| Literature DB >> 33386993 |
Smrithi Salian1, Hind Benkerroum1, Thi Tuyet Mai Nguyen1, Sheela Nampoothiri2, Taroh Kinoshita3, Têmis Maria Félix4, Fiona Stewart5, Sanjay M Sisodiya6, Yoshiko Murakami3, Philippe M Campeau7.
Abstract
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.Entities:
Year: 2021 PMID: 33386993 DOI: 10.1007/s00439-020-02251-2
Source DB: PubMed Journal: Hum Genet ISSN: 0340-6717 Impact factor: 4.132