Literature DB >> 28232779

De novo KAT6B Mutation Identified with Whole-Exome Sequencing in a Girl with Say-Barber/Biesecker/Young-Simpson Syndrome.

Malene Lundsgaard1, Vang Q Le2, Anja Ernst2, Hans C Laugaard-Jacobsen3, Kirsten Rasmussen1, Inge S Pedersen2, Michael B Petersen4.   

Abstract

Say-Barber/Biesecker/Young-Simpson syndrome (SBBYSS; OMIM 603736) is a rare syndrome with multiple congenital anomalies/malformations. The clinical diagnosis is usually based on a phenotype with a mask-like face and severe blepharophimosis and ptosis as well as other distinctive facial traits. We present a girl with dysmorphic features, an atrial septal defect, and developmental delay. Previous genetic testing (array-CGH, 22q11 deletion, PTPN11 and MLL2 mutation analysis) gave normal results. We performed whole-exome sequencing (WES) and identified a heterozygous nonsense mutation in the KAT6B gene, NM_001256468.1: c.4943C>G (p.S1648*). The mutation led to a premature stop codon and occurred de novo. KAT6B sequence variants have previously been identified in patients with SBBYSS, and the phenotype of the girl is similar to other patients diagnosed with SBBYSS. This case report provides additional evidence for the correlation between the KAT6B mutation and SBBYSS. If a patient is suspected of having a blepharophimosis syndrome or SBBYSS, we recommend sequencing the KAT6B gene. This is a further example showing that WES can assist diagnosis.

Entities:  

Keywords:  Blepharophimosis; Developmental delay; Epicanthus inversus; KAT6B; Ohdo syndrome; Whole-exome sequencing; Young-Simpson syndrome

Year:  2016        PMID: 28232779      PMCID: PMC5260604          DOI: 10.1159/000452258

Source DB:  PubMed          Journal:  Mol Syndromol        ISSN: 1661-8769


  11 in total

1.  Whole-exome-sequencing identifies mutations in histone acetyltransferase gene KAT6B in individuals with the Say-Barber-Biesecker variant of Ohdo syndrome.

Authors:  Jill Clayton-Smith; James O'Sullivan; Sarah Daly; Sanjeev Bhaskar; Ruth Day; Beverley Anderson; Anne K Voss; Tim Thomas; Leslie G Biesecker; Philip Smith; Alan Fryer; Kate E Chandler; Bronwyn Kerr; May Tassabehji; Sally-Ann Lynch; Malgorzata Krajewska-Walasek; Shane McKee; Janine Smith; Elizabeth Sweeney; Sahar Mansour; Shehla Mohammed; Dian Donnai; Graeme Black
Journal:  Am J Hum Genet       Date:  2011-11-11       Impact factor: 11.025

2.  A clinical and genetic study of the Say/Barber/Biesecker/Young-Simpson type of Ohdo syndrome.

Authors:  R Day; B Beckett; D Donnai; A Fryer; M Heidenblad; P Howard; B Kerr; S Mansour; U Maye; S McKee; S Mohammed; E Sweeney; M Tassabehji; B B A de Vries; J Clayton-Smith
Journal:  Clin Genet       Date:  2008-09-16       Impact factor: 4.438

3.  De novo mutations of the gene encoding the histone acetyltransferase KAT6B in two patients with Say-Barber/Biesecker/Young-Simpson syndrome.

Authors:  Katalin Szakszon; Carmelo Salpietro; Naseebullah Kakar; Alida C Knegt; Éva Oláh; Bruno Dallapiccola; Guntram Borck
Journal:  Am J Med Genet A       Date:  2013-02-22       Impact factor: 2.802

4.  An individual with blepharophimosis-ptosis-epicanthus inversus syndrome (BPES) and additional features expands the phenotype associated with mutations in KAT6B.

Authors:  Hung-Chun Yu; Elizabeth A Geiger; Livija Medne; Elaine H Zackai; Tamim H Shaikh
Journal:  Am J Med Genet A       Date:  2014-01-23       Impact factor: 2.802

5.  Blepharophimosis-mental retardation (BMR) syndromes: A proposed clinical classification of the so-called Ohdo syndrome, and delineation of two new BMR syndromes, one X-linked and one autosomal recessive.

Authors:  Alain Verloes; Dominique Bremond-Gignac; Bertrand Isidor; Albert David; Clarisse Baumann; Marie-Anne Leroy; René Stevens; Yves Gillerot; Delphine Héron; Bénédicte Héron; Brigitte Benzacken; Didier Lacombe; Han Brunner; Pierre Bitoun
Journal:  Am J Med Genet A       Date:  2006-06-15       Impact factor: 2.802

6.  Complex phenotypes blur conventional borders between Say-Barber-Biesecker-Young-Simpson syndrome and genitopatellar syndrome.

Authors:  J Radvanszky; M Hyblova; D Durovcikova; M Hikkelova; E Fiedler; L Kadasi; J Turna; G Minarik; T Szemes
Journal:  Clin Genet       Date:  2016-09-29       Impact factor: 4.438

7.  dbNSFP v2.0: a database of human non-synonymous SNVs and their functional predictions and annotations.

