Literature DB >> 27625340

Baraitser-Winter cerebrofrontofacial syndrome.

T M Yates1, C L Turner2, H V Firth3, J Berg4, D T Pilz5.   

Abstract

Baraitser-Winter cerebrofrontofacial syndrome (BWCFF) (BRWS; MIM #243310, 614583) is a rare developmental disorder affecting multiple organ systems. It is characterised by intellectual disability (mild to severe) and distinctive facial appearance (metopic ridging/trigonocephaly, bilateral ptosis, hypertelorism). The additional presence of cortical malformations (pachygyria/lissencephaly) and ocular colobomata are also suggestive of this syndrome. Other features include moderate short stature, contractures, congenital cardiac disease and genitourinary malformations. BWCFF is caused by missense mutations in the cytoplasmic beta- and gamma-actin genes ACTB and ACTG1. We provide an overview of the clinical characteristics (including some novel findings in four recently diagnosed patients), diagnosis, management, mutation spectrum and genetic counselling.
© 2016 John Wiley & Sons A/S. Published by John Wiley & Sons Ltd.

Entities:  

Keywords:  ACTB; ACTG1; Baraitser-Winter cerebrofrontofacial syndrome; Baraitser-Winter syndrome; coloboma; pachygyria

Mesh:

Substances:

Year:  2016        PMID: 27625340     DOI: 10.1111/cge.12864

Source DB:  PubMed          Journal:  Clin Genet        ISSN: 0009-9163            Impact factor:   4.438


  10 in total

1.  Could Dissimilar Phenotypic Effects of ACTB Missense Mutations Reflect the Actin Conformational Change? Two Novel Mutations and Literature Review.

Authors:  Anna Sandestig; Anna Green; Jon Jonasson; Hartmut Vogt; Johan Wahlström; Alexander Pepler; Katarina Ellnebo; Saskia Biskup; Margarita Stefanova
Journal:  Mol Syndromol       Date:  2018-08-09

2.  Post-zygotic ACTB mutations underlie congenital smooth muscle hamartomas.

Authors:  Lihi Atzmony; Nelson Ugwu; Theodore D Zaki; Richard J Antaya; Keith A Choate
Journal:  J Cutan Pathol       Date:  2020-04-06       Impact factor: 1.587

3.  ACTB Loss-of-Function Mutations Result in a Pleiotropic Developmental Disorder.

Authors:  Sara Cuvertino; Helen M Stuart; Kate E Chandler; Neil A Roberts; Ruth Armstrong; Laura Bernardini; Sanjeev Bhaskar; Bert Callewaert; Jill Clayton-Smith; Cristina Hernando Davalillo; Charu Deshpande; Koenraad Devriendt; Maria C Digilio; Abhijit Dixit; Matthew Edwards; Jan M Friedman; Antonio Gonzalez-Meneses; Shelagh Joss; Bronwyn Kerr; Anne Katrin Lampe; Sylvie Langlois; Rachel Lennon; Philippe Loget; David Y T Ma; Ruth McGowan; Maryse Des Medt; James O'Sullivan; Sylvie Odent; Michael J Parker; Céline Pebrel-Richard; Florence Petit; Zornitza Stark; Sylvia Stockler-Ipsiroglu; Sigrid Tinschert; Pradeep Vasudevan; Olaya Villa; Susan M White; Farah R Zahir; Adrian S Woolf; Siddharth Banka
Journal:  Am J Hum Genet       Date:  2017-12-07       Impact factor: 11.025

4.  Post-mortem Characterisation of a Case With an ACTG1 Variant, Agenesis of the Corpus Callosum and Neuronal Heterotopia.

Authors:  Regina Vontell; Veena G Supramaniam; Alice Davidson; Claire Thornton; Andreas Marnerides; Muriel Holder-Espinasse; Suzanne Lillis; Shu Yau; Mattias Jansson; Henrik E Hagberg; Mary A Rutherford
Journal:  Front Physiol       Date:  2019-05-24       Impact factor: 4.566

5.  Building Bridges Between the Clinic and the Laboratory: A Meeting Review - Brain Malformations: A Roadmap for Future Research.

Authors:  Tamar Sapir; Tahsin Stefan Barakat; Mercedes F Paredes; Tally Lerman-Sagie; Eleonora Aronica; Wlodzimierz Klonowski; Laurent Nguyen; Bruria Ben Zeev; Nadia Bahi-Buisson; Richard Leventer; Noa Rachmian; Orly Reiner
Journal:  Front Cell Neurosci       Date:  2019-09-27       Impact factor: 5.505

6.  Identification of a De Novo Heterozygous Missense ACTB Variant in Baraitser-Winter Cerebrofrontofacial Syndrome.

Authors:  Kailai Nie; Junting Huang; Longqian Liu; Hongbin Lv; Danian Chen; Wei Fan
Journal:  Front Genet       Date:  2022-03-24       Impact factor: 4.599

7.  A de novo ACTB gene pathogenic variant in identical twins with phenotypic variation for hydrops and jejunal atresia.

Authors:  Kristina Sibbin; Patrick Yap; Denis Nyaga; Raoul Heller; Stephen Evans; Kate Strachan; Salam Alburaiky; Han M Alex Nguyen; Sylvie Hermann-Le Denmat; Austen R D Ganley; Justin M O'Sullivan; Frank H Bloomfield
Journal:  Am J Med Genet A       Date:  2021-12-31       Impact factor: 2.578

Review 8.  Human deafness-associated variants alter the dynamics of key molecules in hair cell stereocilia F-actin cores.

Authors:  Takushi Miyoshi; Inna A Belyantseva; Shin-Ichiro Kitajiri; Hiroki Miyajima; Shin-Ya Nishio; Shin-Ichi Usami; Bong Jik Kim; Byung Yoon Choi; Koichi Omori; Hari Shroff; Thomas B Friedman
Journal:  Hum Genet       Date:  2021-07-07       Impact factor: 4.132

Review 9.  Genome-wide association studies of structural birth defects: A review and commentary.

Authors:  Philip J Lupo; Laura E Mitchell; Mary M Jenkins
Journal:  Birth Defects Res       Date:  2019-10-25       Impact factor: 2.661

Review 10.  Actin Mutations and Their Role in Disease.

Authors:  Francine Parker; Thomas G Baboolal; Michelle Peckham
Journal:  Int J Mol Sci       Date:  2020-05-10       Impact factor: 6.208

  10 in total

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