Schmitz-Abe Klaus, Ciesielski Szymon J, Schmidt Paul J, Campagna Dean R, Rahimov Fedik, Schilke Brenda A, Cuijpers Marloes, Rieneck Klaus, Lausen Birgitte, Linenberger Michael L, Sendamarai Anoop K, Guo Chaoshe, Hofmann Inga, Newburger Peter E, Matthews Dana, Shimamura Akiko, Snijders Pieter J L M, Towne Meghan C, Niemeyer Charlotte M, Watson Henry G, Dziegiel Morten H, Heeney Matthew M, May Alison, Bottomley Sylvia S, Swinkels Dorine W, Markianos Kyriacos, Craig Elizabeth A, Fleming Mark D
Division of Genetics and Genomics, Boston Children's Hospital, Boston, MA; Harvard Medical School, Boston, MA;
Department of Biochemistry, University of Wisconsin, Madison, WI;
Blood. 2015 Dec 17;126(25):2734-8. doi: 10.1182/blood-2015-09-659854. Epub 2015 Oct 21.
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.
先天性铁粒幼细胞贫血(CSA)是相对罕见的疾病,其特征是线粒体血红素合成、铁硫(Fe-S)簇生物合成或蛋白质合成存在缺陷。我们在此证明,位于染色体5q缺失综合征5q33关键缺失区间且参与线粒体Fe-S生物合成的线粒体HSP70同源物HSPA9发生突变,会导致CSA以常染色体隐性性状遗传。在一部分只有1个严重功能丧失等位基因的患者中,临床表型的表达与反式常见编码单核苷酸多态性相关,该多态性与信使核糖核酸表达降低相关,并导致假显性遗传模式。