Literature DB >> 9843047

Radiographic and morphologic findings in a previously undescribed type of mesomelic dysplasia resembling atelosteogenesis type II.

S G Brodie1, R S Lachman, B F Crandall, M A Fox, D L Rimoin, D H Cohn, W R Wilcox.   

Abstract

The mesomelic chondrodysplasias are a heterogeneous group of dwarfing disorders characterized by shortness of the middle segments of limbs. We report on a 25-week fetus with disproportionate shortness of limbs with an apparently distinct form of mesomelic dysplasia. Radiographic findings at necropsy included ulnar deviation of hands, talipes equinovarus, distal tapering of the humeri, and hypoplastic fibulae, radii, and ulnae. Chondro-osseous morphology showed mild shortness of the physeal columns, overgrowth of perichondral bone, peripheral ingrowth of mesenchymal cells into the physis, and numerous areas of fibrillar degeneration with rings of collagen surrounding the chondrocytes. Ultrastructural findings included a degenerated territorial matrix, pericellular halos of collagen, and dilated loops of rough endoplasmic reticulum in chondrocytes. The radiographic appearance of the long bones is distinct from that of previously described mesomelic dysplasias. The chondro-osseous morphologic findings and the distal tapering of the humerus are somewhat reminiscent of atelosteogenesis type II, but the pattern of matrix degeneration and the presence of inclusion bodies in the chondrocytes distinguish it from disorders of sulfate transport.

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Year:  1998        PMID: 9843047

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


  2 in total

1.  A recurrent RNA-splicing mutation in the SEDL gene causes X-linked spondyloepiphyseal dysplasia tarda.

Authors:  G E Tiller; V L Hannig; D Dozier; L Carrel; K C Trevarthen; W R Wilcox; S Mundlos; J L Haines; A K Gedeon; J Gecz
Journal:  Am J Hum Genet       Date:  2001-04-26       Impact factor: 11.025

2.  Identification of a de novo heterozygous missense FLNB mutation in lethal atelosteogenesis type I by exome sequencing.

Authors:  Ga Won Jeon; Mi-Na Lee; Ji Mi Jung; Seong Yeon Hong; Young Nam Kim; Jong Beom Sin; Chang-Seok Ki
Journal:  Ann Lab Med       Date:  2014-02-13       Impact factor: 3.464

  2 in total

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