RDH5

UniProt ID: Q92781
Organism: Homo sapiens
Review Status: INITIALIZED
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Gene Description

RDH5 (retinol dehydrogenase 5), also known as 11-cis retinol dehydrogenase, is a microsomal NAD(H)-dependent short-chain dehydrogenase/reductase (SDR family, SDR9C5) anchored in the endoplasmic reticulum membrane as a multi-pass membrane protein with a lumenal-facing catalytic domain, and it functions as a homodimer. It oxidizes cis-isomers of retinol (11-cis-, 9-cis-, and 13-cis-retinol) but has no activity toward all-trans retinoids. Its principal physiological role is in the visual (retinoid) cycle of the retinal pigment epithelium, where it catalyzes the step following the isomerase RPE65: the oxidation of 11-cis-retinol to 11-cis-retinal, the chromophore that recombines with opsins to regenerate rod and cone visual pigments. Beyond the eye RDH5 is widely expressed and also displays 3-alpha-hydroxysteroid dehydrogenase activity, oxidizing androsterone and 5-alpha-androstane-3-alpha,17-beta-diol, contributing to androgen (dihydrotestosterone) metabolism. Loss-of-function variants in RDH5 cause fundus albipunctatus, a form of congenital stationary night blindness with delayed dark adaptation and white retinal dots.

Existing Annotations Review

GO Term Evidence Action Reason
GO:0005789 endoplasmic reticulum membrane
IBA
GO_REF:0000033
ACCEPT
Summary: Phylogenetic (PANTHER) inference that RDH5 is active in the ER membrane. This agrees with experimental data (IDA, PMID:11675386) and UniProt subcellular location. Correct and part of the core localization.
Supporting Evidence:
file:human/RDH5/RDH5-uniprot.txt
SUBCELLULAR LOCATION: Endoplasmic reticulum membrane
GO:0005788 endoplasmic reticulum lumen
IBA
GO_REF:0000033
ACCEPT
Summary: Phylogenetic inference that the active site faces the ER lumen. Consistent with UniProt (multi-pass membrane protein with lumenal-side catalytic domain) and with the IDA annotation from PMID:11675386. Correct.
Supporting Evidence:
file:human/RDH5/RDH5-uniprot.txt
Lumenal side
GO:0001523 retinoid metabolic process
IBA
GO_REF:0000033
ACCEPT
Summary: Phylogenetic inference of a retinoid metabolic role, correct for RDH5, which oxidizes cis-retinols within the retinoid (visual) cycle. Accurate but relatively general; the more specific retinol metabolic process and visual perception capture the core role.
Supporting Evidence:
file:human/RDH5/RDH5-uniprot.txt
Catalyzes the oxidation of cis-isomers of retinol, including
GO:0004745 all-trans-retinol dehydrogenase (NAD+) activity
IBA
GO_REF:0000033
MODIFY
Summary: Family-level (IBA) assignment of all-trans-retinol dehydrogenase activity. This is the wrong substrate for RDH5: UniProt states the enzyme has no activity toward all-trans retinal and acts specifically on cis-isomers of retinol. The physiologically relevant and biochemically demonstrated activity is 11-cis-retinol dehydrogenase (GO:0106429). Modify to the cis-specific term.
Supporting Evidence:
file:human/RDH5/RDH5-uniprot.txt
no activity towards all-trans retinal (By similarity). Plays a
GO:0004022 alcohol dehydrogenase (NAD+) activity
IEA
GO_REF:0000116
MARK AS OVER ANNOTATED
Summary: Rhea-mapped electronic annotation to the broad parent alcohol dehydrogenase (NAD+) activity. Not incorrect (the cis-retinol reactions are formally alcohol oxidations) but uninformative compared with the specific 11-cis-retinol dehydrogenase term. Over-annotation.
Supporting Evidence:
file:human/RDH5/RDH5-uniprot.txt
11-cis-, 9-cis-, and 13-cis-retinol in an NAD-dependent manner
GO:0005789 endoplasmic reticulum membrane
IEA
GO_REF:0000044
ACCEPT
Summary: Electronic annotation from the UniProt subcellular-location keyword. Correct and redundant with the IDA/IBA ER membrane annotations. Core localization.
Supporting Evidence:
file:human/RDH5/RDH5-uniprot.txt
SUBCELLULAR LOCATION: Endoplasmic reticulum membrane
GO:0016616 oxidoreductase activity, acting on the CH-OH group of donors, NAD or NADP as acceptor
IEA
GO_REF:0000117
MARK AS OVER ANNOTATED
Summary: ARBA machine-learning annotation to a very general oxidoreductase parent. True but uninformative relative to the specific 11-cis-retinol dehydrogenase activity. Over-annotation.
Supporting Evidence:
file:human/RDH5/RDH5-uniprot.txt
11-cis-, 9-cis-, and 13-cis-retinol in an NAD-dependent manner
GO:0047023 androsterone dehydrogenase [NAD(P)+] activity
IEA
GO_REF:0000120
KEEP AS NON CORE
Summary: Electronic (EC/Rhea) annotation of androsterone dehydrogenase activity. This is experimentally supported (see EXP annotation from PMID:29541409 and IDA from PMID:9931293) but represents an extra-ocular steroid-metabolizing (3-alpha-HSD) activity, not the core visual-cycle function.
