PTPN3

Chr 9

protein tyrosine phosphatase non-receptor type 3

Also known as: PTP-H1, PTPH1

The protein encoded by PTPN3 is a tyrosine phosphatase that dephosphorylates substrate proteins and acts at membrane-cytoskeleton junctions, regulating cellular processes including cell growth, differentiation, and cell cycle progression. Mutations cause disease through a dominant-negative mechanism, as indicated by the gene's extremely low tolerance to loss-of-function variants. However, the specific pediatric neurological phenotypes associated with PTPN3 mutations are not established in the provided data.

OMIMResearchSummary from RefSeq, UniProt, Mechanism
MultiplemechanismLOEUF 0.57
Clinical SummaryPTPN3
Population Constraint (gnomAD)
Low constraint (pLI 0.00) — loss-of-function variants are relatively tolerated in the population.
📋
ClinVar Variants
24 unique Pathogenic / Likely Pathogenic· 139 VUS of 200 total submissions

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Moderate LoF intolerance
LoF Constraint
0.57LOEUF
pLI 0.000
Z-score 4.31
OE 0.41 (0.290.57)
Moderately constrained

More LoF-intolerant than ~75% of genes

Missense Constraint
0.71Z-score
OE missense 0.91 (0.850.98)
495 obs / 541.2 exp
Tolerant

Mild missense constraint

Observed / Expected Ratios
LoF OE0.41 (0.290.57)
00.351.4
Missense OE0.91 (0.850.98)
00.61.4
Synonymous OE1.02
01.21.6
LoF obs/exp: 25 / 61.5Missense obs/exp: 495 / 541.2Syn Z: -0.26
DN
0.74top 25%
GOF
0.72top 25%
LOF
0.2873th %ile

This gene has evidence for multiple mechanisms of pathogenicity (dominant-negative and gain-of-function). Both the Badonyi & Marsh prediction and the broader genomic evidence point to dominant-negative as the predominant mechanism. Different variants in this gene may act through different mechanisms — interpret in context of the specific variant.

DNprediction above median
GOFprediction above median

Note: In-silico variant effect predictors (SIFT, PolyPhen, REVEL, CADD) may underestimate pathogenicity of missense variants in genes with GOF or DN mechanisms. Consider functional evidence and clinical context.

Predictions from Badonyi M, Marsh JA. PLoS ONE. 2024;19(8):e0307312.

ClinVar Variant Classifications

200 submitted variants in ClinVar

Classification Summary

Pathogenic19
Likely Pathogenic5
VUS139
Likely Benign6
Benign1
19
Pathogenic
5
Likely Pathogenic
139
VUS
6
Likely Benign
1
Benign

Curated Variants Distribution

Classified variants from ClinVar · 5 ACMG categories

ClassificationLoFMissense + InframeNon-codingSynonymousTotal
Pathogenic
0
0
19
0
19
Likely Pathogenic
0
0
5
0
5
VUS
0
138
1
0
139
Likely Benign
0
3
0
3
6
Benign
0
0
0
1
1
Total0141254170

LoF = frameshift, stop gained/lost, canonical splice · Counts from ClinVar esearch · Updated hourly

View in ClinVar →

Protein Context — Lollipop Plot

PTPN3 · protein map & ClinVar variants

Showing all ClinVar variants across the protein. Search a specific variant to highlight its position.

3D Protein StructureAlphaFold

Clinical Trials

Active and recruiting trials from ClinicalTrials.gov

No active trials found for this gene.

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Clinical Literature
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