GLTPD2

Chr 17

glycolipid transfer protein domain containing 2

The protein is predicted to bind and transfer ceramide 1-phosphate between cellular membranes within the cytoplasm, facilitating ceramide transport and intermembrane lipid transfer. This gene is highly constrained against loss-of-function variants (pLI = 0.00006), suggesting that mutations would likely cause severe disease, but no definitive disease associations have been established in humans. Further clinical and functional studies are needed to determine the phenotypic consequences of GLTPD2 mutations.

OMIMResearchSummary from RefSeq
MultiplemechanismLOEUF 1.54
Clinical SummaryGLTPD2
Population Constraint (gnomAD)
Low constraint (pLI 0.00) — loss-of-function variants are relatively tolerated in the population.

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Tolerant — LoF & missense variants common in population
LoF Constraint
1.54LOEUF
pLI 0.000
Z-score 0.44
OE 0.84 (0.471.54)
Tolerant

Highly tolerant — LoF variants common in population

Missense Constraint
-0.16Z-score
OE missense 1.04 (0.911.19)
143 obs / 137.8 exp
Tolerant

Tolerant to missense variation

Observed / Expected Ratios
LoF OE0.84 (0.471.54)
00.351.4
Missense OE1.04 (0.911.19)
00.61.4
Synonymous OE0.98
01.21.6
LoF obs/exp: 7 / 8.4Missense obs/exp: 143 / 137.8Syn Z: 0.11
DN
0.6745th %ile
GOF
0.6444th %ile
LOF
0.2387th %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

0 submitted variants in ClinVar

Protein Context — Lollipop Plot

GLTPD2 · 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.

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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

No open access results found