Literature DB >> 187366

Sclerosteosis - an autosomal recessive disorder.

P Beighton, J Davidson, L Durr, H Hamersma.   

Abstract

Sclerosteosis is a rare, potentially lethal skeletal disorder in which massive bony over-growth leads to facial distortion, cranial nerve compression and progressive rise in intracranial pressure. Gigantism and syndactyly of the 2nd and 3rd fingers are associated features. In a nationwide investigation in South Africa, 25 affected individuals in 15 Afrikaner kindreds have been studied. The minimum prevalence of the contition in this community is 1 in 75,000. Analysis of pedigree data confirms that sclerosteosis is an autosomal recessive condition. The gene frequency in the Afrikaner people is estimated at 0.0035, with 10,000 clinically normal heterozygotes in this population. Heterozygote detection may be possible on a basis of recognition of minor changes which are apparent on skull radiographs.

Entities:  

Mesh:

Year:  1977        PMID: 187366     DOI: 10.1111/j.1399-0004.1977.tb01269.x

Source DB:  PubMed          Journal:  Clin Genet        ISSN: 0009-9163            Impact factor:   4.438


  20 in total

Review 1.  Role of sclerostin in bone and cartilage and its potential as a therapeutic target in bone diseases.

Authors:  E Michael Lewiecki
Journal:  Ther Adv Musculoskelet Dis       Date:  2014-04       Impact factor: 5.346

Review 2.  Sclerostin and skeletal health.

Authors:  Maryam Sharifi; Lisa Ereifej; E Michael Lewiecki
Journal:  Rev Endocr Metab Disord       Date:  2015-06       Impact factor: 6.514

3.  Genomic deletion of a long-range bone enhancer misregulates sclerostin in Van Buchem disease.

Authors:  Gabriela G Loots; Michaela Kneissel; Hansjoerg Keller; Myma Baptist; Jessie Chang; Nicole M Collette; Dmitriy Ovcharenko; Ingrid Plajzer-Frick; Edward M Rubin
Journal:  Genome Res       Date:  2005-06-17       Impact factor: 9.043

4.  Sclerosteosis.

Authors:  P Beighton
Journal:  J Med Genet       Date:  1988-03       Impact factor: 6.318

Review 5.  TGF-β Family Signaling in Connective Tissue and Skeletal Diseases.

Authors:  Elena Gallo MacFarlane; Julia Haupt; Harry C Dietz; Eileen M Shore
Journal:  Cold Spring Harb Perspect Biol       Date:  2017-11-01       Impact factor: 10.005

6.  Van Buchem disease (hyperostosis corticalis generalisata) maps to chromosome 17q12-q21.

Authors:  W Van Hul; W Balemans; E Van Hul; F G Dikkers; H Obee; R J Stokroos; P Hildering; F Vanhoenacker; G Van Camp; P J Willems
Journal:  Am J Hum Genet       Date:  1998-02       Impact factor: 11.025

Review 7.  Craniotubular bone disorders.

Authors:  R J Gorlin
Journal:  Pediatr Radiol       Date:  1994

Review 8.  Alterations in vitamin D metabolite, parathyroid hormone and fibroblast growth factor-23 concentrations in sclerostin-deficient mice permit the maintenance of a high bone mass.

Authors:  Zachary C Ryan; Theodore A Craig; Meghan McGee-Lawrence; Jennifer J Westendorf; Rajiv Kumar
Journal:  J Steroid Biochem Mol Biol       Date:  2014-11-22       Impact factor: 4.292

Review 9.  Wnt modulators in the biotech pipeline.

Authors:  Jean-Philippe Rey; Debra L Ellies
Journal:  Dev Dyn       Date:  2010-01       Impact factor: 3.780

10.  A host of hypercholesterolaemic homozygotes in South Africa.

Authors:  H C Seftel; S G Baker; M P Sandler; M B Forman; B I Joffe; D Mendelsohn; T Jenkins; C J Mieny
Journal:  Br Med J       Date:  1980-09-06
View more

北京卡尤迪生物科技股份有限公司 © 2022-2023.