Literature DB >> 3717210

Achondrogenesis II-hypochondrogenesis: variability versus heterogeneity.

Z Borochowitz, A Ornoy, R Lachman, D L Rimoin.   

Abstract

Recently hypochondrogenesis was described as a form of neonatally lethal dwarfism said to resemble spondyloepiphyseal dysplasia congenita radiographically and achondrogenesis II morphologically. Because of the difficulty in distinguishing radiographically between mild achondrogenesis II and severe hypochondrogenesis, we performed a clinical, radiographic, and morphologic study of 24 cases originally classified as either achondrogenesis II or hypochondrogenesis, in an attempt to distinguish between heterogeneity and clinical variability. Review of the radiographic findings in these cases show a fairly continuous spectrum of bony defects, rather than two distinct radiographic syndromes. Chondro-osseous histology and ultrastructure was similar in all cases regardless of severity and was characterized by hypervascularity and hypercellularity of the cartilage with multiple small, round dilated cysternae of rough endoplasmic reticulum. These findings suggest that hypochondrogenesis and achondrogenesis type II represent a spectrum with marked phenotypic variability.

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Mesh:

Year:  1986        PMID: 3717210     DOI: 10.1002/ajmg.1320240208

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


  8 in total

1.  Quantitative histology of cartilage vascular canals in the human rib. Findings in normal neonates and children and in achondrogenesis II-hypochondrogenesis.

Authors:  H E Gruber; R S Lachman; D L Rimoin
Journal:  J Anat       Date:  1990-12       Impact factor: 2.610

2.  Visceral manifestations of hypochondrogenesis.

Authors:  Helen Wainwright; Peter Beighton
Journal:  Virchows Arch       Date:  2008-07-19       Impact factor: 4.064

3.  Tandem duplication within a type II collagen gene (COL2A1) exon in an individual with spondyloepiphyseal dysplasia.

Authors:  G E Tiller; D L Rimoin; L W Murray; D H Cohn
Journal:  Proc Natl Acad Sci U S A       Date:  1990-05       Impact factor: 11.205

4.  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

5.  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

6.  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

7.  An RNA-splicing mutation (G+5IVS20) in the type II collagen gene (COL2A1) in a family with spondyloepiphyseal dysplasia congenita.

Authors:  G E Tiller; M A Weis; P A Polumbo; H E Gruber; D L Rimoin; D H Cohn; D R Eyre
Journal:  Am J Hum Genet       Date:  1995-02       Impact factor: 11.025

8.  Substitution of aspartic acid for glycine at position 310 in type II collagen produces achondrogenesis II, and substitution of serine at position 805 produces hypochondrogenesis: analysis of genotype-phenotype relationships.

Authors:  J Bonaventure; L Cohen-Solal; P Ritvaniemi; L Van Maldergem; N Kadhom; A L Delezoide; P Maroteaux; D J Prockop; L Ala-Kokko
Journal:  Biochem J       Date:  1995-05-01       Impact factor: 3.857

  8 in total

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