Journal of clinical immunology · 2015
IL2RG reversion event in a common lymphoid progenitor leads to delayed diagnosis and milder phenotype.
Клинический случайСтатьяПоддержка NIH: внешняя программаResearch Support, N.I.H., IntramuralНазвание и аннотация: Перевод пока не подготовлен
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Hsu Amy P, Pittaluga Stefania, Martinez Bianca, Rump Amy P, Raffeld Mark, Uzel Gulbu, Puck Jennifer M, Freeman Alexandra F, Holland Steven M
Аннотация
Severe combined immunodeficiency (SCID) is most frequently caused by mutations in the cytokine receptor common gamma chain, CD132, encoded by the X-linked gene, IL2RG. Most patients present in the first year of life with failure to thrive, severe, opportunistic infections and absence of CD3+ T cells. We present a patient with pediatric illness and a diagnosis of combined variable immune deficiency (CVID) who was diagnosed at age 23 with an inherited IL2RG mutation causing loss of signal transduction through CD132. His peripheral blood included CD3/CD4 and CD3/CD8 positive cells as well as low levels of CD19+ B cells containing a reversion to the wildtype IL2RG allele. The reversion, which was not present at birth, may account for his mild phenotype and late diagnosis.
Предметные рубрики MeSH
AdultAntigens, CD19B-LymphocytesBronchiectasisCD4-Positive T-LymphocytesCD8-Positive T-LymphocytesDelayed DiagnosisHumansInfant, NewbornInfectionsInterleukin Receptor Common gamma SubunitLymphoid Progenitor CellsMaleMutationPedigreePhenotypeSevere Combined ImmunodeficiencySignal TransductionSkinYoung Adult