Literature DB >> 19205729

Multiple malformations: a possible Sonic hedgehog phenotype?

Helen Wainwright1, Peter Beighton.   

Abstract

A malformed foetus delivered after 21-week gestation had multiple gross craniofacial, midline and limb abnormalities. At autopsy, obstructive hydrocephalus and colonic atresia were observed. Radiographs revealed extensive epiphyseal stippling which might indicate abnormalities of sterol synthesis. The pattern of malformation in this foetus is similar to the phenotype for mutations in the Sonic hedgehog gene in animal models. It can be speculated that the abnormalities in the foetus could have resulted from disruption of the Sonic hedgehog pathway by molecular, chromosomal or environmental factors.

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Year:  2009        PMID: 19205729     DOI: 10.1007/s00428-009-0735-9

Source DB:  PubMed          Journal:  Virchows Arch        ISSN: 0945-6317            Impact factor:   4.064


  6 in total

1.  Abnormal sterol metabolism in patients with Conradi-Hünermann-Happle syndrome and sporadic lethal chondrodysplasia punctata.

Authors:  R I Kelley; W G Wilcox; M Smith; L E Kratz; A Moser; D S Rimoin
Journal:  Am J Med Genet       Date:  1999-03-19

Review 2.  Essential roles of Gli3 and sonic hedgehog in pattern formation and developmental anomalies caused by their dysfunction.

Authors:  Jun Motoyama
Journal:  Congenit Anom (Kyoto)       Date:  2006-09       Impact factor: 1.409

3.  Severe facial clefting in Insig-deficient mouse embryos caused by sterol accumulation and reversed by lovastatin.

Authors:  Luke J Engelking; Bret M Evers; James A Richardson; Joseph L Goldstein; Michael S Brown; Guosheng Liang
Journal:  J Clin Invest       Date:  2006-09       Impact factor: 14.808

4.  Cyclopia and defective axial patterning in mice lacking Sonic hedgehog gene function.

Authors:  C Chiang; Y Litingtung; E Lee; K E Young; J L Corden; H Westphal; P A Beachy
Journal:  Nature       Date:  1996-10-03       Impact factor: 49.962

Review 5.  Human malformation syndromes due to inborn errors of cholesterol synthesis.

Authors:  Forbes D Porter
Journal:  Curr Opin Pediatr       Date:  2003-12       Impact factor: 2.856

Review 6.  Defects of blastogenesis.

Authors:  John M Opitz; Ginevra Zanni; James F Reynolds; Enid Gilbert-Barness
Journal:  Am J Med Genet       Date:  2002-12-30
  6 in total
  1 in total

1.  Lethal epiphyseal stippling in the foetus and neonate; pathological implications.

Authors:  Helen Wainwright; Peter Beighton
Journal:  Virchows Arch       Date:  2010-03       Impact factor: 4.064

  1 in total

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