13-32363178-G-T
Variant summary
The NM_000059.4(BRCA2):c.7977-1G>T variant causes a splice acceptor, intron change involving the alteration of a conserved nucleotide. The variant is predicted to lead to nonsense-mediated mRNA decay (NMD). The variant allele was found at a cumulative frequency of 0.00000658 (AC=1) in the gnomAD database across 152,072 control chromosomes (no homozygotes observed). The grpmax filtering allele frequency (95% CI) is 0.000192. In-silico predictor (BayesDel (noAF)) classifies this variant as likely damaging/oncogenic. The affected nucleotide is highly conserved across species (PhyloP 100-way vertebrate score: 9.52). Variant has been reported in ClinVar as Pathogenic/Likely Pathogenic (★★). ClinVar reports functional evidence for this variant: "SCV000253757: Studies have shown that disruption of this splice site alters mRNA splicing and is expected to lead to the loss of protein expression (PMID:16211554, 22006311, 28339459)." and additional evidence is available in ClinVar.
Frequency
Consequence
NM_000059.4 splice_acceptor, intron
Scores
Clinical Significance
Conservation
Publications
- BRCA2-related cancer predispositionInheritance: AD Classification: DEFINITIVE Submitted by: ClinGen
- breast-ovarian cancer, familial, susceptibility to, 2Inheritance: AD Classification: DEFINITIVE, STRONG Submitted by: Labcorp Genetics (formerly Invitae), Genomics England PanelApp, Ambry Genetics
- Fanconi anemia complementation group D1Inheritance: AR Classification: DEFINITIVE, STRONG Submitted by: G2P, Labcorp Genetics (formerly Invitae), ClinGen, Ambry Genetics
- pancreatic cancer, susceptibility to, 2Inheritance: AD Classification: STRONG Submitted by: Genomics England PanelApp
- sarcomaInheritance: AD Classification: MODERATE Submitted by: Genomics England PanelApp
- hereditary breast ovarian cancer syndromeInheritance: AD Classification: SUPPORTIVE Submitted by: Orphanet
- Fanconi anemiaInheritance: AR Classification: SUPPORTIVE Submitted by: Orphanet
- medulloblastomaInheritance: AD Classification: LIMITED Submitted by: Ambry Genetics
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Classification according to ACMG Germline Pathogenicity v2019
Our verdict: Pathogenic. The variant received 20 points.
| Apply | Criterion Code & Title / Strength | Rationale / Clinical Context | |
|---|---|---|---|
| PVS1Null variant in LOF gene | Splice site frameshift, NMD predicted, LoF disease mechanism — very strong (PVS1); Splice acceptor variant predicted to cause a frameshift, triggering NMD. Loss of function is a known disease mechanism for this gene. | ||
| PS1Same amino acid change as known pathogenic | Splice site has a more-destructive ClinVar pathogenic — strong (PS1); A more pathogenic splice-site variant at the same position has been reported in ClinVar. | ||
| PS3Functional studies show damaging effect | Well-established functional study supports damaging effect (PS3); SCV000253757: Studies have shown that disruption of this splice site alters mRNA splicing and is expected to lead to the loss of protein expression (PMID: 16211554, 22006311, 28339459).; SCV004060668: RNA studies have demonstrated that this alteration results in abnormal splicing in the set of samples tested (Fraile-Bethencourt E et al. PLoS Genet, 2017 Mar;13:e1006691; Mesman RLS et al. Genet Med, 2020 Aug;22:1355-1365; Ambry internal data). In addition, this alteration failed to rescue the loss of Brca2 activity in an assay of cell survival in murine embryonic stem cells (Mesman RLS et al. Genet Med, 2020 Aug;22:1355-1365). | ||
| PP5Reputable source reports pathogenic | ClinVar 2-star pathogenic — strong (PP5); ClinVar germline classification: Pathogenic/Likely Pathogenic, 2 star(s). |
Variant Effect in Transcripts
Automated classification analysis was done for transcript: NM_000059.4. You can select a different transcript below to see updated classification assignments.
RefSeq Transcripts
| Sel. | Gene / Transcript | Tags | HGVSc / HGVSp | Effect | Exon rank | Protein / UniProt |
|---|---|---|---|---|---|---|
BRCA2 | MANE Select | c.7977-1G>T | splice_acceptorintron | Intron 17 of 26 | Prot: NP_000050.3 UniProt: | |
BRCA2 | c.7977-1G>T | splice_acceptorintron | Intron 17 of 26 | Prot: NP_001419006.1 UniProt: | ||
BRCA2 | c.7977-1G>T | splice_acceptorintron | Intron 17 of 26 | Prot: NP_001393649.1 UniProt: |
Ensembl Transcripts
| Sel. | Gene / Transcript | Tags | HGVSc / HGVSp | Effect | Exon rank | Protein / UniProt |
|---|---|---|---|---|---|---|
BRCA2 | TSL:5MANE Select | c.7977-1G>T | splice_acceptorintron | Intron 17 of 26 | Prot: ENSP00000369497.3 UniProt: | |
BRCA2 | TSL:1 | c.7977-1G>T | splice_acceptorintron | Intron 17 of 26 | Prot: ENSP00000439902.1 UniProt: | |
BRCA2 | TSL:1 | c.7608-1G>T | splice_acceptorintron | Intron 17 of 26 | Prot: ENSP00000499438.2 UniProt: |
Frequencies
Allele frequencies (AF), counts (AC/AN), homozygotes and coverage
| Source / population | AF | AC | Hom | AN | Coverage |
|---|---|---|---|---|---|
Global population databases 4 sources | |||||
GnomAD3 genomes | AF: 0.00000658 | AC:1 | Hom:0 | AN:152072 | Cov:32 |
GnomAD4 exome | Cov:31 | ||||
GnomAD4 genome | AF: 0.00000658 | AC:1 | Hom:0 | AN:152072 | Cov:32 |
ALFA (dbGaP/dbSNP Allele Frequency Aggregator) | AF: 0.00 | AC:0 | Hom:0 | AN:36924 | |
ClinVar
Computational Scores
| Algorithm | Calibrated pred. | Score | Prediction |
|---|---|---|---|
AlphaGenome AVI | Pathogenic | 32 | |
BayesDel_addAF | Pathogenic | 0.39 | D |
BayesDel_noAF | Pathogenic | 0.33 | |
CADD | Pathogenic | 32 | |
DANN | Uncertain | 1.0 | |
Eigen | Pathogenic | 1.2 | |
Eigen_PC | Pathogenic | 1.0 | |
FATHMM_MKL | Pathogenic | 0.99 | D |
GPN-Star LLR | N/A | -10 | |
GPN-Star score | N/A | 10 | |
Mutation Taster | N/A | 0/100 | disease causing (ClinVar) |
PhyloP100 | Pathogenic | 9.5 |
Splicing Scores
| Algorithm | Calibrated pred. | Score | Prediction |
|---|---|---|---|
dbscSNV1_ADA | Pathogenic | 1.0 | |
dbscSNV1_RF | Pathogenic | 0.94 | |
DS_AL_spliceai | Pathogenic | 0.98 | Position offset: 1 |
Pangolin (max) | Pathogenic | 0.91 | |
Pangolin gain | Uncertain | 0.31 | Position offset: 22 |
Pangolin loss | Pathogenic | 0.91 | Position offset: 1 |
SpliceAI score (max) | Pathogenic | 0.98 | Details are displayed if max score is > 0.2 |
Find out detailed SpliceAI scores and Pangolin per-transcript scores at
MaxEntScan Visualizer can be used to analyze the impact of this mutation on the neighboring sequence.