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Severe combined immunodeficiency, microcephaly, growth retardation, sensitivity to ionizing radiation syndrome

MedGen UID:
929461
Concept ID:
C4303792
Disease or Syndrome
Synonyms: Cernunnos-XLF deficiency; NHEJ1 (non-homologous end joining factor) syndrome; Severe combined immunodeficiency due to Cernunnos protein deficiency; Severe Combined Immunodeficiency due to NHEJ1 Deficiency
SNOMED CT: Severe combined immunodeficiency, microcephaly, growth retardation, sensitivity to ionizing radiation syndrome (720853005); Cernunnos-XLF deficiency (720853005); Severe combined immunodeficiency due to Cernunnos protein deficiency (720853005); NHEJ1 (non-homologous end joining factor) syndrome (720853005)

Definition

A rare form of combined immunodeficiency with characteristics of microcephaly, growth retardation and T and B cell lymphopenia. Patients present in childhood with growth retardation, microcephaly, urogenital and bone malformations, dysmorphic features, including bird-like facial dysmorphism, and features of combined immunodeficiency. Some patients may also present with autoimmune cytopenia. This disease is caused by mutations in the NHEJ1 (or Cernunos) gene (2q35). The resulting defect of Cernunnos/XLF, a core protein of the non-homologous end-joining (NHEJ) pathway, affects the major mechanism of DNA double-strand break repair. Transmission is autosomal recessive. [from SNOMEDCT_US]

Term Hierarchy

CClinical test,  RResearch test,  OOMIM,  GGeneReviews,  VClinVar  
  • CROGVSevere combined immunodeficiency, microcephaly, growth retardation, sensitivity to ionizing radiation syndrome

Recent clinical studies

Etiology

Chrzanowska KH, Gregorek H, Dembowska-Bagińska B, Kalina MA, Digweed M
Orphanet J Rare Dis 2012 Feb 28;7:13. doi: 10.1186/1750-1172-7-13. PMID: 22373003Free PMC Article

Diagnosis

Chrzanowska KH, Gregorek H, Dembowska-Bagińska B, Kalina MA, Digweed M
Orphanet J Rare Dis 2012 Feb 28;7:13. doi: 10.1186/1750-1172-7-13. PMID: 22373003Free PMC Article
Rucci F, Notarangelo LD, Fazeli A, Patrizi L, Hickernell T, Paganini T, Coakley KM, Detre C, Keszei M, Walter JE, Feldman L, Cheng HL, Poliani PL, Wang JH, Balter BB, Recher M, Andersson EM, Zha S, Giliani S, Terhorst C, Alt FW, Yan CT
Proc Natl Acad Sci U S A 2010 Feb 16;107(7):3024-9. Epub 2010 Feb 1 doi: 10.1073/pnas.0914865107. PMID: 20133615Free PMC Article

Prognosis

Chrzanowska KH, Gregorek H, Dembowska-Bagińska B, Kalina MA, Digweed M
Orphanet J Rare Dis 2012 Feb 28;7:13. doi: 10.1186/1750-1172-7-13. PMID: 22373003Free PMC Article

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