Supporting Evidence:
file:human/RDH5/RDH5-uniprot.txt
Also recognizes steroids (androsterone,
GO:0047044 androstan-3-alpha,17-beta-diol dehydrogenase (NAD+) activity
IEA
GO_REF:0000120
KEEP AS NON CORE
Summary: Electronic (EC/Rhea) annotation of 5-alpha-androstane-3-alpha,17-beta-diol dehydrogenase activity, supported experimentally by the IDA from PMID:9931293. A genuine secondary 3-alpha-HSD activity, extra-ocular and non-core.
Supporting Evidence:
file:human/RDH5/RDH5-uniprot.txt
androstanediol) as its substrates (PubMed:29541409, PubMed:9931293).
GO:0106429 11-cis-retinol dehydrogenase (NAD+) activity
IEA
GO_REF:0000003
ACCEPT
Summary: EC-based electronic annotation of 11-cis-retinol dehydrogenase (NAD+) activity (EC 1.1.1.315). This is the correct, specific molecular function of RDH5 and its core evolved activity, supported by direct biochemistry (PubMed:10588954, 11675386, 9931293).
Supporting Evidence:
file:human/RDH5/RDH5-uniprot.txt
Reaction=11-cis-retinol + NAD(+) = 11-cis-retinal + NADH + H(+);
GO:0001523 retinoid metabolic process
IEA
GO_REF:0000107
ACCEPT
Summary: Ensembl orthology projection (from mouse Rdh5) of a retinoid metabolic role. Correct and consistent with the IBA and IDA retinoid-metabolic-process annotations.
Supporting Evidence:
file:human/RDH5/RDH5-uniprot.txt
Catalyzes the oxidation of cis-isomers of retinol, including
GO:0004745 all-trans-retinol dehydrogenase (NAD+) activity
IEA
GO_REF:0000107
MODIFY
Summary: Ensembl orthology projection of all-trans-retinol dehydrogenase activity from the mouse ortholog. As with the IBA version, this is the wrong substrate: RDH5 acts on cis-retinols and has no all-trans activity. Modify to the cis-specific term.
Supporting Evidence:
file:human/RDH5/RDH5-uniprot.txt
no activity towards all-trans retinal (By similarity). Plays a
GO:0005788 endoplasmic reticulum lumen
IEA
GO_REF:0000107
ACCEPT
Summary: Ensembl orthology projection of ER lumen localization. Consistent with the IDA annotation (PMID:11675386) and UniProt (lumenal-side active site). Correct.
Supporting Evidence:
file:human/RDH5/RDH5-uniprot.txt
Lumenal side
GO:0044297 cell body
IEA
GO_REF:0000107
REMOVE
Summary: Ensembl orthology projection of "cell body" localization from a rat ortholog (UniProtKB:A0A0G2K1R4 / ENSRNOP00000071951). There is no experimental support for RDH5 residing in a neuronal cell body; RDH5 is an ER-membrane enzyme. This is a spurious cross-species electronic projection and should be removed.
Supporting Evidence:
file:human/RDH5/RDH5-uniprot.txt
SUBCELLULAR LOCATION: Endoplasmic reticulum membrane
GO:0001523 retinoid metabolic process
TAS
Reactome:R-HSA-2453902
ACCEPT
Summary: Reactome (canonical retinoid cycle in rods) traceable assignment of a retinoid metabolic role. Correct: RDH5 oxidizes 11-cis-retinol to 11-cis-retinal within the retinoid/visual cycle in the RPE.
Supporting Evidence:
Reactome:R-HSA-2454081
RDH5, can (reversibly) catalyse the oxidation of 11-cis-retinol (11cROL) to 11-cis-retinal
GO:0042572 retinol metabolic process
IEA
GO_REF:0000041
ACCEPT
Summary: UniPathway-based electronic annotation of retinol metabolic process. Correct and the most substrate-precise process term for RDH5 (its substrate is a retinol). Core process.
Supporting Evidence:
file:human/RDH5/RDH5-uniprot.txt
PATHWAY: Cofactor metabolism; retinol metabolism.
GO:0004745 all-trans-retinol dehydrogenase (NAD+) activity
TAS
Reactome:R-HSA-2454081
MODIFY
Summary: Reactome maps RDH5 catalysis to the generic all-trans-retinol dehydrogenase GO term, but the Reactome reaction it annotates ("RDH5 oxidises 11cROL to 11cRAL") is explicitly the 11-cis reaction, and UniProt shows RDH5 has no all-trans activity. Modify to the cis-specific term.
Supporting Evidence:
Reactome:R-HSA-2454081
RDH5, can (reversibly) catalyse the oxidation of 11-cis-retinol (11cROL) to 11-cis-retinal
GO:0004745 all-trans-retinol dehydrogenase (NAD+) activity
TAS
Reactome:R-HSA-2466832
MODIFY
Summary: Reactome reaction ("Defective RDH5 does not oxidise 11cROL to 11cRAL") again describes the 11-cis reaction but is mapped to the generic all-trans GO term. Same wrong-substrate issue; modify to the 11-cis-specific molecular function.
