RBP3

Chr 10AR

retinol binding protein 3

Also known as: D10S64, D10S65, D10S66, IRBP, RBPI, RP66

Interphotoreceptor retinol-binding protein shuttles 11-cis and all-trans retinoids between the retinal pigment epithelium and photoreceptor cells, playing a critical role in the visual cycle. Mutations cause retinitis pigmentosa 66, inherited in an autosomal recessive pattern. The gene is highly intolerant to loss-of-function variants (pLI near 1.0, LOEUF 0.647), indicating strong evolutionary constraint.

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

?Retinitis pigmentosa 66MIM #615233
AR
0
Active trials
9
Pubs (1 yr)
22
P/LP submissions
5%
P/LP missense
0.65
LOEUF
LOF
Mechanism· G2P
Clinical SummaryRBP3
Population Constraint (gnomAD)
Low constraint (pLI 0.00) — loss-of-function variants are relatively tolerated in the population.
📋
ClinVar Variants
22 unique Pathogenic / Likely Pathogenic· 257 VUS of 400 total submissions
📖
GeneReview available — RBP3
Authoritative clinical overview · Recommended first read
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Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Tolerant — LoF & missense variants common in population
LoF Constraint
0.65LOEUF
pLI 0.000
Z-score 3.05
OE 0.40 (0.260.65)
Tolerant

Typical tolerance to LoF variation

Missense Constraint
-0.42Z-score
OE missense 1.04 (0.981.11)
785 obs / 752.6 exp
Tolerant

Tolerant to missense variation

Observed / Expected Ratios
LoF OE0.40 (0.260.65)
00.351.4
Missense OE1.04 (0.981.11)
00.61.4
Synonymous OE1.28
01.21.6
LoF obs/exp: 12 / 30.1Missense obs/exp: 785 / 752.6Syn Z: -4.02
Curated Mechanism (G2P)Gene2Phenotype (DDG2P) ↗
strongRBP3-related retinitis pigmentosaLOFAR

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

DN
0.74top 25%
GOF
0.7126th %ile
LOF
0.2581th %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

400 submitted variants in ClinVar

Classification Summary

Pathogenic16
Likely Pathogenic6
VUS257
Likely Benign118
Conflicting2
16
Pathogenic
6
Likely Pathogenic
257
VUS
118
Likely Benign
2
Conflicting

Curated Variants Distribution

Classified variants from ClinVar · 5 ACMG categories

ClassificationLoFMissense + InframeNon-codingSynonymousTotal
Pathogenic
5
0
11
0
16
Likely Pathogenic
1
1
3
1
6
VUS
2
240
10
5
257
Likely Benign
0
5
5
108
118
Benign
0
0
0
0
0
Conflicting
2
Total824629114399

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

View in ClinVar →

Protein Context — Lollipop Plot

RBP3 · protein map & ClinVar variants

Showing all ClinVar variants across the protein. Search a specific variant to highlight its position.

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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