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GeneE
10 sources retrieved Β· Most recent: April 2026 Β· Index updated 15 days ago
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CDHR1
cadherin related family member 1
Chromosome 10 Β· 10q23.1
NCBI Gene: 92211Ensembl: ENSG00000148600.16HGNC: HGNC:14550UniProt: F1T0L2
30PubMed Papers
21Diseases
0Drugs
100Pathogenic Variants
FUNCTIONAL ROLE
Receptor
RESEARCH IMPACT
Variant-Rich
CLINICAL
OMIM Disease Gene
DATA QUALITY
βœ“ Experimental GO Evidenceβœ“ Swiss-Prot Reviewed
photoreceptor cell maintenancephotoreceptor cell morphogenesisphotoreceptor cell outer segment organizationcell adhesioncone-rod dystrophy 15retinitis pigmentosaCone rod dystrophyretinitis pigmentosa 65
✦AI Summary

CDHR1 (cadherin related family member 1) is a specialized protocadherin essential for photoreceptor structural integrity and function. It is highly expressed in cone and rod photoreceptors where it maintains the structural organization of outer segments 1. CDHR1 functions as a calcium-dependent cell-adhesion protein critical for photoreceptor cell morphogenesis and outer segment organization. Biallelic CDHR1 variants cause autosomal recessive retinal dystrophy presenting with marked clinical heterogeneity 2. The disease manifests as cone-rod dystrophy, rod-cone dystrophy, or maculopathy, typically with symptom onset between the fourth and sixth decades of life 3. Pathologically, mutations result in shortened photoreceptor outer segments and progressive photoreceptor cell death 1. Electrophysiologically, both rod and cone function are severely compromised, with electroretinography showing reduced scotopic and photopic responses 34. CDHR1 is among the most frequent causes of inherited macular dystrophy, accounting for 6.1% of genetically explained cases 5. Notably, some CDHR1 variants demonstrate incomplete penetrance, complicating genetic diagnosis 5. Gene therapy targeting CDHR1 has demonstrated durable morphological and functional rescue in murine models, sustaining improvement for 23 months and warranting clinical trial development 1.

Sources cited
1
CDHR1 is a specialized protocadherin highly expressed in cone and rod photoreceptors; biallelic variants cause shortened photoreceptor outer segments and progressive photoreceptor death; gene therapy achieves sustained rescue
PMID: 38504520
2
CDHR1-related retinal dystrophy presents with clinical heterogeneity including rod-cone dystrophy, cone-rod dystrophy, and maculopathy phenotypes
PMID: 35627310
3
CDHR1 mutations cause autosomal recessive retinopathy with symptom onset in fourth-sixth decades; characterized by macular atrophy, decreased cone-rod ERG responses, and nyctalopia
PMID: 28885867
4
CDHR1 is the third most frequent cause of inherited macular dystrophy (6.1%); some CDHR1 variants exhibit incomplete penetrance
PMID: 38540785
5
Cdhr1-/- mice show outer retinal thinning, shortened outer segments, and severe functional deficits in rod and cone photoreceptors, validating the model for therapeutic evaluation
PMID: 33964272
6
CDHR1 mutations identified in patients with macular and cone/cone-rod dystrophies as part of broad genetic heterogeneity
PMID: 29555955
Disease Associationsβ“˜21
cone-rod dystrophy 15Open Targets
0.71Strong
retinitis pigmentosaOpen Targets
0.66Moderate
Cone rod dystrophyOpen Targets
0.66Moderate
retinitis pigmentosa 65Open Targets
0.63Moderate
Retinal dystrophyOpen Targets
0.56Moderate
cone-rod dystrophyOpen Targets
0.55Moderate
macular dystrophy, retinal, 5Open Targets
0.47Moderate
autosomal recessive cone rod dystrophyOpen Targets
0.42Moderate
Rod-cone dystrophyOpen Targets
0.37Weak
eye diseaseOpen Targets
0.37Weak
cone-rod dystrophy 12Open Targets
0.34Weak
Leber congenital amaurosisOpen Targets
0.34Weak
placental retentionOpen Targets
0.32Weak
rib fractureOpen Targets
0.31Weak
myopiaOpen Targets
0.30Weak
optic atrophyOpen Targets
0.29Weak
PhlebitisOpen Targets
0.27Weak
ThrombophlebitisOpen Targets
0.27Weak
HypermetropiaOpen Targets
0.26Weak
genetic disorderOpen Targets
0.19Weak
