HomeAboutRankingsData Sources
Β© 2026 GeneE
🧬
GeneE
10 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.
RDH5
retinol dehydrogenase 5
Chromosome 12 Β· 12q13.2
NCBI Gene: 5959Ensembl: ENSG00000135437.11HGNC: HGNC:9940UniProt: Q92781
60PubMed Papers
21Diseases
0Drugs
58Pathogenic Variants
RESEARCH IMPACT
Variant-Rich
CLINICAL
OMIM Disease Gene
DATA QUALITY
βœ“ Experimental GO Evidenceβœ“ Swiss-Prot Reviewed
steroid metabolic processendoplasmic reticulum membraneprotein homodimerization activityandrosterone dehydrogenase [NAD(P)+] activityfundus albipunctatusRetinal dystrophycongenital stationary night blindnessmyopia
✦AI Summary

RDH5 encodes retinol dehydrogenase 5, a rate-limiting enzyme of the visual cycle that catalyzes NAD-dependent oxidation of cis-isomers of retinol, including 11-cis-, 9-cis-, and 13-cis-retinol, with particular importance in the retinal pigment epithelium 1. The enzyme also recognizes steroid substrates such as androsterone and androstanediol 2. RDH5 localizes to the endoplasmic reticulum, where its stability is regulated by interaction with the heat shock protein HSP90, which prevents autophagic degradation 3. Mutations in RDH5 cause fundus albipunctatus, a congenital stationary night blindness characterized by numerous retinal white-yellow lesions and rod system impairment 45. RDH5-associated retinopathy presents with congenital night blindness and progressive macular atrophy in adult patients, with estimated annual macular volume loss of -0.007 mmΒ³/year 6. Pathogenic variants include missense mutations affecting conserved residues, frameshift deletions, and nonsense mutations that impair protein function and localization 78. Some RDH5 mutations may also cause progressive cone dystrophy beyond rod dysfunction 5. RDH5 mutations account for a minority of macular and cone/cone-rod dystrophy cases among identified genetic causes 9.

Sources cited
1
RDH5 catalyzes oxidation of cis-retinol isomers and participates in the visual cycle with large quantities of 11-cis-retinal
PMID: 35162940
2
RDH5 recognizes steroids including androsterone and androstanediol as substrates
PMID: 29541409
3
RDH5 localizes to endoplasmic reticulum, is stabilized by HSP90, and undergoes autophagic degradation when HSP90 function is impaired
PMID: 39725039
4
RDH5 mutations cause fundus albipunctatus with congenital night blindness and rod system impairment
PMID: 25820994
5
RDH5 mutations can cause progressive cone dystrophy and macular dystrophy in addition to night blindness
PMID: 12906118
6
RDH5-associated retinopathy presents with progressive macular atrophy and annual macular volume loss rate of -0.007 mmΒ³/year
PMID: 38945349
7
RDH5 mutations include frameshift deletions and missense variants that segregate with fundus albipunctatus in autosomal recessive inheritance
PMID: 35148716
8
RDH5 compound heterozygous mutations including missense and nonsense variants cause fundus albipunctatus with characteristic retinal imaging findings
PMID: 22669287
9
RDH5 mutations account for a portion of macular and cone/cone-rod dystrophy cases among identified genetic causes
PMID: 29555955
Disease Associationsβ“˜21
fundus albipunctatusOpen Targets
0.83Strong
Retinal dystrophyOpen Targets
0.51Moderate
congenital stationary night blindnessOpen Targets
0.50Moderate
myopiaOpen Targets
0.43Moderate
retinitis punctata albescensOpen Targets
0.43Moderate
age-related macular degenerationOpen Targets
0.43Moderate
refractive errorOpen Targets
0.40Weak
HypermetropiaOpen Targets
0.39Weak
retinopathyOpen Targets
0.38Weak
COVID-19Open Targets
0.38Weak
RDH5-related retinopathyOpen Targets
0.37Weak
Abnormality of refractionOpen Targets
0.35Weak
degeneration of macula and posterior poleOpen Targets
0.34Weak
macular degenerationOpen Targets
0.34Weak
lens diseaseOpen Targets
0.28Weak
Progressive visual lossOpen Targets
0.27Weak
eye diseaseOpen Targets
0.27Weak
optic atrophyOpen Targets
0.27Weak
atrophic macular degenerationOpen Targets
0.26Weak
wet macular degenerationOpen Targets
0.26Weak
Fundus albipunctatusUniProt
Pathogenic Variants58
NM_002905.5(RDH5):c.839G>A (p.Arg280His)Pathogenic
Fundus albipunctatus, autosomal recessive|not provided|Pigmentary retinal dystrophy|Congenital stationary night blindness
