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特发性丑角面容综合征患者 1 例:病例报告。

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

机构信息

Department of Neurology, Addis Ababa University School of Medicine, Addis Ababa, Ethiopia.

Yehulshet Neurology specialty Clinic, Addis Ababa, Ethiopia.

出版信息

Ethiop J Health Sci. 2023 Mar;33(2):383-386. doi: 10.4314/ejhs.v33i2.24.

DOI:10.4314/ejhs.v33i2.24
PMID:37484168
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC10358395/
Abstract

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.

摘要

背景

哈姆林格综合征是一种罕见的自主神经系统功能障碍引起的疾病。其表现为不对称的面部阵发性潮红和出汗,同时对侧无汗。它可能是原发性(特发性)的,具有良性过程,也可能继发于结构异常或医源性因素。据我们所知,目前还没有来自埃塞俄比亚的特发性哈姆林格综合征的报告。我们报告这个病例是因为它表明在我们的环境中存在特发性哈姆林格综合征,需要正确诊断这种疾病。

病例介绍

我们报告了一名 29 岁的埃塞俄比亚亚的斯亚贝巴女性,她因 8 年前出现左侧半脸多汗,且在日常家务和运动后加重而就诊。体格检查显示左侧半脸多汗,右侧无汗。适当的检查发现无明显异常,诊断为特发性哈姆林格综合征。对症治疗没有明显改善,也对患者进行了疾病实体的咨询。

结论

这里描述的患者是一名埃塞俄比亚女性的特发性哈姆林格综合征。这个病例突出了特发性哈姆林格综合征在我们环境中的存在,需要正确诊断这种疾病。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/a670/10358395/85f005e31115/EJHS3302-0383Fig2.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/a670/10358395/3691f5ed3a0f/EJHS3302-0383Fig1.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/a670/10358395/85f005e31115/EJHS3302-0383Fig2.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/a670/10358395/3691f5ed3a0f/EJHS3302-0383Fig1.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/a670/10358395/85f005e31115/EJHS3302-0383Fig2.jpg

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Australas J Dermatol. 2021 Nov;62(4):504-505. doi: 10.1111/ajd.13665. Epub 2021 Jul 27.
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Idiopathic Harlequin Syndrome Manifesting during Exercise: A Case Report and Review of the Literature.运动期间出现的特发性丑角综合征:一例报告并文献复习
Case Rep Med. 2017;2017:5342593. doi: 10.1155/2017/5342593. Epub 2017 Feb 21.
3
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Korean J Pediatr. 2016 Nov;59(Suppl 1):S125-S128. doi: 10.3345/kjp.2016.59.11.S125. Epub 2016 Nov 30.
4
Harlequin syndrome in two athletes.两名运动员的丑角综合征
Br J Sports Med. 2005 Jan;39(1):e1. doi: 10.1136/bjsm.2004.012195.