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Abstract

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.

Details

Title
Germline copy number variants in RUNX1: An updated case report and a decade-old red herring
Author
Deuitch, Natalie T. 1 ; Kajdic, Amra 1 ; Bresciani, Erica 1 ; Horwitz, Marshall S. 2 ; Scott, Hamish S. 3 ; Craft, Katie 1 ; Chong, Shawn 1 ; Young, David J. 4 ; Godley, Lucy A. 5 ; Liu, Paul P. 1 

 NIH, Oncogenesis and Development Section, Translational and Functional Genomics Branch, National Human Genome Research Institute, Bethesda, USA (GRID:grid.280128.1) (ISNI:0000 0001 2233 9230) 
 University of Washington School of Medicine, Laboratory Medicine & Pathology, Seattle, USA (GRID:grid.34477.33) (ISNI:0000000122986657) 
 SA Pathology and University of South Australia, Centre for Cancer Biology, Adelaide, Australia (GRID:grid.470344.0) (ISNI:0000 0004 0450 082X) 
 NIH, Translational Stem Cell Biology Branch, National Heart Lung and Blood Institute, Bethesda, USA (GRID:grid.279885.9) (ISNI:0000 0001 2293 4638) 
 Northwestern University, Robert H. Lurie Comprehensive Cancer Center, Division of Hematology/Oncology, Evanston, USA (GRID:grid.16753.36) (ISNI:0000 0001 2299 3507) 
Pages
18
Publication year
2025
Publication date
Dec 2025
Publisher
Nature Publishing Group
e-ISSN
27319377
Source type
Scholarly Journal
Language of publication
English
ProQuest document ID
3225865413
Copyright
Copyright Nature Publishing Group Dec 2025