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A novel deletion mutation involving TMEM38B in a patient with autosomal recessive osteogenesis imperfecta

Authors :
Angela D'Eustacchio
Elisa Rubinato
Paolo Gasparini
Vanna Pecile
Flavio Faletra
Anna Morgan
Giulia Gortani
Elisa, Rubinato
Morgan, Anna
Angela, D'Eustacchio
Vanna, Pecile
Giulia, Gortani
Gasparini, Paolo
Flavio, Faletra
Source :
Gene. 545:290-292
Publication Year :
2014
Publisher :
Elsevier BV, 2014.

Abstract

Osteogenesis imperfecta (OI) is a hereditary bone disease characterized by decreased bone density and multiple fractures, usually inherited in an autosomal dominant manner. Several gene encoding proteins related to collagen metabolism have been described in some cases of autosomal recessive OI (including CRTAP, LEPRE1, PPIB, FKBP65, SERPINF1, BMP1, WNT1, FKBP10). Recently, TMEM38B, a gene that encodes TRIC-B, a monovalent cation-specific channel involved in calcium flux from intracellular stores and in cell differentiation, has been associated with autosomal recessive OI. Here, we describe the second deletion-mutation involving the TMEM38B gene in an 11 year-old Albanian female with a clinical phenotype of OI, born to parents with suspected consanguinity. SNP array analysis revealed a homozygous region larger than 2 Mb that overlapped with the TMEM38B locus and was characterized by a 35 kb homozygous deletion involving exons 1 and 2 of TMEM38B gene.

Details

ISSN :
03781119
Volume :
545
Database :
OpenAIRE
Journal :
Gene
Accession number :
edsair.doi.dedup.....bbef522dcc49ce2a3f844328ad2da3be