Literature DB >> 3041834

Antley-Bixler syndrome from a prognostic perspective: report of a case and review of the literature.

L F Escobar1, D Bixler, M Sadove, M J Bull.   

Abstract

The Antley-Bixler syndrome (ABS) is characterized by craniosynostosis, radiohumeral synostosis, and femoral bowing. Other findings include a trapezoid-shaped head, deformed ears, severe midface hypoplasia, choanal atresia or stenosis, and long bone fractures. Most ABS cases have died in the first months of life from respiratory complications. The poor prognosis in this condition makes counseling difficult and early termination of pregnancy a consideration. The medical and surgical management information presented here can be used as a guide for counseling parents in the future. We report on a new patient with ABS who now at age 3 yr, has been followed by the medical staff of Riley Children's Hospital since birth. She has had successful medical and surgical management. Although the multisynostoses seen in this disorder is undoubtedly related to the soft tissue malformations such as choanal stenosis and midface hypoplasia, the cause remains unknown. The literature is also reviewed in this condition.

Entities:  

Mesh:

Year:  1988        PMID: 3041834     DOI: 10.1002/ajmg.1320290412

Source DB:  PubMed          Journal:  Am J Med Genet        ISSN: 0148-7299


  6 in total

Review 1.  Malformation syndromes caused by disorders of cholesterol synthesis.

Authors:  Forbes D Porter; Gail E Herman
Journal:  J Lipid Res       Date:  2010-10-07       Impact factor: 5.922

2.  Picture of the month. Antley-Bixler syndrome.

Authors:  M G Butler; S Hassell; M Feingold; W W Tunnessen
Journal:  Am J Dis Child       Date:  1991-06

3.  The Antley-Bixler syndrome: report of two familial cases with severe renal and anal anomalies.

Authors:  B P LeHeup; J P Masutti; P Droullé; J Tisserand
Journal:  Eur J Pediatr       Date:  1995-02       Impact factor: 3.183

4.  Antley-Bixler syndrome: description of two patients.

Authors:  E Bianchi; S Cordini; P Fiori; F Torcetta; G Beluffi
Journal:  Skeletal Radiol       Date:  1991       Impact factor: 2.199

Review 5.  Antley-Bixler syndrome: report of a patient and review of literature.

Authors:  S Hassell; M G Butler
Journal:  Clin Genet       Date:  1994-11       Impact factor: 4.438

6.  Rdh10 loss-of-function and perturbed retinoid signaling underlies the etiology of choanal atresia.

Authors:  Hiroshi Kurosaka; Qi Wang; Lisa Sandell; Takashi Yamashiro; Paul A Trainor
Journal:  Hum Mol Genet       Date:  2017-04-01       Impact factor: 6.150

  6 in total

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