RAB5IF

Chr 20AR

RAB5 interacting factor

Also known as: C20orf24, CFSMR2, OPTI, PNAS-11, RCAF1, RIP5

The protein is a component of the multi-pass translocon complex that mediates insertion of multi-pass membrane proteins into the ER membrane and acts as an assembly factor for mitochondrial respiratory complexes. Autosomal recessive mutations cause craniofacial dysmorphism, skeletal anomalies, and impaired intellectual development syndrome 2. The gene shows moderate constraint against loss-of-function variants (LOEUF 0.602), and the condition involves craniofacial, skeletal, and neurodevelopmental systems.

OMIMResearchSummary from RefSeq, OMIM, UniProt
MultiplemechanismARLOEUF 0.601 OMIM phenotype
Clinical SummaryRAB5IF
Population Constraint (gnomAD)
Moderately constrained gene (pLI 0.67) — some intolerance to loss-of-function variants.

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Moderate LoF intolerance
LoF Constraint
0.60LOEUF
pLI 0.670
Z-score 2.27
OE 0.13 (0.040.60)
Moderately constrained

Typical tolerance to LoF variation

Missense Constraint
0.51Z-score
OE missense 0.83 (0.671.03)
60 obs / 72.2 exp
Tolerant

Mild missense constraint

Observed / Expected Ratios
LoF OE0.13 (0.040.60)
00.351.4
Missense OE0.83 (0.671.03)
00.61.4
Synonymous OE1.41
01.21.6
LoF obs/exp: 1 / 7.9Missense obs/exp: 60 / 72.2Syn Z: -1.72
DN
0.6453th %ile
GOF
0.74top 25%
LOF
0.2968th %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

0 submitted variants in ClinVar

Protein Context — Lollipop Plot

RAB5IF · 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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