Your browser doesn't support javascript.
loading
Agammaglobulinemia associated to nasal polyposis due to a hypomorphic RAG1 mutation in a 12 years old boy.
Cifaldi, Cristina; Scarselli, Alessia; Petricone, Davide; Di Cesare, Silvia; Chiriaco, Maria; Claps, Alessia; Rossi, Paolo; Calzoni, Enrica; Yamazaki, Yasuhiro; Notarangelo, Luigi Daniele; Di Matteo, Gigliola; Cancrini, Caterina; Finocchi, Andrea.
Afiliación
  • Cifaldi C; Department of Systems Medicine, "University of Rome Tor Vergata", Rome, Italy.
  • Scarselli A; University Department of Pediatrics, Unit of Immune and Infectious Diseases, Childrens' Hospital Bambino Gesù, Italy.
  • Petricone D; Department of Systems Medicine, "University of Rome Tor Vergata", Rome, Italy.
  • Di Cesare S; University Department of Pediatrics, Unit of Immune and Infectious Diseases, Childrens' Hospital Bambino Gesù, Italy.
  • Chiriaco M; University Department of Pediatrics, Unit of Immune and Infectious Diseases, Childrens' Hospital Bambino Gesù, Italy.
  • Claps A; University Department of Pediatrics, Unit of Immune and Infectious Diseases, Childrens' Hospital Bambino Gesù, Italy.
  • Rossi P; Department of Systems Medicine, "University of Rome Tor Vergata", Rome, Italy; University Department of Pediatrics, Unit of Immune and Infectious Diseases, Childrens' Hospital Bambino Gesù, Italy.
  • Calzoni E; Division of Immunology, Boston Children's Hospital, Boston, MA, USA.
  • Yamazaki Y; Division of Immunology, Boston Children's Hospital, Boston, MA, USA.
  • Notarangelo LD; Division of Immunology, Boston Children's Hospital, Boston, MA, USA; Harvard Medical School, Boston, MA, USA.
  • Di Matteo G; Department of Systems Medicine, "University of Rome Tor Vergata", Rome, Italy.
  • Cancrini C; Department of Systems Medicine, "University of Rome Tor Vergata", Rome, Italy; University Department of Pediatrics, Unit of Immune and Infectious Diseases, Childrens' Hospital Bambino Gesù, Italy.
  • Finocchi A; Department of Systems Medicine, "University of Rome Tor Vergata", Rome, Italy; University Department of Pediatrics, Unit of Immune and Infectious Diseases, Childrens' Hospital Bambino Gesù, Italy. Electronic address: andrea.finocchi@uniroma2.it.
Clin Immunol ; 173: 121-123, 2016 Dec.
Article en En | MEDLINE | ID: mdl-27713031
Recombination-activating gene (RAG) 1 and 2 mutations in humans cause T- B- NK+ SCID and Omenn syndrome, but milder phenotypes associated with residual protein activity have been recently described. We report a male patient with a diagnosis of common variable immunodeficiency (CVID) born from non-consanguineous parents, whose immunological phenotype was characterized by severe reduction of B cells and agammaglobulinemia for which several candidate genes were excluded by targeted Sanger sequencing. Next Generation Sequencing revealed two compound heterozygous mutations in the RAG1 gene: the previously described p.R624H, and the novel p.Y728H mutation, as well as the known polymorphism p.H249R. This case reinforces the notion of large phenotypic spectrum in RAG deficiency and opens questions on the management and follow-up of these patients.
Asunto(s)
Palabras clave

Texto completo: 1 Banco de datos: MEDLINE Asunto principal: Pólipos Nasales / Inmunodeficiencia Variable Común / Proteínas de Homeodominio / Agammaglobulinemia Tipo de estudio: Risk_factors_studies Límite: Child / Humans / Male Idioma: En Revista: Clin Immunol Asunto de la revista: ALERGIA E IMUNOLOGIA Año: 2016 Tipo del documento: Article País de afiliación: Italia

Texto completo: 1 Banco de datos: MEDLINE Asunto principal: Pólipos Nasales / Inmunodeficiencia Variable Común / Proteínas de Homeodominio / Agammaglobulinemia Tipo de estudio: Risk_factors_studies Límite: Child / Humans / Male Idioma: En Revista: Clin Immunol Asunto de la revista: ALERGIA E IMUNOLOGIA Año: 2016 Tipo del documento: Article País de afiliación: Italia