ARL6IP1

Chr 16AR

ARL6 interacting reticulophagy regulator 1

Also known as: AIP1, ARL6IP, ARMER, SPG61

The protein regulates glutamate transport by enhancing SLC1A1/EAAC1 transporter activity, forms and stabilizes endoplasmic reticulum tubules, and negatively regulates apoptosis. Mutations cause spastic paraplegia 61, an autosomal recessive disorder affecting the corticospinal tracts and leading to progressive lower limb spasticity. The gene shows relatively low constraint to loss-of-function variation (pLI 0.02, LOEUF 0.78).

OMIMResearchSummary from RefSeq, OMIM, UniProt
MultiplemechanismARLOEUF 0.781 OMIM phenotype
Clinical SummaryARL6IP1
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Gene-Disease Validity (ClinGen)
hereditary spastic paraplegia · ARDefinitive

Definitive — sufficient evidence for diagnostic panels

Population Constraint (gnomAD)
Low constraint (pLI 0.02) — loss-of-function variants are relatively tolerated in the population.

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Tolerant — LoF & missense variants common in population
LoF Constraint
0.78LOEUF
pLI 0.022
Z-score 2.15
OE 0.37 (0.190.78)
Tolerant

Typical tolerance to LoF variation

Missense Constraint
1.07Z-score
OE missense 0.71 (0.590.86)
79 obs / 110.7 exp
Tolerant

Mild missense constraint

Observed / Expected Ratios
LoF OE0.37 (0.190.78)
00.351.4
Missense OE0.71 (0.590.86)
00.61.4
Synonymous OE1.15
01.21.6
LoF obs/exp: 5 / 13.5Missense obs/exp: 79 / 110.7Syn Z: -0.72
DN
0.79top 25%
GOF
0.7125th %ile
LOF
0.1796th %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

ARL6IP1 · 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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