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一名患有白内障的女孩出现毛囊性鱼鳞病伴脱发和畏光:组织学和电子显微镜检查结果

Ichthyosis follicularis with alopecia and photophobia in a girl with cataract: histological and electron microscopy findings.

作者信息

Tsolia Maria, Aroni Kyriaki, Konstantopoulou Ioanna, Karpathios Themistokles, Tsoukatou Theano, Paraskevakou Helen, Stavrinadis Christodoulos, Fretzayas Andrew

机构信息

Second Department of Paediatrics, P. and A. Kyriakou Children's Hospital, University of Athen School of Medicine, Greece.

出版信息

Acta Derm Venereol. 2005;85(1):51-5. doi: 10.1080/00015550410022230.

Abstract

A rare congenital ectodermal disorder characterized by ichthyosis follicularis, alopecia and photophobia has been designated the acronym IFAP. An X-linked recessive mode of inheritance was initially proposed but a few recent reports in girls suggested genetic heterogeneity of this syndrome. We herein describe a 3-year-old girl with clinical and histological features typical of IFAP. In addition to the already known features of the syndrome the patient also developed bilateral cataract. Electron microscopy examination of the skin showed partial disruption of the intercellular bridges, spongiotic changes and decrease in the number and size of desmosomes supporting the notion that IFAP may be a cell-to-cell adhesion disorder.

摘要

一种以毛囊性鱼鳞病、脱发和畏光为特征的罕见先天性外胚层疾病被命名为IFAP(首字母缩略词)。最初提出其为X连锁隐性遗传模式,但最近有一些关于女孩的报道提示该综合征存在遗传异质性。我们在此描述一名3岁女孩,其具有IFAP典型的临床和组织学特征。除了该综合征已知的特征外,该患者还出现了双侧白内障。皮肤的电子显微镜检查显示细胞间桥部分破坏、海绵状改变以及桥粒数量和大小减少,这支持了IFAP可能是一种细胞间黏附障碍的观点。

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