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PIGF deficiency causes a phenotype overlapping with DOORS syndrome.
Salian, Smrithi; Benkerroum, Hind; Nguyen, Thi Tuyet Mai; Nampoothiri, Sheela; Kinoshita, Taroh; Félix, Têmis Maria; Stewart, Fiona; Sisodiya, Sanjay M; Murakami, Yoshiko; Campeau, Philippe M.
Afiliación
  • Salian S; Department of Pediatrics, CHU Sainte-Justine Research Center, University of Montreal, 3175, Côte-Sainte-Catherine, Montreal, QC, H3T 1C5, Canada.
  • Benkerroum H; Department of Pediatrics, CHU Sainte-Justine Research Center, University of Montreal, 3175, Côte-Sainte-Catherine, Montreal, QC, H3T 1C5, Canada.
  • Nguyen TTM; Department of Pediatrics, CHU Sainte-Justine Research Center, University of Montreal, 3175, Côte-Sainte-Catherine, Montreal, QC, H3T 1C5, Canada.
  • Nampoothiri S; Pediatric Genetics, Amrita Institute of Medical Sciences and Research Centre (AIMS), Kochi, 682041, India.
  • Kinoshita T; Yabumoto Department of Intractable Disease Research, Research Institute for Microbial Diseases, Osaka University, Suita, Osaka, 565-0871, Japan.
  • Félix TM; Medical Genetics Service, Hospital de Clinicas de Porto Alegre, Porto Alegre, Brazil.
  • Stewart F; Northern Ireland Regional Genetics Centre, Belfast, BT97AB, Northern Ireland.
  • Sisodiya SM; NIHR University College London Hospitals Biomedical Research Centre, UCL Queen Square Institute of Neurology, Queen Square, London, WC1N 3BG, UK.
  • Murakami Y; Yabumoto Department of Intractable Disease Research, Research Institute for Microbial Diseases, Osaka University, Suita, Osaka, 565-0871, Japan.
  • Campeau PM; Department of Pediatrics, CHU Sainte-Justine Research Center, University of Montreal, 3175, Côte-Sainte-Catherine, Montreal, QC, H3T 1C5, Canada. p.campeau@umontreal.ca.
Hum Genet ; 140(6): 879-884, 2021 Jun.
Article en En | MEDLINE | ID: mdl-33386993
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.
Asunto(s)

Texto completo: 1 Colección: 01-internacional Banco de datos: MEDLINE Asunto principal: Convulsiones / Deformidades Congénitas de la Mano / Glicosilfosfatidilinositoles / Anomalías Craneofaciales / Mutación Missense / Pérdida Auditiva Sensorineural / Proteínas de la Membrana / Discapacidad Intelectual / Uñas Malformadas Tipo de estudio: Etiology_studies Límite: Adolescent / Animals / Female / Humans / Infant / Male Idioma: En Revista: Hum Genet Año: 2021 Tipo del documento: Article País de afiliación: Canadá

Texto completo: 1 Colección: 01-internacional Banco de datos: MEDLINE Asunto principal: Convulsiones / Deformidades Congénitas de la Mano / Glicosilfosfatidilinositoles / Anomalías Craneofaciales / Mutación Missense / Pérdida Auditiva Sensorineural / Proteínas de la Membrana / Discapacidad Intelectual / Uñas Malformadas Tipo de estudio: Etiology_studies Límite: Adolescent / Animals / Female / Humans / Infant / Male Idioma: En Revista: Hum Genet Año: 2021 Tipo del documento: Article País de afiliación: Canadá