Literature DB >> 30914273

Phenotype and response to growth hormone therapy in siblings with B4GALT7 deficiency.

Carla Sandler-Wilson1, Jennifer A Wambach2, Bess A Marshall3, Daniel J Wegner1, William McAlister4, F Sessions Cole3, Marwan Shinawi1.   

Abstract

B4GALT7 encodes beta-1,4-galactosyltransferase which links glycosaminoglycans to proteoglycans in connective tissues. Rare, biallelic variants in B4GALT7 have been associated with spondylodysplastic Ehlers-Danlos and Larsen of Reunion Island syndromes. Thirty patients with B4GALT7-related disorders have been reported to date with phenotypic variability. Using whole exome sequencing, we identified male and female siblings with biallelic, pathogenic B4GALT7 variants and phenotypic features of spondylodysplastic Ehlers-Danlos syndrome as well as previously unreported skeletal characteristics. We also provide detailed radiological characterization and describe the siblings' responses to growth hormone treatment. Our report extends the phenotypic spectrum of B4GALT7-associated spondylodysplastic Ehlers-Danlos syndrome and reports results of growth hormone treatment for patients with this rare disorder.
Copyright © 2019 Elsevier Inc. All rights reserved.

Entities:  

Keywords:  B4GALT7; Growth hormone; Larsen of Reunion Island syndrome; Proteoglycan; Spondylodysplastic Ehlers-Danlos syndrome

Year:  2019        PMID: 30914273      PMCID: PMC6551519          DOI: 10.1016/j.bone.2019.03.029

Source DB:  PubMed          Journal:  Bone        ISSN: 1873-2763            Impact factor:   4.398


  33 in total

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5.  Mutant B3GALT6 in a Multiplex Family: A Dominant Variant Co-Segregated With Moderate Malformations.

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