Title | An ELF4 hypomorphic variant results in NK cell deficiency. |
Publication Type | Journal Article |
Year of Publication | 2022 |
Authors | Salinas, SAndrea, Mace, EM, Conte, MI, Park, CShik, Li, Y, Rosario-Sepulveda, JI, Mahapatra, S, Moore, EK, Hernandez, ER, Chinn, IK, Reed, AE, Lee, BJ, Frumovitz, A, Gibbs, RA, Posey, JE, Satter, LRForbes, Thatayatikom, A, Allenspach, EJ, Wensel, TG, Lupski, JR, H Lacorazza, D, Orange, JS |
Journal | JCI Insight |
Volume | 7 |
Issue | 23 |
Date Published | 2022 Dec 08 |
ISSN | 2379-3708 |
Keywords | Animals, DNA-Binding Proteins, Humans, Killer Cells, Natural, Mice, Transcription Factors |
Abstract | NK cell deficiencies (NKD) are a type of primary immune deficiency in which the major immunologic abnormality affects NK cell number, maturity, or function. Since NK cells contribute to immune defense against virally infected cells, patients with NKD experience higher susceptibility to chronic, recurrent, and fatal viral infections. An individual with recurrent viral infections and mild hypogammaglobulinemia was identified to have an X-linked damaging variant in the transcription factor gene ELF4. The variant does not decrease expression but disrupts ELF4 protein interactions and DNA binding, reducing transcriptional activation of target genes and selectively impairing ELF4 function. Corroborating previous murine models of ELF4 deficiency (Elf4-/-) and using a knockdown human NK cell line, we determined that ELF4 is necessary for normal NK cell development, terminal maturation, and function. Through characterization of the NK cells of the proband, expression of the proband's variant in Elf4-/- mouse hematopoietic precursor cells, and a human in vitro NK cell maturation model, we established this ELF4 variant as a potentially novel cause of NKD. |
DOI | 10.1172/jci.insight.155481 |
Alternate Journal | JCI Insight |
PubMed ID | 36477361 |
PubMed Central ID | PMC9746917 |
Grant List | R01 CA207086 / CA / NCI NIH HHS / United States R01 AI120989 / AI / NIAID NIH HHS / United States T32 GM007367 / GM / NIGMS NIH HHS / United States T32 GM008280 / GM / NIGMS NIH HHS / United States R56 AI067946 / AI / NIAID NIH HHS / United States R25 GM056929 / GM / NIGMS NIH HHS / United States R01 EY026545 / EY / NEI NIH HHS / United States S10 OD026845 / OD / NIH HHS / United States UM1 HG006542 / HG / NHGRI NIH HHS / United States R01 AI067946 / AI / NIAID NIH HHS / United States K08 HG008986 / HG / NHGRI NIH HHS / United States R01 AI137275 / AI / NIAID NIH HHS / United States T32 GM088129 / GM / NIGMS NIH HHS / United States U01 HG011758 / HG / NHGRI NIH HHS / United States |
An ELF4 hypomorphic variant results in NK cell deficiency.
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