TIMP4

Chr 3

TIMP metallopeptidase inhibitor 4

Also known as: TIMP-4

The protein inhibits matrix metalloproteinases by binding to their catalytic zinc cofactor, regulating extracellular matrix degradation and playing roles in platelet aggregation and endometrial remodeling. Mutations cause autosomal recessive Joubert syndrome and related cerebellar disorders, typically presenting in infancy with characteristic brain malformations affecting the cerebellum and brainstem. The gene shows tolerance to loss-of-function variants (low constraint), which is consistent with the recessive inheritance pattern observed in affected individuals.

OMIMResearchSummary from RefSeq, UniProt
MultiplemechanismLOEUF 1.11
Clinical SummaryTIMP4
Population Constraint (gnomAD)
Low constraint (pLI 0.00) — loss-of-function variants are relatively tolerated in the population.
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ClinVar Variants
28 unique Pathogenic / Likely Pathogenic· 53 VUS of 88 total submissions
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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
1.11LOEUF
pLI 0.000
Z-score 1.30
OE 0.59 (0.341.11)
Tolerant

Highly tolerant — LoF variants common in population

Missense Constraint
-0.61Z-score
OE missense 1.15 (1.011.32)
147 obs / 127.7 exp
Tolerant

Tolerant to missense variation

Observed / Expected Ratios
LoF OE0.59 (0.341.11)
00.351.4
Missense OE1.15 (1.011.32)
00.61.4
Synonymous OE1.39
01.21.6
LoF obs/exp: 7 / 11.8Missense obs/exp: 147 / 127.7Syn Z: -2.21
DN
0.6261th %ile
GOF
0.6930th %ile
LOF
0.2774th %ile

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

GOFprediction above median
DNprediction 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

88 submitted variants in ClinVar

Classification Summary

Pathogenic27
Likely Pathogenic1
VUS53
Likely Benign2
Benign1
27
Pathogenic
1
Likely Pathogenic
53
VUS
2
Likely Benign
1
Benign

Curated Variants Distribution

Classified variants from ClinVar · 5 ACMG categories

ClassificationLoFMissense + InframeNon-codingSynonymousTotal
Pathogenic
0
0
27
0
27
Likely Pathogenic
0
0
1
0
1
VUS
0
53
0
0
53
Likely Benign
0
1
0
1
2
Benign
0
0
0
1
1
Total05428284

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

View in ClinVar →

Protein Context — Lollipop Plot

TIMP4 · protein map & ClinVar variants

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

3D Protein StructureAlphaFold
Clinical Literature
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