Diagnosis of GATA2 Deficiency in a Young Woman with Hemophagocytic Lymphohistiocytosis Triggered by Acute Systemic Cytomegalovirus Infection.
Journal
The American journal of case reports
ISSN: 1941-5923
Titre abrégé: Am J Case Rep
Pays: United States
ID NLM: 101489566
Informations de publication
Date de publication:
08 Mar 2021
08 Mar 2021
Historique:
entrez:
8
3
2021
pubmed:
9
3
2021
medline:
11
3
2021
Statut:
epublish
Résumé
BACKGROUND Hemophagocytic lymphohistiocytosis (HLH) is a life-threatening disease characterized by an intense immunologic response that results in multiorgan dysfunction. It typically manifests as a result of a familial genetic immunodeficiency disorder or secondary to a trigger such as an infection, malignancy, or autoimmune disease. The major factors involved in the development of the disease are an individual's genetic propensity to develop HLH, such as rare associated mutations, or inflammatory processes that trigger the immune system to go haywire. CASE REPORT Before the COVID-19 pandemic, a 22-year-old woman with a history of congenital absence of the right kidney, right-sided hearing loss, and leukopenia presented with a 3-week history of generalized malaise, fever, chest pain, cough, and shortness of breath. She developed an acute systemic cytomegalovirus infection further complicated by HLH. Based on her history and clinical course, an underlying primary immunodeficiency was suspected. An immunodeficiency gene panel revealed a monoallelic mutation in GATA2, a gene that encodes zinc-transcription factors responsible for the regulation of hematopoiesis. CONCLUSIONS GATA2 deficiency encompasses a large variety of mutations in the GATA2 gene and leads to disorders associated with hematologic and immunologic manifestations of monocytopenia and B-, and natural killer-cell deficiency. Over time, affected individuals are at high risk of developing life-threatening infections and serious hematologic complications, such as myelodysplastic syndromes and/or leukemias. We aimed to illustrate the importance of identifying an underlying genetic disorder associated with secondary HLH to help guide acute and long-term management.
Identifiants
pubmed: 33684095
pii: 927087
doi: 10.12659/AJCR.927087
pmc: PMC7959100
doi:
Types de publication
Journal Article
Case Reports
Langues
eng
Sous-ensembles de citation
IM
Pagination
e927087Références
Eur J Microbiol Immunol (Bp). 2013 Mar;3(1):81-9
pubmed: 24265923
Medicine (Baltimore). 1988 Nov;67(6):369-88
pubmed: 3054418
Hematology Am Soc Hematol Educ Program. 2015;2015:183-9
pubmed: 26637719
Pediatr Blood Cancer. 2008 Jun;50(6):1227-35
pubmed: 18085676
Haematologica. 2015 Jul;100(7):978-88
pubmed: 26022711
Blood. 2019 Jun 6;133(23):2465-2477
pubmed: 30992265
J Infect Chemother. 2020 Feb;26(2):252-256
pubmed: 31350183
Biol Blood Marrow Transplant. 2018 Jun;24(6):1250-1259
pubmed: 29412158
Pediatr Blood Cancer. 2019 Nov;66(11):e27929
pubmed: 31339233
J Allergy Clin Immunol. 2016 Feb;137(2):638-40
pubmed: 26395816
Curr Opin Allergy Clin Immunol. 2015 Feb;15(1):104-9
pubmed: 25397911