17-43115726-T-G

Variant summary

Our verdict is Uncertain significance. Variant got 1 ACMG points: 2P and 1B. PM2BP6

The NM_001407889.1(BRCA1):​c.-171A>C variant causes a 5 prime UTR premature start codon gain change involving the alteration of a non-conserved nucleotide. The variant allele was found at a frequency of 0.00000657 in 152,138 control chromosomes in the GnomAD database, with no homozygous occurrence. Variant has been reported in ClinVar as Conflicting classifications of pathogenicity (no stars).

Frequency

Genomes: 𝑓 0.0000066 ( 0 hom., cov: 32)

Consequence

BRCA1
NM_001407889.1 5_prime_UTR_premature_start_codon_gain

Scores

3
10
6
Splicing: ADA: 0.000005988
2

Clinical Significance

Conflicting classifications of pathogenicity criteria provided, conflicting classifications U:4B:6O:1

Conservation

PhyloP100: 0.475
Variant links:
Genes affected
BRCA1 (HGNC:1100): (BRCA1 DNA repair associated) This gene encodes a 190 kD nuclear phosphoprotein that plays a role in maintaining genomic stability, and it also acts as a tumor suppressor. The BRCA1 gene contains 22 exons spanning about 110 kb of DNA. The encoded protein combines with other tumor suppressors, DNA damage sensors, and signal transducers to form a large multi-subunit protein complex known as the BRCA1-associated genome surveillance complex (BASC). This gene product associates with RNA polymerase II, and through the C-terminal domain, also interacts with histone deacetylase complexes. This protein thus plays a role in transcription, DNA repair of double-stranded breaks, and recombination. Mutations in this gene are responsible for approximately 40% of inherited breast cancers and more than 80% of inherited breast and ovarian cancers. Alternative splicing plays a role in modulating the subcellular localization and physiological function of this gene. Many alternatively spliced transcript variants, some of which are disease-associated mutations, have been described for this gene, but the full-length natures of only some of these variants has been described. A related pseudogene, which is also located on chromosome 17, has been identified. [provided by RefSeq, May 2020]

Genome browser will be placed here

ACMG classification

Classification made for transcript

Verdict is Uncertain_significance. Variant got 1 ACMG points.

PM2
Very rare variant in population databases, with high coverage;
BP6
Variant 17-43115726-T-G is Benign according to our data. Variant chr17-43115726-T-G is described in ClinVar as [Conflicting_classifications_of_pathogenicity]. Clinvar id is 37402.We mark this variant Likely_benign, oryginal submissions are: {Likely_benign=6, Uncertain_significance=2, not_provided=1}.

Transcripts

RefSeq

Gene Transcript HGVSc HGVSp Effect #exon/exons MANE Protein UniProt
BRCA1NM_007294.4 linkuse as main transcriptc.134A>C p.Lys45Thr missense_variant, splice_region_variant 3/23 ENST00000357654.9 NP_009225.1 P38398-1

Ensembl

Gene Transcript HGVSc HGVSp Effect #exon/exons TSL MANE Protein Appris UniProt
BRCA1ENST00000357654.9 linkuse as main transcriptc.134A>C p.Lys45Thr missense_variant, splice_region_variant 3/231 NM_007294.4 ENSP00000350283.3 P38398-1

Frequencies

GnomAD3 genomes
AF:
0.00000657
AC:
1
AN:
152138
Hom.:
0
Cov.:
32
show subpopulations
Gnomad AFR
AF:
0.0000241
Gnomad AMI
AF:
0.00
Gnomad AMR
AF:
0.00
Gnomad ASJ
AF:
0.00
Gnomad EAS
AF:
0.00
Gnomad SAS
AF:
0.00
Gnomad FIN
AF:
0.00
Gnomad MID
AF:
0.00
Gnomad NFE
AF:
0.00
Gnomad OTH
AF:
0.00
GnomAD3 exomes
AF:
0.00000798
AC:
2
AN:
250502
Hom.:
0
AF XY:
0.00000739
AC XY:
1
AN XY:
135398
show subpopulations
Gnomad AFR exome
AF:
0.00
Gnomad AMR exome
AF:
0.00
Gnomad ASJ exome
AF:
0.00
Gnomad EAS exome
AF:
0.00
Gnomad SAS exome
AF:
0.00
Gnomad FIN exome
AF:
0.00
Gnomad NFE exome
AF:
0.0000177
Gnomad OTH exome
AF:
0.00
GnomAD4 exome
Cov.:
30
GnomAD4 genome
AF:
0.00000657
AC:
1
AN:
152138
Hom.:
0
Cov.:
32
AF XY:
0.0000135
AC XY:
1
AN XY:
74330
show subpopulations
Gnomad4 AFR
AF:
0.0000241
Gnomad4 AMR
AF:
0.00
Gnomad4 ASJ
AF:
0.00
Gnomad4 EAS
AF:
0.00
Gnomad4 SAS
AF:
0.00
Gnomad4 FIN
AF:
0.00
Gnomad4 NFE
AF:
0.00
Gnomad4 OTH
AF:
0.00
Bravo
AF:
0.0000113
ExAC
AF:
0.0000165
AC:
2
EpiCase
AF:
0.0000545
EpiControl
AF:
0.00

