Literature DB >> 34645992

Further delineation of the clinical spectrum of White-Sutton syndrome: 12 new individuals and a review of the literature.

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.
© 2021. The Author(s), under exclusive licence to European Society of Human Genetics.

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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


  1 in total

Review 1.  POGZ-related epilepsy: Case report and review of the literature.

Authors:  Alessandro Ferretti; Sabina Barresi; Marina Trivisano; Andrea Ciolfi; Maria L Dentici; Francesca C Radio; Federico Vigevano; Marco Tartaglia; Nicola Specchio
Journal:  Am J Med Genet A       Date:  2019-05-28       Impact factor: 2.578

  1 in total
  3 in total

1.  New year, new issue.

Authors:  Alisdair McNeill
Journal:  Eur J Hum Genet       Date:  2022-01       Impact factor: 4.246

2.  A novel, de novo intronic variant in POGZ causes White-Sutton syndrome.

Authors:  Ashanta Merriweather; David R Murdock; Jill A Rosenfeld; Hongzheng Dai; Shamika Ketkar; Lisa Emrick; Sarah Nicholas; Richard A Lewis; Carlos A Bacino; Daryl A Scott; Brendan Lee; Vernon Reid Sutton; Lorraine Potocki; Lindsay C Burrage
Journal:  Am J Med Genet A       Date:  2022-04-09       Impact factor: 2.578

3.  Genotype-Phenotype Comparison in POGZ-Related Neurodevelopmental Disorders by Using Clinical Scoring.

Authors:  Dóra Nagy; Sarah Verheyen; Kristen M Wigby; Artem Borovikov; Artem Sharkov; Valerie Slegesky; Austin Larson; Christina Fagerberg; Charlotte Brasch-Andersen; Maria Kibæk; Ingrid Bader; Rebecca Hernan; Frances A High; Wendy K Chung; Jolanda H Schieving; Jana Behunova; Mateja Smogavec; Franco Laccone; Martina Witsch-Baumgartner; Joachim Zobel; Hans-Christoph Duba; Denisa Weis
Journal:  Genes (Basel)       Date:  2022-01-15       Impact factor: 4.141

  3 in total

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