Identification of a novel nonsense variant in FYCO1 gene associated with infantile cataract and cortical atrophy

Ophthalmic Genet. 2021 Dec;42(6):744-746. doi: 10.1080/13816810.2021.1955277. Epub 2021 Jul 20.

Abstract

Introduction: Cataract is a major condition characterized by ocular lens opacification, resulting from alteration in the lens architecture, lens proteins or both. It is responsible for about one-third of infants' blindness worldwide. Variants in the FYCO1 gene have been associated with autosomal recessive infantile cataract.

Material and methods: We conducted whole exome sequencing (WES) in a nine months old male patient who was referred for genetic investigation because of infantile cataract. WES analysis revealed the presence of a homozygous pathogenic variant (c.2365C>T) in exon 8 of the FYCO1 gene.

Results and discussion: This is the first report on a Lebanese infant with infantile cataract and cortical atrophy which was not previously reported, resulting from a novel homozygous FYCO1 variant; thus expanding the clinical phenotypic spectrum of FYCO1 involvement.

Keywords: FYCO1 gene; Infantile cataract; Lebanese; cortical atrophy; whole exome sequencing.

Publication types

  • Case Reports

MeSH terms

  • Atrophy
  • Cataract / congenital
  • Cataract / diagnosis
  • Cataract / genetics*
  • Codon, Nonsense / genetics*
  • Consanguinity
  • Exome Sequencing
  • Exons / genetics
  • Genes, Recessive
  • Homozygote
  • Humans
  • Infant
  • Lens Cortex, Crystalline / pathology*
  • Male
  • Microtubule-Associated Proteins / genetics*
  • Mutation*
  • Pedigree
  • Polymerase Chain Reaction

Substances

  • Codon, Nonsense
  • FYCO1 protein, human
  • Microtubule-Associated Proteins