| Literature DB >> 26491070 |
Klaus Schmitz-Abe1, Szymon J Ciesielski2, Paul J Schmidt3, Dean R Campagna4, Fedik Rahimov1, Brenda A Schilke2, Marloes Cuijpers5, Klaus Rieneck6, Birgitte Lausen7, Michael L Linenberger8, Anoop K Sendamarai3, Chaoshe Guo3, Inga Hofmann9, Peter E Newburger10, Dana Matthews11, Akiko Shimamura11, Pieter J L M Snijders12, Meghan C Towne13, Charlotte M Niemeyer14, Henry G Watson15, Morten H Dziegiel6, Matthew M Heeney9, Alison May16, Sylvia S Bottomley17, Dorine W Swinkels18, Kyriacos Markianos1, Elizabeth A Craig2, Mark D Fleming3.
Abstract
The congenital sideroblastic anemias (CSAs) are relatively uncommon diseases characterized by defects in mitochondrial heme synthesis, iron-sulfur (Fe-S) cluster biogenesis, or protein synthesis. Here we demonstrate that mutations in HSPA9, a mitochondrial HSP70 homolog located in the chromosome 5q deletion syndrome 5q33 critical deletion interval and involved in mitochondrial Fe-S biogenesis, result in CSA inherited as an autosomal recessive trait. In a fraction of patients with just 1 severe loss-of-function allele, expression of the clinical phenotype is associated with a common coding single nucleotide polymorphism in trans that correlates with reduced messenger RNA expression and results in a pseudodominant pattern of inheritance.Entities:
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Year: 2015 PMID: 26491070 PMCID: PMC4683334 DOI: 10.1182/blood-2015-09-659854
Source DB: PubMed Journal: Blood ISSN: 0006-4971 Impact factor: 22.113