| Literature DB >> 29597095 |
Xiang Chen1, Yanyan Gao2, Lin Yang3, Bingbing Wu3, Xinran Dong3, Bo Liu3, Yulan Lu3, Wenhao Zhou4, Huijun Wang5.
Abstract
Primordial dwarfism (PD) is mainly characterized by growth deficiency with heterogeneous phenotypes. A group of genes are known to be associated with PD or PD-related syndrome. WD repeat domain 4 (WDR4) is recently reported to be responsible for PD. Here we report a 6-year-old boy from a non-consanguineous couple with motor and speech delay as well as intellectual disability. Whole exome sequencing (WES) identified a missense mutation (NM_033661.4:c.491A > C; p.(Asp164Ala)) and a small insertion (NM_033661.4:c.940dupC; p.(Leu314Profs*16)) of WDR4 in this patient. Two novel mutations confirmed by Sanger sequencing are from father and mother respectively according to a recessive inheritance pattern. Asp164Ala located in functional region is predicted to be deleterious by two kinds of algorithm. The small insertion causing a frameshift mutation leads to truncated protein. In this study, we present two novel WDR4 mutations responsible for PD in a 6-year-old patient, expanding the molecular and phenotype spectrum of WDR4-related PD.Entities:
Keywords: Mutation; Primordial dwarfism (PD); WDR4; Whole-exome sequencing
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Year: 2018 PMID: 29597095 DOI: 10.1016/j.ejmg.2018.03.007
Source DB: PubMed Journal: Eur J Med Genet ISSN: 1769-7212 Impact factor: 2.708