反复鼻衄和鼻腔血管畸形患者的 RASGRP2 基因剪接突变。

IF 2.5 3区 医学 Q3 CELL BIOLOGY
Platelets Pub Date : 2024-12-01 Epub Date: 2024-11-08 DOI:10.1080/09537104.2024.2425664
Zhong-Yu Shi, Qing-Ling Lei, Shao-Qin Duan, Yan Zhou, Ting-Ting Cui, Yun-Bi Lin, Chun-Hui Yang, Chun-Yan Song, Chun-Lian Fang, Xin Tian, Xian-Wen Zhang, Ti-Long Huang
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引用次数: 0

摘要

由Ras鸟苷释放蛋白2(RASGRP2)突变引起的血小板型出血障碍-18(BDPLT18)是一种相对罕见的新型常染色体隐性遗传疾病。在此,我们报告了一名反复鼻衄和鼻腔血管畸形患者的 RASGRP2 基因剪接突变。患者是一名 8 岁女孩,因经常反复发作严重鼻衄而导致贫血,需要每 2-3 个月定期输血。血液学检查显示中度贫血(血红蛋白:89 克/升),血小板数量、形态和血小板糖蛋白正常。该患者花生四烯酸和二磷酸腺苷诱导的血小板聚集明显减少。通过下一代测序,我们检测到 RASGRP2 基因中位于第 3 外显子的同源剪接变异(C.74-1 G>C)。有趣的是,在治疗顽固性鼻衄期间,我们通过经皮超选择性血管造影术发现了鼻腔血管畸形。我们的病例进一步证明,对于一些原因不明的出血性疾病,应进行基因检测。
本文章由计算机程序翻译,如有差异,请以英文原文为准。
A splice mutation in RASGRP2 gene in the patient with recurrent epistaxis and nasal vascular malformation.

Platelet type bleeding disorder-18 (BDPLT18) caused by mutations of Ras guanyl releasing protein 2 (RASGRP2) is a relatively rare, new autosomal recessive disorder. Here, we reported a splice mutation in RASGRP2 gene in the patient with recurrent epistaxis and nasal vascular malformation. The patient, an 8-year-old girl, suffered from anemia due to frequently severe recurrent epistaxis, requiring regular blood transfusions every 2-3 months. Hematological investigations showed moderate anemia (Hb: 89 g/L), normal platelet count, morphology, and platelet glycoproteins. Arachidonic acid and adenosine diphosphate induced platelet aggregation was markedly reduced in the patient. A homozygous splice variant (C.74-1 G>C) in RASGRP2 gene, located within the exon 3, was detected by next-generation sequencing. Interestingly, we identified nasal vascular malformation by percutaneous super-selective angiography during the treatment of an intractable epistaxis. Our case further support that genetic testing should be performed for some unexplained bleeding diseases.

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来源期刊
Platelets
Platelets 医学-细胞生物学
CiteScore
6.70
自引率
3.00%
发文量
79
审稿时长
1 months
期刊介绍: Platelets is an international, peer-reviewed journal covering all aspects of platelet- and megakaryocyte-related research. Platelets provides the opportunity for contributors and readers across scientific disciplines to engage with new information about blood platelets. The journal’s Methods section aims to improve standardization between laboratories and to help researchers replicate difficult methods. Research areas include: Platelet function Biochemistry Signal transduction Pharmacology and therapeutics Interaction with other cells in the blood vessel wall The contribution of platelets and platelet-derived products to health and disease The journal publishes original articles, fast-track articles, review articles, systematic reviews, methods papers, short communications, case reports, opinion articles, commentaries, gene of the issue, and letters to the editor. Platelets operates a single-blind peer review policy. Authors can choose to publish gold open access in this journal.
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