Literature DB >> 4080742

Pattern recognition in bone dysplasias.

J Spranger.   

Abstract

Genetically different bone dysplasias may manifest themselves in similar patterns of skeletal abnormalities. It is proposed to group these similar dysplasias in 'families' for two reasons: 1 The knowledge of developmental patterns shared by different genetic disorders cautions the diagnostician and encourages a two-step procedure: a) provisional recognition of a pattern and b) more careful analysis of the pattern to reach a final, specific diagnosis. 2 Families of bone dysplasias may be the result of similar pathogenetic mechanisms. Once the mechanism is discovered in one member of the family, a search for similar mechanisms in others may be rewarding. An example of such a pattern is dysostosis multiplex. It is found in a family of disorders caused by defects of complex carbohydrate degradation. The present study delineates four more patterns and their families: the achondroplasia pattern, spondyloepiphyseal dysplasia congenita pattern, the Larsen/OPD pattern and the Stickler/Kniest pattern.

Entities:  

Mesh:

Year:  1985        PMID: 4080742

Source DB:  PubMed          Journal:  Prog Clin Biol Res        ISSN: 0361-7742


  17 in total

1.  Report of five novel and one recurrent COL2A1 mutations with analysis of genotype-phenotype correlation in patients with a lethal type II collagen disorder.

Authors:  G R Mortier; M Weis; L Nuytinck; L M King; D J Wilkin; A De Paepe; R S Lachman; D L Rimoin; D R Eyre; D H Cohn
Journal:  J Med Genet       Date:  2000-04       Impact factor: 6.318

2.  Type II achondrogenesis-hypochondrogenesis: identification of abnormal type II collagen.

Authors:  M Godfrey; D W Hollister
Journal:  Am J Hum Genet       Date:  1988-12       Impact factor: 11.025

3.  Of mice and men: heritable skeletal disorders.

Authors:  O Jacenko; B R Olsen; M L Warman
Journal:  Am J Hum Genet       Date:  1994-02       Impact factor: 11.025

4.  Type II achondrogenesis-hypochondrogenesis: morphologic and immunohistopathologic studies.

Authors:  M Godfrey; D R Keene; E Blank; H Hori; L Y Sakai; L A Sherwin; D W Hollister
Journal:  Am J Hum Genet       Date:  1988-12       Impact factor: 11.025

Review 5.  Genetic analysis of skeletal dysplasia: recent advances and perspectives in the post-genome-sequence era.

Authors:  Shiro Ikegawa
Journal:  J Hum Genet       Date:  2006-05-03       Impact factor: 3.172

6.  Severe short-limb dwarfism resembling Grebe chondrodysplasia.

Authors:  A S Teebi; S A Al-Awadi; J M Opitz; J Spranger
Journal:  Hum Genet       Date:  1986-12       Impact factor: 4.132

7.  Skeletal dysplasias associated with mild myopathy-a clinical and molecular review.

Authors:  Katarzyna A Piróg; Michael D Briggs
Journal:  J Biomed Biotechnol       Date:  2010-05-24

Review 8.  Achondroplasia: a comprehensive clinical review.

Authors:  Richard M Pauli
Journal:  Orphanet J Rare Dis       Date:  2019-01-03       Impact factor: 4.123

Review 9.  Skeletal ciliopathies: a pattern recognition approach.

Authors:  Atsuhiko Handa; Ulrika Voss; Anna Hammarsjö; Giedre Grigelioniene; Gen Nishimura
Journal:  Jpn J Radiol       Date:  2020-01-21       Impact factor: 2.374

10.  Acromesomelic dysplasia Maroteaux type maps to human chromosome 9.

Authors:  S G Kant; A Polinkovsky; S Mundlos; B Zabel; R T Thomeer; H M Zonderland; L Shih; A van Haeringen; M L Warman
Journal:  Am J Hum Genet       Date:  1998-07       Impact factor: 11.025

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