Literature DB >> 21635882

Endocrine profile and phenotype-genotype correlation in unrelated patients with non-classical congenital adrenal hyperplasia.

Nicos Skordis1, Christos Shammas, Elisavet Efstathiou, Katerina Kaffe, Vassos Neocleous, Leonidas A Phylactou.   

Abstract

OBJECTIVES: The aim of this study was to identify the molecular defect in a group of 37 unrelated Greek Cypriot patients affected by NC-CAH and evaluate the relationship between the genotype, phenotype and adrenal androgen levels. DESIGN AND METHODS: Clinical evaluation, biochemical analysis of 17-OHP, Testosterone, Androstenedione, DHEA-S, direct DNA sequencing and MLPA analyses.
RESULTS: Eleven known mutations were identified with the p.V281L being the most predominant and observed in 68.9% of the alleles. There was no difference between the two genotypes (mild/mild and mild/severe) with clinical presentation, whereas a proportional relationship between the type of mutation and adrenal androgen levels was found.
CONCLUSION: The frequency of the underlying genetic defect in our patients with NC-CAH is similar to that observed in most Mediterranean populations. Although the genotype cannot solely explain the clinical expression of NC-CAH, discrimination between mild and severe alleles is crucial in antenatal diagnosis and genetic counselling.
Copyright © 2011 The Canadian Society of Clinical Chemists. Published by Elsevier Inc. All rights reserved.

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Year:  2011        PMID: 21635882     DOI: 10.1016/j.clinbiochem.2011.05.013

Source DB:  PubMed          Journal:  Clin Biochem        ISSN: 0009-9120            Impact factor:   3.281


  6 in total

Review 1.  Adopting orphans: comprehensive genetic testing of Mendelian diseases of childhood by next-generation sequencing.

Authors:  Stephen F Kingsmore; Darrell L Dinwiddie; Neil A Miller; Sarah E Soden; Carol J Saunders
Journal:  Expert Rev Mol Diagn       Date:  2011-11       Impact factor: 5.225

2.  Genetic defects of the CYP21A2 gene in girls with premature adrenarche.

Authors:  N Skordis; C Shammas; A A P Phedonos; A Kyriakou; M Toumba; V Neocleous; L A Phylactou
Journal:  J Endocrinol Invest       Date:  2014-12-07       Impact factor: 4.256

3.  Phenotypic variability of hyperandrogenemia in females heterozygous for CYP21A2 mutations.

Authors:  Vassos Neocleous; Christos Shammas; Alexia Ap Phedonos; Leonidas A Phylactou; Nicos Skordis
Journal:  Indian J Endocrinol Metab       Date:  2014-11

4.  Variations in the 3'UTR of the CYP21A2 Gene in Heterozygous Females with Hyperandrogenaemia.

Authors:  Vassos Neocleous; Pavlos Fanis; Meropi Toumba; Alexia A P Phedonos; Michalis Picolos; Elena Andreou; Tassos C Kyriakides; George A Tanteles; Christos Shammas; Leonidas A Phylactou; Nicos Skordis
Journal:  Int J Endocrinol       Date:  2017-04-12       Impact factor: 3.257

Review 5.  Metabolic Perspectives for Non-classical Congenital Adrenal Hyperplasia With Relation to the Classical Form of the Disease.

Authors:  Djuro Macut; Vera Zdravković; Jelica Bjekić-Macut; George Mastorakos; Duarte Pignatelli
Journal:  Front Endocrinol (Lausanne)       Date:  2019-10-02       Impact factor: 5.555

6.  Prevalence of nonclassic congenital adrenal hyperplasia in Turkish children presenting with premature pubarche, hirsutism, or oligomenorrhoea.

Authors:  Cigdem Binay; Enver Simsek; Oguz Cilingir; Zafer Yuksel; Ozden Kutlay; Sevilhan Artan
Journal:  Int J Endocrinol       Date:  2014-03-23       Impact factor: 3.257

  6 in total

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