TPP2

Chr 13AR

tripeptidyl peptidase 2

Also known as: IMD78, TPP-2, TPP-II, TPPII

This cytosolic tripeptidyl-peptidase removes N-terminal tripeptides from polypeptides as part of the proteolytic cascade downstream of the 26S proteasome and plays an important role in MHC class I antigen presentation and intracellular amino acid homeostasis. Biallelic mutations cause autosomal recessive immunodeficiency 78 with autoimmunity and developmental delay. The gene is highly constrained against loss-of-function variants (pLI 0.999, LOEUF 0.263), indicating intolerance to protein-truncating mutations.

Summary from RefSeq, OMIM, UniProt
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Primary Disease Associations & Inheritance

Immunodeficiency 78 with autoimmunity and developmental delayMIM #619220
AR
0
Active trials
3
Pubs (1 yr)
45
P/LP submissions
0%
P/LP missense
0.26
LOEUF· LoF intol.
LOF
Mechanism· G2P
Clinical SummaryTPP2
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Gene-Disease Validity (ClinGen)
immunodeficiency 78 with autoimmunity and developmental delay · ARDefinitive

Definitive — sufficient evidence for diagnostic panels

Population Constraint (gnomAD)
Highly constrained gene — heterozygous loss-of-function variants are very rare in the population (pLI 1.00). One damaged copy is likely sufficient to cause disease.
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ClinVar Variants
45 unique Pathogenic / Likely Pathogenic· 177 VUS of 600 total submissions

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Dual constrained — LoF & missense intolerant
LoF Constraint
0.26LOEUF
pLI 0.999
Z-score 6.28
OE 0.16 (0.100.26)
Highly constrained

Highly LoF-intolerant (top ~10% of genes)

Missense Constraint
3.86Z-score
OE missense 0.57 (0.530.63)
374 obs / 651.3 exp
Constrained

Highly missense-constrained (top ~0.1%)

Observed / Expected Ratios
LoF OE0.16 (0.100.26)
00.351.4
Missense OE0.57 (0.530.63)
00.61.4
Synonymous OE1.03
01.21.6
LoF obs/exp: 10 / 64.4Missense obs/exp: 374 / 651.3Syn Z: -0.39
Curated Mechanism (G2P)Gene2Phenotype (DDG2P) ↗
definitiveTPP2-related immune deficiency, autoimmune disease and intellectual disabilityLOFAR

Predictions shown for reference only — model trained on dominant genes, not applicable to AR conditions.

DN
0.3594th %ile
GOF
0.2597th %ile
LOF
0.67top 25%

The Badonyi & Marsh prediction model was trained exclusively on dominant disease genes. Predictions are not reliable for genes with autosomal recessive inheritance and are shown at reduced opacity for reference only.

Predictions from Badonyi M, Marsh JA. PLoS ONE. 2024;19(8):e0307312. Mechanism ranking also informed by gnomAD constraint, ClinVar, and ClinGen data.

ClinVar Variant Classifications

600 submitted variants in ClinVar

Classification Summary

Pathogenic43
Likely Pathogenic2
VUS177
Likely Benign318
Benign26
Conflicting1
43
Pathogenic
2
Likely Pathogenic
177
VUS
318
Likely Benign
26
Benign
1
Conflicting

Curated Variants Distribution

Classified variants from ClinVar · 5 ACMG categories

ClassificationLoFMissense + InframeNon-codingSynonymousTotal
Pathogenic
4
0
39
0
43
Likely Pathogenic
1
0
1
0
2
VUS
1
155
16
5
177
Likely Benign
0
1
152
165
318
Benign
0
0
26
0
26
Conflicting
1
Total6156234170567

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

View in ClinVar →

Protein Context — Lollipop Plot

TPP2 · protein map & ClinVar variants

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

Clinical Trials

Active and recruiting trials from ClinicalTrials.gov

No active trials found for this gene.

Search ClinicalTrials.gov →
Clinical Literature
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