient from Ethiopia: A Case Report
title_sort idiopathic harlequin syndrome in a patient from ethiopia: a case report
topic Case Report
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10358395/
https://www.ncbi.nlm.nih.gov/pubmed/37484168
http://dx.doi.org/10.4314/ejhs.v33i2.24
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