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.2577C>T
Protein Change
N859=
Location
Exon 17 (Exon 17 of )
17
5'Exon Structure3'
Functional Consequence
Loss of Function
Alternate Identifiers

Clinical & Population Data

Population Frequency

gnomAD
Global Frequency
0.00141 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
Likely Benign
2 publications
Clinical Statement

"This variant has been reported in ClinVar as Likely benign (7 clinical laboratories) and as Benign (2 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 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) 0.0 -110 bp
- Donor Loss (DL) 0.0 -206 bp
+ Acceptor Gain (AG) 0.0 -462 bp
+ Donor Gain (DG) 0.0 -33 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, ATM PVS1 Decision Tree applies only to predicted loss-of-function variants. The evidence for this variant shows it is synonymous with no predicted splice impact (SpliceAI Δ scores = 0). Therefore, PVS1 is not applied as the variant does not meet any loss-of-function criterion.

PS1

PS1 (Not Applied)

According to standard ACMG guidelines, PS1 applies when a variant results in the same amino acid change as a known pathogenic variant. This variant is synonymous (N859=) and does not change the amino acid. Therefore, PS1 is not applied.

PS2

PS2 (Not Applied)

According to standard ACMG guidelines, PS2 applies to confirmed de novo occurrences. Parental testing data are not available. Therefore, PS2 is not applied.

PS3

PS3 (Not Applied)

According to standard ACMG guidelines, PS3 applies when well-established functional studies show a damaging effect. No functional studies have been performed for this variant. Therefore, PS3 is not applied.

PS4

PS4 (Not Applied)

According to standard ACMG guidelines, PS4 applies when case-control data demonstrate a significant association (OR ≥2). No case-control or cohort data are available. Therefore, PS4 is not applied.

PM1

PM1 (Not Applied)

According to standard ACMG guidelines, PM1 applies to variants in mutational hot spots or critical domains. This synonymous variant is outside of any known functional domain. Therefore, PM1 is not applied.

PM2

PM2 (Not Applied)

According to VCEP guidelines, PM2_supporting applies if frequency ≤0.001% AND single observation. The evidence shows MAF=0.00141% with four alleles observed. Therefore, PM2 is not applied.

PM3

PM3 (Not Applied)

According to VCEP guidelines, PM3 applies to recessive disorders when variants are observed in trans with a pathogenic variant. No trans observation data are available. Therefore, PM3 is not applied.

PM4

PM4 (Not Applied)

According to standard ACMG guidelines, PM4 applies to protein length changes (in-frame indels). This variant is synonymous and does not alter protein length. Therefore, PM4 is not applied.

PM5

PM5 (Not Applied)

According to VCEP guidelines, PM5 applies to novel missense changes at residues with known pathogenic missense. This variant is synonymous. Therefore, PM5 is not applied.

PM6

PM6 (Not Applied)

According to standard ACMG guidelines, PM6 applies to assumed de novo cases without confirmation. No de novo data are available. Therefore, PM6 is not applied.

PP1

PP1 (Not Applied)

According to standard ACMG guidelines, PP1 applies to segregation in affected family members. No segregation data are available. Therefore, PP1 is not applied.

PP2

PP2 (Not Applied)

According to standard ACMG guidelines, PP2 applies when a missense variant occurs in a gene with low benign missense rate. This variant is synonymous. Therefore, PP2 is not applied.

PP3

PP3 (Not Applied)

According to VCEP guidelines, PP3 applies when in silico predictors support a deleterious effect. The evidence shows SpliceAI predicts no splicing impact and there is no protein change. Therefore, PP3 is not applied.

PP4

PP4 (Not Applied)

According to standard ACMG guidelines, PP4 applies when the patient phenotype is highly specific for a disease. No phenotype data are provided. Therefore, PP4 is not applied.

PP5

PP5 (Not Applied)

According to standard ACMG guidelines, PP5 applies when a reputable source reports a variant as pathogenic. The variant is reported as benign/likely benign in ClinVar. Therefore, PP5 is not applied.

BA1

BA1 (Not Applied)

According to VCEP guidelines, BA1 applies for filtering allele frequency >0.5%. The evidence shows MAF=0.00141%, well below threshold. Therefore, BA1 is not applied.

BS1

BS1 (Not Applied)

According to VCEP guidelines, BS1 applies for filtering allele frequency >0.05%. The evidence shows MAF=0.00141%, below threshold. Therefore, BS1 is not applied.

BS2

BS2 (Not Applied)

According to standard ACMG guidelines, BS2 applies when variant observed in healthy adult individuals for dominant disorders. ATM is recessive and no appropriate healthy cohort data are available. Therefore, BS2 is not applied.

BS3

BS3 (Not Applied)

According to VCEP guidelines, BS3 applies when functional studies show rescue of ATM-specific features. No such studies exist. Therefore, BS3 is not applied.

BS4

BS4 (Not Applied)

According to standard ACMG guidelines, BS4 applies when segregation analysis shows lack of cosegregation. No segregation data are available. Therefore, BS4 is not applied.

BP1

BP1 (Not Applied)

According to standard ACMG guidelines, BP1 applies when a missense variant occurs in a gene where only truncating variants cause disease. This variant is synonymous. Therefore, BP1 is not applied.

BP2

BP2 (Not Applied)

According to VCEP guidelines, BP2 applies when variant is observed in cis with a pathogenic variant. No data are available. Therefore, BP2 is not applied.

BP3

BP3 (Not Applied)

According to standard ACMG guidelines, BP3 applies to in-frame indels in repetitive regions. This variant is synonymous. Therefore, BP3 is not applied.

BP4

BP4 (Not Applied)

According to VCEP guidelines, BP4 for benign computational evidence may not be applied in conjunction with BP7 for RNA in synonymous variants. We have chosen to apply BP7. Therefore, BP4 is not applied.

BP5

BP5 (Not Applied)

According to standard ACMG guidelines, BP5 applies when variant is found in a case with an alternate molecular basis for disease. No such case data are available. Therefore, BP5 is not applied.

BP6

BP6 (Supporting)

According to standard ACMG guidelines, BP6: "Reputable source recently reports variant as benign, but the evidence is not available for independent evaluation." The evidence shows ClinVar reports this variant as Likely Benign (7 labs) and Benign (2 labs). Therefore, BP6 is applied at Supporting strength.

BP7

BP7 (Supporting)

According to VCEP guidelines, "BP7 (Supporting): Use for synonymous variants with no predicted splice impact (further than +7/-40)." SpliceAI Δ scores = 0 predicting no impact on splicing for this synonymous variant. Therefore, BP7 is applied at Supporting strength.