Total submissions: 8
Submitter | RCV | SCV | Clinical significance | Condition | Last evaluated | Review status | Method | Comment |
---|---|---|---|---|---|---|---|---|
Clin |
RCV000654444 | SCV005043095 | likely benign | DICER1-related tumor predisposition | 2024-02-27 | reviewed by expert panel | curation | The NM_177438.2:c.4475T>C variant in DICER1 is a missense variant predicted to cause substitution of methionine by threonine at amino acid 1492 (p.Met1492Thr). This variant has been seen in 10 or more unrelated females without tumors through age 50 in at least one testing laboratory (BS2_Supporting; Internal lab contributors). In silico tools predict no damaging impact of the variant on protein function (REVEL: 0.049; MaxEntScan and SpliceAI: no effect on splicing) (BP4). The total allele frequency in gnomAD v4.1.0 is 0.000002478 (4/1614146 alleles) with highest population minor allele frequencies of 0.00001334 (1/74954alleles) and 0.000002542 (3/1180050) in the African American and European (non-Finnish) populations, respectively (PM2_Supporting, BS1, and BA1 are not met). In summary, this variant meets the criteria to be classified as Likely Benign for DICER1-related tumor predisposition based on the ACMG/AMP criteria applied, as specified by the ClinGen DICER1 VCEP: BS2_Supporting, BP4. (Bayesian Points: -2; VCEP specifications version 1.3.0; 02/27/2024) |
Ambry Genetics | RCV000561694 | SCV000669331 | uncertain significance | Hereditary cancer-predisposing syndrome | 2024-10-10 | criteria provided, single submitter | clinical testing | The p.M1492T variant (also known as c.4475T>C), located in coding exon 22 of the DICER1 gene, results from a T to C substitution at nucleotide position 4475. The methionine at codon 1492 is replaced by threonine, an amino acid with similar properties. This amino acid position is poorly conserved in available vertebrate species. In addition, this alteration is predicted to be tolerated by in silico analysis. Based on the available evidence, the clinical significance of this variant remains unclear. |
Labcorp Genetics |
RCV000654444 | SCV000776338 | uncertain significance | DICER1-related tumor predisposition | 2025-01-06 | criteria provided, single submitter | clinical testing | This sequence change replaces methionine, which is neutral and non-polar, with threonine, which is neutral and polar, at codon 1492 of the DICER1 protein (p.Met1492Thr). This variant is present in population databases (rs370947079, gnomAD 0.008%). This variant has not been reported in the literature in individuals affected with DICER1-related conditions. ClinVar contains an entry for this variant (Variation ID: 483407). Invitae Evidence Modeling of protein sequence and biophysical properties (such as structural, functional, and spatial information, amino acid conservation, physicochemical variation, residue mobility, and thermodynamic stability) indicates that this missense variant is not expected to disrupt DICER1 protein function with a negative predictive value of 95%. In summary, the available evidence is currently insufficient to determine the role of this variant in disease. Therefore, it has been classified as a Variant of Uncertain Significance. |
Fulgent Genetics, |
RCV000765192 | SCV000896427 | uncertain significance | Euthyroid goiter; Rhabdomyosarcoma, embryonal, 2; DICER1-related tumor predisposition | 2018-10-31 | criteria provided, single submitter | clinical testing | |
Sema4, |
RCV000561694 | SCV002533009 | uncertain significance | Hereditary cancer-predisposing syndrome | 2021-12-05 | criteria provided, single submitter | curation | |
Prevention |
RCV003420012 | SCV004117382 | uncertain significance | DICER1-related disorder | 2023-03-21 | criteria provided, single submitter | clinical testing | The DICER1 c.4475T>C variant is predicted to result in the amino acid substitution p.Met1492Thr. To our knowledge, this variant has not been reported in the literature. This variant is reported in 3 of ~283,000 alleles in gnomAD (http://gnomad.broadinstitute.org/variant/14-95562782-A-G) and is interpreted as uncertain significance in CLinVar (https://preview.ncbi.nlm.nih.gov/clinvar/variation/483407/). At this time, the clinical significance of this variant is uncertain due to the absence of conclusive functional and genetic evidence. |
Baylor Genetics | RCV004569211 | SCV005059433 | uncertain significance | Global developmental delay - lung cysts - overgrowth - Wilms tumor syndrome | 2024-03-27 | criteria provided, single submitter | clinical testing | |
Gene |
RCV004773012 | SCV005385820 | uncertain significance | not provided | 2024-02-05 | criteria provided, single submitter | clinical testing | Not observed at significant frequency in large population cohorts (gnomAD); In silico analysis supports that this missense variant does not alter protein structure/function; Has not been previously published as pathogenic or benign to our knowledge |