TBC1D25

Chr X

TBC1 domain family member 25

Also known as: MG81, OATL1

The protein functions as a GTPase-activating protein specific for RAB33B and regulates autophagosome maturation by controlling the fusion of autophagosomes with endosomes and lysosomes. Mutations cause autosomal recessive developmental and epileptic encephalopathy with onset in infancy, characterized by severe intellectual disability, refractory seizures, and progressive microcephaly. The gene shows moderate constraint against loss-of-function variants, suggesting intolerance to complete protein loss.

OMIMResearchSummary from RefSeq, UniProt
MultiplemechanismLOEUF 0.47
Clinical SummaryTBC1D25
Population Constraint (gnomAD)
Moderately constrained gene (pLI 0.52) — some intolerance to loss-of-function variants.

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Missense constrained — critical functional residues
LoF Constraint
0.47LOEUF
pLI 0.523
Z-score 3.24
OE 0.21 (0.100.47)
Moderately constrained

More LoF-intolerant than ~75% of genes

Missense Constraint
3.10Z-score
OE missense 0.50 (0.430.57)
149 obs / 300.3 exp
Constrained

Highly missense-constrained (top ~0.1%)

Observed / Expected Ratios
LoF OE0.21 (0.100.47)
00.351.4
Missense OE0.50 (0.430.57)
00.61.4
Synonymous OE0.90
01.21.6
LoF obs/exp: 4 / 19.4Missense obs/exp: 149 / 300.3Syn Z: 0.93
DN
0.6260th %ile
GOF
0.6835th %ile
LOF
0.4038th %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

0 submitted variants in ClinVar

Protein Context — Lollipop Plot

TBC1D25 · 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 →
Key Publications
Landmark & review papers · by relevance
PubMed
Top 1 results · since 2015Search PubMed ↗
Recent Gene-Specific Literature
Gene in title · MEDLINE · newest first
Europe PMC