Literature DB >> 1468459

Craniofrontonasal dysplasia.

L Kapusta1, H G Brunner, B C Hamel.   

Abstract

We report on nine patients with craniofrontonasal dysplasia (CFND). Seven classical cases had facial features suggestive of frontonasal dysplasia and coronal craniosynostosis. Extracranial abnormalities such as brittle nails with prominent longitudinal grooves or syndactyly of fingers and toes were observed in individual patients. In two families the father of classical cases showed a milder pattern of abnormalities, consistent with the diagnosis. We present a 2- to 13-year follow-up on our patients. Hypotonia and laxity of joints are common and may necessitate supportive measures. Mild developmental delay was noted in three out of six classical cases studied in detail. Unlike almost all other X-linked disorders, clinical expression in CFND is generally much more severe in females than in males. In contrast to previous reports of this condition, one of our severely affected cases is a male.

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Year:  1992        PMID: 1468459     DOI: 10.1007/bf01957936

Source DB:  PubMed          Journal:  Eur J Pediatr        ISSN: 0340-6199            Impact factor:   3.183


  24 in total

1.  Craniofrontonasal dysplasia.

Authors:  J Hurst; M Baraitser
Journal:  J Med Genet       Date:  1988-02       Impact factor: 6.318

2.  Frontonasal dysplasia.

Authors:  H O Sedano; M M Cohen; J Jirasek; R J Gorlin
Journal:  J Pediatr       Date:  1970-06       Impact factor: 4.406

3.  Frontonasal dysplasia with coronal craniosynostosis in three sibs.

Authors:  R Slover; E Sujansky
Journal:  Birth Defects Orig Artic Ser       Date:  1979

4.  Congenital hypothalamic hamartoma associated with severe midline defect: a developmental field defect. Report of a case.

Authors:  R C Hennekam; F A Beemer; F Van Merrienboer; B A Van Ketel; P P Kramer
Journal:  Am J Med Genet Suppl       Date:  1986

Review 5.  Frontonasal "dysplasia," cerebral anomalies, and polydactyly: report of a new syndrome and discussion from a developmental field perspective.

Authors:  H V Toriello; L L Radecki; J Sharda; D Looyenga; R Mann
Journal:  Am J Med Genet Suppl       Date:  1986

Review 6.  The central nervous system in the Apert syndrome.

Authors:  M M Cohen; S Kreiborg
Journal:  Am J Med Genet       Date:  1990-01

7.  A previously undescribed autosomal recessive multiple congenital anomalies/mental retardation (MCA/MR) syndrome with fronto-nasal dysostosis, cleft lip/palate, limb hypoplasia, and postaxial poly-syndactyly: acro-fronto-facio-nasal dysostosis syndrome.

Authors:  A Richieri-Costa; G M Colletto; T R Gollop; D Masiero
Journal:  Am J Med Genet       Date:  1985-04

8.  Craniofrontonasal dysplasia in a three-generation kindred.

Authors:  J F Reynolds; R J Haas; M T Edgerton; T E Kelly
Journal:  J Craniofac Genet Dev Biol       Date:  1982

9.  New autosomal dominant syndrome resembling craniofrontonasal dysplasia.

Authors:  A S Teebi
Journal:  Am J Med Genet       Date:  1987-11

Review 10.  Craniosynostosis update 1987.

Authors:  M M Cohen
Journal:  Am J Med Genet Suppl       Date:  1988
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  5 in total

1.  The origin of EFNB1 mutations in craniofrontonasal syndrome: frequent somatic mosaicism and explanation of the paucity of carrier males.

Authors:  Stephen R F Twigg; Kazuya Matsumoto; Alexa M J Kidd; Anne Goriely; Indira B Taylor; Richard B Fisher; A Jeannette M Hoogeboom; Irene M J Mathijssen; M Teresa Lourenco; Jenny E V Morton; Elizabeth Sweeney; Louise C Wilson; Han G Brunner; John B Mulliken; Steven A Wall; Andrew O M Wilkie
Journal:  Am J Hum Genet       Date:  2006-04-28       Impact factor: 11.025

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

Authors:  M E P van den Elzen; S R F Twigg; J A C Goos; A J M Hoogeboom; A M W van den Ouweland; A O M Wilkie; I M J Mathijssen
Journal:  Eur J Hum Genet       Date:  2013-11-27       Impact factor: 4.246

3.  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

4.  mRNA expression analysis of the hippocampus in a vervet monkey model of fetal alcohol spectrum disorder.

Authors:  Rob F Gillis; Roberta M Palmour
Journal:  J Neurodev Disord       Date:  2022-03-19       Impact factor: 4.025

5.  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

  5 in total

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