Literature DB >> 25355368

4q12-4q21.21 deletion genotype-phenotype correlation and the absence of piebaldism in presence of KIT haploinsufficiency.

Parisa Hemati1, Christèle du Souich, Cornelius F Boerkoel.   

Abstract

Chromosome 4q deletion syndrome is a rare intellectual disability disorder caused by a variety of non-recurrent deletions of 4q. We describe the evolution of the phenotypic features of a female patient with a previously unreported deletion of 4q12-4q21.21 (hg 18; 54,711,575-79,601,919). By review reported individuals with interstitial deletions extending telomeric from 4q12 have syndromic intellectual disability with variable piebaldism. We expand the phenotype to include dolichocephaly, pectus excavatum, hip dysplasia, pes planus, myopia, lens opacities, and an absence of spoken language but not of communication through sign. The proposita also did not have piebaldism suggesting again that piebaldism arises from a mechanism more complex than simple haploinsufficiency of KIT. Comparing deletions among affected individuals localizes the critical interval within 4q12-4q13.1, although the absence of molecular boundaries for nearly all reported cases precludes precise delineation and genotype-phenotype correlation.
© 2014 Wiley Periodicals, Inc.

Entities:  

Keywords:  4q proximal deletion; 4q21 deletion; haploinsufficiency; hip dysplasia; incomplete penetrance; piebaldism

Mesh:

Substances:

Year:  2014        PMID: 25355368     DOI: 10.1002/ajmg.a.36821

Source DB:  PubMed          Journal:  Am J Med Genet A        ISSN: 1552-4825            Impact factor:   2.802


  3 in total

1.  Phenotypic variability in a Hungarian patient with the 4q21 microdeletion syndrome.

Authors:  Katalin Komlósi; Balázs Duga; Kinga Hadzsiev; Márta Czakó; György Kosztolányi; András Fogarasi; Béla Melegh
Journal:  Mol Cytogenet       Date:  2015-03-03       Impact factor: 2.009

2.  An Interstitial 4q Deletion with a Mosaic Complementary Ring Chromosome in a Child with Dysmorphism, Linear Skin Pigmentation, and Hepatomegaly.

Authors:  J Carter; H Brittain; D Morrogh; N Lench; J J Waters
Journal:  Case Rep Genet       Date:  2017-07-27

3.  A case report of familial 4q13.3 microdeletion in three individuals with syndromic intellectual disability.

Authors:  Živilė Maldžienė; Evelina M Vaitėnienė; Beata Aleksiūnienė; Algirdas Utkus; Eglė Preikšaitienė
Journal:  BMC Med Genomics       Date:  2020-04-16       Impact factor: 3.063

  3 in total

北京卡尤迪生物科技股份有限公司 © 2022-2023.