RIMS2

Chr 8AR

regulating synaptic membrane exocytosis 2

Also known as: CRSDS, OBOE, RAB3IP3, RIM2

RIMS2 encodes a presynaptic scaffold protein that regulates neurotransmitter release by interacting with RAB3 and other synaptic proteins involved in exocytosis. Biallelic mutations cause cone-rod synaptic disorder syndrome, a congenital nonprogressive condition affecting retinal photoreceptor synapses and leading to visual impairment from birth. This gene is highly constrained against loss-of-function variants (pLI ~1.0, LOEUF 0.21) and follows autosomal recessive inheritance.

Summary from RefSeq, OMIM, UniProt
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Primary Disease Associations & Inheritance

Cone-rod synaptic disorder syndrome, congenital nonprogressiveMIM #618970
AR
0
Active trials
10
Pubs (1 yr)
47
P/LP submissions
0%
P/LP missense
0.21
LOEUF· LoF intol.
LOF
Mechanism· G2P
Clinical SummaryRIMS2
Population Constraint (gnomAD)
Highly constrained gene — heterozygous loss-of-function variants are very rare in the population (pLI 1.00). One damaged copy is likely sufficient to cause disease.
📋
ClinVar Variants
46 unique Pathogenic / Likely Pathogenic· 204 VUS of 294 total submissions

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

LoF intolerant — likely haploinsufficient
LoF Constraint
0.21LOEUF
pLI 1.000
Z-score 7.38
OE 0.12 (0.070.21)
Highly constrained

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

Missense Constraint
0.78Z-score
OE missense 0.92 (0.860.98)
697 obs / 757.1 exp
Tolerant

Mild missense constraint

Observed / Expected Ratios
LoF OE0.12 (0.070.21)
00.351.4
Missense OE0.92 (0.860.98)
00.61.4
Synonymous OE1.04
01.21.6
LoF obs/exp: 10 / 82.2Missense obs/exp: 697 / 757.1Syn Z: -0.56
Curated Mechanism (G2P)Gene2Phenotype (DDG2P) ↗
strongRIMS2-related syndromic congenital cone-rod synaptic diseaseLOFAR

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

DN
0.4983th %ile
GOF
0.5269th %ile
LOF
0.76top 10%

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. Mechanism ranking also informed by gnomAD constraint, ClinVar, and ClinGen data.

ClinVar Variant Classifications

294 submitted variants in ClinVar

Classification Summary

Pathogenic44
Likely Pathogenic2
VUS204
Likely Benign19
Benign7
Conflicting2
44
Pathogenic
2
Likely Pathogenic
204
VUS
19
Likely Benign
7
Benign
2
Conflicting

Curated Variants Distribution

Classified variants from ClinVar · 5 ACMG categories

ClassificationLoFMissense + InframeNon-codingSynonymousTotal
Pathogenic
1
0
43
0
44
Likely Pathogenic
1
0
1
0
2
VUS
1
191
11
1
204
Likely Benign
0
3
7
9
19
Benign
0
3
0
4
7
Conflicting
2
Total31976214278

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

View in ClinVar →

Protein Context — Lollipop Plot

RIMS2 · protein map & ClinVar variants

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

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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