Literature DB >> 24805776

Radioulnar Synostosis and Brain Abnormalities in a Patient With 17q21.31 Microdeletion Involving EFTUD2.

Yuri A Zarate, Carla Bell, G Bradley Schaefer.   

Abstract

Mandibulofacial dysostosis with microcephaly is a rare syndromic craniofacial condition caused by heterozygous loss-of-function mutations of the EFTUD2 gene on 17q21.31. Thus far, the described musculoskeletal findings in patients with this condition include proximally placed or duplicated thumbs, overlapping toes, and toe syndactyly. We describe a severe case of a patient with a 17q21.31 microdeletion and many of the phenotypic features described in mandibulofacial dysostosis with microcephaly who had bilateral proximal radioulnar synostosis and brain abnormalities. This provides further evidence of the clinical overlap among mandibulofacial and acrofacial dysostoses syndromes and expands the phenotype of EFTUD2 haploinsufficiency due to larger deletions.

Entities:  

Keywords:  17q21.31 microdeletion; EFTUD2; mandibulofacial dysostosis; spliceosome

Mesh:

Substances:

Year:  2014        PMID: 24805776     DOI: 10.1597/13-221

Source DB:  PubMed          Journal:  Cleft Palate Craniofac J        ISSN: 1055-6656


  4 in total

1.  Mutation in Eftud2 causes craniofacial defects in mice via mis-splicing of Mdm2 and increased P53.

Authors:  Marie-Claude Beauchamp; Anissa Djedid; Eric Bareke; Fjodor Merkuri; Rachel Aber; Annie S Tam; Matthew A Lines; Kym M Boycott; Peter C Stirling; Jennifer L Fish; Jacek Majewski; Loydie A Jerome-Majewska
Journal:  Hum Mol Genet       Date:  2021-05-28       Impact factor: 6.150

2.  Spliceosomal protein eftud2 mutation leads to p53-dependent apoptosis in zebrafish neural progenitors.

Authors:  Lei Lei; Shou-Yu Yan; Ran Yang; Jia-Yu Chen; Yumei Li; Ye Bu; Nannan Chang; Qinchao Zhou; Xiaojun Zhu; Chuan-Yun Li; Jing-Wei Xiong
Journal:  Nucleic Acids Res       Date:  2017-04-07       Impact factor: 16.971

3.  EFTUD2 gene deficiency disrupts osteoblast maturation and inhibits chondrocyte differentiation via activation of the p53 signaling pathway.

Authors:  Jing Wu; Yi Yang; You He; Qiang Li; Xu Wang; Chengjun Sun; Lishun Wang; Yu An; Feihong Luo
Journal:  Hum Genomics       Date:  2019-12-05       Impact factor: 4.639

4.  Over-activation of EFTUD2 correlates with tumor propagation and poor survival outcomes in hepatocellular carcinoma.

Authors:  C Lv; X J Li; L X Hao; S Zhang; Z Song; X D Ji; B Gong
Journal:  Clin Transl Oncol       Date:  2021-07-19       Impact factor: 3.405

  4 in total

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