RHPN1

Chr 8

rhophilin Rho GTPase binding protein 1

Also known as: ODF5, RHOPHILIN, RHPN

RHPN1 encodes a protein that serves as a target for Rho signaling and interacts with cytoskeletal components to regulate stress fiber assembly and focal adhesion formation. Mutations cause autosomal recessive nephronophthisis with retinal dystrophy, typically presenting in childhood with progressive kidney disease and vision loss. The gene shows very low constraint against loss-of-function variants, consistent with a recessive inheritance pattern.

Summary from RefSeq, UniProt
Research Assistant →
0
Active trials
7
Pubs (1 yr)
61
P/LP submissions
0%
P/LP missense
1.11
LOEUF
Mechanism
Clinical SummaryRHPN1
Population Constraint (gnomAD)
Low constraint (pLI 0.00) — loss-of-function variants are relatively tolerated in the population.
📋
ClinVar Variants
60 unique Pathogenic / Likely Pathogenic· 152 VUS of 257 total submissions

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Tolerant — LoF & missense variants common in population
LoF Constraint
1.11LOEUF
pLI 0.000
Z-score 1.08
OE 0.78 (0.561.11)
Tolerant

Highly tolerant — LoF variants common in population

Missense Constraint
0.41Z-score
OE missense 0.94 (0.871.03)
388 obs / 411.2 exp
Tolerant

Mild missense constraint

Observed / Expected Ratios
LoF OE0.78 (0.561.11)
00.351.4
Missense OE0.94 (0.871.03)
00.61.4
Synonymous OE1.09
01.21.6
LoF obs/exp: 22 / 28.2Missense obs/exp: 388 / 411.2Syn Z: -0.96

ClinVar Variant Classifications

257 submitted variants in ClinVar

Classification Summary

Pathogenic55
Likely Pathogenic5
VUS152
Likely Benign16
55
Pathogenic
5
Likely Pathogenic
152
VUS
16
Likely Benign

Curated Variants Distribution

Classified variants from ClinVar · 5 ACMG categories

ClassificationLoFMissense + InframeNon-codingSynonymousTotal
Pathogenic
0
0
55
0
55
Likely Pathogenic
0
0
5
0
5
VUS
0
142
10
0
152
Likely Benign
0
13
1
2
16
Benign
0
0
0
0
0
Total0155712228

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

View in ClinVar →

Protein Context — Lollipop Plot

RHPN1 · 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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