Literature DB >> 8357004

New form of spondyloepimetaphyseal dysplasia (SEMD) in Jewish family of Iraqi origin.

M Shohat1, R Lachman, R Carmi, J Bar Ziv, D Rimoin.   

Abstract

We report a distinct type of spondyloepimetaphyseal dysplasia seen in 2 sibs and their second cousin, characterized by early onset severe short stature, small chest, and distended abdomen. They had short neck, severe lumbar lordosis, and marked genu varum due to fibular overgrowth and joint laxity. Radiographically, the patients had platyspondyly, initially noted during the first years of life, with central hypoplasia of the vertebral bodies. At a later age, the vertebrae appear squared with mild interpedicular narrowing. The long bone changes, which at early age resemble those seen in achondroplasia, later include general metaphyseal irregularities and significant epiphyseal ossification delay. These patients present a previously undescribed form of spondyloepimetaphyseal dysplasia, most probably transmitted as an autosomal recessive tract.

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Year:  1993        PMID: 8357004     DOI: 10.1002/ajmg.1320460403

Source DB:  PubMed          Journal:  Am J Med Genet        ISSN: 0148-7299


  2 in total

1.  Loss of DDRGK1 modulates SOX9 ubiquitination in spondyloepimetaphyseal dysplasia.

Authors:  Adetutu T Egunsola; Yangjin Bae; Ming-Ming Jiang; David S Liu; Yuqing Chen-Evenson; Terry Bertin; Shan Chen; James T Lu; Lisette Nevarez; Nurit Magal; Annick Raas-Rothschild; Eric C Swindell; Daniel H Cohn; Richard A Gibbs; Philippe M Campeau; Mordechai Shohat; Brendan H Lee
Journal:  J Clin Invest       Date:  2017-03-06       Impact factor: 14.808

2.  DDRGK1 is required for the proper development and maintenance of the growth plate cartilage.

Authors:  Monika Weisz-Hubshman; Adetutu T Egunsula; Brian Dawson; Alexis Castellon; Ming-Ming Jiang; Yuqing Chen-Evenson; Yu Zhiyin; Brendan Lee; Yangjin Bae
Journal:  Hum Mol Genet       Date:  2022-08-23       Impact factor: 5.121

  2 in total

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