Cone-rod dystrophy 15UniProt
Pathogenic Variants100
NM_033100.4(CDHR1):c.193_194dup (p.Asp65fs)Pathogenic
not provided
β˜…β˜…β˜†β˜†2025β†’ Residue 65
NM_033100.4(CDHR1):c.1744A>T (p.Lys582Ter)Pathogenic
not provided|Cone-rod dystrophy
β˜…β˜…β˜†β˜†2025β†’ Residue 582
NM_033100.4(CDHR1):c.1463del (p.Gly488fs)Pathogenic
Retinitis pigmentosa 65|Retinal dystrophy|not provided|Cone-rod dystrophy 15
β˜…β˜…β˜†β˜†2025β†’ Residue 488
NM_033100.4(CDHR1):c.954_958del (p.His319fs)Pathogenic
Retinal dystrophy|not provided
β˜…β˜…β˜†β˜†2025β†’ Residue 319
NM_033100.4(CDHR1):c.2522_2528del (p.Ile841fs)Pathogenic
not provided|Retinitis pigmentosa|Cone dystrophy|Retinal dystrophy|Cone-rod dystrophy 15|Macular dystrophy, retinal, 5|Retinitis pigmentosa 65
β˜…β˜…β˜†β˜†2025β†’ Residue 841
NM_033100.4(CDHR1):c.55+1G>ALikely pathogenic
Retinal dystrophy|not provided|CDHR1-related disorder
β˜…β˜…β˜†β˜†2025
NM_033100.4(CDHR1):c.863-1G>APathogenic
not provided|Cone-rod dystrophy 15
β˜…β˜…β˜†β˜†2025
NM_033100.4(CDHR1):c.1503_1507del (p.Gly502fs)Pathogenic
not provided|Cone-rod dystrophy 15|Retinal dystrophy
β˜…β˜…β˜†β˜†2025β†’ Residue 502
NM_033100.4(CDHR1):c.525+1G>APathogenic
not provided|Cone-rod dystrophy 15
β˜…β˜…β˜†β˜†2025
NM_033100.4(CDHR1):c.1485+2T>CPathogenic
not provided|Retinal dystrophy
β˜…β˜…β˜†β˜†2025
NM_033100.4(CDHR1):c.1342dup (p.Thr448fs)Pathogenic
not provided|CDHR1-related disorder
β˜…β˜…β˜†β˜†2025β†’ Residue 448
NM_033100.4(CDHR1):c.295del (p.Glu99fs)Pathogenic
not provided|Retinitis pigmentosa
β˜…β˜…β˜†β˜†2025β†’ Residue 99
NM_033100.4(CDHR1):c.713del (p.Asp238fs)Pathogenic
not provided|Cone-rod dystrophy 15
β˜…β˜…β˜†β˜†2025β†’ Residue 238
NM_033100.4(CDHR1):c.338del (p.Gly113fs)Pathogenic
Retinitis pigmentosa 65|Retinal dystrophy|not provided
β˜…β˜…β˜†β˜†2024β†’ Residue 113
NM_033100.4(CDHR1):c.1381C>T (p.Gln461Ter)Pathogenic
not provided|Cone-rod dystrophy
β˜…β˜…β˜†β˜†2024β†’ Residue 461
NM_033100.4(CDHR1):c.56-1G>APathogenic
not provided|Cone-rod dystrophy 15
β˜…β˜…β˜†β˜†2024
NM_033100.4(CDHR1):c.2087_2090del (p.Asp696fs)Pathogenic
not provided|Cone-rod dystrophy 15
β˜…β˜…β˜†β˜†2024β†’ Residue 696
NM_033100.4(CDHR1):c.524dup (p.Asn176fs)Pathogenic
Cone-rod dystrophy 15|Leber congenital amaurosis|not provided|Retinitis pigmentosa
β˜…β˜…β˜†β˜†2024β†’ Residue 176
NM_033100.4(CDHR1):c.768C>G (p.Tyr256Ter)Pathogenic
not provided
β˜…β˜…β˜†β˜†2024β†’ Residue 256
NM_033100.4(CDHR1):c.616del (p.His206fs)Pathogenic
Cone-rod dystrophy 15|not provided
β˜…β˜…β˜†β˜†2023β†’ Residue 206
View on ClinVar β†—
Related Genes
TM4SF5Protein interaction78%PROM1Protein interaction76%ABCA4Protein interaction75%GUCY2DProtein interaction75%AIPL1Protein interaction75%RPGRIP1Protein interaction75%
Tissue Expression6 tissues
Bone Marrow
100%
Brain
85%
Heart
25%
Ovary
15%
Liver
14%
Lung
7%
Gene Interaction Network
Click a node to explore
CDHR1TM4SF5PROM1ABCA4GUCY2DAIPL1RPGRIP1
PROTEIN STRUCTURE
Preparing viewer…
AlphaFoldAI-predicted Β· UniProt Q96JP9
View on AlphaFold β†—
Constraintβ“˜
LOEUFβ“˜
1.00LoF Tolerant
pLIβ“˜
0.00Tolerant
Observed/Expected LoF0.81 [0.66–1.00]
RankingsWhere CDHR1 stands among ~20K protein-coding genes
  • #11,875of 20,598
    Most Researched30
  • #780of 5,498
    Most Pathogenic Variants100 Β· top quartile
  • #9,734of 17,882
    Most Constrained (LOEUF)1.00
Genes detectedCDHR1
Sources retrieved10 papers
Response timeβ€”
πŸ“„ Sources
10β–Ό
1
Clinical and genetic characteristics of 251 consecutive patients with macular and cone/cone-rod dystrophy.
PMID: 29555955
Sci Rep Β· 2018
1.00
2
Towards Uncovering the Role of Incomplete Penetrance in Maculopathies through Sequencing of 105 Disease-Associated Genes.
PMID: 38540785
Biomolecules Β· 2024
0.90
3
Rescue of cone and rod photoreceptor function in a CDHR1-model of age-related retinal degeneration.
PMID: 38504520
Mol Ther Β· 2024
0.80
4
Clinical characteristics of recessive retinal degeneration due to mutations in the CDHR1 gene and a review of the literature.
PMID: 28885867
Ophthalmic Genet Β· 2018
0.70
5
Clinical Phenotypes of
PMID: 35627310
Genes (Basel) Β· 2022
0.60