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PIGF deficiency causes a phenotype overlapping with DOORS syndrome.

Smrithi SalianHind BenkerroumThi Tuyet Mai NguyenSheela NampoothiriTaroh KinoshitaTêmis Maria FélixFiona StewartSanjay M SisodiyaYoshiko MurakamiPhilippe M Campeau
Published in: Human genetics (2021)
DOORS syndrome is characterized by deafness, onychodystrophy, osteodystrophy, intellectual disability, and seizures. In this study, we report two unrelated individuals with DOORS syndrome without deafness. Exome sequencing revealed a homozygous missense variant in PIGF (NM_173074.3:c.515C>G, p.Pro172Arg) in both. We demonstrate impaired glycosylphosphatidylinositol (GPI) biosynthesis through flow cytometry analysis. We thus describe the causal role of a novel disease gene, PIGF, in DOORS syndrome and highlight the overlap between this condition and GPI deficiency disorders. For each gene implicated in DOORS syndrome and/or inherited GPI deficiencies, there is considerable clinical variability so a high index of suspicion is warranted even though not all features are noted.
Keyphrases
  • intellectual disability
  • case report
  • flow cytometry
  • copy number
  • autism spectrum disorder
  • single cell
  • photodynamic therapy
  • transcription factor