A possible genotype-phenotype correlation in Ashkenazi-Jewish individuals with Aicardi-Goutières syndrome associated with SAMHD1 mutation

J Child Neurol. 2015 Mar;30(4):490-5. doi: 10.1177/0883073814549241. Epub 2014 Sep 22.

Abstract

Aicardi-Goutières syndrome is a genetic neurodegenerative disorder with clinical symptoms mimicking a congenital viral infection. Mutations in 6 genes are known to cause the disease: 3 prime repair exonuclease1, ribonucleases H2A, B, and C, SAM domain and HD domain 1, and most recently ADAR1. HD domain 1 mutations were previously reported in the Ashkenazi-Jewish community. We report an additional patient of Ashkenazi-Jewish descent and review the other 3 cases affected with Aicardi-Goutières syndrome due to SAM domain and HD domain 1 (SAMHD1) mutations described in Israel. We propose that there may be a phenotypic-genotypic correlation in accordance with the type of mutations inherited in the SAMHD1 genotype and suggest that Aicardi-Goutières syndrome may not be a rare disease in the Ashkenazi-Jewish population.

Keywords: Aicardi-Goutières; Ashkenazi-Jewish; FTT; irritability; microcephaly; seizures.

Publication types

  • Case Reports

MeSH terms

  • Autoimmune Diseases of the Nervous System / diagnostic imaging
  • Autoimmune Diseases of the Nervous System / genetics*
  • Autoimmune Diseases of the Nervous System / pathology
  • Brain / pathology
  • Cerebral Angiography
  • Child
  • Echoencephalography
  • Female
  • Genetic Association Studies
  • Humans
  • Infant
  • Jews / genetics*
  • Magnetic Resonance Imaging
  • Male
  • Monomeric GTP-Binding Proteins / genetics*
  • Mutation*
  • Nervous System Malformations / diagnostic imaging
  • Nervous System Malformations / genetics*
  • Nervous System Malformations / pathology
  • SAM Domain and HD Domain-Containing Protein 1
  • Tomography, X-Ray Computed

Substances

  • SAM Domain and HD Domain-Containing Protein 1
  • SAMHD1 protein, human
  • Monomeric GTP-Binding Proteins

Supplementary concepts

  • Aicardi-Goutieres syndrome