Genetic Information

Gene & Transcript Details

Gene
ATM
Transcript
NM_000051.4 MANE Select
Total Exons
Reference Sequence
NC_000011.9
Alternative Transcripts
IDStatusDetails
NM_000051.3 RefSeq Select 13147 nt | 386–9556
NM_000051.4 MANE Select 12915 nt | 151–9321

Variant Details

HGVS Notation
NM_000051.4:c.4777-13T>A
Protein Change
?
Location
Exon 31 (Exon 31 of )
31
5'Exon Structure3'
Functional Consequence
Loss of Function
Alternate Identifiers

Clinical & Population Data

Population Frequency

gnomAD
Global Frequency
0.00159 in 100,000
Extremely Rare
ACMG Criteria Applied PM2
This variant is absent or extremely rare in population databases (PM2 criteria applies).

ClinVar

Open
Classification
Uncertain Significance (VUS)
2 publications
Publications List
PMID: 25085752

This variant is considered likely benign. This variant is intronic and is not expected to impact mRNA splicing. This variant is strongly associated with less severe personal and family histories of cancer, typical for individuals without pathogenic variants in this gene [PMID: 25085752].

Clinical Statement

"This variant has been reported in ClinVar as Likely benign (4 clinical laboratories) and as Uncertain significance (1 clinical laboratories)."

COSMIC Somatic Evidence

Open
COSMIC ID
Recurrence
0 occurrences
PM1 Criteria
Not Applied
COSMIC Database Preview
COSMIC Preview
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Functional Impact & Domains

Functional Domain

Hotspot Status
Not a hotspot
Domain Summary
This variant is not located in a mutational hotspot or critical domain.
Related Variants in This Domain
No evidence of other pathogenic variants at this position in gene ATM.

Functional Studies & Therapeutic Relevance

Functional Summary

The ATM 4777-13T>A variant has not been functionally characterized.

Database Previews
OncoKB
OncoKB Preview
JAX-CKB
JAX-CKB Preview

Click on previews to view full database entries. External databases may require institutional access.

Computational Analysis

Pathogenicity Predictions

SpliceAI
Predictor Consensus
Mixed/VUS
PP3 Applied
No
REVEL Score
0.0
Threshold: ≥0.75 = PP3 applied

SpliceAI Scores

Window: ±500bp
Effect Type Score Position
- Acceptor Loss (AL) 0.09 13 bp
- Donor Loss (DL) 0.12 145 bp
+ Acceptor Gain (AG) 0.05 -45 bp
+ Donor Gain (DG) 0.0 13 bp
High impact (≥0.5) Medium impact (0.2-0.49) Low impact (<0.2)

VCEP Guidelines

Applied ACMG/AMP Criteria (VCEP Specific)

Filter Criteria:
PVS1

PVS1 (Not Applied)

According to VCEP guidelines, the rule for PVS1 is: "Use ATM PVS1 Decision Tree Modification Type: Gene-specific,Strength". The evidence for this variant shows it is an intronic change at position -13, outside the +1/+2 canonical splice sites. Therefore, this criterion is not applied because the variant does not meet the requirement for a null (loss-of-function) variant at canonical splice sites.

PS1

PS1 (Not Applied)

According to VCEP guidelines, the rule for PS1 is: "Use for protein changes as long as splicing is ruled-out for both alterations. Use ATM PS1 Splicing table for splicing variants with similar predictions or observations of splice defect." The evidence for this variant shows no amino acid change and no known protein consequence. Therefore, this criterion is not applied because there is no established pathogenic protein change to compare.

PS2

PS2 (Not Applied)

According to standard ACMG guidelines, the rule for PS2 is: "De novo (both maternity and paternity confirmed) in a patient with the disease and no family history." The evidence for this variant shows no data on de novo occurrence. Therefore, this criterion is not applied due to lack of de novo confirmation.

PS3

PS3 (Not Applied)

According to VCEP gene-specific guidelines for PS3, functional assays must show failure to rescue ATM-specific features. The evidence for this variant shows no functional study results. Therefore, this criterion is not applied because functional data are absent.

PS4

PS4 (Not Applied)

According to standard ACMG guidelines, the rule for PS4 is: "Case-control studies; p-value ≤ .05 AND (Odds ratio ≥2 or lower 95% CI ≥1.5)." The evidence for this variant shows no case-control or statistical association data. Therefore, this criterion is not applied due to lack of case-control evidence.

PM1

PM1 (Not Applied)

According to standard ACMG guidelines, the rule for PM1 is: "Located in a mutational hotspot and/or critical and well-established functional domain." The evidence for this variant shows it is intronic and not within a characterized hotspot or domain. Therefore, this criterion is not applied.

PM2

PM2 (Not Applied)

According to VCEP guidelines, the rule for PM2 (Supporting) is: "Frequency ≤.001% if n=1 in a single sub-population." The evidence for this variant shows MAF=0.00159% with n=4 in gnomAD. Therefore, this criterion is not applied because the allele frequency and counts exceed the threshold.

