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患者无颅面发育不全的 Nager 综合征:扩大 SF3B4 相关疾病的表型谱。

Nager syndrome in patient lacking acrofacial dysostosis: Expanding the phenotypic spectrum of SF3B4-related disease.

机构信息

Department of Pathology and Laboratory Medicine, Children's Mercy Hospitals, Kansas City, Missouri, USA.

Division of Clinical Genetics, Children's Mercy Hospital, Kansas City, Missouri, USA.

出版信息

Am J Med Genet A. 2021 May;185(5):1515-1518. doi: 10.1002/ajmg.a.62113. Epub 2021 Feb 8.

Abstract

Nager syndrome epitomizes the acrofacial dysostoses, which are characterized by craniofacial and limb defects. The craniofacial defects include midfacial retrusion, downslanting palpebral fissures, prominent nasal bridge, and micrognathia. Limb malformations typically include hypoplasia or aplasia of radial elements including the thumb. Nager syndrome is caused by haploinsufficiency of SF3B4, encoding a spliceosomal protein called SAP49. Here, we report a patient with a loss of function variant in SF3B4 without acrofacial dysostosis or limb defects, whose reason for referral was developmental and growth delay. This patient is evidence of a broader phenotypic spectrum associated with SF3B4 variants than previously appreciated.

摘要

Nager 综合征是颅面发育不全的典型代表,其特征为颅面和四肢的缺陷。颅面缺陷包括中面部后缩、下斜的睑裂、高鼻、小下颌。肢体畸形通常包括桡骨元素包括拇指的发育不良或发育不全。Nager 综合征是由 SF3B4 的单倍不足引起的,SF3B4 编码一种剪接体蛋白,称为 SAP49。在这里,我们报告了一名 SF3B4 功能丧失变异患者,无颅面发育不全或肢体缺陷,其转诊原因是发育和生长迟缓。该患者证明了与 SF3B4 变异相关的表型谱比以前认识到的更广泛。

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