[局灶性真皮发育不全与致病性 PORCN 基因变异有关,为后代单侧镶嵌型]。

Dermatologie (Heidelberg, Germany) Pub Date : 2024-06-01 Epub Date: 2024-02-16 DOI:10.1007/s00105-024-05308-9
Eleni Koutra, Elke Lusmöller, Judith Fischer, Katalin Komlosi, Rudolf Stadler, Ralf Gutzmer
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引用次数: 0

摘要

我们报告了一例 29 岁女性的病例,她自幼身体右侧沿布拉什科纹出现部分红斑、部分色素沉着的条纹。皮肤和血液检测的原始 DNA 结果诊断为镶嵌型局灶性真皮发育不全。PORCN 基因的杂交后突变只在受影响的皮肤中检测到,在血液检测中没有检测到。这篇文章说明,临床上非常不连续的色素沉着症和布拉斯克氏线型皮肤病应引起人们对强有力的诊断分析的重视,以便识别这种可变的多系统疾病,并确保对皮肤外异常和人类遗传咨询进行适当的搜索,最好是在怀孕前进行。临床、组织学和遗传学特征的仔细关联,以及人类遗传学、皮肤病学、儿科学、整形外科和眼科学等领域专家的多学科密切合作,对于最终诊断、评估预后和为患者提供有针对性的遗传咨询至关重要。
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[Focal dermal hypoplasia associated with pathogenic PORCN gene variant in postzygotic, unilateral mosaic form].

We report a case of a 29-year-old woman with subtle partial erythematous, partial hyperpigmented streaks along the Blaschko's lines on the right side of the body since early childhood. Primary DNA results of the skin and blood assay diagnosed focal dermal hypoplasia in mosaic form. The postzygotic mutation in the PORCN gene was only detectable in the affected skin and not in the blood assay. This article illustrates that clinically very discrete hypopigmentation and poikiloderma along Blaschko lines should raise awareness for robust diagnostic analysis in order to recognize this variable multisystem disease and to ensure an appropriate search for extracutaneous abnormalities and human genetic counseling, ideally before pregnancy. Careful correlation of clinical, histological, and genetic features along with close multidisciplinary cooperation of specialists from the fields of human genetics, dermatology, pediatrics, orthopedics and ophthalmology is crucial for final diagnosis, assessment of the prognosis and targeted genetic counseling of affected individuals.

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