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一组患有腓骨发育不全、胫骨弯曲和少指(趾)畸形(FATCO)综合征个体的基因组测序。

Genomic sequencing in a cohort of individuals with fibular aplasia, tibial campomelia, and oligosyndactyly (FATCO) syndrome.

作者信息

Matalon Dena R, Bhoj Elizabeth J, Li Dong, McDougall Carey, Schindewolf Erica, Khalek Nahla, Wilkens Alisha, McManus Morgan, Deardorff Matthew A, Zackai Elaine H

机构信息

Division of Medical Genetics, Stanford University, Stanford, California, USA.

Division of Human Genetics and Molecular Biology, Children's Hospital of Philadelphia, Philadelphia, Pennsylvania, USA.

出版信息

Am J Med Genet A. 2023 Apr;191(4):977-982. doi: 10.1002/ajmg.a.63105. Epub 2023 Jan 6.

Abstract

Fibular aplasia, tibial campomelia, and oligosyndactyly (FATCO) syndrome (MIM 246570) is a rare disorder characterized by specific skeletal findings (fibular aplasia, shortened or bowed tibia, and oligosyndactyly of the foot and/or hand). Typically, no other anomalies, craniofacial dysmorphism, or developmental delays are associated. Here we report three unrelated individuals with limb anomalies consistent with FATCO syndrome who have been followed clinically for 5 years. Genetic testing of previously reported individuals with FATCO syndrome has not revealed a genetic diagnosis. However, no broader sequencing approaches have been reported. We describe the results of the three individuals with FATCO syndrome from exome and genome sequencing, all of which was nondiagnostic. Our study suggests that FATCO syndrome is not the result of a simple monogenic etiology.

摘要

腓骨发育不全、胫骨弯曲和少指(趾)畸形(FATCO)综合征(MIM 246570)是一种罕见的疾病,其特征为特定的骨骼表现(腓骨发育不全、胫骨缩短或弯曲以及足和/或手的少指[趾]畸形)。通常,不伴有其他异常、颅面畸形或发育迟缓。在此,我们报告了三名患有与FATCO综合征相符的肢体异常的无关个体,他们已接受了5年的临床随访。对先前报道的患有FATCO综合征的个体进行的基因检测尚未得出基因诊断结果。然而,尚未有更广泛的测序方法的报道。我们描述了这三名患有FATCO综合征的个体外显子组和基因组测序的结果,所有结果均未得出诊断。我们的研究表明,FATCO综合征并非简单的单基因病因所致。

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