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[与致病性PORCN基因变异相关的局限性皮肤发育不全,呈合子后单侧镶嵌形式]

[Focal dermal hypoplasia associated with pathogenic PORCN gene variant in postzygotic, unilateral mosaic form].

作者信息

Koutra Eleni, Lusmöller Elke, Fischer Judith, Komlosi Katalin, Stadler Rudolf, Gutzmer Ralf

机构信息

Universitätsklinik für Dermatologie, Venerologie, Allergologie und Phlebologie, Johannes Wesling Klinikum Minden, Hans-Nolte-Str. 1, 32429, Minden, Deutschland.

Institut für Humangenetik, Universitätsklinikum Freiburg, Freiburg, Deutschland.

出版信息

Dermatologie (Heidelb). 2024 Jun;75(6):486-491. doi: 10.1007/s00105-024-05308-9. Epub 2024 Feb 16.

DOI:10.1007/s00105-024-05308-9
PMID:38366244
Abstract

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.

摘要

我们报告了一例29岁女性病例,自幼年起,其身体右侧沿布拉斯科线出现细微的部分红斑、部分色素沉着条纹。皮肤和血液检测的初步DNA结果诊断为镶嵌型局灶性真皮发育不全。PORCN基因的合子后突变仅在受影响的皮肤中可检测到,而在血液检测中未检测到。本文表明,临床上沿布拉斯科线非常离散的色素减退和皮肤异色症应提高对进行全面诊断分析的认识,以便识别这种多变的多系统疾病,并确保在理想情况下,在怀孕前对皮肤外异常进行适当检查并提供人类遗传咨询。临床、组织学和遗传学特征的仔细关联,以及人类遗传学、皮肤科、儿科、骨科和眼科领域专家的密切多学科合作,对于最终诊断、预后评估以及对受影响个体进行针对性的遗传咨询至关重要。

相似文献

1
[Focal dermal hypoplasia associated with pathogenic PORCN gene variant in postzygotic, unilateral mosaic form].[与致病性PORCN基因变异相关的局限性皮肤发育不全,呈合子后单侧镶嵌形式]
Dermatologie (Heidelb). 2024 Jun;75(6):486-491. doi: 10.1007/s00105-024-05308-9. Epub 2024 Feb 16.
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本文引用的文献

1
Mosaicism due to postzygotic mutations in women with focal dermal hypoplasia.女性局灶性皮肤发育不良的合子后突变导致镶嵌现象。
Br J Dermatol. 2019 Mar;180(3):657-661. doi: 10.1111/bjd.17024. Epub 2018 Sep 30.
2
[Focal dermal hypoplasia (Goltz-Gorlin syndrome) : The cause is now known].
Hautarzt. 2016 Jul;67(7):583-5. doi: 10.1007/s00105-016-3823-1.
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Dermatologic findings of focal dermal hypoplasia (Goltz syndrome).局灶性真皮发育不全(戈尔茨综合征)的皮肤表现。
Am J Med Genet C Semin Med Genet. 2016 Mar;172C(1):44-51. doi: 10.1002/ajmg.c.31472. Epub 2016 Feb 9.
4
Phenotypic and molecular characterization of focal dermal hypoplasia in 18 individuals.18例患者局灶性真皮发育不全的表型和分子特征分析
Am J Med Genet C Semin Med Genet. 2016 Mar;172C(1):9-20. doi: 10.1002/ajmg.c.31473. Epub 2016 Feb 7.
5
Deficiency of PORCN, a regulator of Wnt signaling, is associated with focal dermal hypoplasia.Wnt信号通路的调节因子PORCN的缺乏与局灶性真皮发育不全相关。
Nat Genet. 2007 Jul;39(7):833-5. doi: 10.1038/ng2052. Epub 2007 Jun 3.
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The evolutionarily conserved porcupine gene family is involved in the processing of the Wnt family.进化上保守的豪猪基因家族参与Wnt家族的加工过程。
Eur J Biochem. 2000 Jul;267(13):4300-11. doi: 10.1046/j.1432-1033.2000.01478.x.
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Focal dermal hypoplasia syndrome. An update.局灶性真皮发育不全综合征。最新进展。
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