TTLL7

Chr 1

tubulin tyrosine ligase like 7

The protein functions as a polyglutamylase that modifies tubulin by generating polyglutamate side chains on tubulin tails, preferentially targeting beta-tubulin and playing a critical role in neurite growth and postnatal neuronal maturation. Mutations cause autosomal recessive retinal dystrophy and cone-rod dystrophy, affecting vision typically in childhood or adolescence. The gene is highly constrained against loss-of-function variants, indicating that complete loss of protein function is likely incompatible with normal development.

Summary from RefSeq, UniProt
Research Assistant →
0
Active trials
0
Pubs (1 yr)
16
P/LP submissions
0%
P/LP missense
0.43
LOEUF
Multiple*
Mechanism· predicted
Clinical SummaryTTLL7
Population Constraint (gnomAD)
Constrained for loss-of-function variants (OE-LoF 0.28) despite low pLI — interpret in context.
📋
ClinVar Variants
16 unique Pathogenic / Likely Pathogenic· 94 VUS of 121 total submissions

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Moderate LoF intolerance
LoF Constraint
0.43LOEUF
pLI 0.004
Z-score 5.04
OE 0.28 (0.190.43)
Moderately constrained

More LoF-intolerant than ~75% of genes

Missense Constraint
2.58Z-score
OE missense 0.67 (0.610.73)
316 obs / 474.0 exp
Mild constraint

Moderately missense-constrained (top ~2.5%)

Observed / Expected Ratios
LoF OE0.28 (0.190.43)
00.351.4
Missense OE0.67 (0.610.73)
00.61.4
Synonymous OE1.01
01.21.6
LoF obs/exp: 16 / 57.0Missense obs/exp: 316 / 474.0Syn Z: -0.13
DN
0.6356th %ile
GOF
0.6930th %ile
LOF
0.3551th %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

121 submitted variants in ClinVar

Classification Summary

Pathogenic14
Likely Pathogenic2
VUS94
Likely Benign4
14
Pathogenic
2
Likely Pathogenic
94
VUS
4
Likely Benign

Curated Variants Distribution

Classified variants from ClinVar · 5 ACMG categories

ClassificationLoFMissense + InframeNon-codingSynonymousTotal
Pathogenic
0
0
14
0
14
Likely Pathogenic
0
0
2
0
2
VUS
0
91
3
0
94
Likely Benign
0
2
1
1
4
Benign
0
0
0
0
0
Total093201114

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

View in ClinVar →

Protein Context — Lollipop Plot

TTLL7 · 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.

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Clinical Literature
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