Genetic Information

Gene & Transcript Details

Gene
PMS2
Transcript
NM_000535.7 MANE Select
Total Exons
Reference Sequence
NC_000007.13
Alternative Transcripts
IDStatusDetails
NM_000535.6 Alternative 5156 nt | 88–2676
NM_000535.4 Alternative 2836 nt | 88–2676
NM_000535.7 MANE Select 5093 nt | 31–2619
NM_000535.5 Alternative 2851 nt | 88–2676
NM_000535.3 Alternative 2820 nt | 57–2645

Variant Details

HGVS Notation
NM_000535.7:c.1362G>A
Protein Change
L454=
Location
Exon 11 (Exon 11 of )
11
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
Likely Benign
2 publications
Clinical Statement

"This variant has been reported in ClinVar as Likely benign (1 clinical laboratories) and as Benign (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 PMS2.

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) 0.02 256 bp
- Donor Loss (DL) 0.0 -60 bp
+ Acceptor Gain (AG) 0.02 -281 bp
+ Donor Gain (DG) 0.05 8 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 for PVS1: "Very Strong Nonsense/frameshift variant introducing Premature Termination Codon (PTC) ≤ codon 798 in PMS2…". The evidence for this variant shows it is a synonymous substitution (no PTC). Therefore, this criterion is not applied.

PS1

PS1 (Not Applied)

According to VCEP guidelines for PS1: "Strong: A predicted missense substitution that encodes the same amino acid change with a different underlying nucleotide change previously established as Pathogenic…". The evidence for this variant shows no amino acid change. Therefore, this criterion is not applied.

PS2

PS2 (Not Applied)

According to standard ACMG for PS2: requires confirmed de novo occurrence. No de novo data are available. Therefore, this criterion is not applied.

PS3

PS3 (Not Applied)

According to standard ACMG for PS3: "Functional studies supportive of a damaging effect on the gene or gene product." No functional assay data are available. Therefore, this criterion is not applied.

PS4

PS4 (Not Applied)

According to standard ACMG for PS4: requires case–control or family-based statistical evidence of pathogenicity. No such data are available. Therefore, this criterion is not applied.

PM1

PM1 (Not Applied)

According to standard ACMG for PM1: requires variant in a mutational hot spot or well-established functional domain without benign variation. This variant is synonymous outside known hotspots. Therefore, this criterion is not applied.

PM2

PM2 (Supporting)

According to VCEP guidelines for PM2: "Supporting Absent/extremely rare (<1 in 50,000 alleles) in gnomAD v4 dataset." The evidence for this variant shows absence from population databases (MAF=0%). Therefore, this criterion is applied at Supporting strength.

PM3

PM3 (Not Applied)

According to VCEP guidelines for PM3: requires observed in trans with a pathogenic variant in a recessive disorder. No such data are available. Therefore, this criterion is not applied.

PM4

PM4 (Not Applied)

According to standard ACMG for PM4: pertains to protein length changes. This is a synonymous variant. Therefore, this criterion is not applied.

PM5

PM5 (Not Applied)

According to VCEP guidelines for PM5: requires missense change at a residue with a different pathogenic missense. This is synonymous. Therefore, this criterion is not applied.

PM6

PM6 (Not Applied)

According to standard ACMG for PM6: unconfirmed de novo. No de novo evidence. Therefore, this criterion is not applied.

PP1

PP1 (Not Applied)

According to standard ACMG for PP1: requires co-segregation with disease. No segregation data. Therefore, this criterion is not applied.

PP2

PP2 (Not Applied)

According to standard ACMG for PP2: pertains to missense variants in genes with low benign variation. This is synonymous. Therefore, this criterion is not applied.

PP3

PP3 (Not Applied)

According to VCEP guidelines for PP3: supporting for non-canonical splice or high prior missense. SpliceAI score is 0.05 (no impact) and variant is synonymous. Therefore, this criterion is not applied.

PP4

PP4 (Not Applied)

According to standard ACMG for PP4: requires specific phenotype or family history. No phenotype data. Therefore, this criterion is not applied.

PP5

PP5 (Not Applied)

According to standard ACMG for PP5: requires reputable source reporting pathogenic. No such report. Therefore, this criterion is not applied.

BA1

BA1 (Not Applied)

According to VCEP guidelines for BA1: "Stand Alone: GnomAD v4 grpmax filtering allele frequency ≥0.0028." Variant MAF=0%. Therefore, this criterion is not applied.

BS1

BS1 (Not Applied)

According to VCEP guidelines for BS1: "Strong: GnomAD v4 grpmax filtering allele frequency ≥0.0001 and <0.001." Variant MAF=0%. Therefore, this criterion is not applied.

BS2

BS2 (Not Applied)

According to VCEP guidelines for BS2: requires co-occurrence in trans with a known pathogenic variant. No such data. Therefore, this criterion is not applied.

BS3

BS3 (Not Applied)

According to VCEP guidelines for BS3: "Strong: Synonymous substitutions with no associated mRNA aberration as determined by laboratory assays." No laboratory mRNA assay data are available. Therefore, this criterion is not applied.

BS4

BS4 (Not Applied)

According to standard ACMG for BS4: requires lack of segregation with disease. No segregation data. Therefore, this criterion is not applied.

BP1

BP1 (Not Applied)

According to standard ACMG for BP1: pertains to missense in genes where only truncating variants cause disease. This is synonymous. Therefore, not applied.

BP2

BP2 (Not Applied)

According to standard ACMG for BP2: observed in trans with a pathogenic variant in a dominant disorder. No such data. Therefore, this criterion is not applied.

BP3

BP3 (Not Applied)

According to standard ACMG for BP3: in-frame indels in repetitive regions. Not applicable to synonymous SNV. Therefore, not applied.

BP4

BP4 (Supporting)

According to VCEP guidelines for BP4: "Supporting: For intronic and synonymous variants: SpliceAI predicts no splicing impact with delta score ≤0.1." SpliceAI max delta=0.05. Therefore, this criterion is applied at Supporting strength.

BP5

BP5 (Not Applied)

According to standard ACMG for BP5: requires variant found in a case with an alternate molecular cause. No such case data. Therefore, not applied.

BP6

BP6 (Supporting)

According to standard ACMG for BP6: "Supporting: Reputable source recently reports variant as benign but evidence not available to laboratory." ClinVar reports Likely benign/Benign. Therefore, this criterion is applied at Supporting strength.

BP7

BP7 (Supporting)

According to standard ACMG for BP7: "Supporting: A synonymous variant at or beyond -21/+7 exonic with no predicted splice impact." SpliceAI delta ≤0.1. Therefore, this criterion is applied at Supporting strength.