Literature DB >> 8305967

Craniofrontonasal dysplasia in two male sibs.

U Natarajan1, M Baraitser, K Nicolaides, C Gosden.   

Abstract

Craniofrontonasal dysplasia (CFND) was diagnosed in a male child who had bilateral coronal craniosynostosis, midline facial clefting with cleft lip and palate, a broad and high forehead, and hypertelorism. The parents were normal and there was no family history suggestive of CFND. A small recurrence risk was counselled and prenatal ultrasound was recommended in the next pregnancy. At 23 weeks, the ultrasound scan detected a number of dysmorphic features and the pregnancy was terminated. Autopsy of the aborted male foetus showed the following features: High arched palate, a sloping forehead, flattened nose and receding chin, multiple joint contractures, particularly of the mid phalangeal joints, elbows and ankles. This report highlights the occurrence of CFND in two male siblings born to normal parents, and therefore the need to perform prenatal ultrasound in a subsequent pregnancy, even if CFND is diagnosed in an isolated case.

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Year:  1993        PMID: 8305967

Source DB:  PubMed          Journal:  Clin Dysmorphol        ISSN: 0962-8827            Impact factor:   0.816


  3 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
  3 in total

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