Literature DB >> 6837632

Brief clinical report: skeletal dysplasia with short, angulated femora (kyphomelic dysplasia).

R N Maclean, W K Prater, C B Lozzio.   

Abstract

We report on an infant with broad and severely angulated short femora as the most salient manifestation of a generalized skeletal dysplasia. Other findings include congenital bowing of other long bones, narrow thorax, platyspondyly, micrognathia, and skin dimples. A marked improvement of the bowing and of the irregular flare of the metaphyses was noted over a period of 6 mo. Congenital bowing of long bones can be an isolated finding or associated with other anomalies, so the purpose of reporting all cases is important for further nosologic and pathogenetic elucidation. Because of the severity of the femoral involvement, the condition has been called kyphomelic dysplasia. It may be an autosomal recessive trait. recessive trait.

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Year:  1983        PMID: 6837632     DOI: 10.1002/ajmg.1320140218

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


  6 in total

1.  Kyphomelic dysplasia: the first 10 cases.

Authors:  P D Turnpenny; R A Dakwar; F N Boulos
Journal:  J Med Genet       Date:  1990-04       Impact factor: 6.318

2.  Familial congenital bowing with short thick bones and metaphyseal changes, a distinct entity. Report of the clinical and radiological findings in two siblings.

Authors:  E Rezza; G Iannaccone; D Lendvai
Journal:  Pediatr Radiol       Date:  1984

Review 3.  Kyphomelic dysplasia in two sib fetuses.

Authors:  C P Chen; S R Chern; S L Shih; C Y Chuang; F Y Huang
Journal:  J Med Genet       Date:  1998-01       Impact factor: 6.318

4.  Isolated femoral hypoplasia: an intrauterine differential diagnosis to campomelia.

Authors:  Friederike Körber; Eckard Schönau; A Eldad Horwitz; Gabriele Benz-Bohm
Journal:  Pediatr Radiol       Date:  2005-01-06

5.  Oto-palato-digital syndrome type II. Report of two related cases.

Authors:  P W Gendall; K Kozlowski
Journal:  Pediatr Radiol       Date:  1992

6.  Kyphomelic dysplasia.

Authors:  I K Temple; E M Thompson; C M Hall; G Bridgeman; M E Pembrey
Journal:  J Med Genet       Date:  1989-07       Impact factor: 6.318

  6 in total

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