Literature DB >> 29450859

Congenital Adrenal Hyperplasia (CAH) due to 21-Hydroxylase Deficiency: A Comprehensive Focus on 233 Pathogenic Variants of CYP21A2 Gene.

Paola Concolino1,2, Alessandra Costella3.   

Abstract

Congenital adrenal hyperplasia (CAH) comprises a group of autosomal recessive disorders caused by complete or partial defects in one of the several steroidogenic enzymes involved in the synthesis of cortisol from cholesterol in the adrenal glands. More than 95-99% of all cases of CAH are caused by deficiency of steroid 21-hydroxylase, an enzyme encoded by the CYP21A2 gene. Currently, CYP21A2 genotyping is considered a valuable complement to biochemical investigations in the diagnosis of 21-hydroxylase deficiency. More than 200 mutations have been described in literature reports, and much energy is still focused on the clinical classification of new variants. In this review, we focus on molecular genetic features of 21-hydroxylase deficiency, performing an extensive survey of all clinical pathogenic variants modifying the whole sequence of the CYP21A2 gene. Our aim is to offer a very useful tool for clinical and genetic specialists in order to ease clinical diagnosis and genetic counseling.

Entities:  

Mesh:

Substances:

Year:  2018        PMID: 29450859     DOI: 10.1007/s40291-018-0319-y

Source DB:  PubMed          Journal:  Mol Diagn Ther        ISSN: 1177-1062            Impact factor:   4.074


  82 in total

1.  Modular variations of the human major histocompatibility complex class III genes for serine/threonine kinase RP, complement component C4, steroid 21-hydroxylase CYP21, and tenascin TNX (the RCCX module). A mechanism for gene deletions and disease associations.

Authors:  Z Yang; A R Mendoza; T R Welch; W B Zipf; C Y Yu
Journal:  J Biol Chem       Date:  1999-04-23       Impact factor: 5.157

Review 2.  Impact of molecular genetics on congenital adrenal hyperplasia management.

Authors:  A Balsamo; L Baldazzi; S Menabò; A Cicognani
Journal:  Sex Dev       Date:  2010-07-15       Impact factor: 1.824

3.  Functional characterization of two novel point mutations in the CYP21 gene causing simple virilizing forms of congenital adrenal hyperplasia due to 21-hydroxylase deficiency.

Authors:  Nils Krone; Felix G Riepe; Joachim Grötzinger; Carl-Joachim Partsch; Wolfgang G Sippell
Journal:  J Clin Endocrinol Metab       Date:  2004-10-13       Impact factor: 5.958

4.  Duplications of the functional CYP21A2 gene are primarily restricted to Q318X alleles: evidence for a founder effect.

Authors:  S Kleinle; R Lang; G F Fischer; H Vierhapper; F Waldhauser; M Födinger; S M Baumgartner-Parzer
Journal:  J Clin Endocrinol Metab       Date:  2009-09-22       Impact factor: 5.958

5.  Microconversion between CYP21A2 and CYP21A1P promoter regions causes the nonclassical form of 21-hydroxylase deficiency.

Authors:  Rogério S Araújo; Berenice B Mendonca; Angela S Barbosa; Chin J Lin; José A M Marcondes; Ana Elisa C Billerbeck; Tânia A S S Bachega
Journal:  J Clin Endocrinol Metab       Date:  2007-07-31       Impact factor: 5.958

Review 6.  Steroid 21-hydroxylase deficiency in congenital adrenal hyperplasia.

Authors:  Alan A Parsa; Maria I New
Journal:  J Steroid Biochem Mol Biol       Date:  2016-07-02       Impact factor: 4.292

Review 7.  Congenital adrenal hyperplasia.

Authors:  Diala El-Maouche; Wiebke Arlt; Deborah P Merke
Journal:  Lancet       Date:  2017-05-30       Impact factor: 79.321

8.  Functional studies of novel CYP21A2 mutations detected in Norwegian patients with congenital adrenal hyperplasia.

Authors:  Ingeborg Brønstad; Lars Breivik; Paal Methlie; Anette S B Wolff; Eirik Bratland; Ingrid Nermoen; Kristian Løvås; Eystein S Husebye
Journal:  Endocr Connect       Date:  2014-04-15       Impact factor: 3.335

Review 9.  Recent advances in biochemical and molecular analysis of congenital adrenal hyperplasia due to 21-hydroxylase deficiency.

