OSBPL1A

Chr 18

oxysterol binding protein like 1A

Also known as: ORP-1, ORP1, OSBPL1B

The protein binds phospholipids and cholesterol metabolites, stabilizes RAB7A on late endosomes/lysosomes, and alters the functional properties of late endocytic compartments. OSBPL1A is extremely intolerant to loss-of-function variants (LOEUF 0.522), suggesting that mutations likely cause significant clinical phenotypes, though specific disease associations have not yet been established. The inheritance pattern and associated phenotypes remain to be determined.

Summary from RefSeq, UniProt
Research Assistant →
0
Active trials
2
Pubs (1 yr)
36
P/LP submissions
0%
P/LP missense
0.52
LOEUF
Mechanism
Clinical SummaryOSBPL1A
Population Constraint (gnomAD)
Low constraint (pLI 0.00) — loss-of-function variants are relatively tolerated in the population.
📋
ClinVar Variants
34 unique Pathogenic / Likely Pathogenic· 105 VUS of 168 total submissions

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Moderate LoF intolerance
LoF Constraint
0.52LOEUF
pLI 0.000
Z-score 4.62
OE 0.37 (0.260.52)
Moderately constrained

More LoF-intolerant than ~75% of genes

Missense Constraint
1.56Z-score
OE missense 0.80 (0.740.87)
405 obs / 503.3 exp
Tolerant

Mild missense constraint

Observed / Expected Ratios
LoF OE0.37 (0.260.52)
00.351.4
Missense OE0.80 (0.740.87)
00.61.4
Synonymous OE1.00
01.21.6
LoF obs/exp: 23 / 62.4Missense obs/exp: 405 / 503.3Syn Z: 0.01

ClinVar Variant Classifications

168 submitted variants in ClinVar

Classification Summary

Pathogenic33
Likely Pathogenic1
VUS105
Likely Benign3
33
Pathogenic
1
Likely Pathogenic
105
VUS
3
Likely Benign

Curated Variants Distribution

Classified variants from ClinVar · 5 ACMG categories

ClassificationLoFMissense + InframeNon-codingSynonymousTotal
Pathogenic
0
0
33
0
33
Likely Pathogenic
0
0
1
0
1
VUS
0
99
6
0
105
Likely Benign
0
2
0
1
3
Benign
0
0
0
0
0
Total0101401142

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

View in ClinVar →

Protein Context — Lollipop Plot

OSBPL1A · 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
Open Research Assistant →