Literature DB >> 17466088

Congenital adrenal hyperplasia: focus on the molecular basis of 21-hydroxylase deficiency.

João Gonçalves1, Ana Friães, Luís Moura.   

Abstract

Congenital adrenal hyperplasia (CAH) is an autosomal recessive disorder caused by defects in one of several steroidogenic enzymes involved in the synthesis of cortisol from cholesterol in the adrenal glands. More than 90% of cases are caused by 21-hydroxylase deficiency, and the severity of the resulting clinical symptoms varies according to the level of 21-hydroxylase activity. 21-Hydroxylase deficiency is usually caused by mutations in the CYP21A2 gene, which is located on the RCCX module, a chromosomal region highly prone to genetic recombination events that can result in a wide variety of complex rearrangements, such as gene duplications, gross deletions and gene conversions of variable extensions. Molecular genotyping of CYP21A2 and the RCCX module has proved useful for a more accurate diagnosis of the disease, and prenatal diagnosis. This article summarises the clinical features of 21-hydroxylase deficiency, explains current understanding of the disease at the molecular level, and highlights recent developments, particularly in diagnosis.

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Year:  2007        PMID: 17466088     DOI: 10.1017/S1462399407000300

Source DB:  PubMed          Journal:  Expert Rev Mol Med        ISSN: 1462-3994            Impact factor:   5.600


  8 in total

1.  Mutation analysis of the CYP21A2 gene in the Iranian population.

Authors:  Bahareh Rabbani; Nejat Mahdieh; Mohammad Tahgi Haghi Ashtiani; Bagher Larijani; Mohammad Taghi Akbari; Maria New; Alan Parsa; Jan P Schouten; Ali Rabbani
Journal:  Genet Test Mol Biomarkers       Date:  2011-10-21

2.  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

3.  A large view of CYP21 locus among Sicilians and other populations: identification of a novel CYP21A2 variant in Sicily.

Authors:  M Niceta; M Bono; C Fabiano; F Pojero; F Niceta; P Sammarco; G Corsello; P Garofalo
Journal:  J Endocrinol Invest       Date:  2010-12-15       Impact factor: 4.256

4.  Great genotypic and phenotypic diversities associated with copy-number variations of complement C4 and RP-C4-CYP21-TNX (RCCX) modules: a comparison of Asian-Indian and European American populations.

Authors:  Kapil Saxena; Kathryn J Kitzmiller; Yee Ling Wu; Bi Zhou; Nazreen Esack; Leena Hiremath; Erwin K Chung; Yan Yang; C Yung Yu
Journal:  Mol Immunol       Date:  2009-01-09       Impact factor: 4.407

5.  Molecular genetic study of congenital adrenal hyperplasia in Serbia: novel p.Leu129Pro and p.Ser165Pro CYP21A2 gene mutations.

Authors:  I Milacic; M Barac; T Milenkovic; M Ugrin; K Klaassen; A Skakic; M Jesic; I Joksic; K Mitrovic; S Todorovic; S Vujovic; S Pavlovic; M Stojiljkovic
Journal:  J Endocrinol Invest       Date:  2015-08-02       Impact factor: 4.256

6.  A novel 9 bp deletion (c.1271_1279delGTGCCCGCG) in exon 10 of CYP21A2 gene causing severe congenital adrenal hyperplasia.

Authors:  Violeta Anastasovska; Mirjana Kocova; Nikolina Zdraveska; Maja Stojiljkovic; Anita Skakic; Kristel Klaassen; Sonja Pavlovic
Journal:  Endocrine       Date:  2021-03-14       Impact factor: 3.633

7.  Molecular Diagnosis of Congenital Adrenal Hyperplasia in Iran: Focusing on CYP21A2 Gene.

Authors:  Bahareh Rabbani; Nejat Mahdieh; Mohammad-Taghi Haghi Ashtiani; Mohammad-Taghi Akbari; Ali Rabbani
Journal:  Iran J Pediatr       Date:  2011-06       Impact factor: 0.364

8.  Novel p53 target genes secreted by the liver are involved in non-cell-autonomous regulation.

Authors:  M Charni; A Molchadsky; I Goldstein; H Solomon; P Tal; N Goldfinger; P Yang; Z Porat; G Lozano; V Rotter
Journal:  Cell Death Differ       Date:  2015-09-11       Impact factor: 15.828

  8 in total

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