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初诊或复诊:常染色体显性遗传性皮肤病镶嵌表现的临床特征-病例系列。

At first sight or second glance: clinical presentation of mosaic manifestations of autosomal dominant skin disorders - a case series.

机构信息

Department of Dermatology, Venerology, and Allergology, University Medical Center, Ruprecht-Karls-University, Heidelberg, Germany.

Department of Dermatology, University Medical Center Freiburg, Freiburg, Germany.

出版信息

J Eur Acad Dermatol Venereol. 2017 Nov;31(11):1912-1915. doi: 10.1111/jdv.14242. Epub 2017 Apr 10.

Abstract

BACKGROUND

Several autosomal dominant disorders may manifest in mosaic patterns with cutaneous involvement. Genomic mosaicism results from postzygotic autosomal mutations, giving rise to clonal proliferation of two genetically distinct cell groups, which clinically present as lesions following the lines of Blaschko.

OBJECTIVE

To increase the awareness of the clinical variability of mosaic manifestations in autosomal dominant skin disorders in order to avoid delayed diagnosis.

METHODS

Clinicopathologic correlation in a case series including three patients with mosaic manifestations of different autosomal dominant skin diseases.

RESULTS

Here, we describe a patient with type 1 segmental mosaicism of epidermolytic ichthyosis (case 1) and two patients with either type 1 (case 2) or type 2 (case 3) segmental neurofibromatosis 1 (NF1).

CONCLUSION

Dermatologists should be familiar with mosaic manifestations of autosomal dominant skin diseases to ensure appropriate guidance of the affected patient. Genetic counselling is mandatory as even limited forms of mosaicism may involve the patient's germline with a moderately increased risk to transmit the mutation to their offspring, resulting in a more severe, generalized form of the respective disease.

摘要

背景

一些常染色体显性遗传疾病可能以镶嵌模式表现,并伴有皮肤受累。基因组镶嵌性是由合子后常染色体突变引起的,导致两个遗传上不同的细胞群克隆性增殖,临床上表现为沿着 Blaschko 线的病变。

目的

提高对常染色体显性遗传皮肤疾病镶嵌表现临床变异性的认识,以避免延迟诊断。

方法

对包括三例不同常染色体显性皮肤疾病镶嵌表现的患者进行临床病理相关性研究。

结果

我们在此描述了一例 1 型节段性表皮松解性鱼鳞病(病例 1)和两例 1 型(病例 2)或 2 型(病例 3)节段性神经纤维瘤病 1(NF1)的患者。

结论

皮肤科医生应熟悉常染色体显性遗传皮肤疾病的镶嵌表现,以确保对受影响患者进行适当的指导。遗传咨询是强制性的,因为即使是有限形式的镶嵌性也可能涉及患者的生殖系,使其后代有中度增加的突变传递风险,导致各自疾病的更严重、更广泛的形式。

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