RPGRIP1L

Chr 16AR

RPGRIP1 like

Also known as: COACH3, CORS3, FTM, JBTS7, MKS5, NPHP8, PPP1R134

The protein localizes to basal bodies, centrosomes, and primary cilia where it regulates signaling pathways and is involved in establishing planar cell polarity and organizing apical junctions. Mutations cause Joubert syndrome 7, Meckel syndrome 5, and COACH syndrome 3, which are ciliopathies affecting the central nervous system, kidneys, liver, and other organ systems with onset typically in the neonatal period. The gene follows autosomal recessive inheritance.

OMIMResearchSummary from RefSeq, OMIM, UniProt
LOFmechanismARLOEUF 0.963 OMIM phenotypes
Clinical SummaryRPGRIP1L
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Gene-Disease Validity (ClinGen)
ciliopathy · ARDefinitive

Definitive — sufficient evidence for diagnostic panels

Population Constraint (gnomAD)
Low constraint (pLI 0.00) — loss-of-function variants are relatively tolerated in the population.

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Tolerant — LoF & missense variants common in population
LoF Constraint
0.96LOEUF
pLI 0.000
Z-score 1.86
OE 0.77 (0.620.96)
Tolerant

Typical tolerance to LoF variation

Missense Constraint
-0.11Z-score
OE missense 1.01 (0.951.08)
696 obs / 688.0 exp
Tolerant

Tolerant to missense variation

Observed / Expected Ratios
LoF OE0.77 (0.620.96)
00.351.4
Missense OE1.01 (0.951.08)
00.61.4
Synonymous OE1.00
01.21.6
LoF obs/exp: 57 / 74.3Missense obs/exp: 696 / 688.0Syn Z: -0.00

ClinVar Variant Classifications

0 submitted variants in ClinVar

Protein Context — Lollipop Plot

RPGRIP1L · 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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Key Publications
Landmark & review papers · by relevance
PubMed
Top 5 results · since 2015Search PubMed ↗