Literature DB >> 24719364

Blepharo-cheilo-dontic (BCD) syndrome: expanding the phenotype, case report and review of literature.

Farouq K Ababneh1, Abdulrahman Al-Swaid, Ahmed Elhag, Talaat Youssef, Saif Alsaif.   

Abstract

The combination of lagophthalmia, euryblepharon, ectropion of lower eyelids, distichiasis, bilateral cleft lip and palate, and oligodontia comprises the blepharo-cheilo-dontic (BCD) syndrome. This combination has been found sporadically or with positive family history and inherited as an autosomal dominant condition with variable expression. We described a Saudi boy with the cardinal signs consistent with the BCD syndrome. In addition to the common components of BCD syndrome that involve eyelids, lip, and teeth abnormalities, this patient is the third reported BCD case with imperforate anus, the second with thyroid agenesis, and the first with lumbosacral meningomyelocele.
© 2014 Wiley Periodicals, Inc.

Entities:  

Keywords:  anal atresia; blepharo-cheilo-dontic; cleft lip and palate; euryblepharon; lagophthalmia; spina bifida; thyroid agenesis

Mesh:

Year:  2014        PMID: 24719364     DOI: 10.1002/ajmg.a.36465

Source DB:  PubMed          Journal:  Am J Med Genet A        ISSN: 1552-4825            Impact factor:   2.802


  4 in total

1.  Variants in members of the cadherin-catenin complex, CDH1 and CTNND1, cause blepharocheilodontic syndrome.

Authors:  Anneke Kievit; Federico Tessadori; Hannie Douben; Ingrid Jordens; Madelon Maurice; Jeannette Hoogeboom; Raoul Hennekam; Sheela Nampoothiri; Hülya Kayserili; Marco Castori; Margo Whiteford; Connie Motter; Catherine Melver; Michael Cunningham; Anne Hing; Nancy M Kokitsu-Nakata; Siulan Vendramini-Pittoli; Antonio Richieri-Costa; Annette F Baas; Corstiaan C Breugem; Karen Duran; Maarten Massink; Patrick W B Derksen; Wilfred F J van IJcken; Leontine van Unen; Fernando Santos-Simarro; Pablo Lapunzina; Vera L Gil-da Silva Lopes; Elaine Lustosa-Mendes; Max Krall; Anne Slavotinek; Victor Martinez-Glez; Jeroen Bakkers; Koen L I van Gassen; Annelies de Klein; Marie-José H van den Boogaard; Gijs van Haaften
Journal:  Eur J Hum Genet       Date:  2018-01-18       Impact factor: 4.246

2.  CDH1 germline mutations: different syndromes, same management?

Authors:  Patrick R Benusiglio
Journal:  Genet Med       Date:  2017-04-20       Impact factor: 8.822

3.  Blepharocheilodontic syndrome is a CDH1 pathway-related disorder due to mutations in CDH1 and CTNND1.

Authors:  Jamal Ghoumid; Morgane Stichelbout; Anne-Sophie Jourdain; Frederic Frenois; Sophie Lejeune-Dumoulin; Marie-Pierre Alex-Cordier; Marine Lebrun; Pierre Guerreschi; Veronique Duquennoy-Martinot; Matthieu Vinchon; Joel Ferri; Matthieu Jung; Serge Vicaire; Clemence Vanlerberghe; Fabienne Escande; Florence Petit; Sylvie Manouvrier-Hanu
Journal:  Genet Med       Date:  2017-03-16       Impact factor: 8.822

4.  Novel truncating mutations in CTNND1 cause a dominant craniofacial and cardiac syndrome.

Authors:  Reham Alharatani; Athina Ververi; Ana Beleza-Meireles; Weizhen Ji; Emily Mis; Quinten T Patterson; John N Griffin; Nabina Bhujel; Caitlin A Chang; Abhijit Dixit; Monica Konstantino; Christopher Healy; Sumayyah Hannan; Natsuko Neo; Alex Cash; Dong Li; Elizabeth Bhoj; Elaine H Zackai; Ruth Cleaver; Diana Baralle; Meriel McEntagart; Ruth Newbury-Ecob; Richard Scott; Jane A Hurst; Ping Yee Billie Au; Marie Therese Hosey; Mustafa Khokha; Denise K Marciano; Saquib A Lakhani; Karen J Liu
Journal:  Hum Mol Genet       Date:  2020-07-21       Impact factor: 6.150

  4 in total

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