Natalie T Deuitch, Amra Kajdic, Erica Bresciani, Marshall S Horwitz, Hamish S Scott, Katie Craft, Shawn Chong, David J Young, Lucy A Godley, Paul P Liu
{"title":"RUNX1的生殖系拷贝数变异:一份最新的病例报告和十年来的转移注意力。","authors":"Natalie T Deuitch, Amra Kajdic, Erica Bresciani, Marshall S Horwitz, Hamish S Scott, Katie Craft, Shawn Chong, David J Young, Lucy A Godley, Paul P Liu","doi":"10.1038/s44276-024-00117-y","DOIUrl":null,"url":null,"abstract":"<p><p>Pathogenic/likely pathogenic (P/LP) germline variants in RUNX1 cause familial platelet disorder with associated myeloid malignancies (FPDMM), also known as RUNX1-Familial Platelet Disorder (RUNX1-FPD, or FPD), a condition characterized by qualitative and quantitative platelet defects and predisposition to hematopoietic malignancies. Here, we present follow up to a case of a woman with acute myeloid leukemia and lifelong thrombocytopenia which had previously been attributed to presumptive pathogenic (P) GATA2 missense variants. However, re-evaluation with updated molecular technology sensitive for detection of copy number variants (CNVs) led to the identification of a P deletion of exons 5-6 in RUNX1, which had been undetected when examined at first presentation. This case highlights the importance of comprehensive molecular evaluation and careful variant interpretation, especially regarding CNVs.</p>","PeriodicalId":519964,"journal":{"name":"BJC reports","volume":"3 1","pages":"18"},"PeriodicalIF":0.0000,"publicationDate":"2025-03-27","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11950377/pdf/","citationCount":"0","resultStr":"{\"title\":\"Germline copy number variants in RUNX1: An updated case report and a decade-old red herring.\",\"authors\":\"Natalie T Deuitch, Amra Kajdic, Erica Bresciani, Marshall S Horwitz, Hamish S Scott, Katie Craft, Shawn Chong, David J Young, Lucy A Godley, Paul P Liu\",\"doi\":\"10.1038/s44276-024-00117-y\",\"DOIUrl\":null,\"url\":null,\"abstract\":\"<p><p>Pathogenic/likely pathogenic (P/LP) germline variants in RUNX1 cause familial platelet disorder with associated myeloid malignancies (FPDMM), also known as RUNX1-Familial Platelet Disorder (RUNX1-FPD, or FPD), a condition characterized by qualitative and quantitative platelet defects and predisposition to hematopoietic malignancies. Here, we present follow up to a case of a woman with acute myeloid leukemia and lifelong thrombocytopenia which had previously been attributed to presumptive pathogenic (P) GATA2 missense variants. However, re-evaluation with updated molecular technology sensitive for detection of copy number variants (CNVs) led to the identification of a P deletion of exons 5-6 in RUNX1, which had been undetected when examined at first presentation. This case highlights the importance of comprehensive molecular evaluation and careful variant interpretation, especially regarding CNVs.</p>\",\"PeriodicalId\":519964,\"journal\":{\"name\":\"BJC reports\",\"volume\":\"3 1\",\"pages\":\"18\"},\"PeriodicalIF\":0.0000,\"publicationDate\":\"2025-03-27\",\"publicationTypes\":\"Journal Article\",\"fieldsOfStudy\":null,\"isOpenAccess\":false,\"openAccessPdf\":\"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11950377/pdf/\",\"citationCount\":\"0\",\"resultStr\":null,\"platform\":\"Semanticscholar\",\"paperid\":null,\"PeriodicalName\":\"BJC reports\",\"FirstCategoryId\":\"1085\",\"ListUrlMain\":\"https://doi.org/10.1038/s44276-024-00117-y\",\"RegionNum\":0,\"RegionCategory\":null,\"ArticlePicture\":[],\"TitleCN\":null,\"AbstractTextCN\":null,\"PMCID\":null,\"EPubDate\":\"\",\"PubModel\":\"\",\"JCR\":\"\",\"JCRName\":\"\",\"Score\":null,\"Total\":0}","platform":"Semanticscholar","paperid":null,"PeriodicalName":"BJC reports","FirstCategoryId":"1085","ListUrlMain":"https://doi.org/10.1038/s44276-024-00117-y","RegionNum":0,"RegionCategory":null,"ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":null,"EPubDate":"","PubModel":"","JCR":"","JCRName":"","Score":null,"Total":0}
Germline copy number variants in RUNX1: An updated case report and a decade-old red herring.
Pathogenic/likely pathogenic (P/LP) germline variants in RUNX1 cause familial platelet disorder with associated myeloid malignancies (FPDMM), also known as RUNX1-Familial Platelet Disorder (RUNX1-FPD, or FPD), a condition characterized by qualitative and quantitative platelet defects and predisposition to hematopoietic malignancies. Here, we present follow up to a case of a woman with acute myeloid leukemia and lifelong thrombocytopenia which had previously been attributed to presumptive pathogenic (P) GATA2 missense variants. However, re-evaluation with updated molecular technology sensitive for detection of copy number variants (CNVs) led to the identification of a P deletion of exons 5-6 in RUNX1, which had been undetected when examined at first presentation. This case highlights the importance of comprehensive molecular evaluation and careful variant interpretation, especially regarding CNVs.