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.1219_1221dup
Protein Change
F407dup
Location
Exon 9 (Exon 9 of )
9
5'Exon Structure3'
Functional Consequence
Loss of Function
Alternate Identifiers

Clinical & Population Data

Population Frequency

gnomAD
Global Frequency
0.0 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)
1 publications
Clinical Statement

"This variant has been reported in ClinVar as Uncertain significance (8 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 F407dup 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
Unknown
PP3 Applied
No
REVEL Score
0.0
Threshold: ≥0.75 = PP3 applied

SpliceAI Scores

Window: ±500bp
Effect Type Score Position
- Acceptor Loss (AL) 0.19 -151 bp
- Donor Loss (DL) 0.17 18 bp
+ Acceptor Gain (AG) 0.06 79 bp
+ Donor Gain (DG) 0.0 -85 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: c.1219_1221dupTTT is an in‐frame duplication, not predicted to cause loss of function. Therefore, this criterion is not applied because the variant does not meet the threshold for a null effect.

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'. The evidence for this variant shows: F407dup is not the same amino acid change as any previously established pathogenic variant. Therefore, this criterion is not applied.

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 de novo data are available. Therefore, this criterion is not applied due to lack of de novo confirmation.

PS3

PS3 (Not Applied)

According to VCEP guidelines, the rule for PS3 is: 'Use when a variant fails to rescue both an ATM specific feature AND radiosensitivity for Strong, or only an ATM feature for Supporting'. The evidence for this variant shows: no functional studies exist for F407dup. Therefore, this criterion is not applied due to missing functional rescue data.

PS4

PS4 (Not Applied)

According to standard ACMG guidelines, the rule for PS4 is: 'Case-control studies; p≤0.05 AND OR≥2 or lower 95% CI≥1.5'. The evidence for this variant shows: no case–control data are available. Therefore, this criterion is not applied.

PM1

PM1 (Not Applied)

According to standard ACMG guidelines, the rule for PM1 is: 'Located in a mutational hot spot and/or critical and well-established functional domain'. The evidence for this variant shows: F407dup lies outside known ATM functional domains/hot spots. Therefore, this criterion is not applied.

PM2

PM2 (Supporting)

According to VCEP guidelines, the rule for PM2 is: 'Supporting Strength: Frequency ≤.001% if n=1 in a single subpopulation'. The evidence for this variant shows: absent from gnomAD and other population databases (MAF=0%). Therefore, this criterion is applied at Supporting strength because the variant is absent in controls.

PM3

PM3 (Not Applied)

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

PM4

PM4 (Not Applied)

According to VCEP guidelines, the rule for PM4 is: 'Use for stop-loss variants at Moderate strength'. The evidence for this variant shows: F407dup is an in‐frame duplication, not a stop-loss. Therefore, this criterion is not applied.

PM5

PM5 (Not Applied)

According to VCEP guidelines, the rule for PM5 is: 'Supporting for frameshift/truncating variants upstream of p.R3047 or splice variants with PTC upstream'. The evidence for this variant shows: F407dup is neither a truncating nor a splice variant with PTC. 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 and maternity'. The evidence for this variant shows: no de novo data available. Therefore, this criterion is not applied.

PP1

PP1 (Not Applied)

According to standard ACMG guidelines, the rule for PP1 is: 'Co‐segregation with disease in multiple affected family members'. The evidence for this variant shows: no segregation data reported. 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 a low rate of benign missense variation'. The evidence for this variant shows: ATM has both missense and truncating pathogenic variants and is not constrained for missense variation. Therefore, this criterion is not applied.

PP3

PP3 (Not Applied)

According to VCEP guidelines, the rule for PP3 is: 'Supporting if REVEL>0.7333 or splicing predictor shows impact'. The evidence for this variant shows: SpliceAI score=0.19 (no impact), REVEL unavailable. 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 provided. Therefore, this criterion is not applied.

PP5

PP5 (Not Applied)

According to standard ACMG guidelines, the rule for PP5 is: 'Reputable source reports variant as pathogenic'. The evidence for this variant shows: ClinVar reports uncertain significance. Therefore, this criterion is not applied.

BA1

BA1 (Not Applied)

According to VCEP guidelines, the rule for BA1 is: 'Filtering Allele Frequency >0.5%'. The evidence for this variant shows: MAF=0%. Therefore, this criterion is not applied.

BS1

BS1 (Not Applied)

According to VCEP guidelines, the rule for BS1 is: 'Filtering Allele Frequency >0.05%'. The evidence for this variant shows: MAF=0%. 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 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: 'Moderate if variant rescues both ATM features and radiosensitivity, Supporting if either'. The evidence for this variant shows: no functional rescue 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'. 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 for which only truncating variants cause disease'. The evidence for this variant shows: ATM missense variants can be pathogenic. 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 variants in cis with a pathogenic variant'. The evidence for this variant shows: no phase 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 a repetitive region without a known function'. The evidence for this variant shows: region is not repetitive. Therefore, this criterion is not applied.

BP4

BP4 (Supporting)

According to standard ACMG guidelines, the rule for BP4 is: 'Multiple lines of computational evidence suggest no impact on gene or gene product'. The evidence for this variant shows: SpliceAI predicts no impact on splicing; no in silico evidence of deleterious effect. Therefore, this criterion is applied at Supporting strength because computational tools do not predict a functional impact.

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 case reported. Therefore, this criterion is not applied.

BP6

BP6 (Not Applied)

According to standard ACMG guidelines, the rule for BP6 is: 'Reputable source reports variant as benign'. The evidence for this variant shows: ClinVar reports uncertain. Therefore, this criterion is not applied.

BP7

BP7 (Not Applied)

According to standard ACMG guidelines, the rule for BP7 is: 'Synonymous variant with no predicted splice impact'. The evidence for this variant shows: it is not synonymous. Therefore, this criterion is not applied.