β˜…β˜…β˜†β˜†2026β†’ Residue 280
NM_002905.5(RDH5):c.625C>T (p.Arg209Ter)Pathogenic
Retinal dystrophy|not provided|Pigmentary retinal dystrophy
β˜…β˜…β˜†β˜†2026β†’ Residue 209
NM_002905.5(RDH5):c.712G>T (p.Gly238Trp)Pathogenic
Fundus albipunctatus, autosomal recessive|not provided|Pigmentary retinal dystrophy|See cases|RDH5-related disorder|Congenital stationary night blindness|Retinal dystrophy|Retinitis punctata albescens
β˜…β˜…β˜†β˜†2025β†’ Residue 238
NM_002905.5(RDH5):c.71_74del (p.Leu24fs)Pathogenic
Pigmentary retinal dystrophy|not provided
β˜…β˜…β˜†β˜†2025β†’ Residue 24
NM_002905.5(RDH5):c.160C>T (p.Arg54Ter)Pathogenic
not provided|Pigmentary retinal dystrophy
β˜…β˜…β˜†β˜†2025β†’ Residue 54
NM_002905.5(RDH5):c.469C>T (p.Arg157Trp)Pathogenic
Fundus albipunctatus, autosomal recessive|not provided|Pigmentary retinal dystrophy
β˜…β˜…β˜†β˜†2025β†’ Residue 157
NM_002905.5(RDH5):c.285G>A (p.Trp95Ter)Pathogenic
Pigmentary retinal dystrophy|not provided|Fundus albipunctatus, autosomal recessive
β˜…β˜…β˜†β˜†2025β†’ Residue 95
NM_002905.5(RDH5):c.382G>A (p.Asp128Asn)Pathogenic
Pigmentary retinal dystrophy|not provided
β˜…β˜…β˜†β˜†2025β†’ Residue 128
NM_002905.5(RDH5):c.388C>T (p.Gln130Ter)Pathogenic
not provided|Pigmentary retinal dystrophy
β˜…β˜…β˜†β˜†2025β†’ Residue 130
NM_002905.5(RDH5):c.95del (p.Phe32fs)Pathogenic
not provided|Pigmentary retinal dystrophy
β˜…β˜…β˜†β˜†2025β†’ Residue 32
NM_002905.5(RDH5):c.832C>T (p.Arg278Ter)Pathogenic
Pigmentary retinal dystrophy|not provided
β˜…β˜…β˜†β˜†2025β†’ Residue 278
NM_002905.5(RDH5):c.470G>A (p.Arg157Gln)Likely pathogenic
not provided|Pigmentary retinal dystrophy
β˜…β˜…β˜†β˜†2025β†’ Residue 157
NM_002905.5(RDH5):c.103G>A (p.Gly35Ser)Pathogenic
not provided|Pigmentary retinal dystrophy
β˜…β˜…β˜†β˜†2025β†’ Residue 35
NM_002905.5(RDH5):c.928delinsGAAG (p.Leu310delinsGluVal)Pathogenic
Fundus albipunctatus, autosomal recessive|not provided|Pigmentary retinal dystrophy
β˜…β˜…β˜†β˜†2024β†’ Residue 310
NM_002905.5(RDH5):c.814_815del (p.Leu272fs)Pathogenic
not provided
β˜…β˜…β˜†β˜†2024β†’ Residue 272
NM_002905.5(RDH5):c.758T>G (p.Met253Arg)Pathogenic
Pigmentary retinal dystrophy|not provided
β˜…β˜…β˜†β˜†2024β†’ Residue 253
NM_002905.5(RDH5):c.500G>A (p.Arg167His)Pathogenic
not provided|Pigmentary retinal dystrophy
β˜…β˜…β˜†β˜†2024β†’ Residue 167
NM_002905.5(RDH5):c.536A>G (p.Lys179Arg)Pathogenic
Retinal dystrophy|not provided|Pigmentary retinal dystrophy
β˜…β˜…β˜†β˜†2024β†’ Residue 179
NM_002905.5(RDH5):c.632_633del (p.Pro211fs)Pathogenic
not provided|Retinal dystrophy
β˜…β˜…β˜†β˜†2022β†’ Residue 211
NM_002905.5(RDH5):c.796C>T (p.Arg266Ter)Pathogenic
not provided
β˜…β˜†β˜†β˜†2026β†’ Residue 266
View on ClinVar β†—
Related Genes
ALDH1A2Protein interaction98%RGRProtein interaction97%ALDH1A1Protein interaction95%ALDH1A3Protein interaction95%DGAT1Protein interaction92%AOX1Protein interaction91%
Tissue Expression6 tissues
Liver
100%
Heart
44%
Ovary
27%
Lung
22%
Bone Marrow
13%
Brain
3%
Gene Interaction Network
Click a node to explore
RDH5ALDH1A2RGRALDH1A1ALDH1A3DGAT1AOX1
PROTEIN STRUCTURE
Preparing viewer…
AlphaFoldAI-predicted Β· UniProt Q92781
View on AlphaFold β†—
Constraintβ“˜
LOEUFβ“˜
1.30LoF Tolerant
pLIβ“˜
0.00Tolerant
Observed/Expected LoF0.96 [0.72–1.30]
RankingsWhere RDH5 stands among ~20K protein-coding genes
  • #7,703of 20,598
    Most Researched60
  • #1,203of 5,498
    Most Pathogenic Variants58 Β· top quartile
  • #13,679of 17,882
    Most Constrained (LOEUF)1.30
Genes detectedRDH5
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
The Role of Vitamin A in Retinal Diseases.
PMID: 35162940
Int J Mol Sci Β· 2022
0.90
3
RDH5 and RLBP1-Associated Inherited Retinal Diseases: Refining the Spectrum of Stationary and Progressive Phenotypes.
PMID: 38945349
Am J Ophthalmol Β· 2024
0.80
4
Fundus albipunctatus: review of the literature and report of a novel RDH5 gene mutation affecting the invariant tyrosine (p.Tyr175Phe).
PMID: 25820994
J Appl Genet Β· 2015
0.70
5
RDH5 gene mutations and electroretinogram in fundus albipunctatus with or without macular dystrophy: RDH5 mutations and ERG in fundus albipunctatus.
PMID: 12906118
Doc Ophthalmol Β· 2003
0.60