ClinVar Miner

Submissions for variant NM_000529.2(MC2R):c.409C>T (p.Arg137Trp)

gnomAD frequency: 0.00003  dbSNP: rs104894660
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Total submissions: 5
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Submitter RCV SCV Clinical significance Condition Last evaluated Review status Method Comment
GeneDx RCV001851615 SCV006088101 likely pathogenic not provided 2024-12-12 criteria provided, single submitter clinical testing Published functional studies demonstrate very low activity when stimulated with high doses of ACTH (PMID: 12213892); In silico analysis supports that this missense variant has a deleterious effect on protein structure/function; This variant is associated with the following publications: (PMID: 18840636, 12213892, 26523528, 32952553, 10971458, 15673970, 38430736)
Women's Health and Genetics/Laboratory Corporation of America, LabCorp RCV000003421 SCV005727071 pathogenic Glucocorticoid deficiency 1 2024-11-06 criteria provided, single submitter clinical testing Variant summary: MC2R c.409C>T (p.Arg137Trp) results in a non-conservative amino acid change in the encoded protein sequence. Four of five in-silico tools predict a damaging effect of the variant on protein function. The variant allele was found at a frequency of 6e-05 in 251244 control chromosomes. This frequency is not significantly higher than estimated for a pathogenic variant in MC2R causing Glucocorticoid Deficiency, Due To ACTH Unresponsiveness, allowing no conclusion about variant significance. c.409C>T has been reported in the literature in individuals affected with Glucocorticoid Deficiency (example: Ishii_2000, Fluck_2002, Guran_2015). These data indicate that the variant is likely to be associated with disease. At least one publication reports experimental evidence that variant reduced normal activity (Fluck_2002). The following publications have been ascertained in the context of this evaluation (PMID: 12213892, 26523528, 10971458). ClinVar contains an entry for this variant (Variation ID: 3265). Based on the evidence outlined above, the variant was classified as pathogenic.
Labcorp Genetics (formerly Invitae), Labcorp RCV001851615 SCV002146499 pathogenic not provided 2021-09-02 criteria provided, single submitter clinical testing
Clinical Molecular Genetics Laboratory, Johns Hopkins All Children's Hospital RCV000584506 SCV000692309 pathogenic Glucocorticoid Deficiency 2008-04-16 no assertion criteria provided clinical testing
OMIM RCV000003421 SCV000023579 pathogenic Glucocorticoid deficiency 1 2002-09-01 no assertion criteria provided literature only

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