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一例罕见的 HIV 感染者三叉神经营养综合征伴眶周蜂窝织炎和全层上睑缺损:病例报告。

A rare case of trigeminal trophic syndrome with periorbital cellulitis and full-thickness upper eyelid defect in an undiagnosed patient with human immunodeficiency virus: a case report.

机构信息

Department of Community Health and Preventive Medicine, School of Medicine, University for Development Studies, Tamale, Ghana.

Yizura Hospital Limited, Kintampo, Ghana.

出版信息

J Med Case Rep. 2024 Jul 22;18(1):335. doi: 10.1186/s13256-024-04621-0.

DOI:10.1186/s13256-024-04621-0
PMID:39034418
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC11265085/
Abstract

BACKGROUND

Trigeminal trophic syndrome is a rare cranial and facial condition caused by damage to the central or peripheral branches of the trigeminal nerve. This syndrome consists of a triad of anesthesia, paresthesia, and crescent-shaped facial ulcer involving the ala nasi and sometimes extending to the upper lip. Although previous screening for human immunodeficiency virus in some patients with trigeminal trophic syndrome was negative, we present a unique case of trigeminal trophic syndrome who tested positive for human immunodeficiency virus with eye complications.

CASE PRESENTATION

We present a rare case of trigeminal trophic syndrome in a 44-year-old Black African woman who tested positive for human immunodeficiency virus. She presented with a 6-week history of progressive, persistent, and painless left sided facial and scalp ulcerations that started as small skin erosion. Diagnosis of trigeminal trophic syndrome was made on clinical grounds based on the triad of anesthesia, paresthesia, and unilateral crescent-shaped ulcer in the trigeminal dermatome and her past medical history. The ulcer healed completely after counseling and pharmacological therapy, but she later developed left periorbital cellulitis and left upper eyelid full-thickness defect.

CONCLUSION

This is by far the first documented case of trigeminal trophic syndrome with a positive human immunodeficiency virus test. Testing for human immunodeficiency virus in patients with trigeminal trophic syndrome is necessary as this can help improve clinical management and treatment outcomes. Seeking the services of specialists remotely in resource constraint settings is beneficial for managing complications associated with trigeminal trophic syndrome.

摘要

背景

三叉神经营养综合征是一种罕见的颅面疾病,由三叉神经的中枢或周围分支损伤引起。该综合征由三联征组成,即麻醉、感觉异常和新月形面部溃疡,累及鼻翼,有时延伸至上唇。尽管先前对一些三叉神经营养综合征患者进行的人类免疫缺陷病毒筛查结果为阴性,但我们报告了一例独特的人类免疫缺陷病毒阳性三叉神经营养综合征病例,该病例伴有眼部并发症。

病例介绍

我们报告了一例罕见的 44 岁黑人女性三叉神经营养综合征病例,该患者人类免疫缺陷病毒检测结果为阳性。她有 6 周的左面部和头皮进行性、持续性、无痛性溃疡病史,最初是小的皮肤糜烂。根据临床三联征(麻醉、感觉异常和单侧三叉神经皮节的新月形溃疡)和既往病史,诊断为三叉神经营养综合征。在进行咨询和药物治疗后,溃疡完全愈合,但随后她出现左眼眶蜂窝织炎和左上眼睑全层缺损。

结论

这是迄今为止第一例有记录的人类免疫缺陷病毒阳性三叉神经营养综合征病例。对三叉神经营养综合征患者进行人类免疫缺陷病毒检测是必要的,因为这有助于改善临床管理和治疗结果。在资源有限的环境中远程寻求专家服务,有利于管理三叉神经营养综合征相关并发症。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/dd4b/11265085/da1b6b9013f8/13256_2024_4621_Fig3_HTML.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/dd4b/11265085/6bc0c85f07b1/13256_2024_4621_Fig1_HTML.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/dd4b/11265085/47b456458feb/13256_2024_4621_Fig2_HTML.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/dd4b/11265085/da1b6b9013f8/13256_2024_4621_Fig3_HTML.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/dd4b/11265085/6bc0c85f07b1/13256_2024_4621_Fig1_HTML.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/dd4b/11265085/47b456458feb/13256_2024_4621_Fig2_HTML.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/dd4b/11265085/da1b6b9013f8/13256_2024_4621_Fig3_HTML.jpg

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本文引用的文献

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Trigeminal Trophic Syndrome as a Complication of Herpes Zoster Ophthalmicus.三叉神经营养综合征作为眼部带状疱疹的一种并发症
Cureus. 2022 Oct 17;14(10):e30382. doi: 10.7759/cureus.30382. eCollection 2022 Oct.
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Trigeminal trophic syndrome: Strange evolution of maxillofacial surgery.三叉神经营养综合征:颌面外科的奇特演变。
EXCLI J. 2019 Oct 16;18:931-935. doi: 10.17179/excli2019-1846. eCollection 2019.
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Trigeminal Trophic Syndrome-A Unique Clinical Presentation of a Rare Condition.三叉神经营养综合征——一种罕见病症的独特临床表现
Ear Nose Throat J. 2019 Dec;98(10):606-608. doi: 10.1177/0145561319850818. Epub 2019 May 23.
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Trigeminal trophic syndrome: an updated review.三叉神经营养综合征:更新的综述。
Int J Dermatol. 2019 May;58(5):530-537. doi: 10.1111/ijd.14098. Epub 2018 Jun 21.
5
Trigeminal Trophic Syndrome Leading to Orbital Cellulitis.三叉神经营养综合征导致眶蜂窝织炎。
Clin Pract Cases Emerg Med. 2018 Mar 14;2(2):121-124. doi: 10.5811/cpcem.2018.1.36622. eCollection 2018 May.
6
Periocular manifestations of trigeminal trophic syndrome: A case series and literature review.三叉神经营养综合征的眼周表现:病例系列及文献综述
Orbit. 2018 Feb;37(1):32-35. doi: 10.1080/01676830.2017.1353117. Epub 2017 Aug 16.
7
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