Literature DB >> 9508243

Autosomal-recessive omodysplasia: prenatal diagnosis and histomorphometric assessment of the physeal plates of the long bones.

Z Borochowitz1, E Sabo, I Misselevitch, J H Boss.   

Abstract

Second-semester ultrasonography of a female fetus documented short femora and humeri and dislocation of the radii. Based on the clinical and postmortem radiological findings, autosomal-recessive omodysplasia was diagnosed. The physeal plates of the long tubular bones were assessed by computer-assisted image analysis. The dimensions and orientation of the chondrocytic lacunae in the physeal plates of the omodysplastic fetus were compared with those in the physeal plates of fetuses without gross limb abnormalities (oligohydramnios, n = 2; hydrocephalus, n = 2; Down syndrome, n = 1). The pathological characteristics of the omodysplastic physeal plates were an expanded zone of proliferating cartilage and an increased number of closely packed, small chondrocytes. We propose that a genetic, functional deficiency of the physeal cells, underlying the short-limbed dwarfism of autosomal-recessive omodysplasia, is partially compensated, albeit ineffectively, by an increased number of small chondrocytes in the proliferating zone of the physeal plate.

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Year:  1998        PMID: 9508243     DOI: 10.1002/(sici)1096-8628(19980319)76:3<238::aid-ajmg7>3.0.co;2-m

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


  4 in total

1.  A mutation in FRIZZLED2 impairs Wnt signaling and causes autosomal dominant omodysplasia.

Authors:  Howard M Saal; Cynthia A Prows; Iris Guerreiro; Milene Donlin; Luke Knudson; Kristen L Sund; Ching-Fang Chang; Samantha A Brugmann; Rolf W Stottmann
Journal:  Hum Mol Genet       Date:  2015-03-10       Impact factor: 6.150

Review 2.  Recessive omodysplasia: five new cases and review of the literature.

Authors:  Nursel H Elçioglu; Karl H Gustavson; Andrew O M Wilkie; Memune Yüksel-Apak; Jürgen W Spranger
Journal:  Pediatr Radiol       Date:  2003-10-18

3.  Mutations in the heparan-sulfate proteoglycan glypican 6 (GPC6) impair endochondral ossification and cause recessive omodysplasia.

Authors:  Ana Belinda Campos-Xavier; Danielle Martinet; John Bateman; Dan Belluoccio; Lynn Rowley; Tiong Yang Tan; Alica Baxová; Karl-Henrik Gustavson; Zvi U Borochowitz; A Micheil Innes; Sheila Unger; Jacques S Beckmann; Lauréane Mittaz; Diana Ballhausen; Andrea Superti-Furga; Ravi Savarirayan; Luisa Bonafé
Journal:  Am J Hum Genet       Date:  2009-05-28       Impact factor: 11.025

4.  Glypican-6 promotes the growth of developing long bones by stimulating Hedgehog signaling.

Authors:  Mariana Capurro; Tomomi Izumikawa; Philippe Suarez; Wen Shi; Marzena Cydzik; Tomoyuki Kaneiwa; Jean Gariepy; Luisa Bonafe; Jorge Filmus
Journal:  J Cell Biol       Date:  2017-07-10       Impact factor: 10.539

  4 in total

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