Department of Clinical Laboratory Sciences, Prince Sultan Military College of Health Sciences, Dhahran, Saudi Arabia.
Vice Deanship of Postgraduate Studies and Research, Prince Sultan Military College of Health Sciences, Dhahran, Saudi Arabia -.
Gene. 2023 Jan 30;851:146977. doi: 10.1016/j.gene.2022.146977. Epub 2022 Oct 17.
Iron-refractory iron deficiency anemia (IRIDA) is considered an autosomal recessive iron deficiency anemia due to mutations in the transmembrane protease serine 6 (TMPRSS6) gene. Variations in iron parameters and a higher risk of iron deficiency have been linked to the TMPRSS6 mutations. Furthermore, human genome-wide association studies (GWAS) identified a common mutation (rs855791) linked to abnormal hematological parameters, highlighting the importance of the TMPRSS6 gene in the regulation of iron homeostasis. This is the first study to investigate TMPRSS6 gene mutation in six Saudi families of probands with iron deficiency anemia unresponsive to oral iron and partially responsive to parenteral iron administration. Each participant provided a vacutainer tube with three blood samples (2.5 ml each) and analyzed based on hematological, biochemical iron profiles, and followed by genotyping by PCR. The TMPRSS6 gene was amplified, sequenced, and analyzed in all probands and family members. Statistical analysis was done using SPSS and SHEsis software. Few functional mutations in these families were suggested (p.W73X, p.E523K and p.V736A). The proband of family 6 presented numerous hematological abnormalities upon initial consultation, including normocytic anemia accompanied by low Hb, normal MCV, low serum iron, low serum ferritin, and normal TIBC. While the p.W73X variant was only found in 2 families, the p.V736A variant was found in all examined Saudi families with IRIDA. Given the evidence outlined for these six cases, future genotype-phenotype correlation studies in a large number of IRIDA patients in Saudi Arabia may be very informative for patient management, in addition to increasing knowledge of TMPRSS6 function during development as well as factors in the regulation of TMPRSS6 and its effect on iron levels in the body.
铁难治性缺铁性贫血(IRIDA)被认为是一种常染色体隐性铁缺乏性贫血,是由于跨膜丝氨酸蛋白酶 6(TMPRSS6)基因突变所致。铁参数的变化和缺铁风险的增加与 TMPRSS6 突变有关。此外,人类全基因组关联研究(GWAS)发现了与异常血液学参数相关的常见突变(rs855791),突出了 TMPRSS6 基因在铁稳态调节中的重要性。这是第一项研究,调查了 6 个沙特家庭的 TMPRSS6 基因突变,这些家庭的先证者患有对口服铁治疗无反应的缺铁性贫血,部分对静脉铁治疗有反应。每位参与者提供一个含有 3 份血样的真空管(每份 2.5ml),并根据血液学、生化铁谱进行分析,然后进行 PCR 基因分型。在所有先证者和家族成员中扩增、测序和分析 TMPRSS6 基因。使用 SPSS 和 SHEsis 软件进行统计分析。这些家庭中存在少数功能突变(p.W73X、p.E523K 和 p.V736A)。家族 6 的先证者在初次就诊时表现出许多血液学异常,包括正细胞性贫血伴有低 Hb、正常 MCV、低血清铁、低血清铁蛋白和正常 TIBC。虽然仅在 2 个家庭中发现 p.W73X 变体,而 p.V736A 变体在所有检查的沙特 IRIDA 家庭中均发现。考虑到这些 6 个病例的证据,在沙特阿拉伯大量的 IRIDA 患者中进行未来的基因型-表型相关性研究,除了增加对 TMPRSS6 功能在发育过程中的认识以及调节 TMPRSS6 的因素及其对体内铁水平的影响外,对患者管理也非常有意义。