PPT1

Chr 1AR

palmitoyl-protein thioesterase 1

Also known as: CLN1, INCL, PPT

The protein removes thioester-linked fatty acyl groups from cysteine residues during lysosomal degradation of lipid-modified proteins. Mutations cause neuronal ceroid lipofuscinosis 1 (CLN1), also known as infantile neuronal ceroid lipofuscinosis, inherited in an autosomal recessive pattern. Disease results from loss of enzyme function leading to accumulation of lipofuscin-like material in lysosomes.

OMIMResearchSummary from RefSeq, OMIM, UniProt, Mechanism
LOFmechanismARLOEUF 0.891 OMIM phenotype
Clinical SummaryPPT1
🧬
Gene-Disease Validity (ClinGen)
neuronal ceroid lipofuscinosis · ARDefinitive

Definitive — sufficient evidence for diagnostic panels

Population Constraint (gnomAD)
Low constraint (pLI 0.00) — loss-of-function variants are relatively tolerated in the population.
💊
Clinical Trials
1 active or recruiting trial — potential therapeutic options may be available

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Tolerant — LoF & missense variants common in population
LoF Constraint
0.89LOEUF
pLI 0.000
Z-score 1.94
OE 0.52 (0.320.89)
Tolerant

Typical tolerance to LoF variation

Missense Constraint
0.03Z-score
OE missense 0.99 (0.871.13)
163 obs / 164.2 exp
Tolerant

Mild missense constraint

Observed / Expected Ratios
LoF OE0.52 (0.320.89)
00.351.4
Missense OE0.99 (0.871.13)
00.61.4
Synonymous OE1.18
01.21.6
LoF obs/exp: 10 / 19.2Missense obs/exp: 163 / 164.2Syn Z: -1.10
Curated Mechanism (G2P)Gene2Phenotype (DDG2P) ↗
definitivePPT1-related neuronal ceroid lipofuscinosisLOFAR

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

DN
0.6551th %ile
GOF
0.5660th %ile
LOF
0.2385th %ile

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.

ClinVar Variant Classifications

0 submitted variants in ClinVar

Protein Context — Lollipop Plot

PPT1 · protein map & ClinVar variants

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

3D Protein StructureAlphaFold
Clinical Literature
Open Research Assistant →