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A Case of Salt-Wasting Congenital Adrenal Hyperplasia Caused by a Rare Intronic Variant in the CYP21A2 Gene

Title: A Case of Salt-Wasting Congenital Adrenal Hyperplasia Caused by a Rare Intronic Variant in the CYP21A2 Gene
Authors: Zoia Antysheva; Anton Esibov; Ekaterina Avsievich; Ekaterina Petriaikina; Vladimir Yudin; Anton Keskinov; Sergey Yudin; Dmitry Svetlichnyy; Julia Krupinova; Aleksey Ivashechkin; Yulia Katsaran; Mary Woroncow; Veronika Skvortsova; Viktor Bogdanov; Pavel Volchkov
Source: International Journal of Molecular Sciences ; Volume 26 ; Issue 14 ; Pages: 6648
Publisher Information: Multidisciplinary Digital Publishing Institute
Publication Year: 2025
Collection: MDPI Open Access Publishing
Subject Terms: CYP21A2; congenital adrenal hyperplasia; intronic variant; splicing mutations; adrenal insufficiency; NGS; amplicon sequencing
Description: This case report describes a novel intronic mutation, CYP21A2:c.738+75C>T (rs1463196531), identified in a 4-year-old male with congenital adrenal insufficiency, and expands the known mutation spectrum associated with this condition. The patient, born full-term to unrelated parents, presented with adrenal failure within the first month of life, characterized by acute adrenal crisis symptoms such as vomiting, dehydration, weight loss, hypotension, and electrolyte imbalances. Hormonal evaluations confirmed primary adrenocortical insufficiency, necessitating ongoing hydrocortisone and fludrocortisone therapy. Using family trio-based amplicon sequencing of the CYP21A2 gene, we identified compound heterozygosity consisting of a full gene deletion and a novel pathogenic intronic mutation. Additionally, analysis of WGS data was performed to rule out pathogenic variants in genes that might lead to a similar phenotype, thereby eliminating the possibility of other genes contributing to the proband’s disease. This case demonstrates the potential of using amplicon sequencing in molecular genetic diagnostic testing to detect rare intronic variants in the CYP21A2 gene in cases of early-onset adrenal failure. It also contributes to a better understanding of the genetic basis of congenital adrenal hyperplasia (CAH), which remains a significant autosomal recessive disorder affecting cortisol and aldosterone production, with an incidence of 1 in 10,000 to 1 in 15,000 globally.
Document Type: text
File Description: application/pdf
Language: English
Relation: Molecular Genetics and Genomics; https://dx.doi.org/10.3390/ijms26146648
DOI: 10.3390/ijms26146648
Availability: https://doi.org/10.3390/ijms26146648
Rights: https://creativecommons.org/licenses/by/4.0/
Accession Number: edsbas.416723EC
Database: BASE