Literature DB >> 9714007

Smith-Lemli-Opitz syndrome: phenotypic extreme with minimal clinical findings.

M J Nowaczyk1, D T Whelan, R E Hill.   

Abstract

Smith-Lemli-Opitz syndrome (SLO) is caused by inherited enzymatic deficiency of 7-dehydrocholesterol-delta7-reductase and resultant cholesterol deficiency. It comprises a characteristic combination of facial features, malformations, and mental retardation. We report on three related patients (two brothers and their first cousin) with mental retardation and minimal physical signs in whom the diagnosis of SLO was delayed for a number of years. The presence of a third-degree relative in the absence of consanguinity in this family supports the proposed high population carrier frequency. Our report suggests that cases of mild SLO remain undiagnosed and untreated, and that awareness of this common cause of mental retardation is low.

Entities:  

Mesh:

Substances:

Year:  1998        PMID: 9714007     DOI: 10.1002/(sici)1096-8628(19980806)78:5<419::aid-ajmg5>3.0.co;2-g

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


  9 in total

Review 1.  Malformation syndromes caused by disorders of cholesterol synthesis.

Authors:  Forbes D Porter; Gail E Herman
Journal:  J Lipid Res       Date:  2010-10-07       Impact factor: 5.922

2.  Smith-Lemli-Opitz syndrome with a classical phenotype, oesophageal achalasia and borderline plasma sterol concentrations.

Authors:  D Haas; S Armbrust; J-P Haas; J Zschocke; K Mühlmann; C Fusch; L M Neumann
Journal:  J Inherit Metab Dis       Date:  2005       Impact factor: 4.982

Review 3.  The Smith-Lemli-Opitz syndrome.

Authors:  R I Kelley; R C Hennekam
Journal:  J Med Genet       Date:  2000-05       Impact factor: 6.318

4.  The cytochrome P450scc system opens an alternate pathway of vitamin D3 metabolism.

Authors:  Andrzej Slominski; Igor Semak; Jordan Zjawiony; Jacobo Wortsman; Wei Li; Andre Szczesniewski; Robert C Tuckey
Journal:  FEBS J       Date:  2005-08       Impact factor: 5.542

Review 5.  Smith-Lemli-Opitz syndrome: a treatable inherited error of metabolism causing mental retardation.

Authors:  M J Nowaczyk; D T Whelan; T W Heshka; R E Hill
Journal:  CMAJ       Date:  1999-07-27       Impact factor: 8.262

6.  Normal cognition and behavior in a Smith-Lemli-Opitz syndrome patient who presented with Hirschsprung disease.

Authors:  C Mueller; S Patel; M Irons; K Antshel; G Salen; G S Tint; C Bay
Journal:  Am J Med Genet A       Date:  2003-11-15       Impact factor: 2.802

7.  Biochemical and Physiological Improvement in a Mouse Model of Smith-Lemli-Opitz Syndrome (SLOS) Following Gene Transfer with AAV Vectors.

Authors:  Lee Ying; Xavier Matabosch; Montserrat Serra; Berna Watson; Cedric Shackleton; Gordon Watson
Journal:  Mol Genet Metab Rep       Date:  2014

8.  Reduced cholesterol levels impair Smoothened activation in Smith-Lemli-Opitz syndrome.

Authors:  Robert Blassberg; James I Macrae; James Briscoe; John Jacob
Journal:  Hum Mol Genet       Date:  2015-12-18       Impact factor: 6.150

9.  Sequential metabolism of 7-dehydrocholesterol to steroidal 5,7-dienes in adrenal glands and its biological implication in the skin.

Authors:  Andrzej T Slominski; Michal A Zmijewski; Igor Semak; Trevor Sweatman; Zorica Janjetovic; Wei Li; Jordan K Zjawiony; Robert C Tuckey
Journal:  PLoS One       Date:  2009-02-03       Impact factor: 3.240

  9 in total

北京卡尤迪生物科技股份有限公司 © 2022-2023.