Literature DB >> 14715874

Molecular genetic analysis of Tunisian patients with a classic form of 21-hydroxylase deficiency: identification of four novel mutations and high prevalence of Q318X mutation.

Maher Kharrat1, Véronique Tardy, Ridha M'Rad, Faouzi Maazoul, Lamia Ben Jemaa, Mohamed Refaï, Yves Morel, Habiba Chaabouni.   

Abstract

Congenital adrenal hyperplasia (CAH) is a group of autosomal recessive disorders mainly due to defects in the steroid 21-hydroxylase (CYP21) gene. To determine the mutational spectrum in the Tunisian CAH population, the CYP21 active gene was analyzed in 51 unrelated patients using our cascade strategy (digestion by restriction enzyme, sequencing). All patients had a classical form of 21-hydroxylase deficiency. Mutations were detected in over 94% of the chromosomes examined. The most frequent mutation in the Tunisian CAH population was found to be Q318X, with large prevalence (35.3%), in contrast to 0.5-13.8% described in other series. Incidence of other mutations does not differ, as previously described: large deletions (19.6%), mutation in intron 2 (17.6%), and I172N (10.8%). Four novel mutations were found in four patients with the salt-wasting form. These four novel mutations include three point mutations that have not been reported to occur in the CYP21P pseudogene: R483W, W19X, 2669insC, and one small conversion of DNA sequence from exon 5 to exon 8. Our results have shown a good genotype/phenotype correlation in the case of most mutations. This is the first report of screening for mutations of 21-hydroxylase gene in the Tunisian population and even in the Arab population.

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Year:  2004        PMID: 14715874     DOI: 10.1210/jc.2003-031056

Source DB:  PubMed          Journal:  J Clin Endocrinol Metab        ISSN: 0021-972X            Impact factor:   5.958


  24 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.  Characterization of novel missense mutations in CYP21 causing congenital adrenal hyperplasia.

Authors:  Tiina Robins; Christine Bellanne-Chantelot; Michela Barbaro; Sylvie Cabrol; Anna Wedell; Svetlana Lajic
Journal:  J Mol Med (Berl)       Date:  2006-11-21       Impact factor: 4.599

3.  Identification and functional characterization of a novel mutation P459H and a rare mutation R483W in the CYP21A2 gene in two Chinese patients with simple virilizing form of congenital adrenal hyperplasia.

Authors:  L Jiang; L L Song; H Wang; J L Wang; P P Wang; H B Zhou; X L Zhang
Journal:  J Endocrinol Invest       Date:  2011-07-12       Impact factor: 4.256

4.  A unique haplotype of RCCX copy number variation: from the clinics of congenital adrenal hyperplasia to evolutionary genetics.

Authors:  Márton Doleschall; Andrea Luczay; Klára Koncz; Kinga Hadzsiev; Éva Erhardt; Ágnes Szilágyi; Zoltán Doleschall; Krisztina Németh; Dóra Török; Zoltán Prohászka; Balázs Gereben; György Fekete; Edit Gláz; Péter Igaz; Márta Korbonits; Miklós Tóth; Károly Rácz; Attila Patócs
Journal:  Eur J Hum Genet       Date:  2017-04-12       Impact factor: 4.246

5.  Mutational characterization of congenital adrenal hyperplasia due to 21-hydroxylase deficiency in Malaysia.

Authors:  P Balraj; P G Lim; H Sidek; L L Wu; A S B Khoo
Journal:  J Endocrinol Invest       Date:  2012-10-01       Impact factor: 4.256

6.  The residue E351 is essential for the activity of human 21-hydroxylase: evidence from a naturally occurring novel point mutation compared with artificial mutants generated by single amino acid substitutions.

Authors:  Nils Krone; Felix G Riepe; Joachim Grötzinger; Carl-Joachim Partsch; Jürgen Brämswig; Wolfgang G Sippell
Journal:  J Mol Med (Berl)       Date:  2005-04-14       Impact factor: 4.599

7.  Genetic profiling of CAH Egyptian children: rapid guide to clinical interpretation of common mutations.

Authors:  F Elmougy; M Elsharkawy; M Hafez; S A Atty; H Baz; A Ibrahim; H Soliman; S Ekladious; M Abdullatif; G Thabet; N Rady; A Afif; A Tolba; Z Zaki; N Musa
Journal:  J Endocrinol Invest       Date:  2020-05-01       Impact factor: 4.256

Review 8.  21-Hydroxylase deficiency: from molecular genetics to clinical presentation.

Authors:  E Trakakis; D Laggas; E Salamalekis; G Creatsas
Journal:  J Endocrinol Invest       Date:  2005-02       Impact factor: 4.256

9.  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

10.  Salt-wasting congenital adrenal hyperplasia: genotypical peculiarities in a Sicilian ethnic group.

Authors:  M Wasniewska; M Caruso; S Indovina; G Crisafulli; S Mirabelli; G Salzano; T Arrigo; F De Luca
Journal:  J Endocrinol Invest       Date:  2008-07       Impact factor: 4.256

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