ERLIN1

Chr 10AR

ER lipid raft associated 1

Also known as: C10orf69, Erlin-1, KE04, KEO4, SPFH1, SPG62

ERLIN1 encodes a protein that mediates endoplasmic reticulum-associated degradation of inositol 1,4,5-trisphosphate receptors and regulates cellular cholesterol homeostasis through the SREBP signaling pathway. Mutations cause spastic paraplegia 62, an autosomal recessive disorder affecting the pyramidal motor system. The gene shows moderate tolerance to loss-of-function variants (LOEUF 0.539).

OMIMResearchSummary from RefSeq, OMIM, UniProt
MultiplemechanismARLOEUF 0.541 OMIM phenotype
Clinical SummaryERLIN1
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Gene-Disease Validity (ClinGen)
hereditary spastic paraplegia 62 · ARDefinitive

Definitive — sufficient evidence for diagnostic panels

Population Constraint (gnomAD)
Constrained for loss-of-function variants (OE-LoF 0.26) despite low pLI — interpret in context.

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Moderate LoF intolerance
LoF Constraint
0.54LOEUF
pLI 0.178
Z-score 3.04
OE 0.26 (0.130.54)
Moderately constrained

More LoF-intolerant than ~75% of genes

Missense Constraint
1.79Z-score
OE missense 0.63 (0.540.73)
115 obs / 183.1 exp
Tolerant

Mild missense constraint

Observed / Expected Ratios
LoF OE0.26 (0.130.54)
00.351.4
Missense OE0.63 (0.540.73)
00.61.4
Synonymous OE0.96
01.21.6
LoF obs/exp: 5 / 19.5Missense obs/exp: 115 / 183.1Syn Z: 0.24
DN
0.77top 25%
GOF
0.6345th %ile
LOF
0.2873th %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

0 submitted variants in ClinVar

Protein Context — Lollipop Plot

ERLIN1 · 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.

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Clinical Literature
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