TTC39A

Chr 1

tetratricopeptide repeat domain 39A

Also known as: C1orf34, DEME-6

TTC39A encodes a protein located in the centrosome, a cellular structure critical for cell division and cilia formation. Mutations in this gene cause autosomal recessive developmental and epileptic encephalopathy with microcephaly, affecting the nervous system and brain development. The gene is highly constrained against loss-of-function variants, indicating that complete loss of protein function is likely pathogenic.

OMIMResearchSummary from RefSeq
MultiplemechanismLOEUF 0.56
Clinical SummaryTTC39A
Population Constraint (gnomAD)
Constrained for loss-of-function variants (OE-LoF 0.35) despite low pLI — interpret in context.
📋
ClinVar Variants
8 unique Pathogenic / Likely Pathogenic· 63 VUS of 94 total submissions

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Moderate LoF intolerance
LoF Constraint
0.56LOEUF
pLI 0.001
Z-score 3.58
OE 0.35 (0.220.56)
Moderately constrained

More LoF-intolerant than ~75% of genes

Missense Constraint
2.28Z-score
OE missense 0.64 (0.570.72)
203 obs / 317.3 exp
Mild constraint

Moderately missense-constrained (top ~2.5%)

Observed / Expected Ratios
LoF OE0.35 (0.220.56)
00.351.4
Missense OE0.64 (0.570.72)
00.61.4
Synonymous OE0.89
01.21.6
LoF obs/exp: 12 / 34.8Missense obs/exp: 203 / 317.3Syn Z: 0.95
DN
0.6841th %ile
GOF
0.6638th %ile
LOF
0.3068th %ile

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

DNprediction above median
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

94 submitted variants in ClinVar

Classification Summary

Pathogenic5
Likely Pathogenic3
VUS63
Likely Benign4
5
Pathogenic
3
Likely Pathogenic
63
VUS
4
Likely Benign

Curated Variants Distribution

Classified variants from ClinVar · 5 ACMG categories

ClassificationLoFMissense + InframeNon-codingSynonymousTotal
Pathogenic
0
0
5
0
5
Likely Pathogenic
0
0
3
0
3
VUS
0
59
4
0
63
Likely Benign
0
2
0
2
4
Benign
0
0
0
0
0
Total06112275

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

View in ClinVar →

Protein Context — Lollipop Plot

TTC39A · 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
Open Research Assistant →
Recent Gene-Specific Literature
Gene in title · MEDLINE · newest first
Europe PMC