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穆林线性皮肤萎缩症:一例罕见病例报告及文献综述

Linear atrophoderma of Moulin: A rare case report and review of the literature.

作者信息

Hussein Al-Janabi Moatasem, Diab Sdrah, Aljammal Ghina, Kassab Lina, Al-Shehabi Zuheir, Al-Soiufi Lina

机构信息

Department of Pathology Cancer Research Center Tishreen University Hospital Lattakia Syria.

Department of Dermatology and Sexually Transmitted Disease Tishreen University Hospital Lattakia Syria.

出版信息

Skin Health Dis. 2024 Jul 21;4(5):e424. doi: 10.1002/ski2.424. eCollection 2024 Oct.

DOI:10.1002/ski2.424
PMID:39355723
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC11442054/
Abstract

Linear atrophoderma of Moulin (LAM) is an uncommon dermatological disease characterised by linear, depressed plaques typically following Blaschko's lines. LAM generally occurs in adolescence or early adulthood and is more commonly observed in females. The aetiology of LAM is still unclear. We report a rare case of LAM in an 18-year-old female presenting with an atypical Blaschkoid distribution (multiple band-like pattern) on her right trunk. A clinical examination and histopathological analysis were performed to make the diagnosis. Partial improvement was obtained with calcipotriol and colchicine. LAM is a rare condition; we were only able to identify 23 case reports in the published literature. The findings of this report contribute to the limited literature on LAM, highlighting the clinical variability of LAM and suggesting potential novel variants beyond the classic presentation, emphasising the importance of recognising diverse manifestations for accurate diagnosis and management. Early recognition of LAM is crucial for appropriate treatment and improved patient outcomes. Further research is needed to elucidate LAM's aetiology and underlying mechanisms to facilitate the development of more targeted therapeutic strategies.

摘要

穆林线性皮肤萎缩症(LAM)是一种罕见的皮肤病,其特征为通常沿布拉斯科线分布的线性凹陷斑块。LAM一般发生于青春期或成年早期,女性更为常见。LAM的病因仍不清楚。我们报告了一例罕见的LAM病例,患者为一名18岁女性,其右侧躯干出现非典型布拉斯科样分布(多条带状图案)。进行了临床检查和组织病理学分析以明确诊断。使用卡泊三醇和秋水仙碱后病情有部分改善。LAM是一种罕见病症;我们在已发表的文献中仅能找到23例病例报告。本报告的结果丰富了关于LAM的有限文献,突出了LAM的临床变异性,并提示了经典表现之外可能存在的新变体,强调了认识多种表现形式对于准确诊断和管理的重要性。早期识别LAM对于恰当治疗和改善患者预后至关重要。需要进一步研究以阐明LAM的病因和潜在机制,从而推动更具针对性治疗策略的发展。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/5ac0/11442054/0c66a3c4fc7d/SKI2-4-e424-g001.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/5ac0/11442054/6772622ee290/SKI2-4-e424-g004.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/5ac0/11442054/9b6e0ab90653/SKI2-4-e424-g005.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/5ac0/11442054/3a66ca29458f/SKI2-4-e424-g002.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/5ac0/11442054/0c66a3c4fc7d/SKI2-4-e424-g001.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/5ac0/11442054/6772622ee290/SKI2-4-e424-g004.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/5ac0/11442054/9b6e0ab90653/SKI2-4-e424-g005.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/5ac0/11442054/3a66ca29458f/SKI2-4-e424-g002.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/5ac0/11442054/0c66a3c4fc7d/SKI2-4-e424-g001.jpg

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本文引用的文献

1
Atypical Linear Atrophoderma of Moulin Complicated with Elevated Immunoglobulin M: A Case Report.伴有免疫球蛋白M升高的穆林型非典型线状皮肤萎缩症:一例报告
Clin Cosmet Investig Dermatol. 2023 Jan 21;16:193-196. doi: 10.2147/CCID.S398441. eCollection 2023.
2
Linear atrophoderma of Moulin is due to the decreased dermal thickness.穆林线性皮肤萎缩症是由于真皮厚度减少所致。
Skin Res Technol. 2022 Jul;28(4):646-648. doi: 10.1111/srt.13175. Epub 2022 Jun 10.
3
Linear Atrophoderma of Moulin.穆林线性皮肤萎缩
JAMA Dermatol. 2020 May 1;156(5):581. doi: 10.1001/jamadermatol.2020.0320.
4
A case of linear atrophoderma of Moulin.穆林线性皮肤萎缩症一例。
An Bras Dermatol. 2020 Jan-Feb;95(1):119-121. doi: 10.1016/j.abd.2019.03.005. Epub 2019 Dec 18.
5
Linear atrophoderma of Moulin: A rare entity.穆林线状皮肤萎缩症:一种罕见病症。
Indian J Dermatol Venereol Leprol. 2018 Sep-Oct;84(5):591-594. doi: 10.4103/ijdvl.IJDVL_136_17.
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Linear Atrophoderma of Moulin over Face: An Exceedingly Rare Entity.面部穆林线性皮肤萎缩:一种极其罕见的病症。
Indian J Dermatol. 2017 Mar-Apr;62(2):214-215. doi: 10.4103/ijd.IJD_469_16.
7
Linear atrophoderma of Moulin: a disease related to immunity or a kind of connective tissue disease?穆林线性皮肤萎缩症:一种与免疫相关的疾病还是一种结缔组织病?
Australas J Dermatol. 2017 Aug;58(3):e126-e128. doi: 10.1111/ajd.12514. Epub 2016 Jun 10.
8
Linear atrophoderma of Moulin.穆林线性皮肤萎缩
JAAD Case Rep. 2016 Jan 8;2(1):10-2. doi: 10.1016/j.jdcr.2015.10.005. eCollection 2016 Jan.
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Linear atrophoderma of Moulin.穆林线性皮肤萎缩
Dermatol Online J. 2015 Dec 16;21(12):13030/qt9x28x358.
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