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GeneE
25 sources retrieved Β· Most recent: April 2026 Β· Index updated 14 days ago
β“˜GeneE is for informational purposes only. It is not a substitute for professional medical advice, diagnosis, or treatment.
RPGR
retinitis pigmentosa GTPase regulator
Chromosome X Β· Xp11.4
NCBI Gene: 6103Ensembl: ENSG00000156313.15HGNC: HGNC:10295UniProt: A0A2R8YFT6
183PubMed Papers
24Diseases
0Drugs
794Pathogenic Variants
FUNCTIONAL ROLE
Highly ConstrainedHub Gene
RESEARCH IMPACT
TrendingVariant-Rich
CLINICAL
OMIM Disease Gene
DATA QUALITY
βœ“ Experimental GO Evidenceβœ“ Swiss-Prot Reviewed
RNA bindingguanyl-nucleotide exchange factor activityprotein bindingvisual perceptionretinitis pigmentosaCone rod dystrophyPrimary ciliary dyskinesia - retinitis pigmentosaretinitis pigmentosa 3
✦AI Summary

RPGR (retinitis pigmentosa GTPase regulator) is an X-linked gene encoding a guanine-nucleotide exchange factor (GEF) that activates RAB8A and RAB37 small GTPases by promoting conversion of inactive RAB-GDP to active RAB-GTP 1. Through RAB8A activation, RPGR facilitates ciliary trafficking and intracellular localization within photoreceptor cilia 1. RPGR regulates cilia formation by modulating actin stress filaments and may coordinate microtubule organization and intraflagellar transport 2. These functions are critical for photoreceptor integrity and retinal homeostasis. RPGR mutations cause X-linked retinitis pigmentosa (XLRP) and cone-rod dystrophy, representing approximately 5% of inherited retinal disease cases in clinical cohorts 34. The gene demonstrates genetic heterogeneity, with diverse phenotypes arising from variants in the same gene, partly reflecting limitations in detecting mutations in the repetitive exon 15 region 45. Gene replacement therapy using AAV5-hRKp.RPGR demonstrated clinical efficacy and safety in phase 1/2 trials, with improvements in retinal sensitivity and functional vision, supporting its therapeutic potential 67. These findings establish RPGR dysfunction in progressive photoreceptor degeneration and validate gene therapy as a treatment approach for RPGR-associated retinopathies.

Sources cited
1
RPGR acts as a GEF for RAB8A and RAB37, promoting GDP-to-GTP conversion and facilitating ciliary trafficking
PMID: 20631154
2
RPGR regulates cilia formation through actin stress filaments and microtubule organization
PMID: 21933838
3
RPGR mutations account for approximately 5.01% of solved inherited retinal disease cases in a large Italian cohort
PMID: 36460718
4
Mutations in RPGR exon 15 repetitive region can be missed by NGS and require specific detection methods
PMID: 28559085
5
RPGR-associated retinopathy exhibits diverse phenotypes from variants in the same gene, reflecting genetic heterogeneity
PMID: 32860923
6
AAV5-hRKp.RPGR gene therapy demonstrated safety and efficacy improvements in retinal sensitivity and functional vision in XLRP patients
