SLCO1B3

Chr 12Digenic recessive

solute carrier organic anion transporter family member 1B3

Also known as: HBLRR, LST-2, LST-3TM13, LST3, OATP-8, OATP1B3, OATP8, SLC21A8

This protein is a liver-specific organic anion transporter that mediates sodium-independent uptake of bile acids, bilirubin, and other organic compounds from blood into hepatocytes. Mutations cause Rotor syndrome, a benign form of conjugated hyperbilirubinemia that typically presents in infancy or childhood with jaundice but normal liver function. The condition follows digenic recessive inheritance, requiring mutations in both SLCO1B1 and SLCO1B3 genes.

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

Hyperbilirubinemia, Rotor type, digenicMIM #237450
Digenic recessive
1
Active trials
22
Pubs (1 yr)
59
P/LP submissions
0%
P/LP missense
1.54
LOEUF
Multiple*
Mechanism· predicted
Clinical SummarySLCO1B3
Population Constraint (gnomAD)
Low constraint (pLI 0.00) — loss-of-function variants are relatively tolerated in the population.
📋
ClinVar Variants
51 unique Pathogenic / Likely Pathogenic· 196 VUS of 383 total submissions
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Clinical Trials
1 active or recruiting trial — potential therapeutic options may be available
📖
GeneReview available — SLCO1B3
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
1.54LOEUF
pLI 0.000
Z-score -0.77
OE 1.16 (0.881.54)
Tolerant

Highly tolerant — LoF variants common in population

Missense Constraint
-1.16Z-score
OE missense 1.17 (1.081.27)
412 obs / 350.7 exp
Tolerant

Tolerant to missense variation

Observed / Expected Ratios
LoF OE1.16 (0.881.54)
00.351.4
Missense OE1.17 (1.081.27)
00.61.4
Synonymous OE1.07
01.21.6
LoF obs/exp: 33 / 28.5Missense obs/exp: 412 / 350.7Syn Z: -0.61
DN
0.74top 25%
GOF
0.6931th %ile
LOF
0.2776th %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

383 submitted variants in ClinVar

Classification Summary

Pathogenic45
Likely Pathogenic6
VUS196
Likely Benign55
Benign56
Conflicting12
45
Pathogenic
6
Likely Pathogenic
196
VUS
55
Likely Benign
56
Benign
12
Conflicting

Curated Variants Distribution

Classified variants from ClinVar · 5 ACMG categories

ClassificationLoFMissense + InframeNon-codingSynonymousTotal
Pathogenic
3
0
42
0
45
Likely Pathogenic
3
0
3
0
6
VUS
4
144
44
4
196
Likely Benign
0
11
18
26
55
Benign
0
3
49
4
56
Conflicting
12
Total1015815634370

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

View in ClinVar →

Protein Context — Lollipop Plot

SLCO1B3 · protein map & ClinVar variants

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

Clinical Literature
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