| Literature DB >> 30022420 |
Mehdi Shafaat1, Mohammad Reza Alaee2, Ali Rahmanifar3, Aria Setoodeh4, Maryam Razzaghy-Azar5,6, Hamideh Bagherian7, Samira Dabbagh Bagheri7, Fatemeh Zafarghandi Motlagh7, Mehrdad Hashemi1, Maryam Abiri8,9, Sirous Zeinali10,11.
Abstract
Isolated Methylmalonic acidemia/aciduria (MMA) is a group of inborn errors of metabolism disease which is caused by defect in methylmalonyl-CoA mutase (MCM) enzyme. The enzyme has a key function in the catabolism of branched chain amino acids (BCAA, isoleucine, and valine), methionine, and threonine. MCM is encoded by a single gene named "MUT". Other subtypes of MMA are caused by mutations in cblA (encoded by MMAA) and cblB (encoded by MMAB), which is involved in the synthesis of methylmalonyl-coenzyme A cofactor. Different types of mutations have been identified as the cause of MMA. However, the mutation spectrum of MMA in Iran has not been studied so far. Here, we aimed to investigate the MMA causative mutations in the Iranian population. Using STR (Short Tandem Repeat) markers, we performed autozygosity mapping to identify the potential pathogenic variants in 11 patients with clinical diagnosis of MMA. Nineteen STR markers which are linked to the MUT, MMAA and MMAB genes (the genes with known causative mutations in MMA) were selected for PCR-amplification using two recently designed multiplex PCR panels. Next, the families that were diagnosed with homozygous haplotypes for the candidate genes were directly sequenced. Five novel mutations (c.805delG, c.693delC, c.223A > T, c.668A > G and c.976A > G in MUT) were identified beside other 4 recurrent mutations (c.361insT in MUT, c.571C > T and c.197-1 G > T in MMAB and c.1075C > T in MMAA). In silico analyses were also performed to predict the pathogenicity of the identified variants. The mutation c.571C > T in MMAB was the most common mutation in our study.Entities:
Keywords: Autozygosity mapping; Iran; Methylmalonic acidemia (MMA); Mutation analysis
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Year: 2018 PMID: 30022420 DOI: 10.1007/s11011-018-0277-4
Source DB: PubMed Journal: Metab Brain Dis ISSN: 0885-7490 Impact factor: 3.584