TTLL8

Chr 22

tubulin tyrosine ligase like 8

This protein is a monoglycylase that adds glycine modifications to tubulin and other proteins, which is essential for microtubule stability and maintenance in cilia and sperm flagella. Mutations cause autosomal recessive primary ciliary dyskinesia, a disorder affecting respiratory function, fertility, and sometimes organ positioning due to defective ciliary motility. The gene shows minimal constraint against loss-of-function variants in the general population.

OMIMResearchSummary from RefSeq, UniProt
GOFmechanismLOEUF 1.03
Clinical SummaryTTLL8
Population Constraint (gnomAD)
Low constraint (pLI 0.00) — loss-of-function variants are relatively tolerated in the population.
📋
ClinVar Variants
138 unique Pathogenic / Likely Pathogenic· 12 VUS of 154 total submissions

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Tolerant — LoF & missense variants common in population
LoF Constraint
1.03LOEUF
pLI 0.000
Z-score 1.42
OE 0.75 (0.551.03)
Tolerant

Highly tolerant — LoF variants common in population

Missense Constraint
0.35Z-score
OE missense 0.95 (0.881.03)
451 obs / 472.5 exp
Tolerant

Mild missense constraint

Observed / Expected Ratios
LoF OE0.75 (0.551.03)
00.351.4
Missense OE0.95 (0.881.03)
00.61.4
Synonymous OE0.98
01.21.6
LoF obs/exp: 27 / 36.2Missense obs/exp: 451 / 472.5Syn Z: 0.28
DN
0.5771th %ile
GOF
0.7028th %ile
LOF
0.3550th %ile

The highest-scoring mechanism for this gene is gain-of-function.

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

154 submitted variants in ClinVar

Classification Summary

Pathogenic136
Likely Pathogenic2
VUS12
Likely Benign3
Benign1
136
Pathogenic
2
Likely Pathogenic
12
VUS
3
Likely Benign
1
Benign

Curated Variants Distribution

Classified variants from ClinVar · 5 ACMG categories· variant type breakdown unavailable

ClassificationLoFMissense + InframeNon-codingSynonymousTotal
Pathogenic
136
Likely Pathogenic
2
VUS
12
Likely Benign
3
Benign
1
Total154

Counts from ClinVar esearch · Updated hourly

View in ClinVar →

Protein Context — Lollipop Plot

TTLL8 · 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.

Search ClinicalTrials.gov →
Clinical Literature
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