Genetic Information

Gene & Transcript Details

Gene
PTEN
Transcript
NM_000314.8 MANE Select
Total Exons
Reference Sequence
NC_000010.10
Alternative Transcripts
IDStatusDetails
NM_000314.8 MANE Select 8515 nt | 846–2057
NM_000314.7 RefSeq Select 8514 nt | 845–2056
NM_000314.5 Alternative 8719 nt | 1032–2243
NM_000314.4 Alternative 5572 nt | 1032–2243
NM_000314.3 Alternative 3416 nt | 1032–2243
NM_000314.6 Alternative 8718 nt | 1032–2243

Variant Details

HGVS Notation
NM_000314.8:c.132_135dup
Protein Change
Y46Rfs*7
Location
Exon 2 (Exon 2 of )
2
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
Unknown
0 publications
Clinical Statement

""

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 PTEN.

Functional Studies & Therapeutic Relevance

Functional Summary

The PTEN Y46Rfs*7 variant is a truncating mutation that results in the loss of PTEN phosphatase function, leading to an inability to negatively regulate PI3K/AKT pathway activity. Functional studies have demonstrated that such truncating mutations in PTEN are oncogenic, as they increase genomic fragility and disrupt PTEN's association with chromosomal centromeres. This evidence supports a damaging effect of the variant.

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) 0.04 -58 bp
- Donor Loss (DL) 0.0 33 bp
+ Acceptor Gain (AG) 0.04 -12 bp
+ Donor Gain (DG) 0.0 28 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 (Very Strong)

According to VCEP guidelines the rule for PVS1 is: "Very Strong Strength: Very Strong Use PTEN PVS1 decision tree." The evidence for this variant shows: it is an early frameshift (Y46Rfs*7) predicted to result in loss of function in a gene where LOF is a known disease mechanism and it is not in the last exon. Therefore, this criterion is applied at Very Strong strength because the PTEN-specific decision tree supports PVS1 for truncating variants outside the last exon.

PS1

PS1 (Not Applied)

According to VCEP guidelines the rule for PS1 is: "Strong Strength: Strong Same amino acid change as a previously established pathogenic variant regardless of nucleotide change OR different variant at same nucleotide position as a pathogenic splicing variant..." The evidence for this variant shows: it is a frameshift, not a missense change at a previously characterized position. Therefore, this criterion is not applied.

PS2

PS2 (Not Applied)

According to VCEP guidelines the rule for PS2 is: "Very Strong Strength: Very Strong Two proven OR four assumed OR one proven + two assumed de novo observations 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.

PS3

PS3 (Strong)

According to PTEN Pre-processing the finding for PS3 is: "The PTEN Y46Rfs*7 variant is a truncating mutation that results in the loss of PTEN phosphatase function, leading to an inability to negatively regulate PI3K/AKT pathway activity... Functional studies have demonstrated that such truncating mutations in PTEN are oncogenic..." According to VCEP guidelines the rule for PS3 is: "Strong Strength: Strong Well-established in vitro or in vivo functional studies supportive of a damaging effect on the gene or gene product." The evidence for this variant shows: functional assays demonstrate loss of phosphatase activity and oncogenic behavior. Therefore, this criterion is applied at Strong strength.

PS4

PS4 (Not Applied)

According to VCEP guidelines the rule for PS4 is: "Very Strong Strength: Very Strong Probands with specificity score ≥16..." The evidence for this variant shows: no case or prevalence data are available. Therefore, this criterion is not applied.

PM1

PM1 (Not Applied)

According to VCEP guidelines the rule for PM1 is: "Moderate Strength: Moderate Located in a mutational hot spot and/or critical and well-established functional domain (residues 90-94, 123-130, 166-168)." The evidence for this variant shows: it affects codon 46, outside of defined critical domains. Therefore, this criterion is not applied.

PM2

PM2 (Supporting)

According to VCEP guidelines the rule for PM2 is: "Supporting Strength: Supporting Absent in population Databases present at <0.00001 (0.001%) allele frequency in gnomAD..." The evidence for this variant shows: it is absent from gnomAD and other large population databases. Therefore, this criterion is applied at Supporting strength.

PM3

PM3 (Not Applied)

According to standard ACMG guidelines the rule for PM3 is: "Detected in trans with a pathogenic variant for a recessive gene/disorder." The evidence for this variant shows: PTEN-associated disease is autosomal dominant and no in trans data exist. Therefore, this criterion is not applied.

PM4

PM4 (Not Applied)

According to VCEP guidelines the rule for PM4 is: "Moderate Strength: Moderate Protein length changes due to in-frame deletions/insertions in a non-repeat region or stop-loss variants." The evidence for this variant shows: it is a frameshift, not an in-frame change or stop-loss. Therefore, this criterion is not applied.

