Literature DB >> 24281372

Phenotypes of craniofrontonasal syndrome in patients with a pathogenic mutation in EFNB1.

M E P van den Elzen1, S R F Twigg2, J A C Goos1, A J M Hoogeboom3, A M W van den Ouweland3, A O M Wilkie2, I M J Mathijssen1.   

Abstract

Craniofrontonasal syndrome (CFNS) is an X-linked developmental malformation, caused by mutations in the EFNB1 gene, which have only been described since 2004. A genotype-phenotype correlation seems not to be present. As it is of major importance to adequately counsel patients with EFNB1 mutations and their parents, and to improve diagnosis of new patients, more information about the phenotypic features is needed. This study included 23 patients (2 male, 21 female) with confirmed EFNB1 mutations. All patients underwent a thorough physical examination and photographs were taken. If available, radiological images were also consulted. Hypertelorism, longitudinal ridging and/or splitting of nails, a (mild) webbed neck and a clinodactyly of one or more toes were the only consistent features observed in all patients. Frequently observed phenotypic features were bifid tip of the nose (91%), columellar indentation (91%) and low implantation of breasts (90%). In comparison with anthropometric data of facial proportions, patients with CFNS had a significantly different face in multiple respects. An overview of all phenotypic features is shown. Patients with EFNB1 mutations have a clear phenotype. This study will facilitate genetic counseling of parents and patients, and contribute to the diagnostic and screening process of patients with suspected CFNS.

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Year:  2013        PMID: 24281372      PMCID: PMC4096149          DOI: 10.1038/ejhg.2013.273

Source DB:  PubMed          Journal:  Eur J Hum Genet        ISSN: 1018-4813            Impact factor:   4.246


  34 in total

1.  Control of skeletal patterning by ephrinB1-EphB interactions.

Authors:  Amelia Compagni; Malcolm Logan; Rüdiger Klein; Ralf H Adams
Journal:  Dev Cell       Date:  2003-08       Impact factor: 12.270

2.  A novel EFNB1 mutation (c.712delG) in a family with craniofrontonasal syndrome and diaphragmatic hernia.

Authors:  Jacob Hogue; Suma Shankar; Hazel Perry; Reena Patel; Karin Vargervik; Anne Slavotinek
Journal:  Am J Med Genet A       Date:  2010-10       Impact factor: 2.802

3.  Craniofrontonasal dysplasia associated with Chiari malformation.

Authors:  Amit Mahore; Abhidha Shah; Trimurti Nadkarni; Atul Goel
Journal:  J Neurosurg Pediatr       Date:  2010-04       Impact factor: 2.375

Review 4.  Diverse clinical and genetic aspects of craniofrontonasal syndrome.

Authors:  Dimitrios I Zafeiriou; Efterpi L Pavlidou; Euthymia Vargìami
Journal:  Pediatr Neurol       Date:  2011-02       Impact factor: 3.372

5.  A novel de novo mutation within EFNB1 gene in a young girl with craniofrontonasal syndrome.

Authors:  Despina Apostolopoulou; Alexander Stratoudakis; Angeliki Hatzaki; Olga S Kaxira; Kanaris P Panagopoulos; Panagoula Kollia; Vassiliki Aleporou
Journal:  Cleft Palate Craniofac J       Date:  2011-02-27

6.  The impact of CFNS-causing EFNB1 mutations on ephrin-B1 function.

Authors:  Roman Makarov; Bernhard Steiner; Zoran Gucev; Velibor Tasic; Peter Wieacker; Ilse Wieland
Journal:  BMC Med Genet       Date:  2010-06-17       Impact factor: 2.103

7.  Craniofrontonasal dysplasia: a surgical treatment algorithm.

Authors:  Henry K Kawamoto; Justin B Heller; Misha M Heller; Andres Urrego; Joubin S Gabbay; Kristy L Wasson; James P Bradley
Journal:  Plast Reconstr Surg       Date:  2007-12       Impact factor: 4.730

8.  Dissecting the molecular mechanisms in craniofrontonasal syndrome: differential mRNA expression of mutant EFNB1 and the cellular mosaic.

Authors:  Ilse Wieland; Roman Makarov; William Reardon; Sigrid Tinschert; Alice Goldenberg; Patrick Thierry; Peter Wieacker
Journal:  Eur J Hum Genet       Date:  2007-11-28       Impact factor: 4.246

9.  Mapping of a further locus for X-linked craniofrontonasal syndrome.

Authors:  I Wieland; S Jakubiczka; P Muschke; A Wolf; L Gerlach; M Krawczak; P Wieacker
Journal:  Cytogenet Genome Res       Date:  2002       Impact factor: 1.636

10.  Cellular interference in craniofrontonasal syndrome: males mosaic for mutations in the X-linked EFNB1 gene are more severely affected than true hemizygotes.