ClinVar

Significance: Conflicting classifications of pathogenicity
Submissions summary: Uncertain:4Benign:6Other:1
Revision: criteria provided, conflicting classifications
LINK: link

Submissions by phenotype

Breast-ovarian cancer, familial, susceptibility to, 1 Uncertain:3Benign:1Other:1
Likely benign, criteria provided, single submitterclinical testingAll of Us Research Program, National Institutes of HealthSep 04, 2023This missense variant replaces lysine with threonine at codon 45 of the BRCA1 protein. Computational prediction is inconclusive regarding the impact of this variant on protein structure and function (internally defined REVEL score threshold 0.5 < inconclusive < 0.7, PMID: 27666373). Functional studies reported conflicting variant impact on BRCA1 function from loss of ubiquitin ligase activity (PMID: 16403807) to no impact on a haploid human cell proliferation assay (PMID: 30209399). This variant has been observed in an individual affected with unilateral breast cancer (PMID: 20104584) and an individual age 70 years or older without cancer (FLOSSIES database). This variant also has been reported in a multifactorial analysis with family history likelihood ratio for pathogenicity of 0.0387 (PMID: 31131967). This variant has been identified in 2/250502 chromosomes in the general population by the Genome Aggregation Database (gnomAD). The available evidence is insufficient to determine the role of this variant in disease conclusively. Therefore, this variant is classified as a Variant of Uncertain Significance. -
Uncertain significance, no assertion criteria providedclinical testingSharing Clinical Reports Project (SCRP)May 03, 2010- -
Uncertain significance, criteria provided, single submitterclinical testingCounsylFeb 19, 2016- -
Uncertain significance, no assertion criteria providedclinical testingBreast Cancer Information Core (BIC) (BRCA1)Feb 20, 2004- -
not provided, no classification providedin vitroBrotman Baty Institute, University of Washington-- -
not provided Uncertain:1Benign:1
Uncertain significance, criteria provided, single submitterclinical testingQuest Diagnostics Nichols Institute San Juan CapistranoJun 19, 2020- -
Likely benign, criteria provided, single submitterclinical testingGeneDxJul 23, 2019Not observed at a significant frequency in large population cohorts (Lek et al., 2016); In silico analysis, which includes protein predictors and evolutionary conservation, supports a deleterious effect; This variant is associated with the following publications: (PMID: 20104584, 30209399, 21520273, 15235020, 16403807, 21523855, 000, 19543972, 16267036, 15385441, 25823446, 24489791, 33087888) -
Hereditary cancer-predisposing syndrome Benign:2
Likely benign, criteria provided, single submitterclinical testingAmbry GeneticsNov 19, 2019This alteration is classified as likely benign based on a combination of the following: seen in unaffected individuals, population frequency, intact protein function, lack of segregation with disease, co-occurrence, RNA analysis, in silico models, amino acid conservation, lack of disease association in case-control studies, and/or the mechanism of disease or impacted region is inconsistent with a known cause of pathogenicity. -
Likely benign, criteria provided, single submitterclinical testingColor Diagnostics, LLC DBA Color HealthMar 02, 2023- -
not specified Benign:1
Likely benign, criteria provided, single submitterclinical testingWomen's Health and Genetics/Laboratory Corporation of America, LabCorpAug 05, 2024Variant summary: BRCA1 c.134A>C (p.Lys45Thr) results in a non-conservative amino acid change located in the Zinc finger, RING-type (IPR001841) and Zinc finger, C3HC4 RING-type (IPR018957) of the encoded protein sequence. Four of five in-silico tools predict a damaging effect of the variant on protein function. Consensus agreement among computation tools predict no significant impact on normal splicing. However, these predictions have yet to be confirmed by functional studies. The variant allele was found at a frequency of 8e-06 in 250502 control chromosomes. The available data on variant occurrences in the general population are insufficient to allow any conclusion about variant significance. c.134A>C has been reported in the literature (e.g. Borg 2010). These report(s) do not provide unequivocal conclusions about association of the variant with Hereditary Breast And Ovarian Cancer Syndrome. Co-occurrences with other pathogenic variant(s) have been reported (BRCA2 c.26_26delC, p.Pro9Glnfs, BIC database), providing supporting evidence for a benign role. At least one functional study reports experimental evidence evaluating an impact on protein function and showed no damaging effect of this variant on homology directed repair (HDR) activity (e.g. Findlay_2018). HDR assays qualify as a recognized gold standard on the basis of updated guidance provided by the ClinGen Sequence Variant Interpretation (SVI) working group. In another in-vitro assay, this variant was shown to decrease the ubiquitin ligase activity (Morris_2006, Starita_2015). The following publications have been ascertained in the context of this evaluation (PMID: 16267036, 20104584, 15235020, 21520273, 16403807, 25823446, 30209399). ClinVar contains an entry for this variant (Variation ID: 37402). Based on the evidence outlined above, the variant was classified as likely benign. -
Hereditary breast ovarian cancer syndrome Benign:1
Likely benign, criteria provided, single submitterclinical testingLabcorp Genetics (formerly Invitae), LabcorpJan 12, 2024- -

