| Literature DB >> 33954879 |
Saul Oswaldo Lugo-Reyes1, Nina Pastor2, Edith González-Serrano1, Marco Antonio Yamazaki-Nakashimada1, Selma Scheffler-Mendoza1, Laura Berron-Ruiz1, Guillermo Wakida1, Maria Enriqueta Nuñez-Nuñez3, Ana Paola Macias-Robles4, Aide Tamara Staines-Boone5, Edna Venegas-Montoya5, Carmen Alaez-Verson6, Carolina Molina-Garay6, Luis Leonardo Flores-Lagunes6, Karol Carrillo-Sanchez6, Julie Niemela7, Sergio D Rosenzweig7, Paul Gaytan8, Jorge A Yañez8, Ivan Martinez-Duncker2, Luigi D Notarangelo7, Sara Espinosa-Padilla9, Mario Ernesto Cruz-Munoz10.
Abstract
Mutations in recombinase activating genes 1 and 2 (RAG1/2) result in human severe combined immunodeficiency (SCID). The products of these genes are essential for V(D)J rearrangement of the antigen receptors during lymphocyte development. Mutations resulting in null-recombination activity in RAG1 or RAG2 are associated with the most severe clinical and immunological phenotypes, whereas patients with hypomorphic mutations may develop leaky SCID, including Omenn syndrome (OS). A group of previously unrecognized clinical phenotypes associated with granulomata and/or autoimmunity have been described as a consequence of hypomorphic mutations. Here, we present six patients from unrelated families with missense variants in RAG1 or RAG2. Phenotypes observed in these patients ranged from OS to severe mycobacterial infections and granulomatous disease. Moreover, we report the first evidence of two variants that had not been associated with immunodeficiency. This study represents the first case series of RAG1- or RAG2-deficient patients from Mexico and Latin America.Entities:
Keywords: Omenn syndrome; Primary immunodeficiencies; RAG1/2; SCID; T lymphocytes
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Year: 2021 PMID: 33954879 PMCID: PMC8319791 DOI: 10.1007/s10875-021-01052-0
Source DB: PubMed Journal: J Clin Immunol ISSN: 0271-9142 Impact factor: 8.317