PMID: 38871269
7
AAV8-coRPGR gene therapy showed safety and visual field improvements in RPGR-associated XLRP patients
PMID: 32094925
Disease Associationsβ“˜24
retinitis pigmentosaOpen Targets
0.83Strong
Cone rod dystrophyOpen Targets
0.75Strong
Primary ciliary dyskinesia - retinitis pigmentosaOpen Targets
0.74Strong
retinitis pigmentosa 3Open Targets
0.73Strong
X-linked cone-rod dystrophy 1Open Targets
0.68Moderate
RPGR-related retinopathyOpen Targets
0.68Moderate
primary ciliary dyskinesiaOpen Targets
0.67Moderate
Retinal dystrophyOpen Targets
0.58Moderate
X-linked cone-rod dystrophyOpen Targets
0.50Moderate
cone-rod dystrophyOpen Targets
0.50Moderate
ciliopathyOpen Targets
0.47Moderate
cone dystrophyOpen Targets
0.43Moderate
Rod-cone dystrophyOpen Targets
0.38Weak
eye diseaseOpen Targets
0.37Weak
myopia 25, autosomal dominantOpen Targets
0.35Weak
retinopathyOpen Targets
0.35Weak
cone dystrophy 1, X-linkedOpen Targets
0.33Weak
Macular dystrophyOpen Targets
0.29Weak
congenital stationary night blindnessOpen Targets
0.27Weak
genetic disorderOpen Targets
0.27Weak
Cone-rod dystrophy, X-linked 1UniProt
Macular degeneration, atrophic, X-linkedUniProt
Retinitis pigmentosa 3UniProt
Retinitis pigmentosa, X-linked, and sinorespiratory infections with or without deafnessUniProt
Pathogenic Variants794
NM_001034853.2(RPGR):c.1234C>T (p.Arg412Ter)Pathogenic
Retinitis pigmentosa|Retinal dystrophy|Primary ciliary dyskinesia|not provided|RPGR-related retinopathy
β˜…β˜…β˜…β˜†2026β†’ Residue 412
NM_001034853.2(RPGR):c.1307G>A (p.Gly436Asp)Likely pathogenic
not provided|RPGR-related retinopathy
β˜…β˜…β˜…β˜†2026β†’ Residue 436
NM_001034853.2(RPGR):c.19_25delinsGGAGCCCGTGGCATGAGGGAG (p.Leu7fs)Likely pathogenic
Primary ciliary dyskinesia|RPGR-related retinopathy
β˜…β˜…β˜…β˜†2026β†’ Residue 7
NM_001034853.2(RPGR):c.1573-8A>GLikely pathogenic
not provided|Primary ciliary dyskinesia|RPGR-related retinopathy
β˜…β˜…β˜…β˜†2026
NM_001034853.2(RPGR):c.1835dup (p.Asn612fs)Pathogenic
not provided|RPGR-related retinopathy
β˜…β˜…β˜…β˜†2026β†’ Residue 612
NM_001034853.2(RPGR):c.1573-1G>APathogenic
not provided|Primary ciliary dyskinesia|RPGR-related retinopathy
β˜…β˜…β˜…β˜†2026
NM_001034853.2(RPGR):c.154+3_154+6delPathogenic
Retinal dystrophy|RPGR-related retinopathy
β˜…β˜…β˜…β˜†2026
NM_001034853.2(RPGR):c.1572+3A>TPathogenic
Retinitis pigmentosa|RPGR-related retinopathy
β˜…β˜…β˜…β˜†2026
NM_001034853.2(RPGR):c.1217dup (p.Ser407fs)Pathogenic
not provided|Retinitis pigmentosa|Primary ciliary dyskinesia|Retinal dystrophy|RPGR-related retinopathy|Retinitis pigmentosa 3
β˜…β˜…β˜…β˜†2026β†’ Residue 407
NM_001034853.2(RPGR):c.1572+1G>APathogenic
Retinitis pigmentosa|Primary ciliary dyskinesia|Nonpapillary renal cell carcinoma|RPGR-related retinopathy
β˜…β˜…β˜…β˜†2026
NM_001034853.2(RPGR):c.1872_1873del (p.Glu624fs)Pathogenic
not provided|Retinitis pigmentosa 3|Primary ciliary dyskinesia|Retinal dystrophy|RPGR-related retinopathy
