LRP1B

Chr 2

LDL receptor related protein 1B

Also known as: LRP-1B, LRP-DIT, LRPDIT

The LRP1B protein is a low-density lipoprotein receptor family member that binds and internalizes ligands through receptor-mediated endocytosis at the cell surface. Mutations cause neurodevelopmental disorders with intellectual disability and autism spectrum disorder features, inherited in an autosomal dominant pattern. This gene is highly constrained against loss-of-function variants in the population, indicating intolerance to such mutations.

OMIMResearchSummary from RefSeq, UniProt
MultiplemechanismLOEUF 0.32
Clinical SummaryLRP1B
Population Constraint (gnomAD)
Constrained for loss-of-function variants (OE-LoF 0.26) despite low pLI — interpret in context.
📋
ClinVar Variants
9 unique Pathogenic / Likely Pathogenic· 278 VUS of 400 total submissions
💊
Clinical Trials
1 active or recruiting trial — potential therapeutic options may be available

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

LoF intolerant — likely haploinsufficient
LoF Constraint
0.32LOEUF
pLI 0.000
Z-score 10.77
OE 0.26 (0.210.32)
Highly constrained

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

Missense Constraint
1.93Z-score
OE missense 0.89 (0.860.92)
2148 obs / 2414.6 exp
Tolerant

Mild missense constraint

Observed / Expected Ratios
LoF OE0.26 (0.210.32)
00.351.4
Missense OE0.89 (0.860.92)
00.61.4
Synonymous OE1.11
01.21.6
LoF obs/exp: 63 / 244.9Missense obs/exp: 2148 / 2414.6Syn Z: -2.56
DN
0.6550th %ile
GOF
0.7029th %ile
LOF
0.3941th %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
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.

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

ClinVar Variant Classifications

400 submitted variants in ClinVar

Classification Summary

Pathogenic1
Likely Pathogenic8
VUS278
Likely Benign61
Benign49
1
Pathogenic
8
Likely Pathogenic
278
VUS
61
Likely Benign
49
Benign

Curated Variants Distribution

Classified variants from ClinVar · 5 ACMG categories

ClassificationLoFMissense + InframeNon-codingSynonymousTotal
Pathogenic
0
0
1
0
1
Likely Pathogenic
0
5
2
1
8
VUS
1
274
3
0
278
Likely Benign
0
21
7
33
61
Benign
0
14
13
22
49
Total13142656397

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

View in ClinVar →

Protein Context — Lollipop Plot

LRP1B · protein map & ClinVar variants

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

3D Protein StructureAlphaFold
Clinical Literature
Open Research Assistant →