Authors:  Xiaoming Liu; Xueqiu Jian; Eric Boerwinkle
Journal:  Hum Mutat       Date:  2013-07-10       Impact factor: 4.878

8.  A framework for variation discovery and genotyping using next-generation DNA sequencing data.

Authors:  Mark A DePristo; Eric Banks; Ryan Poplin; Kiran V Garimella; Jared R Maguire; Christopher Hartl; Anthony A Philippakis; Guillermo del Angel; Manuel A Rivas; Matt Hanna; Aaron McKenna; Tim J Fennell; Andrew M Kernytsky; Andrey Y Sivachenko; Kristian Cibulskis; Stacey B Gabriel; David Altshuler; Mark J Daly
Journal:  Nat Genet       Date:  2011-04-10       Impact factor: 38.330

9.  Further delineation of the KAT6B molecular and phenotypic spectrum.

Authors:  Tamsin Gannon; Rahat Perveen; Hélene Schlecht; Simon Ramsden; Beverley Anderson; Bronwyn Kerr; Ruth Day; Siddharth Banka; Mohnish Suri; Siren Berland; Michael Gabbett; Alan Ma; Stan Lyonnet; Valerie Cormier-Daire; Rüstem Yilmaz; Guntram Borck; Dagmar Wieczorek; Britt-Marie Anderlid; Sarah Smithson; Julie Vogt; Heather Moore-Barton; Pelin Ozlem Simsek-Kiper; Isabelle Maystadt; Anne Destrée; Jessica Bucher; Brad Angle; Shehla Mohammed; Emma Wakeling; Sue Price; Amihood Singer; Yves Sznajer; Annick Toutain; Damien Haye; Ruth Newbury-Ecob; Melanie Fradin; Julie McGaughran; Beyhan Tuysuz; Mark Tein; Katelijne Bouman; Tabib Dabir; Jenneke Van den Ende; Ho Ming Luk; Daniela T Pilz; Jacqueline Eason; Sally Davies; Willie Reardon; Livia Garavelli; Orsetta Zuffardi; Koen Devriendt; Ruth Armstrong; Diana Johnson; Martine Doco-Fenzy; Emilia Bijlsma; Sheila Unger; Hermine E Veenstra-Knol; Jürgen Kohlhase; Ivan F M Lo; Janine Smith; Jill Clayton-Smith
Journal:  Eur J Hum Genet       Date:  2014-11-26       Impact factor: 4.246

Review 10.  The KAT6B-related disorders genitopatellar syndrome and Ohdo/SBBYS syndrome have distinct clinical features reflecting distinct molecular mechanisms.

Authors:  Philippe M Campeau; James T Lu; Brian C Dawson; Ivo F A C Fokkema; Stephen P Robertson; Richard A Gibbs; Brendan H Lee
Journal:  Hum Mutat       Date:  2012-07-12       Impact factor: 4.878

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  2 in total

Review 1.  Further delineation of the clinical spectrum of KAT6B disorders and allelic series of pathogenic variants.

Authors:  Li Xin Zhang; Gabrielle Lemire; Claudia Gonzaga-Jauregui; Sirinart Molidperee; Carolina Galaz-Montoya; David S Liu; Alain Verloes; Amelle G Shillington; Kosuke Izumi; Alyssa L Ritter; Beth Keena; Elaine Zackai; Dong Li; Elizabeth Bhoj; Jennifer M Tarpinian; Emma Bedoukian; Mary K Kukolich; A Micheil Innes; Grace U Ediae; Sarah L Sawyer; Karippoth Mohandas Nair; Para Chottil Soumya; Kinattinkara R Subbaraman; Frank J Probst; Jennifer A Bassetti; Reid V Sutton; Richard A Gibbs; Chester Brown; Philip M Boone; Ingrid A Holm; Marco Tartaglia; Giovanni Battista Ferrero; Marcello Niceta; Maria Lisa Dentici; Francesca Clementina Radio; Boris Keren; Constance F Wells; Christine Coubes; Annie Laquerrière; Jacqueline Aziza; Charlotte Dubucs; Sheela Nampoothiri; David Mowat; Millan S Patel; Ana Bracho; Francisco Cammarata-Scalisi; Alper Gezdirici; Alberto Fernandez-Jaen; Natalie Hauser; Yuri A Zarate; Katherine A Bosanko; Klaus Dieterich; John C Carey; Jessica X Chong; Deborah A Nickerson; Michael J Bamshad; Brendan H Lee; Xiang-Jiao Yang; James R Lupski; Philippe M Campeau
Journal:  Genet Med       Date:  2020-05-19       Impact factor: 8.822

Review 2.  Impaired Regulation of Histone Methylation and Acetylation Underlies Specific Neurodevelopmental Disorders.

Authors:  Merrick S Fallah; Dora Szarics; Clara M Robson; James H Eubanks
Journal:  Front Genet       Date:  2021-01-08       Impact factor: 4.599

  2 in total

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