Literature DB >> 1897572

Osteochondrodysplasia with rhizomelia, platyspondyly, callosal agenesis, thrombocytopenia, hydrocephalus, and hypertension.

O M Faye-Petersen1, K Ward, J C Carey, A S Knisely.   

Abstract

A female infant born at term to phenotypically normal nonconsanguinous parents had hypertension, thrombocytopenia, hydrocephalus, callosal agenesis, and nonlethal rhizomelic osteochondrodysplasia. Her osteochondrodysplasia was characterized roentgenographically by shortening and metaphyseal broadening of long bones, without bowing, and by platyspondyly, with deficient ossification of dorsal and central portions of vertebral bodies. By light microscopy, the iliac crest growth plate showed expansion of the zone of chondrocyte hypertrophy and degeneration, with faulty columnar alignment, sparse vascular ingrowth, and irregular mineralization at the zone of chondroosseous transformation. These findings appear to define a novel osteochondrodysplasia, which in association with hypertension, thrombocytopenia, hydrocephalus, and callosal agenesis may constitute a new syndrome.

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Year:  1991        PMID: 1897572     DOI: 10.1002/ajmg.1320400212

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


  2 in total

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2.  Clinical and molecular findings in three Japanese patients with N-acetylneuraminic acid synthetase-congenital disorder of glycosylation (NANS-CDG).

Authors:  Yohei Masunaga; Gen Nishimura; Koji Takahashi; Tomiyuki Hishiyama; Masatoshi Imamura; Kenichi Kashimada; Machiko Kadoya; Yoshinao Wada; Nobuhiko Okamoto; Daiju Oba; Hirofumi Ohashi; Mitsuru Ikeno; Yuko Sakamoto; Maki Fukami; Hirotomo Saitsu; Tsutomu Ogata
Journal:  Sci Rep       Date:  2022-10-12       Impact factor: 4.996

  2 in total

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