TVP23C-CDRT4

Chr 17

TVP23C-CDRT4 readthrough

Also known as: FAM18B2-CDRT4

This locus encodes a readthrough fusion protein that combines sequences from the trans-Golgi network vesicle protein TVP23C with CDRT4, though the specific function of this fusion protein remains unclear. Mutations cause autosomal recessive developmental and epileptic encephalopathy with microcephaly, typically presenting in early infancy with severe intellectual disability and refractory seizures. The gene shows very low constraint against loss-of-function variants, suggesting haploinsufficiency is likely tolerated.

ResearchSummary from RefSeq
LOEUF 1.48
Clinical SummaryTVP23C-CDRT4
Population Constraint (gnomAD)
Low constraint (pLI 0.00) — loss-of-function variants are relatively tolerated in the population.
📋
ClinVar Variants
119 unique Pathogenic / Likely Pathogenic· 25 VUS of 153 total submissions

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Tolerant — LoF & missense variants common in population
LoF Constraint
1.48LOEUF
pLI 0.000
Z-score 0.38
OE 0.88 (0.541.48)
Tolerant

Highly tolerant — LoF variants common in population

Missense Constraint
0.06Z-score
OE missense 0.98 (0.821.18)
83 obs / 84.6 exp
Tolerant

Mild missense constraint

Observed / Expected Ratios
LoF OE0.88 (0.541.48)
00.351.4
Missense OE0.98 (0.821.18)
00.61.4
Synonymous OE0.99
01.21.6
LoF obs/exp: 10 / 11.4Missense obs/exp: 83 / 84.6Syn Z: 0.02

ClinVar Variant Classifications

153 submitted variants in ClinVar

Classification Summary

Pathogenic116
Likely Pathogenic3
VUS25
Likely Benign8
116
Pathogenic
3
Likely Pathogenic
25
VUS
8
Likely Benign

Curated Variants Distribution

Classified variants from ClinVar · 5 ACMG categories

ClassificationLoFMissense + InframeNon-codingSynonymousTotal
Pathogenic
0
0
116
0
116
Likely Pathogenic
0
0
3
0
3
VUS
0
20
5
0
25
Likely Benign
0
5
2
1
8
Benign
0
0
0
0
0
Total0251261152

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

View in ClinVar →

Protein Context — Lollipop Plot

TVP23C-CDRT4 · 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 2 results · since 2015Search PubMed ↗
Recent Gene-Specific Literature
Gene in title · MEDLINE · newest first
Europe PMC

No open access results found