Authors:  Stephen R F Twigg; Christian Babbs; Marijke E P van den Elzen; Anne Goriely; Stephen Taylor; Simon J McGowan; Eleni Giannoulatou; Lorne Lonie; Jiannis Ragoussis; Elham Sadighi Akha; Samantha J L Knight; Roseli M Zechi-Ceide; Jeannette A M Hoogeboom; Barbara R Pober; Helga V Toriello; Steven A Wall; M Rita Passos-Bueno; Han G Brunner; Irene M J Mathijssen; Andrew O M Wilkie
Journal:  Hum Mol Genet       Date:  2013-01-17       Impact factor: 6.150

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  11 in total

Review 1.  Frontonasal Dysplasia: Towards an Understanding of Molecular and Developmental Aetiology.

Authors:  Peter G Farlie; Naomi L Baker; Patrick Yap; Tiong Y Tan
Journal:  Mol Syndromol       Date:  2016-10-29

2.  A Family with Craniofrontonasal Syndrome and a Mutation (p.G151S) in the EFNB1 Gene: Expanding the Phenotype.

Authors:  Jaime Toral-López; Luz M González-Huerta; Olga Messina Baas; Sergio A Cuevas-Covarrubias
Journal:  Mol Syndromol       Date:  2016-03-19

Review 3.  Syndromic Craniosynostosis: Complexities of Clinical Care.

Authors:  Justine O'Hara; Federica Ruggiero; Louise Wilson; Greg James; Graeme Glass; Owase Jeelani; Juling Ong; Richard Bowman; Michelle Wyatt; Robert Evans; Martin Samuels; Richard Hayward; David J Dunaway
Journal:  Mol Syndromol       Date:  2019-01-16

4.  Evaluation of Sporadic and Familial Cases with Craniofrontonasal Syndrome: A Wide Clinical Spectrum and Identification of a Novel EFNB1 Gene Mutation.

Authors:  Semra Gürsoy; Filiz Hazan; Tülay Öztürk; Rüya Çolak; Şebnem Çalkavur
Journal:  Mol Syndromol       Date:  2021-07-12

5.  Isolated Sagittal Synostosis in a Boy with Craniofrontonasal Dysplasia and a Novel EFNB1 Mutation.

Authors:  Bharesh K Chauhan; Jacqueline M Hoover; Hannah Scanga; Anagha Medsinge; Georgianne L Arnold; Ken K Nischal
Journal:  Plast Reconstr Surg Glob Open       Date:  2015-07-08

6.  Mllt10 knockout mouse model reveals critical role of Af10-dependent H3K79 methylation in midfacial development.

Authors:  Honami Ogoh; Kazutsune Yamagata; Tomomi Nakao; Lisa L Sandell; Ayaka Yamamoto; Aiko Yamashita; Naomi Tanga; Mai Suzuki; Takaya Abe; Issay Kitabayashi; Toshio Watanabe; Daisuke Sakai
Journal:  Sci Rep       Date:  2017-09-20       Impact factor: 4.379

Review 7.  Eph-Ephrin Signaling Mediates Cross-Talk Within the Bone Microenvironment.

Authors:  Agnieszka Arthur; Stan Gronthos
Journal:  Front Cell Dev Biol       Date:  2021-02-09

8.  Clinical and molecular characterization of craniofrontonasal syndrome: new symptoms and novel pathogenic variants in the EFNB1 gene.

Authors:  Ewelina Bukowska-Olech; Paweł Gawliński; Anna Jakubiuk-Tomaszuk; Maria Jędrzejowska; Ewa Obersztyn; Michał Piechota; Marta Bielska; Aleksander Jamsheer
Journal:  Orphanet J Rare Dis       Date:  2021-06-26       Impact factor: 4.123

9.  The osteoprogenitor-specific loss of ephrinB1 results in an osteoporotic phenotype affecting the balance between bone formation and resorption.

Authors:  Agnieszka Arthur; Thao M Nguyen; Sharon Paton; Ana Klisuric; Andrew C W Zannettino; Stan Gronthos
Journal:  Sci Rep       Date:  2018-08-24       Impact factor: 4.379

10.  Aberrant cell segregation in the craniofacial primordium and the emergence of facial dysmorphology in craniofrontonasal syndrome.

Authors:  Terren K Niethamer; Teng Teng; Melanie Franco; Yu Xin Du; Christopher J Percival; Jeffrey O Bush
Journal:  PLoS Genet       Date:  2020-02-24       Impact factor: 5.917

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