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GATA2 deficiency: a protean disorder of hematopoiesis, lymphatics, and immunity
- 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.
- Subjects :
- Adult
Male
Adolescent
Immunology
Plenary Paper
Haploinsufficiency
Monocytopenia
Biochemistry
Lymphatic System
Young Adult
hemic and lymphatic diseases
Humans
Medicine
Primary lymphedema
Prospective Studies
Child
Genetic Association Studies
Immunodeficiency
Aged
GATA2 Deficiency
business.industry
Myelodysplastic syndromes
Immunologic Deficiency Syndromes
Infant
Cell Biology
Hematology
Middle Aged
Prognosis
medicine.disease
Hematopoiesis
MonoMAC
GATA2 Transcription Factor
Survival Rate
Leukemia, Myeloid, Acute
Lymphedema
Myelodysplastic Syndromes
Female
Lymphocytopenia
business
Subjects
Details
- ISSN :
- 15280020 and 00064971
- Volume :
- 123
- Database :
- OpenAIRE
- Journal :
- Blood
- Accession number :
- edsair.doi.dedup.....4a39ad32626bd7c8df8e25926f7135a3