β˜…β˜…β˜…β˜†2026β†’ Residue 624
NM_001034853.2(RPGR):c.1377_1378del (p.Leu460fs)Pathogenic
not provided|Retinal dystrophy|Primary ciliary dyskinesia|X-linked cone-rod dystrophy 1|RPGR-related retinopathy
β˜…β˜…β˜…β˜†2026β†’ Residue 460
NM_001034853.2(RPGR):c.1345C>T (p.Arg449Ter)Pathogenic
Retinal dystrophy|not provided|Primary ciliary dyskinesia|RPGR-related retinopathy|See cases
β˜…β˜…β˜…β˜†2026β†’ Residue 449
NM_001034853.2(RPGR):c.1573-12A>GLikely pathogenic
not provided|Retinal dystrophy|RPGR-related retinopathy
β˜…β˜…β˜…β˜†2026
NM_001034853.2(RPGR):c.154+1G>APathogenic
Retinitis pigmentosa|not provided|Retinal dystrophy|RPGR-related retinopathy
β˜…β˜…β˜…β˜†2026
NM_001034853.2(RPGR):c.1693C>T (p.Gln565Ter)Pathogenic
Primary ciliary dyskinesia|RPGR-related retinopathy
β˜…β˜…β˜…β˜†2026β†’ Residue 565
NM_001034853.2(RPGR):c.706C>T (p.Gln236Ter)Pathogenic
not provided|Primary ciliary dyskinesia|RPGR-related retinopathy
β˜…β˜…β˜…β˜†2025β†’ Residue 236
NM_001034853.2(RPGR):c.934+1G>APathogenic
not provided|RPGR-related retinopathy|Retinal dystrophy
β˜…β˜…β˜…β˜†2025
NM_001034853.2(RPGR):c.3317del (p.Lys1106fs)Pathogenic
Primary ciliary dyskinesia|RPGR-related retinopathy
β˜…β˜…β˜…β˜†2025β†’ Residue 1106
NM_001034853.2(RPGR):c.3212_3218del (p.Glu1071fs)Pathogenic
not provided|RPGR-related retinopathy
β˜…β˜…β˜…β˜†2025β†’ Residue 1071
View on ClinVar β†—
Related Genes
RSPH9Protein interaction100%WHRNProtein interaction100%NPHP1Protein interaction99%DNAI1Protein interaction97%DNAI2Protein interaction96%NPHP4Protein interaction95%
Tissue Expression6 tissues
Bone Marrow
100%
Lung
82%
Brain
29%
Ovary
27%
Heart
25%
Liver
17%
Gene Interaction Network
Click a node to explore
RPGRRSPH9WHRNNPHP1DNAI1DNAI2NPHP4
PROTEIN STRUCTURE
Preparing viewer…
PDB4JHN Β· 1.70 Γ… Β· X-ray
View on RCSB β†—
Constraintβ“˜
LOEUFβ“˜
0.30Highly Constrained
pLIβ“˜
1.00Intolerant
Observed/Expected LoF0.18 [0.11–0.30]
RankingsWhere RPGR stands among ~20K protein-coding genes
  • #2,369of 20,598
    Most Researched183 Β· top quartile
  • #56of 5,498
    Most Pathogenic Variants794 Β· top 5%
  • #1,159of 17,882
    Most Constrained (LOEUF)0.30 Β· top 10%
Genes detectedRPGR
Sources retrieved25 papers
Response timeβ€”
πŸ“„ Sources
25β–Ό
1
Genetic epidemiology of inherited retinal diseases in a large patient cohort followed at a single center in Italy.
PMID: 36460718
Sci Rep Β· 2022
1.00
2
Clinically Focused Molecular Investigation of 1000 Consecutive Families with Inherited Retinal Disease.
PMID: 28559085
Ophthalmology Β· 2017
0.90
3
Genetic characteristics of retinitis pigmentosa in 1204 Japanese patients.
PMID: 31213501
J Med Genet Β· 2019
0.80
4
Phase 1/2 AAV5-hRKp.RPGR (Botaretigene Sparoparvovec) Gene Therapy: Safety and Efficacy in RPGR-Associated X-Linked Retinitis Pigmentosa.
PMID: 38871269
Am J Ophthalmol Β· 2024
0.70
5
Long-Range PCR-Based NGS Applications to Diagnose Mendelian Retinal Diseases.
PMID: 33546218
Int J Mol Sci Β· 2021
0.60