DPM3

Chr 1AR

dolichyl-phosphate mannosyltransferase subunit 3, regulatory

Also known as: CDG1O, MDDGB15, MDDGC15

The protein is a stabilizer subunit of the dolichol-phosphate mannose synthase complex that tethers the catalytic subunit to the endoplasmic reticulum, essential for synthesizing mannose donors required for proper glycosylation of proteins including dystroglycan. Mutations cause autosomal recessive muscular dystrophy-dystroglycanopathy, presenting as either congenital muscular dystrophy with intellectual disability or limb-girdle muscular dystrophy. This affects both muscle and brain development due to defective glycosylation of dystroglycan and other glycoproteins.

OMIMResearchSummary from RefSeq, OMIM, UniProt
LOFmechanismARLOEUF 1.402 OMIM phenotypes
Clinical SummaryDPM3
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Gene-Disease Validity (ClinGen)
DPM3-congenital disorder of glycosylation · ARModerate

Moderate evidence — consider for supplementary testing

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
1.40LOEUF
pLI 0.233
Z-score 1.14
OE 0.31 (0.111.40)
Tolerant

Highly tolerant — LoF variants common in population

Missense Constraint
0.42Z-score
OE missense 0.86 (0.701.07)
59 obs / 68.7 exp
Tolerant

Mild missense constraint

Observed / Expected Ratios
LoF OE0.31 (0.111.40)
00.351.4
Missense OE0.86 (0.701.07)
00.61.4
Synonymous OE0.99
01.21.6
LoF obs/exp: 1 / 3.2Missense obs/exp: 59 / 68.7Syn Z: 0.05
Curated Mechanism (G2P)Gene2Phenotype (DDG2P) ↗
strongDPM3-related congenital disorder of glycosylationLOFAR

Predictions shown for reference only — model trained on dominant genes, not applicable to AR conditions.

DN
0.7132th %ile
GOF
0.7029th %ile
LOF
0.2777th %ile

The Badonyi & Marsh prediction model was trained exclusively on dominant disease genes. Predictions are not reliable for genes with autosomal recessive inheritance and are shown at reduced opacity for reference only.

Predictions from Badonyi M, Marsh JA. PLoS ONE. 2024;19(8):e0307312.

ClinVar Variant Classifications

0 submitted variants in ClinVar

Protein Context — Lollipop Plot

DPM3 · 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
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