Evidence map›Paper›PMID 33954879›Full record

ArticleJournal of clinical immunology2021

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-Reyes, Nina Pastor, Edith González-Serrano, Marco Antonio Yamazaki-Nakashimada, Selma Scheffler-Mendoza, Laura Berron-Ruiz, Guillermo Wakida, Maria Enriqueta Nuñez-Nuñez, Ana Paola Macias-Robles, Aide Tamara Staines-Boone and 13 more

Erratum issuedOpen access · greenAbstract readCase Reports
In one paragraph

Article in Journal of clinical immunology, 2021. The graph could read no effect estimate from its abstract, so it casts no vote on the map. An erratum has been issued. Cited by 3 papers.

0numbers the graph read from it
0cells of the map it votes in
3citing papers in PubMed
0.2field-weighted citation impact, top 56% of its field
1 · What the graph read from it

What it found

Each row is one number read from the abstract, on the scale the paper reported it, with its interval. Left of the dashed line favours the treatment, right favours the comparator. Under each row is the sentence it came from. New to these charts? A ten-minute tutorial.

The abstract states no effect estimate the extractor could read, or names no intervention and outcome on the map, so this paper lights no cell and moves no belief. It is still indexed, cited and linked below.

2 · The registry

The trial behind it

Trials whose registry record cites this paper, or whose number appears in the abstract. A trial that started after this paper was published is citing it as background, not reporting it.

Neither the registry nor the abstract names a trial number. If this is a trial report, that itself is worth knowing.

3 · Its place in the literature

Who cites it

3 citing papers in PubMed, 3 citations in OpenAlex.

  1. Article
  2. Review
  3. Article
4 · The record

Corrections and comments

5 · Who and what money

Authors and funding

23 authors at 6 institutions in 1 country.

Saul Oswaldo Lugo-Reyes *Laboratorio de Inmunodeficiencias, Instituto Nacional de Pediatría, Mexico City, Mexico.
Nina Pastor *Centro de Investigación en Dinámica Celular, Universidad Autónoma del Estado de Morelos, Cuernavaca, Mexico.
Edith González-SerranoLaboratorio de Inmunodeficiencias, Instituto Nacional de Pediatría, Mexico City, Mexico.
Marco Antonio Yamazaki-NakashimadaLaboratorio de Inmunodeficiencias, Instituto Nacional de Pediatría, Mexico City, Mexico.
Selma Scheffler-MendozaLaboratorio de Inmunodeficiencias, Instituto Nacional de Pediatría, Mexico City, Mexico.
Laura Berron-RuizLaboratorio de Inmunodeficiencias, Instituto Nacional de Pediatría, Mexico City, Mexico.
Guillermo WakidaLaboratorio de Inmunodeficiencias, Instituto Nacional de Pediatría, Mexico City, Mexico.
Maria Enriqueta Nuñez-NuñezHospital Civil de Guadalajara Dr. Juan I. Menchaca, Guadalajara, Mexico.
Ana Paola Macias-RoblesCentro Medico Nacional de Oriente IMSS, Mexico City, Mexico.
Aide Tamara Staines-BooneUnidad Médica de Alta Especialidad 25, Instituto Mexicano del Seguro Social, Mexico City, Mexico.
Edna Venegas-MontoyaUnidad Médica de Alta Especialidad 25, Instituto Mexicano del Seguro Social, Mexico City, Mexico.
Carmen Alaez-VersonInstituto Nacional de Medicina Genómica, Mexico City, Mexico.
Carolina Molina-GarayInstituto Nacional de Medicina Genómica, Mexico City, Mexico.
Luis Leonardo Flores-LagunesInstituto Nacional de Medicina Genómica, Mexico City, Mexico.
Karol Carrillo-SanchezInstituto Nacional de Medicina Genómica, Mexico City, Mexico.
Julie NiemelaLaboratory of Clinical Immunology and Microbiology, National Institute of Health, Mexico City, Mexico.
Sergio D RosenzweigLaboratory of Clinical Immunology and Microbiology, National Institute of Health, Mexico City, Mexico.
Paul GaytanInstituto de Biotecnología, Universidad Nacional Autónoma de México, Mexico City, Mexico.
Jorge A YañezInstituto de Biotecnología, Universidad Nacional Autónoma de México, Mexico City, Mexico.
Ivan Martinez-DunckerCentro de Investigación en Dinámica Celular, Universidad Autónoma del Estado de Morelos, Cuernavaca, Mexico.
Luigi D NotarangeloLaboratory of Clinical Immunology and Microbiology, National Institute of Health, Mexico City, Mexico.
Sara Espinosa-PadillaLaboratorio de Inmunodeficiencias, Instituto Nacional de Pediatría, Mexico City, Mexico. saraelvaespino@gmail.com.
Mario Ernesto Cruz-MunozFacultad de Medicina, Universidad Autónoma del Estado de Morelos, Cuernavaca, Mexico. mario.cruz@uaem.mx.ORCID 0000-0001-6851-708X
Instituto Nacional de Pediatria · MXNational Institute of Genomic Medicine · MXUniversidad Autónoma del Estado de Morelos · MXMexican Social Security Institute · MXUniversidad Nacional Autónoma de México · MXHospital Civil de Guadalajara · MX

Funding

Characterization of the cellular and molecular bases of inborn errors of immunityZIAAI001222 · NIAID · NATIONAL INSTITUTE OF ALLERGY AND INFECTIOUS DISEASES · PI NOTARANGELO, LUIGI · 2017 to 2025
$15.2M
Mutation Analysis Of Selected Lymphoid Immune DisordersZIACL010304 · CLC · CLINICAL CENTER · PI ROSENZWEIG, SERGIO · 2009 to 2025
$0k
Intramural NIH HHS ZIA AI001222
6 · The paper itself

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.

Indexed as

AdolescentChildDNA-Binding ProteinsFemaleHomeodomain ProteinsHumansImmunologic Deficiency SyndromesInfantLymphocytesMaleMexicoMutationNuclear ProteinsPhenotypeDNA-Binding ProteinsHomeodomain ProteinsNuclear ProteinsRAG-1 proteinRAG2 protein, humanOmenn syndromePrimary immunodeficienciesRAG1/2SCIDT lymphocytes

Identifiers

PMID33954879
PMCPMC8319791
OpenAlexW3131459613

What OpenQuestion holds

Textmetadata
LicenceTDM
Read underepoch 390

Registered trials

None linked

Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the OpenQuestion graph.