{"title":"反复鼻衄和鼻腔血管畸形患者的 RASGRP2 基因剪接突变。","authors":"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","doi":"10.1080/09537104.2024.2425664","DOIUrl":null,"url":null,"abstract":"<p><p>Platelet type bleeding disorder-18 (BDPLT18) caused by mutations of Ras guanyl releasing protein 2 (<i>RASGRP2</i>) is a relatively rare, new autosomal recessive disorder. Here, we reported a splice mutation in <i>RASGRP2</i> 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 <i>RASGRP2</i> 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.</p>","PeriodicalId":20268,"journal":{"name":"Platelets","volume":"35 1","pages":"2425664"},"PeriodicalIF":2.5000,"publicationDate":"2024-12-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":"0","resultStr":"{\"title\":\"A splice mutation in <i>RASGRP2</i> gene in the patient with recurrent epistaxis and nasal vascular malformation.\",\"authors\":\"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\",\"doi\":\"10.1080/09537104.2024.2425664\",\"DOIUrl\":null,\"url\":null,\"abstract\":\"<p><p>Platelet type bleeding disorder-18 (BDPLT18) caused by mutations of Ras guanyl releasing protein 2 (<i>RASGRP2</i>) is a relatively rare, new autosomal recessive disorder. Here, we reported a splice mutation in <i>RASGRP2</i> 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 <i>RASGRP2</i> 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.</p>\",\"PeriodicalId\":20268,\"journal\":{\"name\":\"Platelets\",\"volume\":\"35 1\",\"pages\":\"2425664\"},\"PeriodicalIF\":2.5000,\"publicationDate\":\"2024-12-01\",\"publicationTypes\":\"Journal Article\",\"fieldsOfStudy\":null,\"isOpenAccess\":false,\"openAccessPdf\":\"\",\"citationCount\":\"0\",\"resultStr\":null,\"platform\":\"Semanticscholar\",\"paperid\":null,\"PeriodicalName\":\"Platelets\",\"FirstCategoryId\":\"3\",\"ListUrlMain\":\"https://doi.org/10.1080/09537104.2024.2425664\",\"RegionNum\":3,\"RegionCategory\":\"医学\",\"ArticlePicture\":[],\"TitleCN\":null,\"AbstractTextCN\":null,\"PMCID\":null,\"EPubDate\":\"2024/11/8 0:00:00\",\"PubModel\":\"Epub\",\"JCR\":\"Q3\",\"JCRName\":\"CELL BIOLOGY\",\"Score\":null,\"Total\":0}","platform":"Semanticscholar","paperid":null,"PeriodicalName":"Platelets","FirstCategoryId":"3","ListUrlMain":"https://doi.org/10.1080/09537104.2024.2425664","RegionNum":3,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":null,"EPubDate":"2024/11/8 0:00:00","PubModel":"Epub","JCR":"Q3","JCRName":"CELL BIOLOGY","Score":null,"Total":0}
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.
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.