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.421C>G
Protein Change
H141D
Location
Exon 5 (Exon 5 of )
5
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 (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 PTEN.

Functional Studies & Therapeutic Relevance

Functional Summary

The PTEN H141D 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
Yes
REVEL Score
0.843
Threshold: ≥0.75 = PP3 applied

SpliceAI Scores

Window: ±500bp
Effect Type Score Position
- Acceptor Loss (AL) 0.03 -185 bp
- Donor Loss (DL) 0.0 -316 bp
+ Acceptor Gain (AG) 0.01 1 bp
+ Donor Gain (DG) 0.0 -51 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, PVS1 applies to null variants in a gene where loss of function is a known mechanism. This variant is missense (H141D), not a null variant. Therefore, this criterion is not applied because the variant type is not a loss-of-function variant.

PS1

PS1 (Not Applied)

According to VCEP guidelines, PS1 applies when a variant results in the same amino acid change as a previously established pathogenic variant. This variant (H141D) does not match any known pathogenic codon change at the nucleotide level. Therefore, this criterion is not applied.

PS2

PS2 (Not Applied)

According to standard ACMG guidelines, PS2 applies to confirmed de novo observations with paternity and maternity confirmed. No such data are available for this variant. Therefore, this criterion is not applied.

PS3

PS3 (Moderate)

According to PTEN Pre-processing guidelines, the rule/finding for PS3 is: "PS3_Moderate evidence added based on high-confidence functional score (-1.4006) < threshold (-1.11)". The evidence for this variant shows a phosphatase activity score of -1.4006, which is below the threshold of -1.11 per Mighell et al. 2018. Therefore, this criterion is applied at Moderate strength because the functional assay result meets the PTEN-specific threshold for damaging effect.

PS4

PS4 (Not Applied)

According to VCEP guidelines, PS4 applies when the variant prevalence is significantly increased in affected individuals compared to controls. No case-control or proband data are available for this variant. Therefore, this criterion is not applied.

PM1

PM1 (Not Applied)

According to VCEP guidelines, PM1 applies when the variant is located in a critical functional domain (e.g., catalytic motifs 90-94, 123-130, 166-168). H141 is not within these motifs. Therefore, this criterion is not applied.

PM2

PM2 (Supporting)

According to VCEP guidelines, the rule for PM2 is: "Supporting Absent in population Databases present at <0.00001 (0.001%) allele frequency in gnomAD or another large sequenced population...". The evidence for this variant shows it is absent from gnomAD (MAF=0%). Therefore, this criterion is applied at Supporting strength because the variant is not present in large population databases.

PM3

PM3 (Not Applied)

According to standard ACMG guidelines, PM3 applies to variants observed in trans with a pathogenic variant in a recessive disorder. PTEN-related disorders are autosomal dominant and no trans observations are relevant. Therefore, this criterion is not applied.

PM4

PM4 (Not Applied)

According to standard ACMG guidelines, PM4 applies to in-frame insertions/deletions. This variant is a missense substitution. Therefore, this criterion is not applied.

PM5

PM5 (Moderate)

According to VCEP guidelines, the rule for PM5 is: "Moderate Missense change at an amino acid residue where a different missense change determined to be pathogenic or likely pathogenic has been seen before. In addition, variant being interrogated must have BLOSUM62 score equal to or less than the known variant." The evidence for this variant shows that H141 is a residue at which a different missense change has been classified as pathogenic and the BLOSUM62 score criterion is met. Therefore, this criterion is applied at Moderate strength.

PM6

PM6 (Not Applied)

According to standard ACMG guidelines, PM6 applies to assumed de novo variants without confirmation. No de novo evidence is available. Therefore, this criterion is not applied.

PP1

PP1 (Not Applied)

According to VCEP guidelines, PP1 applies when there is co-segregation with disease in multiple affected family members. No segregation data are available. Therefore, this criterion is not applied.

PP2

PP2 (Not Applied)

According to standard ACMG guidelines, PP2 applies when missense variants are a common mechanism in a gene with low benign variation rate. PTEN has a range of missense variants, and specific applicability is not established. Therefore, this criterion is not applied.

PP3

PP3 (Supporting)

According to VCEP guidelines, the rule for PP3 is: "Supporting Multiple lines of computational evidence support a deleterious effect on the gene or gene product. Missense variants: REVEL score > 0.7." The evidence for this variant shows a REVEL score of 0.84, exceeding the threshold of 0.7. Therefore, this criterion is applied at Supporting strength because multiple in silico tools predict a damaging effect.

PP4

PP4 (Not Applied)

According to standard ACMG guidelines, PP4 applies when a patient’s phenotype is highly specific for a disease with a single genetic etiology. No phenotype data are provided. Therefore, this criterion is not applied.

PP5

PP5 (Not Applied)

According to standard ACMG guidelines, PP5 applies when a reputable source reports a variant as pathogenic. ClinVar lists this variant as VUS. Therefore, this criterion is not applied.

BA1

BA1 (Not Applied)

According to VCEP guidelines, BA1 applies when allele frequency >0.056% in gnomAD. This variant is absent. Therefore, this criterion is not applied.

BS1

BS1 (Not Applied)

According to VCEP guidelines, BS1 applies when allele frequency is between 0.0043% and 0.056% in gnomAD. This variant is absent. Therefore, this criterion is not applied.

BS2

BS2 (Not Applied)

According to VCEP guidelines, BS2 applies when a variant is observed in homozygous state in healthy individuals. No such observations exist. Therefore, this criterion is not applied.

BS3

BS3 (Not Applied)

According to VCEP guidelines, BS3 applies when functional studies show no damaging effect. Functional data show a damaging effect (PS3). Therefore, this criterion is not applied.

BS4

BS4 (Not Applied)

According to VCEP guidelines, BS4 applies when there is lack of segregation in multiple families. No segregation data are available. Therefore, this criterion is not applied.

BP1

BP1 (Not Applied)

According to standard ACMG guidelines, BP1 applies when missense variants in a gene where only truncating variants cause disease. PTEN disease mechanism includes missense. Therefore, this criterion is not applied.

BP2

BP2 (Not Applied)

According to VCEP guidelines, BP2 applies when variant is observed in trans with a pathogenic variant. Not applicable. Therefore, this criterion is not applied.

BP3

BP3 (Not Applied)

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

BP4

BP4 (Not Applied)

According to VCEP guidelines, BP4 applies when computational evidence suggests no impact (REVEL <0.5). The REVEL score is 0.84, indicating impact. Therefore, this criterion is not applied.

BP5

BP5 (Not Applied)

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

BP6

BP6 (Not Applied)

According to standard ACMG guidelines, BP6 applies when a reputable source reports a variant as benign. ClinVar reports this variant as VUS. Therefore, this criterion is not applied.

BP7

BP7 (Not Applied)

According to VCEP guidelines, BP7 applies to synonymous or intronic variants with no splicing impact. This variant is missense. Therefore, this criterion is not applied.