Literature DB >> 8081391

Mutations in steroid 21-hydroxylase (CYP21).

P C White1, M T Tusie-Luna, M I New, P W Speiser.   

Abstract

The inherited inability to synthesize cortisol is termed congenital adrenal hyperplasia. More than 90% of cases are caused by 21-hydroxylase deficiency. This syndrome is characterized by signs of androgen excess and often mineralocorticoid deficiency. Steroid 21-hydroxylase (P450c21) is a microsomal enzyme expressed in the adrenal gland that catalyzes conversion of 17-hydroxyprogesterone and progesterone to 11-deoxycortisol and deoxycorticosterone respectively. In man, this enzyme is encoded by the CYP21 (CYP21B) gene which is located in the HLA major histocompatibility complex along with a pseudogene, CYP21P (CYP21A). Mutations in CYP21 causing congenital adrenal hyperplasia are almost all generated by recombinations between CYP21 and CYP21P. These recombinations either delete CYP21 or transfer deleterious mutations from CYP21P to CYP21, a process termed apparent gene conversion. The degree of enzymatic compromise caused by each mutation is correlated with the clinical severity of the deficiency observed in patients carrying that mutation.

Entities:  

Mesh:

Substances:

Year:  1994        PMID: 8081391     DOI: 10.1002/humu.1380030408

Source DB:  PubMed          Journal:  Hum Mutat        ISSN: 1059-7794            Impact factor:   4.878


  19 in total

Review 1.  An overview of molecular diagnosis of steroid 21-hydroxylase deficiency.

Authors:  C E Keegan; A A Killeen
Journal:  J Mol Diagn       Date:  2001-05       Impact factor: 5.568

2.  Analysis of CYP21 coding polymorphisms in three ethnic populations: further evidence of nonamplifying CYP21 alleles among whites.

Authors:  I C Ozturk; W L Wei; L Palaniappan; M Rubenfire; A A Killeen
Journal:  Mol Diagn       Date:  2000-03

3.  In Sicilian ethnic group non-classical congenital adrenal hyperplasia is frequently associated with a very mild genotype.

Authors:  M Wasniewska; G Di Pasquale; I Rulli; G Salzano; M Caruso; S Indovina; L Di Pasquale; G Zirilli; F De Luca
Journal:  J Endocrinol Invest       Date:  2007-03       Impact factor: 4.256

4.  Ethnic-specific distribution of mutations in 716 patients with congenital adrenal hyperplasia owing to 21-hydroxylase deficiency.

Authors:  Robert C Wilson; Saroj Nimkarn; Miro Dumic; Jihad Obeid; Maryam Razzaghy Azar; Maryam Azar; Hossein Najmabadi; Fatemeh Saffari; Maria I New
Journal:  Mol Genet Metab       Date:  2007-02-01       Impact factor: 4.797

5.  The clinical and biochemical spectrum of congenital adrenal hyperplasia secondary to 21-hydroxylase deficiency.

Authors:  Tony Huynh; Ivan McGown; David Cowley; Ohn Nyunt; Gary M Leong; Mark Harris; Andrew M Cotterill
Journal:  Clin Biochem Rev       Date:  2009-05

Review 6.  The molecular biology, biochemistry, and physiology of human steroidogenesis and its disorders.

Authors:  Walter L Miller; Richard J Auchus
Journal:  Endocr Rev       Date:  2010-11-04       Impact factor: 19.871

7.  Identification of mutations in the duplicated region of the polycystic kidney disease 1 gene (PKD1) by a novel approach.

Authors:  B Peral; V Gamble; C Strong; A C Ong; J Sloane-Stanley; K Zerres; C G Winearls; P C Harris
Journal:  Am J Hum Genet       Date:  1997-06       Impact factor: 11.025

8.  Comprehensive mutation analysis of the CYP21A2 gene: an efficient multistep approach to the molecular diagnosis of congenital adrenal hyperplasia.

Authors:  Zhi Xu; Wuyan Chen; Deborah P Merke; Nazli B McDonnell
Journal:  J Mol Diagn       Date:  2013-09-23       Impact factor: 5.568

9.  Genotyping of CYP21A2 for congenital adrenal hyperplasia screening using allele-specific primer extension followed by bead array hybridization.

Authors:  Yongtaek Oh; Sung Won Park; Sung-Min Chun; Namkyoo Lim; Ki Sup Ahn; Jong-Ok Ka; Dong-Kyu Jin; Byoung-Don Han
Journal:  Mol Diagn Ther       Date:  2009-12-01       Impact factor: 4.074

Review 10.  Recent advances in diagnosis, treatment, and outcome of congenital adrenal hyperplasia due to 21-hydroxylase deficiency.

Authors:  Felix G Riepe; Wolfgang G Sippell
Journal:  Rev Endocr Metab Disord       Date:  2007-12       Impact factor: 6.514

View more

北京卡尤迪生物科技股份有限公司 © 2022-2023.