DRD5

Chr 4AD

dopamine receptor D5

Also known as: DBDR, DRD1B, DRD1L2

The D5 dopamine receptor is a G-protein coupled receptor that stimulates adenylyl cyclase and is highly expressed in limbic brain regions, with 10-fold higher dopamine affinity than the D1 subtype. Mutations cause autosomal dominant susceptibility to attention deficit-hyperactivity disorder and primary benign blepharospasm. This gene shows very low constraint against loss-of-function variants (pLI near zero), suggesting tolerance to such mutations.

Summary from RefSeq, OMIM, UniProt
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Primary Disease Associations & Inheritance

{Attention deficit-hyperactivity disorder, susceptibility to}MIM #143465
AD
{Blepharospasm, primary benign}MIM #606798
AD
UniProtBenign essential blepharospasm
0
Active trials
18
Pubs (1 yr)
66
P/LP submissions
0%
P/LP missense
1.94
LOEUF
Multiple*
Mechanism· predicted
Clinical SummaryDRD5
Population Constraint (gnomAD)
Low constraint (pLI 0.00) — loss-of-function variants are relatively tolerated in the population.
📋
ClinVar Variants
66 unique Pathogenic / Likely Pathogenic· 114 VUS of 197 total submissions

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Tolerant — LoF & missense variants common in population
LoF Constraint
1.94LOEUF
pLI 0.000
Z-score -1.89
OE 1.57 (1.081.94)
Tolerant

Highly tolerant — LoF variants common in population

Missense Constraint
-0.45Z-score
OE missense 1.07 (0.981.17)
348 obs / 325.4 exp
Tolerant

Tolerant to missense variation

Observed / Expected Ratios
LoF OE1.57 (1.081.94)
00.351.4
Missense OE1.07 (0.981.17)
00.61.4
Synonymous OE1.08
01.21.6
LoF obs/exp: 20 / 12.7Missense obs/exp: 348 / 325.4Syn Z: -0.78
DN
0.6936th %ile
GOF
0.80top 10%
LOF
0.3356th %ile

This gene has evidence for multiple mechanisms of pathogenicity (gain-of-function and dominant-negative). Both the Badonyi & Marsh prediction and the broader genomic evidence point to gain-of-function as the predominant mechanism. Different variants in this gene may act through different mechanisms — interpret in context of the specific variant.

GOFprediction above median · 1 literature citation
DNprediction 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.

Literature Evidence

GOFExpression of Q366R DRD5 was sufficient to induce tumor cell apoptosis, consistent with a gain-of-function.PMID:30559168

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

ClinVar Variant Classifications

197 submitted variants in ClinVar

Classification Summary

Pathogenic63
Likely Pathogenic3
VUS114
Likely Benign12
Benign4
Conflicting1
63
Pathogenic
3
Likely Pathogenic
114
VUS
12
Likely Benign
4
Benign
1
Conflicting

Curated Variants Distribution

Classified variants from ClinVar · 5 ACMG categories

ClassificationLoFMissense + InframeNon-codingSynonymousTotal
Pathogenic
0
0
63
0
63
Likely Pathogenic
0
0
3
0
3
VUS
0
97
16
1
114
Likely Benign
0
4
7
1
12
Benign
0
2
2
0
4
Conflicting
1
Total0103912197

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

View in ClinVar →

Protein Context — Lollipop Plot

DRD5 · 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
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