Authors:  Jin-Ho Choi; Gu-Hwan Kim; Han-Wook Yoo
Journal:  Ann Pediatr Endocrinol Metab       Date:  2016-03-31

10.  Genotype-phenotype correlation in 153 adult patients with congenital adrenal hyperplasia due to 21-hydroxylase deficiency: analysis of the United Kingdom Congenital adrenal Hyperplasia Adult Study Executive (CaHASE) cohort.

Authors:  Nils Krone; Ian T Rose; Debbie S Willis; James Hodson; Sarah H Wild; Emma J Doherty; Stefanie Hahner; Silvia Parajes; Roland H Stimson; Thang S Han; Paul V Carroll; Gerry S Conway; Brian R Walker; Fiona MacDonald; Richard J Ross; Wiebke Arlt
Journal:  J Clin Endocrinol Metab       Date:  2013-01-21       Impact factor: 5.958

View more
  23 in total

Review 1.  Mutational Landscape of Resistance to Thyroid Hormone Beta (RTHβ).

Authors:  Paola Concolino; Alessandra Costella; Rosa Maria Paragliola
Journal:  Mol Diagn Ther       Date:  2019-06       Impact factor: 4.074

2.  Detection of a novel severe mutation affecting the CYP21A2 gene in a Chilean male with salt wasting congenital adrenal hyperplasia.

Authors:  Eugenio Arteaga; Felipe Valenzuela; Carlos F Lagos; Marcela Lagos; Alejandra Martinez; Rene Baudrand; Cristian Carvajal; Carlos E Fardella
Journal:  Endocrine       Date:  2019-09-30       Impact factor: 3.633

3.  Issues with the Detection of Large Genomic Rearrangements in Molecular Diagnosis of 21-Hydroxylase Deficiency.

Authors:  Paola Concolino
Journal:  Mol Diagn Ther       Date:  2019-10       Impact factor: 4.074

4.  Prevalence of CAH-X Syndrome in Italian Patients with Congenital Adrenal Hyperplasia (CAH) Due to 21-Hydroxylase Deficiency.

Authors:  Rosa Maria Paragliola; Alessia Perrucci; Laura Foca; Andrea Urbani; Paola Concolino
Journal:  J Clin Med       Date:  2022-07-01       Impact factor: 4.964

5.  Salt-wasting congenital adrenal hyperplasia phenotype as a result of the TNXA/TNXB chimera 1 (CAH-X CH-1) and the pathogenic IVS2-13A/C > G in CYP21A2 gene.

Authors:  Pavlos Fanis; Nicos Skordis; Leonidas A Phylactou; Vassos Neocleous
Journal:  Hormones (Athens)       Date:  2022-10-20       Impact factor: 3.419

Review 6.  Overcoming challenges and dogmas to understand the functions of pseudogenes.

Authors:  Seth W Cheetham; Geoffrey J Faulkner; Marcel E Dinger
Journal:  Nat Rev Genet       Date:  2019-12-17       Impact factor: 53.242

7.  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

8.  Molecular Analysis of 21-Hydroxylase Deficiency Reveals Two Novel Severe Genotypes in Affected Newborns.

Authors:  Paola Concolino; Rosa Maria Paragliola
Journal:  Mol Diagn Ther       Date:  2021-03-12       Impact factor: 4.074

Review 9.  Molecular Diagnosis of Steroid 21-Hydroxylase Deficiency: A Practical Approach.

Authors:  María Arriba; Begoña Ezquieta
Journal:  Front Endocrinol (Lausanne)       Date:  2022-03-29       Impact factor: 5.555

Review 10.  Clinical outcomes and characteristics of P30L mutations in congenital adrenal hyperplasia due to 21-hydroxylase deficiency.

Authors:  Mirjana Kocova; Violeta Anastasovska; Henrik Falhammar
Journal:  Endocrine       Date:  2020-05-05       Impact factor: 3.633

View more

北京卡尤迪生物科技股份有限公司 © 2022-2023.