FAR1

Chr 11ADAR

fatty acyl-CoA reductase 1

Also known as: CSPSD, MLSTD2, PFCRD, SDR10E1

This peroxisomal membrane protein catalyzes the reduction of fatty acyl-CoA to fatty alcohols, which are essential substrates for synthesizing ether lipids and plasmalogens. Mutations cause a spectrum of neurological disorders including severe intellectual disability, early-onset epilepsy, microcephaly, congenital cataracts, growth retardation, and spastic paraparesis. The condition follows both autosomal dominant and autosomal recessive inheritance patterns.

OMIMResearchSummary from RefSeq, OMIM, UniProt
LOFmechanismAD/ARLOEUF 0.302 OMIM phenotypes
Clinical SummaryFAR1
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Gene-Disease Validity (ClinGen)
fatty acyl-CoA reductase 1 upregulation · ADModerate

Moderate evidence — consider for supplementary testing

2 total gene-disease associations curated

Population Constraint (gnomAD)
Highly constrained gene — heterozygous loss-of-function variants are very rare in the population (pLI 0.99). One damaged copy is likely sufficient to cause disease.
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ClinVar Variants
5 unique Pathogenic / Likely Pathogenic· 107 VUS of 200 total submissions

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Dual constrained — LoF & missense intolerant
LoF Constraint
0.30LOEUF
pLI 0.985
Z-score 4.18
OE 0.12 (0.050.30)
Highly constrained

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

Missense Constraint
3.30Z-score
OE missense 0.44 (0.380.52)
124 obs / 279.4 exp
Constrained

Highly missense-constrained (top ~0.1%)

Observed / Expected Ratios
LoF OE0.12 (0.050.30)
00.351.4
Missense OE0.44 (0.380.52)
00.61.4
Synonymous OE0.93
01.21.6
LoF obs/exp: 3 / 26.0Missense obs/exp: 124 / 279.4Syn Z: 0.53

ClinVar Variant Classifications

200 submitted variants in ClinVar

Classification Summary

Pathogenic2
Likely Pathogenic3
VUS107
Likely Benign78
Benign1
2
Pathogenic
3
Likely Pathogenic
107
VUS
78
Likely Benign
1
Benign

Curated Variants Distribution

Classified variants from ClinVar · 5 ACMG categories

ClassificationLoFMissense + InframeNon-codingSynonymousTotal
Pathogenic
2
0
0
0
2
Likely Pathogenic
2
1
0
0
3
VUS
1
93
8
5
107
Likely Benign
0
1
36
41
78
Benign
0
0
1
0
1
Total5954546191

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

View in ClinVar →

Protein Context — Lollipop Plot

FAR1 · 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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