Azab Belal, Dardas Zain, Hamarsheh Mohannad, Alsalem Mohammad, Kilani Zaid, Kilani Farah, Awidi Abdalla, Jafar Hanan, Amr Sami
Department of Physiology and Biochemistry, School of Medicine, The University of Jordan, Amman 11942, Jordan.
Department of Medical Laboratory Sciences, School of Science, The University of Jordan, Amman 11942, Jordan.
Mol Genet Metab Rep. 2017 Jun 9;12:76-79. doi: 10.1016/j.ymgmr.2017.06.001. eCollection 2017 Sep.
Mucopolysaccharidosis type I (MPS I) is an autosomal recessive storage disorder that result as a consequence of a deficiency in the lysosomal hydrolase, a-L-iduronidase enzyme encoded by IDUA gene. Over a hundred causative variants in IDUA have been identified, which result in a progressive multi-systemic disease with a broad range of severity and disease progression reported across affected individuals. The aim of this study was the detection and interpretation of IDUA mutation in a family with two children affected with lethal MPS I. The IDUA gene was sequenced in the parents of two deceased children who had a clinical diagnosis of MPS I, to assess their carrier status and to help inform on risk in future children. The sequencing analysis was performed by PCR and bidirectional Sanger sequencing of the coding region and exon-intron splice junctions at Labor MVZ Westmecklenburg molecular diagnostics laboratory. A heterozygous c.657delA variant in exon 6 was identified in each parent, which is the most likely explanation for disease in their children. This report represents the first Yemeni family to have a molecular diagnosis for MPS I.
I型黏多糖贮积症(MPS I)是一种常染色体隐性遗传性贮积病,由溶酶体水解酶α-L-艾杜糖醛酸酶缺乏引起,该酶由IDUA基因编码。已在IDUA基因中鉴定出一百多种致病变体,这些变体会导致一种进行性多系统疾病,不同患者的严重程度和疾病进展差异很大。本研究的目的是检测和解读一个有两名儿童患致死性MPS I的家庭中的IDUA突变。对两名临床诊断为MPS I的已故儿童的父母进行IDUA基因测序,以评估他们的携带者状态,并为未来孩子的患病风险提供信息。测序分析在西梅克伦堡MVZ分子诊断实验室通过PCR和对编码区及外显子-内含子剪接位点进行双向桑格测序完成。在每位父母中均鉴定出第6外显子的杂合c.657delA变体,这很可能是他们孩子患病的原因。本报告是首个对MPS I进行分子诊断的也门家庭。