PM3

PM3 (Not Applied)

According to VCEP guidelines, the rule for PM3 is: "Use ATM PM3/BP2 table for recessive inheritance evidence." The evidence for this variant shows no data on trans configuration or recessive inheritance. Therefore, this criterion is not applied.

PM4

PM4 (Not Applied)

According to standard ACMG guidelines, the rule for PM4 is: "Protein length changes due to in-frame deletions/insertions or stop-loss." The evidence for this variant shows no change in coding sequence. Therefore, this criterion is not applied.

PM5

PM5 (Not Applied)

According to VCEP guidelines, the rule for PM5 is: "Use for novel missense at an amino acid residue where a different missense is pathogenic." The evidence for this variant shows no amino acid change. Therefore, this criterion is not applied.

PM6

PM6 (Not Applied)

According to standard ACMG guidelines, the rule for PM6 is: "Assumed de novo without confirmation of paternity/maternity." The evidence for this variant shows no de novo information. Therefore, this criterion is not applied.

PP1

PP1 (Not Applied)

According to standard ACMG guidelines, the rule for PP1 is: "Cosegregation with disease in multiple affected family members." The evidence for this variant shows no segregation data. Therefore, this criterion is not applied.

PP2

PP2 (Not Applied)

According to standard ACMG guidelines, the rule for PP2 is: "Missense variant in a gene with low rate of benign missense variation and where missense is a common mechanism." The evidence for this variant shows it is intronic. Therefore, this criterion is not applied.

PP3

PP3 (Not Applied)

According to VCEP guidelines, the rule for PP3 is: "Supporting if protein REVEL >.7333; RNA: at least one predictor shows impact on splicing." The evidence for this variant shows SpliceAI score 0.12 (below threshold) and no REVEL data. Therefore, this criterion is not applied.

PP4

PP4 (Not Applied)

According to standard ACMG guidelines, the rule for PP4 is: "Patient’s phenotype or family history is highly specific for a disease with a single genetic etiology." The evidence for this variant shows no phenotype data. Therefore, this criterion is not applied.

PP5

PP5 (Not Applied)

According to standard ACMG guidelines, the rule for PP5 is deprecated and not recommended. Therefore, this criterion is not applied.

BA1

BA1 (Not Applied)

According to VCEP guidelines, the rule for BA1 is: "Filtering Allele Frequency >.5%." The evidence for this variant shows MAF=0.00159%. Therefore, this criterion is not applied.

BS1

BS1 (Not Applied)

According to VCEP guidelines, the rule for BS1 is: "Filtering Allele Frequency >.05%." The evidence for this variant shows MAF=0.00159%. Therefore, this criterion is not applied.

BS2

BS2 (Not Applied)

According to standard ACMG guidelines, the rule for BS2 is: "Observed in a healthy adult individual for dominant disorder with full penetrance." The evidence for this variant shows no such observations. Therefore, this criterion is not applied.

BS3

BS3 (Not Applied)

According to VCEP guidelines, the rule for BS3 is: "Use when a variant rescues ATM-specific features in functional assays." The evidence for this variant shows no functional assay data. Therefore, this criterion is not applied.

BS4

BS4 (Not Applied)

According to standard ACMG guidelines, the rule for BS4 is: "Lack of segregation in affected members of a family." The evidence for this variant shows no segregation data. Therefore, this criterion is not applied.

BP1

BP1 (Not Applied)

According to standard ACMG guidelines, the rule for BP1 is: "Missense in gene where only loss-of-function is known mechanism." The evidence for this variant shows it is intronic. Therefore, this criterion is not applied.

BP2

BP2 (Not Applied)

According to VCEP guidelines, the rule for BP2 is: "Use ATM PM3/BP2 table for occurrence in trans with a pathogenic variant." The evidence for this variant shows no trans data. Therefore, this criterion is not applied.

BP3

BP3 (Not Applied)

According to standard ACMG guidelines, the rule for BP3 is: "In-frame indels in repetitive region without a known function." The evidence for this variant shows no indel. Therefore, this criterion is not applied.

BP4

BP4 (Supporting)

According to VCEP guidelines, the rule for BP4 is: "Protein Analysis: REVEL ≤.249; RNA: at least one predictor shows no impact on splicing." The evidence for this variant shows SpliceAI score 0.12, indicating minimal splicing impact. Therefore, this criterion is applied at Supporting strength because computational evidence supports no splicing effect.

BP5

BP5 (Not Applied)

According to standard ACMG guidelines, the rule for BP5 is: "Variant found in a case with an alternate molecular basis for disease." The evidence for this variant shows no such context. Therefore, this criterion is not applied.

BP6

BP6 (Not Applied)

According to standard ACMG guidelines, the rule for BP6 is deprecated and not recommended. Therefore, this criterion is not applied.

BP7

BP7 (Not Applied)

According to standard ACMG guidelines, the rule for BP7 is: "Synonymous or deep intronic (>+7 or <-40) with no splicing impact." The evidence for this variant shows it is at -13, not in the deep intronic region. Therefore, this criterion is not applied.