A brother and sister with intellectual disability and characteristic neuroimaging findings

Eur J Paediatr Neurol. 2018 Sep;22(5):866-869. doi: 10.1016/j.ejpn.2018.06.005. Epub 2018 Jun 25.

Abstract

Leukoencephalopathy with brain calcifications and cysts (LCC) is a genetic white matter disorder, which involves the brain small blood vessels. In the absence of extra-neurological symptoms, LCC has a pathognomonic radiological phenotype. Recently, biallelic mutations in the SNORD118 gene, which is a non-protein coding gene, were discovered to cause LCC. We present here two siblings with developmental delay and a typical MRI pattern, who were diagnosed with LCC. The mutations in the SNORD118 gene were initially missed with whole exome sequencing (WES), but recognition of the MRI patterns of both children raised the suspicion of LCC and led to a genetically proven diagnosis after re-evaluation of the WES data.

Keywords: Developmental disorders; Genetics; Leukodystrophy; Ribosomopathy.

Publication types

  • Case Reports

MeSH terms

  • Brain / diagnostic imaging
  • Calcinosis / diagnostic imaging*
  • Calcinosis / genetics*
  • Central Nervous System Cysts / diagnostic imaging*
  • Central Nervous System Cysts / genetics*
  • Child
  • Child, Preschool
  • Developmental Disabilities / genetics
  • Female
  • Humans
  • Intellectual Disability / genetics
  • Leukoencephalopathies / diagnostic imaging*
  • Leukoencephalopathies / genetics*
  • Magnetic Resonance Imaging
  • Male
  • Mutation
  • Neuroimaging
  • RNA, Small Nucleolar / genetics
  • Siblings

Substances

  • Leukoencephalopathy Brain Calcifications and Cysts
  • RNA, Small Nucleolar
  • SNORD118 RNA, human