Total submissions: 1
Submitter | RCV | SCV | Clinical significance | Condition | Last evaluated | Review status | Method | Comment |
---|---|---|---|---|---|---|---|---|
Victorian Clinical Genetics Services, |
RCV004594931 | SCV005086888 | pathogenic | Classic congenital adrenal hyperplasia due to 21-hydroxylase deficiency | 2023-07-17 | criteria provided, single submitter | clinical testing | Based on the classification scheme VCGS_Germline_v1.3.4, this variant is classified as Pathogenic. Following criteria are met: 0102 - Loss of function is a known mechanism of disease in this gene and is associated with congenital adrenal hyperplasia due to 21-hydroxylase deficiency (MIM#201910) and nonclassic type hyperandrogenism due to 21-hydroxylase deficiency (MIM#201910). (I) 0106 - This gene is associated with autosomal recessive disease. (I) 0204 - Variant is predicted to result in a truncated protein (premature termination codon is located within the first 102 nucleotides of the coding sequence and is predicted to escape nonsense-mediated decay). (SP) 0251 - This variant is heterozygous. (I) 0304 - Variant is present in gnomAD <0.01 for a recessive condition (v3: c.59G>A with 1 heterozygote, 0 homozygotes; c.60G>A with 3 heterozygotes, 0 homozygotes). (SP) 0801 - This variant has strong previous evidence of pathogenicity in unrelated individuals. This nucleotide change and c.60G>A, both predicted to result in p.(Trp20*), have been reported in multiple unrelated individuals with congenital adrenal hyperplasia (PMIDs: 27185867, 30048636, 30816000), and have been reported as pathogenic by a clinical laboratory (ClinVar). Both nucleotide changes have been listed as pathogenic in the EMQN guideline for molecular genetic testing and reporting of 21-hydroxylase deficiency (PMID: 32616876). c.59G>A has also been reported as in cis with p.(Val282Leu) although that is uncommon (PMID: 27185867). (SP) 1208 - Inheritance information for this variant is not currently available in this individual. (I) Legend: (SP) - Supporting pathogenic, (I) - Information, (SB) - Supporting benign |