ACBD5

Chr 10AR

acyl-CoA binding domain containing 5

The protein binds medium- and long-chain acyl-CoA esters and serves as the peroxisome receptor for pexophagy (selective autophagy of peroxisomes). Biallelic mutations cause retinal dystrophy with leukodystrophy, inherited in an autosomal recessive pattern. This gene is highly constrained against loss-of-function variants (LOEUF 0.53), reflecting its essential role in peroxisome homeostasis.

OMIMResearchSummary from RefSeq, OMIM, UniProt
LOFmechanismARLOEUF 0.531 OMIM phenotype
Clinical SummaryACBD5
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Gene-Disease Validity (ClinGen)
acyl-CoA binding domain containing protein 5 deficiency · ARDefinitive

Definitive — sufficient evidence for diagnostic panels

Population Constraint (gnomAD)
Constrained for loss-of-function variants (OE-LoF 0.31) despite low pLI — interpret in context.
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Clinical Trials
1 active or recruiting trial — potential therapeutic options may be available
Some data sources returned errors (1)

ncbi: Error: NCBI fetch failed: 429 https://eutils.ncbi.nlm.nih.gov/entrez/eutils/esearch.fcgi

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Moderate LoF intolerance
LoF Constraint
0.53LOEUF
pLI 0.006
Z-score 3.61
OE 0.31 (0.190.53)
Moderately constrained

More LoF-intolerant than ~75% of genes

Missense Constraint
0.87Z-score
OE missense 0.86 (0.770.95)
246 obs / 287.7 exp
Tolerant

Mild missense constraint

Observed / Expected Ratios
LoF OE0.31 (0.190.53)
00.351.4
Missense OE0.86 (0.770.95)
00.61.4
Synonymous OE1.06
01.21.6
LoF obs/exp: 10 / 32.0Missense obs/exp: 246 / 287.7Syn Z: -0.46
Curated Mechanism (G2P)Gene2Phenotype (DDG2P) ↗
strongACBD5-related deficiencyLOFAR

Predictions shown for reference only — model trained on dominant genes, not applicable to AR conditions.

DN
0.6938th %ile
GOF
0.5366th %ile
LOF
0.3065th %ile

The Badonyi & Marsh prediction model was trained exclusively on dominant disease genes. Predictions are not reliable for genes with autosomal recessive inheritance and are shown at reduced opacity for reference only.

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

ClinVar Variant Classifications

0 submitted variants in ClinVar

Protein Context — Lollipop Plot

ACBD5 · protein map & ClinVar variants

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

3D Protein StructureAlphaFold
Clinical Literature
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