Literature DB >> 29597095

Speech and language delay in a patient with WDR4 mutations.

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.
Copyright © 2018 Elsevier Masson SAS. All rights reserved.

Entities:  

Keywords:  Mutation; Primordial dwarfism (PD); WDR4; Whole-exome sequencing

Mesh:

Substances:

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


  8 in total

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5.  Novel PAK3 gene missense variant associated with two Chinese siblings with intellectual disability: a case report.

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6.  N7-methylguanosine tRNA modification promotes esophageal squamous cell carcinoma tumorigenesis via the RPTOR/ULK1/autophagy axis.

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Journal:  Nat Commun       Date:  2022-03-18       Impact factor: 14.919

7.  METTL1/WDR4-mediated m7G tRNA modifications and m7G codon usage promote mRNA translation and lung cancer progression.

Authors:  Jieyi Ma; Hui Han; Ying Huang; Chunlong Yang; Siyi Zheng; Tiancai Cai; Jiong Bi; Xiaohui Huang; Ruiming Liu; Libin Huang; Yifeng Luo; Wen Li; Shuibin Lin
Journal:  Mol Ther       Date:  2021-08-08       Impact factor: 11.454

8.  Early-onset infant epileptic encephalopathy associated with a de novo PPP3CA gene mutation.

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  8 in total

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