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GATA2 deficiency: a protean disorder of hematopoiesis, lymphatics, and immunity

Authors :
Jordan S. Orange
Janine Daub
Adrian M. Zelazny
Emily M. Mace
Gulbu Uzel
Pamela A. Shaw
Kenneth N. Olivier
Amy P. Hsu
Dennis D. Hickstein
Christine M. Gould
Christine Spalding
Blanche P. Alter
Carmen C. Brewer
Edward W. Cowen
Wenjuan Gu
Jennifer Cuellar-Rodriguez
Neelam Giri
Alexandra F. Freeman
Christa S. Zerbe
Michael A. Spinner
Lauren A. Sanchez
Katherine R. Calvo
Steven M. Holland
Diane C. Arthur
Reginald J. Claypool
Source :
Blood. 123:809-821
Publication Year :
2014
Publisher :
American Society of Hematology, 2014.

Abstract

Haploinsufficiency of the hematopoietic transcription factor GATA2 underlies monocytopenia and mycobacterial infections; dendritic cell, monocyte, B, and natural killer (NK) lymphoid deficiency; familial myelodysplastic syndromes (MDS)/acute myeloid leukemia (AML); and Emberger syndrome (primary lymphedema with MDS). A comprehensive examination of the clinical features of GATA2 deficiency is currently lacking. We reviewed the medical records of 57 patients with GATA2 deficiency evaluated at the National Institutes of Health from January 1, 1992, to March 1, 2013, and categorized mutations as missense, null, or regulatory to identify genotype-phenotype associations. We identified a broad spectrum of disease: hematologic (MDS 84%, AML 14%, chronic myelomonocytic leukemia 8%), infectious (severe viral 70%, disseminated mycobacterial 53%, and invasive fungal infections 16%), pulmonary (diffusion 79% and ventilatory defects 63%, pulmonary alveolar proteinosis 18%, pulmonary arterial hypertension 9%), dermatologic (warts 53%, panniculitis 30%), neoplastic (human papillomavirus+ tumors 35%, Epstein-Barr virus+ tumors 4%), vascular/lymphatic (venous thrombosis 25%, lymphedema 11%), sensorineural hearing loss 76%, miscarriage 33%, and hypothyroidism 14%. Viral infections and lymphedema were more common in individuals with null mutations (P = .038 and P = .006, respectively). Monocytopenia, B, NK, and CD4 lymphocytopenia correlated with the presence of disease (P < .001). GATA2 deficiency unites susceptibility to MDS/AML, immunodeficiency, pulmonary disease, and vascular/lymphatic dysfunction. Early genetic diagnosis is critical to direct clinical management, preventive care, and family screening.

Details

ISSN :
15280020 and 00064971
Volume :
123
Database :
OpenAIRE
Journal :
Blood
Accession number :
edsair.doi.dedup.....4a39ad32626bd7c8df8e25926f7135a3