Literature DB >> 8284113

Mutations in the type II 3 beta-hydroxysteroid dehydrogenase gene in a patient with classic salt-wasting 3 beta-hydroxysteroid dehydrogenase deficiency congenital adrenal hyperplasia.

Y T Chang1, M S Kappy, K Iwamoto, J Wang, X Yang, S Pang.   

Abstract

Inherited adrenal and gonadal 3 beta-hydroxysteroid dehydrogenase (3 beta-HSD) deficiency is most likely caused by a mutation of the type II 3 beta-HSD gene. Cloning and sequencing of exons I-II, III, and IV and portions of the adjacent introns, amplified by polymerase chain reaction using primers specific for the type II gene, in one male pseudohermaphrodite with salt-wasting classic 3 beta-HSD deficiency congenital adrenal hyperplasia revealed the same mutation in all nine clones of exon IV consisting of a missense mutation at codon 248 [GTC(Val)-->AAC(Asn)] followed by a frameshift mutation at codon 249 [CGA (Arg)-->TA], resulting in a stop codon TAG, and normal sequences of exon I-II and III and the adjacent portions of introns. The same codon 248 and 249 mutations were found on one clone of his mother's DNA, but two other clones revealed normal sequences. These data indicate a homozygous combined missense/frameshift mutation in exon IV of the type II 3 beta-HSD gene resulting in severe salt-wasting adrenal and gonadal 3 beta-HSD deficiency in the patient.

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Year:  1993        PMID: 8284113     DOI: 10.1203/00006450-199311000-00026

Source DB:  PubMed          Journal:  Pediatr Res        ISSN: 0031-3998            Impact factor:   3.756


  5 in total

1.  The genetic basis of adrenal gland weight and structure in BXD recombinant inbred mice.

Authors:  Domenico L Di Curzio; Daniel Goldowitz
Journal:  Mamm Genome       Date:  2011-02-24       Impact factor: 2.957

Review 2.  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

3.  Genotype, Mortality, Morbidity, and Outcomes of 3β-Hydroxysteroid Dehydrogenase Deficiency in Algeria.

Authors:  Asmahane Ladjouze; Malcolm Donaldson; Ingrid Plotton; Nacima Djenane; Kahina Mohammedi; Véronique Tardy-Guidollet; Delphine Mallet; Kamélia Boulesnane; Zair Bouzerar; Yves Morel; Florence Roucher-Boulez
Journal:  Front Endocrinol (Lausanne)       Date:  2022-06-10       Impact factor: 6.055

4.  Testicular Adrenal Rest Tumor in Two Brothers with a Novel Mutation in the 3-Beta-Hydroxysteroid Dehydrogenase-2 Gene.

Authors:  Ayla Güven; Seher Polat
Journal:  J Clin Res Pediatr Endocrinol       Date:  2016-07-29

Review 5.  Genetics of congenital adrenal hyperplasia.

Authors:  Nils Krone; Wiebke Arlt
Journal:  Best Pract Res Clin Endocrinol Metab       Date:  2009-04       Impact factor: 4.690

  5 in total

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