Computational scores

Source: dbNSFP v4.3

Name
Calibrated prediction
Score
Prediction
AlphaMissense
Pathogenic
0.81
BayesDel_addAF
Pathogenic
0.23
D
BayesDel_noAF
Uncertain
0.10
CADD
Benign
21
DANN
Uncertain
1.0
DEOGEN2
Uncertain
0.42
.;T;.;.;.;T;.;T;T;.;T;T;.
Eigen
Benign
-0.53
Eigen_PC
Benign
-0.46
FATHMM_MKL
Benign
0.71
D
LIST_S2
Uncertain
0.95
D;D;D;D;D;D;D;D;D;T;D;D;T
M_CAP
Pathogenic
0.77
D
MetaRNN
Uncertain
0.49
T;T;T;T;T;T;T;T;T;T;T;T;T
MetaSVM
Uncertain
0.40
D
MutationAssessor
Benign
1.5
L;L;L;L;L;.;.;.;.;.;.;.;.
PrimateAI
Uncertain
0.50
T
PROVEAN
Benign
-1.1
N;N;N;N;N;N;N;.;N;N;N;D;D
REVEL
Uncertain
0.63
Sift
Uncertain
0.0070
D;D;D;D;D;D;D;.;D;D;D;D;D
Sift4G
Uncertain
0.032
D;D;D;D;D;.;D;.;D;.;D;.;.
Polyphen
0.12, 0.096, 0.41
.;B;.;.;B;.;B;.;.;.;.;.;.
Vest4
0.53
MutPred
0.60
Loss of methylation at K45 (P = 0);Loss of methylation at K45 (P = 0);Loss of methylation at K45 (P = 0);Loss of methylation at K45 (P = 0);Loss of methylation at K45 (P = 0);Loss of methylation at K45 (P = 0);Loss of methylation at K45 (P = 0);Loss of methylation at K45 (P = 0);Loss of methylation at K45 (P = 0);Loss of methylation at K45 (P = 0);Loss of methylation at K45 (P = 0);Loss of methylation at K45 (P = 0);Loss of methylation at K45 (P = 0);
MVP
0.76
MPC
0.19
ClinPred
0.27
T
GERP RS
-0.13
RBP_binding_hub_radar
0.0
RBP_regulation_power_radar
1.7
Varity_R
0.42
gMVP
0.66

Splicing

Name
Calibrated prediction
Score
Prediction
dbscSNV1_ADA
Benign
0.0000060
dbscSNV1_RF
Benign
0.14
SpliceAI score (max)
0.010
Details are displayed if max score is > 0.2

Find out detailed SpliceAI scores and Pangolin per-transcript scores at spliceailookup.broadinstitute.org

Publications

LitVar

Below is the list of publications found by LitVar. It may be empty.

Other links and lift over

dbSNP: rs80356863; hg19: chr17-41267743; API