CDIPT

Chr 16

CDP-diacylglycerol--inositol 3-phosphatidyltransferase

Also known as: PIS, PIS1

The enzyme catalyzes the biosynthesis of phosphatidylinositol and performs phosphatidylinositol:inositol exchange reactions on the endoplasmic reticulum and Golgi apparatus. Mutations cause autosomal recessive congenital cataracts, facial dysmorphism, and neuropathy (CCFDN), a syndrome presenting in infancy with cataracts, distinctive facial features, progressive peripheral neuropathy, and developmental delays. The gene shows moderate tolerance to loss-of-function variants based on population genetics data.

OMIMResearchSummary from RefSeq, UniProt
MultiplemechanismLOEUF 0.79
Clinical SummaryCDIPT
Population Constraint (gnomAD)
Constrained for loss-of-function variants (OE-LoF 0.31) despite low pLI — interpret in context.

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Tolerant — LoF & missense variants common in population
LoF Constraint
0.79LOEUF
pLI 0.130
Z-score 2.01
OE 0.31 (0.140.79)
Tolerant

Typical tolerance to LoF variation

Missense Constraint
0.72Z-score
OE missense 0.82 (0.700.97)
105 obs / 127.9 exp
Tolerant

Mild missense constraint

Observed / Expected Ratios
LoF OE0.31 (0.140.79)
00.351.4
Missense OE0.82 (0.700.97)
00.61.4
Synonymous OE0.87
01.21.6
LoF obs/exp: 3 / 9.8Missense obs/exp: 105 / 127.9Syn Z: 0.73
DN
0.80top 10%
GOF
0.6442th %ile
LOF
0.2680th %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

CDIPT · 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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