Frontiers in Immunology (Jan 2019)
Extreme Phenotypes With Identical Mutations: Two Patients With Same Non-sense NHEJ1 Homozygous Mutation
- Maria J. Recio,
- Maria J. Recio,
- Nerea Dominguez-Pinilla,
- Nerea Dominguez-Pinilla,
- Melina Soledad Perrig,
- Melina Soledad Perrig,
- Carmen Rodriguez Vigil-Iturrate,
- Nerea Salmón-Rodriguez,
- Nerea Salmón-Rodriguez,
- Nerea Salmón-Rodriguez,
- Cristina Martinez Faci,
- María J. Castro-Panete,
- Javier Blas-Espada,
- Javier Blas-Espada,
- Marta López-Nevado,
- Marta López-Nevado,
- Raquel Ruiz-Garcia,
- Raquel Ruiz-Garcia,
- Rebeca Chaparro-García,
- Luis M. Allende,
- Luis M. Allende,
- Luis Ignacio Gonzalez-Granado,
- Luis Ignacio Gonzalez-Granado,
- Luis Ignacio Gonzalez-Granado
Affiliations
- Maria J. Recio
- Department of Immunology, Ophthalmology and ENT, School of Medicine, Complutense University, 12 de Octubre Health Research Institute (imas12), Madrid, Spain
- Maria J. Recio
- Hospital 12 de Octubre Health Research Institute (imas12), Madrid, Spain
- Nerea Dominguez-Pinilla
- Hospital 12 de Octubre Health Research Institute (imas12), Madrid, Spain
- Nerea Dominguez-Pinilla
- Pediatric Hematology and Oncology Unit, University Hospital Virgen de la Salud, Toledo, Spain
- Melina Soledad Perrig
- Department of Immunology, Ophthalmology and ENT, School of Medicine, Complutense University, 12 de Octubre Health Research Institute (imas12), Madrid, Spain
- Melina Soledad Perrig
- Hospital 12 de Octubre Health Research Institute (imas12), Madrid, Spain
- Carmen Rodriguez Vigil-Iturrate
- Pediatric Hematology and Oncology Unit, University Hospital Miguel Servet, Zaragoza, Spain
- Nerea Salmón-Rodriguez
- Hospital 12 de Octubre Health Research Institute (imas12), Madrid, Spain
- Nerea Salmón-Rodriguez
- Immunodeficiencies Unit, Pediatrics, University Hospital 12 octubre, Madrid, Spain
- Nerea Salmón-Rodriguez
- Complutense University School of Medicine, Madrid, Spain
- Cristina Martinez Faci
- Pediatric Hematology and Oncology Unit, University Hospital Miguel Servet, Zaragoza, Spain
- María J. Castro-Panete
- Department of Immunology, University Hospital 12 Octubre, Madrid, Spain
- Javier Blas-Espada
- Hospital 12 de Octubre Health Research Institute (imas12), Madrid, Spain
- Javier Blas-Espada
- Department of Immunology, University Hospital 12 Octubre, Madrid, Spain
- Marta López-Nevado
- Hospital 12 de Octubre Health Research Institute (imas12), Madrid, Spain
- Marta López-Nevado
- Department of Immunology, University Hospital 12 Octubre, Madrid, Spain
- Raquel Ruiz-Garcia
- Hospital 12 de Octubre Health Research Institute (imas12), Madrid, Spain
- Raquel Ruiz-Garcia
- Department of Immunology, University Hospital 12 Octubre, Madrid, Spain
- Rebeca Chaparro-García
- Department of Immunology, Ophthalmology and ENT, School of Medicine, Complutense University, 12 de Octubre Health Research Institute (imas12), Madrid, Spain
- Luis M. Allende
- Hospital 12 de Octubre Health Research Institute (imas12), Madrid, Spain
- Luis M. Allende
- Department of Immunology, University Hospital 12 Octubre, Madrid, Spain
- Luis Ignacio Gonzalez-Granado
- Hospital 12 de Octubre Health Research Institute (imas12), Madrid, Spain
- Luis Ignacio Gonzalez-Granado
- Immunodeficiencies Unit, Pediatrics, University Hospital 12 octubre, Madrid, Spain
- Luis Ignacio Gonzalez-Granado
- Complutense University School of Medicine, Madrid, Spain
- DOI
- https://doi.org/10.3389/fimmu.2018.02959
- Journal volume & issue
-
Vol. 9
Abstract
Cernunnos/XLF deficiency is a rare primary immunodeficiency classified within the DNA repair defects. Patients present with severe growth retardation, microcephaly, lymphopenia and increased cellular sensitivity to ionizing radiation. Here, we describe two unrelated cases with the same non-sense mutation in the NHEJ1 gene showing significant differences in clinical presentation and immunological profile but a similar DNA repair defect.
Keywords
- XLF/Cernunnos
- NHEJ1 mutation
- DNA repair
- severe combined immunodeficiency
- lymphomagenesis
- radiosensitive SCID (RS-SCID)