Literature DB >> 18510828

Clinical and morphological phenotype of geleophysic dysplasia.

Ozlem Giray1, Mustafa Kýr, Elçin Bora, Gül Saylam, Baran Ugurlu, Duygu Gürel.   

Abstract

Geleophysic dysplasia (GD) is a rare, recessively inherited lysosomal storage disorder of unknown origin with a progressive course. A 9-year-old Turkish boy born to consanguineous parents with findings typical of GD is reported. Cardiac abnormalities included mitral and aortic stenosis with aortic insufficiency. There was persistent hypo-uricacidaemia, severe pulmonary hypertension and tricuspid insufficiency. He required aortic and mitral valve replacement but, unfortunately, died of a severe pulmonary infection in the post-operative period. The condition has to be differentiated from lysosomal storage disorders such as mucopolysaccharidosis.

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Year:  2008        PMID: 18510828     DOI: 10.1179/146532808X302206

Source DB:  PubMed          Journal:  Ann Trop Paediatr        ISSN: 0272-4936


  3 in total

1.  Three novel mutations of the FBN1 gene in Chinese children with acromelic dysplasia.

Authors:  Yu Wang; Huiwen Zhang; Jun Ye; Lianshu Han; Xuefan Gu
Journal:  J Hum Genet       Date:  2014-08-21       Impact factor: 3.172

2.  Limb- and tendon-specific Adamtsl2 deletion identifies a role for ADAMTSL2 in tendon growth in a mouse model for geleophysic dysplasia.

Authors:  Dirk Hubmacher; Nandaraj Taye; Zerina Balic; Stetson Thacker; Sheila M Adams; David E Birk; Ronen Schweitzer; Suneel S Apte
Journal:  Matrix Biol       Date:  2019-02-07       Impact factor: 11.583

3.  Adamtsl2 deletion results in bronchial fibrillin microfibril accumulation and bronchial epithelial dysplasia--a novel mouse model providing insights into geleophysic dysplasia.

Authors:  Dirk Hubmacher; Lauren W Wang; Robert P Mecham; Dieter P Reinhardt; Suneel S Apte
Journal:  Dis Model Mech       Date:  2015-03-11       Impact factor: 5.758

  3 in total

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