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STAR综合征附加型:首例致死型女性患者的描述。

STAR syndrome plus: The first description of a female patient with the lethal form.

作者信息

Bedeschi Maria F, Giangiobbe Sara, Paganini Leda, Tabano Silvia, Silipigni Rosamaria, Colombo Lorenzo, Crippa Beatrice L, Lalatta Faustina, Guerneri Silvana, Miozzo Monica

机构信息

Clinical Genetics Unit, Fondazione IRCCS Ca' Granda Ospedale Maggiore Policlinico, Milan, Italy.

Department of Pathophysiology and Transplantation, Università degli Studi di Milano and Fondazione IRCCS Ca' Granda Ospedale Maggiore Policlinico, Milan, Italy.

出版信息

Am J Med Genet A. 2017 Dec;173(12):3226-3230. doi: 10.1002/ajmg.a.38484. Epub 2017 Oct 31.

Abstract

The STAR syndrome is a rare X-linked dominant developmental disorder caused by point mutations in the single FAM58A gene or deletions involving FAM58A and its flanking genes. The STAR phenotype is characterized by a rather homogeneous constellation of facial dysmorphisms and malformations summarized by its acronym, Syndactyly, Telecanthus, Anogenital, and Renal malformations. Here we describe a female patient with STAR syndrome and a 130 kb deletion at Xq28, including the FAM58A gene. She presented with cleft lip palate, omphalocele, and cerebral malformations not previously considered part of the phenotypic spectrum of this syndrome. She died at 6 weeks from respiratory failure.

摘要

STAR综合征是一种罕见的X连锁显性发育障碍,由单个FAM58A基因的点突变或涉及FAM58A及其侧翼基因的缺失引起。STAR表型的特征是一组相当一致的面部畸形和畸形,由其首字母缩写Syndactyly(并指)、Telecanthus(眼距增宽)、Anogenital(肛门生殖器)和Renal(肾脏)畸形概括。在此,我们描述了一名患有STAR综合征且在Xq28处有130 kb缺失(包括FAM58A基因)的女性患者。她表现出唇腭裂、脐膨出和脑畸形,这些以前未被认为是该综合征表型谱的一部分。她在6周时死于呼吸衰竭。

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