| Literature DB >> 34645992 |
Oliver Murch1, Vani Jain2, Andreas Benneche3, Kay Metcalfe4, Emma Hobson5, Katrina Prescott5, Kate Chandler4, Neeti Ghali6, Jenny Carmichael7, Nicola C Foulds8, Julie Paulsen9, Marie F Smeland10, Siren Berland3, Andrew E Fry2,11.
Abstract
White-Sutton syndrome (WHSUS) is a neurodevelopmental disorder caused by heterozygous loss-of-function variants in POGZ. Through the Deciphering Developmental Disorders study and clinical testing, we identified 12 individuals from 10 families with pathogenic or likely pathogenic variants in POGZ (eight de novo and two inherited). Most individuals had delayed development and/or intellectual disability. We analyzed the clinical findings in our series and combined it with data from 89 previously reported individuals. The results demonstrate WHSUS is associated with variable developmental delay or intellectual disability, increased risk of obesity, visual defects, craniofacial dysmorphism, sensorineural hearing loss, feeding problems, seizures, and structural brain malformations. Our series includes further individuals with rod-cone dystrophy, cleft lip and palate, congenital diaphragmatic hernia, and duplicated renal drainage system, suggesting these are rare complications of WHSUS. In addition, we describe an individual with a novel, de novo missense variant in POGZ and features of WHSUS. Our work further delineates the phenotypic spectrum of WHSUS highlighting the variable severity of this disorder and the observation of familial pathogenic POGZ variants.Entities:
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Year: 2021 PMID: 34645992 PMCID: PMC8738758 DOI: 10.1038/s41431-021-00961-3
Source DB: PubMed Journal: Eur J Hum Genet ISSN: 1018-4813 Impact factor: 5.351