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非综合征性先天性铁粒幼细胞性贫血;15 例印度患者的表型和基因型。

Non-syndromic congenital sideroblastic anaemia; phenotype, and genotype of 15 Indian patients.

机构信息

Department of Haematogenetics, ICMR-National Institute of Immunohematology, Indian Council of Medical Research, 13th Floor, NMS Building, King Edward Memorial (KEM.) Hospital Campus, Parel, Mumbai, 400012, India.

Pediatric Hematologist Oncologist, KLE'S Dr Prabhakar Kore Hospital, Belagavi, Karnataka, India.

出版信息

Ann Hematol. 2024 Oct;103(10):3987-3998. doi: 10.1007/s00277-024-05969-5. Epub 2024 Aug 28.

Abstract

Sideroblastic anaemias are a diverse group of congenital and acquired bone marrow failure disorders marked by the presence of ring sideroblasts, ineffective erythropoiesis, and systemic iron overload. Congenital Sideroblastic anaemia (CSA) is mainly caused by gene mutations associated with heme synthesis, iron-sulfur [Fe-S] cluster, and mitochondrial protein synthesis pathways. The most prevalent form of CSA is caused by mutations in the erythroid-specific -amino levulinate synthase (ALAS2) gene, which encodes the first enzyme in the heme synthesis pathway in red blood cells. The second most prevalent form of CSA is caused by a mutation in the Solute carrier family 25 member 38 (SLC25A38) gene, which codes for an erythroid-specific protein of the inner mitochondrial membrane. Additionally, 15-20 genes are altogether associated with CSA. In this study, we aim to identify the CSA patients, understand their genetics and establish genotype-phenotype correlation. We have identified fifteen cases of CSA using our targeted NGS (t-NGS) panel. The major clinical findings in our cohort were microcytic anaemia, ring sideroblasts, and dyserythropoiesis in the bone marrow. Currently, two patients are responsive to pyridoxine, while the rest are on blood transfusion support. We have identified ten variants in three different genes of CSA (ALAS2, SLC25A38 & HSPA9). Five patients harbour four hemizygous variants- p.Ala282Ser, p.Arg170Cys, p.Arg204Gln and exon 2 duplication in the ALAS2 gene. In seven patients, we have identified three homozygous mutations - p.Pro190Arg, p.Arg187Gln and p.Arg134Cys in the SLC25A38 gene. These mutations have been predominantly identified in the European population. Three patients revealed three heterozygous variants p. Thr463Ile, D326Tyr, and Arg284Trp in the HSPA9 gene. PyMoL was used to evaluate the functional effects of these variations and understand their effect on the structure of the protein. We believe that by combining a bone marrow examination with genetic sequencing, CSA patients can acquire a definitive diagnosis.

摘要

铁幼粒细胞性难治性贫血是一组先天性和获得性骨髓衰竭疾病,其特征为存在环形铁幼粒细胞、无效红细胞生成和全身铁过载。先天性铁幼粒细胞性难治性贫血(CSA)主要由与血红素合成、铁-硫 [Fe-S] 簇和线粒体蛋白合成途径相关的基因突变引起。最常见的 CSA 形式是由红细胞特异性 -氨基酮戊酸合酶(ALAS2)基因突变引起的,该基因编码红细胞中血红素合成途径的第一个酶。第二种最常见的 CSA 形式是由溶质载体家族 25 成员 38(SLC25A38)基因突变引起的,该基因编码线粒体内膜的一种红细胞特异性蛋白。此外,总共 15-20 个基因与 CSA 相关。在这项研究中,我们旨在鉴定 CSA 患者,了解他们的遗传学并建立基因型-表型相关性。我们使用靶向 NGS(t-NGS)面板鉴定了 15 例 CSA。我们队列的主要临床发现是小细胞性贫血、环形铁幼粒细胞和骨髓中的红细胞发育异常。目前,有两名患者对吡哆醇有反应,而其余患者则需要输血支持。我们在 CSA 的三个不同基因(ALAS2、SLC25A38 和 HSPA9)中鉴定了 10 个变体。五名患者携带四个半合子变体- ALAS2 基因中的 p.Ala282Ser、p.Arg170Cys、p.Arg204Gln 和外显子 2 重复。在七名患者中,我们鉴定了三个纯合突变- SLC25A38 基因中的 p.Pro190Arg、p.Arg187Gln 和 p.Arg134Cys。这些突变主要在欧洲人群中发现。三名患者在 HSPA9 基因中显示三个杂合变体 p.Thr463Ile、D326Tyr 和 Arg284Trp。PyMoL 用于评估这些变异的功能影响,并了解它们对蛋白质结构的影响。我们相信,通过将骨髓检查与基因测序相结合,CSA 患者可以获得明确的诊断。

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