RP1L1

Chr 8ADAR

RP1 like 1

Also known as: DCDC4B, OCMD, RP88

This gene encodes a member of the doublecortin family. The protein encoded by this gene contains two N-terminal doublecortin domains, which bind microtubules and regulate microtubule polymerization, and two C-terminal large repetitive regions, both of which contain a high percentage of glutamine and glutamic acid residues. This protein is a retinal-specific protein. Its exact length varies among individuals due to the presence of a 16aa repeat in the first C-terminal repetitive region. The 16aa repeat is encoded by the highly polymorphic 48-bp repeat, and 1-6 copies of the 16aa repeat have been identified in normal individuals. The current reference sequence shown here has a single copy of the 16aa repeat. This protein and the RP1 protein, another retinal-specific protein, play essential and synergistic roles in affecting photosensitivity and outer segment morphogenesis of rod photoreceptors. Mutations in this gene cause occult macular dystrophy (OMD). [provided by RefSeq, Sep 2010]

Primary Disease Associations & Inheritance

Occult macular dystrophyMIM #613587
AD
Retinitis pigmentosa 88MIM #618826
AR
0
Active trials
9
Pathogenic / LP
296
ClinVar variants
14
Pubs (1 yr)
-10.1
Missense Z
1.95
LOEUF
Clinical SummaryRP1L1
Population Constraint (gnomAD)
Low constraint (pLI 0.00) — loss-of-function variants are relatively tolerated in the population.
📋
ClinVar Variants
9 Pathogenic / Likely Pathogenic· 254 VUS of 296 total submissions

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Tolerant — LoF & missense variants common in population
LoF Constraint
1.95LOEUF
pLI 0.000
Z-score -1.76
OE 1.72 (1.031.95)
Tolerant

Highly tolerant — LoF variants common in population

Missense Constraint
-10.07Z-score
OE missense 1.77 (1.711.83)
2394 obs / 1351.9 exp
Tolerant

Tolerant to missense variation

Observed / Expected Ratios
LoF OE1.72 (1.031.95)
00.351.4
Missense OE1.77 (1.711.83)
00.61.4
Synonymous OE1.51
01.21.6
LoF obs/exp: 12 / 7.0Missense obs/exp: 2394 / 1351.9Syn Z: -9.81

ClinVar Variant Classifications

296 submitted variants in ClinVar

Classification Summary

Pathogenic4
Likely Pathogenic5
VUS254
Likely Benign31
Benign1
Conflicting1
4
Pathogenic
5
Likely Pathogenic
254
VUS
31
Likely Benign
1
Benign
1
Conflicting

Curated Variants Distribution

Classified variants from ClinVar · 5 ACMG categories

ClassificationLoFMissense + InframeNon-codingSynonymousTotal
Pathogenic
0
0
4
0
4
Likely Pathogenic
1
0
4
0
5
VUS
1
247
6
0
254
Likely Benign
0
14
1
16
31
Benign
0
0
1
0
1
Conflicting
1
Total22611616296

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

View in ClinVar →

RP1L1 · protein map & ClinVar variants

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

Gene2Phenotype Curations

RP1L1-related retinitis pigmentosa

definitive
ARLoss Of FunctionAbsent Gene Product
Eye
G2P ↗

RP1L1-related occult macular dystrophy

definitive
ADUndeterminedAltered Gene Product Structure
Eye
G2P ↗
missense variantinframe deletioninframe insertion

RP1L1-related cone dystrophy

limited
ARUndeterminedAltered Gene Product Structure
Eye
G2P ↗
missense variantinframe deletioninframe insertion

Gene2Phenotype curations · DECIPHER consortium patient cohort (public variants) · deciphergenomics.org

Clinical Trials

Active and recruiting trials from ClinicalTrials.gov

No active trials found for this gene.

Search ClinicalTrials.gov →
Clinical Literature
Landmark / reviewRecent case evidence
Full-Text Mentions
NLP-detected gene mentions in article bodies · via PubTator3
PubTator3
Top 5 full-text resultsSearch PubTator3 ↗