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