Literature DB >> 33954879

Clinical Manifestations, Mutational Analysis, and Immunological Phenotype in Patients with RAG1/2 Mutations: First Cases Series from Mexico and Description of Two Novel Mutations.

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.
© 2021. The Author(s), under exclusive licence to Springer Science+Business Media, LLC, part of Springer Nature.

Entities:  

Keywords:  Omenn syndrome; Primary immunodeficiencies; RAG1/2; SCID; T lymphocytes

Mesh:

Substances:

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


  68 in total

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Journal:  Nucleic Acids Res       Date:  2000-01-01       Impact factor: 16.971

Review 2.  Severe combined immunodeficiency (SCID): from molecular basis to clinical management.

Authors:  Ivonne Sponzilli; Luigi D Notarangelo
Journal:  Acta Biomed       Date:  2011-04

3.  Dominant-negative IKZF1 mutations cause a T, B, and myeloid cell combined immunodeficiency.

Authors:  David Boutboul; Hye Sun Kuehn; Zoé Van de Wyngaert; Julie E Niemela; Isabelle Callebaut; Jennifer Stoddard; Christelle Lenoir; Vincent Barlogis; Catherine Farnarier; Frédéric Vely; Nao Yoshida; Seiji Kojima; Hirokazu Kanegane; Akihiro Hoshino; Fabian Hauck; Ludovic Lhermitte; Vahid Asnafi; Philip Roehrs; Shaoying Chen; James W Verbsky; Katherine R Calvo; Ammar Husami; Kejian Zhang; Joseph Roberts; David Amrol; John Sleaseman; Amy P Hsu; Steven M Holland; Rebecca Marsh; Alain Fischer; Thomas A Fleisher; Capucine Picard; Sylvain Latour; Sergio D Rosenzweig
Journal:  J Clin Invest       Date:  2018-06-11       Impact factor: 14.808

Review 4.  RAG-dependent primary immunodeficiencies.

Authors:  Cristina Sobacchi; Veronica Marrella; Francesca Rucci; Paolo Vezzoni; Anna Villa
Journal:  Hum Mutat       Date:  2006-12       Impact factor: 4.878

5.  ZincBind-the database of zinc binding sites.

Authors:  Sam M Ireland; Andrew C R Martin
Journal:  Database (Oxford)       Date:  2019-01-01       Impact factor: 3.451

6.  The immunophenotypic and immunogenotypic B-cell differentiation arrest in bone marrow of RAG-deficient SCID patients corresponds to residual recombination activities of mutated RAG proteins.

Authors:  Jeroen G Noordzij; Sandra de Bruin-Versteeg; Nicole S Verkaik; Jaak M J J Vossen; Ronald de Groot; Ewa Bernatowska; Anton W Langerak; Dik C van Gent; Jacques J M van Dongen
Journal:  Blood       Date:  2002-09-15       Impact factor: 22.113

Review 7.  Human RAG mutations: biochemistry and clinical implications.

Authors:  Luigi D Notarangelo; Min-Sung Kim; Jolan E Walter; Yu Nee Lee
Journal:  Nat Rev Immunol       Date:  2016-03-21       Impact factor: 53.106

8.  SCID patients with ARTEMIS vs RAG deficiencies following HCT: increased risk of late toxicity in ARTEMIS-deficient SCID.

Authors:  Catharina Schuetz; Benedicte Neven; Christopher C Dvorak; Sandrine Leroy; Markus J Ege; Ulrich Pannicke; Klaus Schwarz; Ansgar S Schulz; Manfred Hoenig; Monika Sparber-Sauer; Susanne A Gatz; Christian Denzer; Stephane Blanche; Despina Moshous; Capucine Picard; Biljana N Horn; Jean-Pierre de Villartay; Marina Cavazzana; Klaus-Michael Debatin; Wilhelm Friedrich; Alain Fischer; Morton J Cowan
Journal:  Blood       Date:  2013-10-21       Impact factor: 22.113

9.  Collaboration of RAG2 with RAG1-like proteins during the evolution of V(D)J recombination.

Authors:  Lina Marcela Carmona; Sebastian D Fugmann; David G Schatz
Journal:  Genes Dev       Date:  2016-04-07       Impact factor: 11.361

10.  Human Inborn Errors of Immunity: 2019 Update on the Classification from the International Union of Immunological Societies Expert Committee.

Authors:  Stuart G Tangye; Waleed Al-Herz; Aziz Bousfiha; Talal Chatila; Charlotte Cunningham-Rundles; Amos Etzioni; Jose Luis Franco; Steven M Holland; Christoph Klein; Tomohiro Morio; Hans D Ochs; Eric Oksenhendler; Capucine Picard; Jennifer Puck; Troy R Torgerson; Jean-Laurent Casanova; Kathleen E Sullivan
Journal:  J Clin Immunol       Date:  2020-01-17       Impact factor: 8.317

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