DOP1A

Chr 6

DOP1 leucine zipper like protein A

Also known as: DOP1, DOPEY1, KIAA1117, dJ202D23.2

The protein is involved in trafficking between the late Golgi and early endosomes and participates in endoplasmic reticulum organization. Mutations cause autosomal recessive developmental delay, intellectual disability, and seizures with onset in infancy or early childhood. The gene is highly constrained against loss-of-function variants (pLI 1.0, LOEUF 0.25), indicating intolerance to functional disruption.

OMIMResearchSummary from RefSeq, UniProt
LOFmechanismLOEUF 0.25
Clinical SummaryDOP1A
Population Constraint (gnomAD)
Highly constrained gene — heterozygous loss-of-function variants are very rare in the population (pLI 1.00). One damaged copy is likely sufficient to cause disease.

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Dual constrained — LoF & missense intolerant
LoF Constraint
0.25LOEUF
pLI 1.000
Z-score 8.37
OE 0.18 (0.120.25)
Highly constrained

Highly LoF-intolerant (top ~10% of genes)

Missense Constraint
3.32Z-score
OE missense 0.74 (0.700.78)
919 obs / 1249.5 exp
Constrained

Highly missense-constrained (top ~0.1%)

Observed / Expected Ratios
LoF OE0.18 (0.120.25)
00.351.4
Missense OE0.74 (0.700.78)
00.61.4
Synonymous OE0.87
01.21.6
LoF obs/exp: 21 / 119.9Missense obs/exp: 919 / 1249.5Syn Z: 2.15
DN
0.4487th %ile
GOF
0.3887th %ile
LOF
0.65top 25%

The highest-scoring mechanism for this gene is loss-of-function (haploinsufficiency).

LOFprediction above median · LOEUF 0.25

Predictions from Badonyi M, Marsh JA. PLoS ONE. 2024;19(8):e0307312. Mechanism ranking also informed by gnomAD constraint, ClinVar, and ClinGen data.

ClinVar Variant Classifications

0 submitted variants in ClinVar

Protein Context — Lollipop Plot

DOP1A · 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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Recent Gene-Specific Literature
Gene in title · MEDLINE · newest first
Europe PMC

No open access results found