PM5

PM5 (Not Applied)

According to VCEP guidelines the rule for PM5 is: "Moderate Strength: Moderate Missense change at an amino acid residue where a different missense change determined to be pathogenic has been seen before." The evidence for this variant shows: it is a frameshift, not a missense substitution. Therefore, this criterion is not applied.

PM6

PM6 (Not Applied)

According to VCEP guidelines the rule for PM6 is: "Very Strong Strength: Very Strong Two proven OR four assumed OR one proven + two assumed de novo observations..." The evidence for this variant shows: no de novo data are available. Therefore, this criterion is not applied.

PP1

PP1 (Not Applied)

According to VCEP guidelines the rule for PP1 is: "Strong Strength: Strong Co-segregation with disease in ≥7 meioses across ≥2 families..." The evidence for this variant shows: no segregation data are available. Therefore, this criterion is not applied.

PP2

PP2 (Not Applied)

According to VCEP guidelines the rule for PP2 is: "Supporting Strength: Supporting Missense variant in a gene with low benign missense variation and where missense is common mechanism." The evidence for this variant shows: it is a frameshift, not missense. Therefore, this criterion is not applied.

PP3

PP3 (Not Applied)

According to VCEP guidelines the rule for PP3 is: "Supporting Strength: Supporting Multiple lines of computational evidence support deleterious effect (REVEL >0.7 or splice algorithms)." The evidence for this variant shows: no strong computational predictions of deleteriousness beyond LOF. Therefore, this criterion is not applied.

PP4

PP4 (Not Applied)

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

PP5

PP5 (Not Applied)

According to standard ACMG guidelines the rule for PP5 is: "Supporting Reputable source reports variant as pathogenic." The evidence for this variant shows: no reputable external classification is available. Therefore, this criterion is not applied.

BA1

BA1 (Not Applied)

According to VCEP guidelines the rule for BA1 is: "Stand Alone Strength: Stand Alone gnomAD Filtering allele frequency >0.00056 (0.056%)." The evidence for this variant shows: allele frequency is 0%. Therefore, this criterion is not applied.

BS1

BS1 (Not Applied)

According to VCEP guidelines the rule for BS1 is: "Strong Strength: Strong gnomAD Filtering allele frequency from 0.000043 to 0.00056..." The evidence for this variant shows: allele frequency is 0%. Therefore, this criterion is not applied.

BS2

BS2 (Not Applied)

According to VCEP guidelines the rule for BS2 is: "Strong Strength: Strong Observed in the homozygous state in a healthy or PHTS-unaffected individual..." The evidence for this variant shows: no homozygous observations in unaffected individuals. Therefore, this criterion is not applied.

BS3

BS3 (Not Applied)

According to VCEP guidelines the rule for BS3 is: "Strong Strength: Strong Well-established functional studies show no damaging effect." The evidence for this variant shows: functional studies demonstrate damaging effect. Therefore, this criterion is not applied.

BS4

BS4 (Not Applied)

According to VCEP guidelines the rule for BS4 is: "Strong Strength: Strong Lack of segregation in affected members of ≥2 families." 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: "Supporting Missense variant in gene where only truncations cause disease." The evidence for this variant shows: it is a truncating variant and truncations are disease-causing. Therefore, this criterion is not applied.

BP2

BP2 (Not Applied)

According to VCEP guidelines the rule for BP2 is: "Supporting Observed in trans with a pathogenic PTEN variant or ≥3 observations in cis..." The evidence for this variant shows: no such observations. Therefore, this criterion is not applied.

BP3

BP3 (Not Applied)

According to VCEP guidelines the rule for BP3 is: "Supporting In-frame indels in repetitive regions without known function." The evidence for this variant shows: it is a frameshift, not an in-frame indel. Therefore, this criterion is not applied.

BP4

BP4 (Not Applied)

According to VCEP guidelines the rule for BP4 is: "Supporting Multiple lines of computational evidence suggest no impact..." The evidence for this variant shows: it is clearly LOF by frameshift. Therefore, this criterion is not applied.

BP5

BP5 (Not Applied)

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

BP6

BP6 (Not Applied)

According to standard ACMG guidelines the rule for BP6 is: "Supporting Reputable source reports variant as benign." The evidence for this variant shows: no such benign classification exists. Therefore, this criterion is not applied.

BP7

BP7 (Not Applied)

According to VCEP guidelines the rule for BP7 is: "Supporting A synonymous or intronic variant at or beyond +7/-21 with no predicted splice impact." The evidence for this variant shows: it is a frameshift, not synonymous or deep intronic. Therefore, this criterion is not applied.