Supporting Evidence:
file:human/RDH5/RDH5-uniprot.txt
Reaction=11-cis-retinol + NAD(+) = 11-cis-retinal + NADH + H(+);
GO:0004745 all-trans-retinol dehydrogenase (NAD+) activity
TAS
Reactome:R-HSA-5362721
MODIFY
Summary: Reactome reaction ("RDH5,RDH11 oxidise 11cROL to 11cRAL") is the 11-cis oxidation mapped to the generic all-trans GO term. Modify to the substrate-correct 11-cis term.
Supporting Evidence:
file:human/RDH5/RDH5-uniprot.txt
Reaction=11-cis-retinol + NAD(+) = 11-cis-retinal + NADH + H(+);
GO:0047023 androsterone dehydrogenase [NAD(P)+] activity
EXP
PMID:29541409
Inhibition of dihydrotestosterone synthesis in prostate canc...
KEEP AS NON CORE
Summary: Experimental annotation: RDH5 is one of four 3-alpha-oxidoreductases shown to convert androsterone (AND) to 5-alpha-dione, with activity abolished by the catalytic Y175F/K179R double mutant. This androsterone dehydrogenase activity is genuine but extra-ocular (prostate androgen "backdoor" metabolism), not the core visual-cycle function. Keep as non-core.
Supporting Evidence:
PMID:29541409
metabolism of 5Ξ±-androstan-3Ξ±-ol-17-one (androsterone; AND) to 5Ξ±-dione
GO:0005789 endoplasmic reticulum membrane
TAS
Reactome:R-HSA-2454081
ACCEPT
Summary: Reactome localization of the RDH5 reaction to the ER membrane. Correct and consistent with IDA (PMID:11675386) and UniProt. Core localization.
Supporting Evidence:
file:human/RDH5/RDH5-uniprot.txt
SUBCELLULAR LOCATION: Endoplasmic reticulum membrane
GO:0005789 endoplasmic reticulum membrane
TAS
Reactome:R-HSA-5362721
ACCEPT
Summary: Reactome localization (RDH5,RDH11 reaction) to the ER membrane. Correct; redundant with other ER membrane annotations. Core localization.
Supporting Evidence:
file:human/RDH5/RDH5-uniprot.txt
SUBCELLULAR LOCATION: Endoplasmic reticulum membrane
GO:0001523 retinoid metabolic process
IDA
PMID:9931293
Activity of human 11-cis-retinol dehydrogenase (Rdh5) with s...
ACCEPT
Summary: Direct-assay annotation of a retinoid metabolic role. PMID:9931293 characterized RDH5 catalysis of 9-cis- and 11-cis-retinol oxidation. Correct; the retinoid/retinol metabolic process is a core process for RDH5.
Supporting Evidence:
PMID:9931293
Rdh5 catalyses 9-cis-retinol metabolism equally efficiently as 11-cis-retinol metabolism
GO:0004745 all-trans-retinol dehydrogenase (NAD+) activity
IDA
PMID:11675386
Biochemical defects in 11-cis-retinol dehydrogenase mutants ...
MODIFY
Summary: Experimental (IDA) annotation labeled as all-trans-retinol dehydrogenase activity, but PMID:11675386 assayed 11-cis-retinol dehydrogenase activity of RDH5 and its fundus-albipunctatus mutants (the paper is titled "Biochemical defects in 11-cis-retinol dehydrogenase mutants"), and UniProt states RDH5 has no all-trans activity. The measured enzyme is real but the substrate label is wrong; modify to the 11-cis-specific term rather than removing an experimental annotation.
Supporting Evidence:
PMID:11675386
Biochemical defects in 11-cis-retinol dehydrogenase mutants associated with
file:human/RDH5/RDH5-uniprot.txt
no activity towards all-trans retinal (By similarity). Plays a
GO:0005788 endoplasmic reticulum lumen
IDA
PMID:11675386
Biochemical defects in 11-cis-retinol dehydrogenase mutants ...
ACCEPT
Summary: Direct experimental evidence that the RDH5 catalytic domain resides on the lumenal side of the ER. Correct core localization (multi-pass ER membrane protein with lumenal active site).
Supporting Evidence:
file:human/RDH5/RDH5-uniprot.txt
Lumenal side
GO:0005789 endoplasmic reticulum membrane
IDA
PMID:11675386
Biochemical defects in 11-cis-retinol dehydrogenase mutants ...
ACCEPT
Summary: Direct experimental evidence for ER membrane localization of RDH5. This is the anchoring IDA that the IBA/IEA/TAS ER-membrane annotations reinforce. Core localization.
Supporting Evidence:
file:human/RDH5/RDH5-uniprot.txt
SUBCELLULAR LOCATION: Endoplasmic reticulum membrane
GO:0008202 steroid metabolic process
IDA
PMID:9931293
Activity of human 11-cis-retinol dehydrogenase (Rdh5) with s...
KEEP AS NON CORE
Summary: Direct evidence that RDH5 metabolizes steroids (3-alpha-hydroxysteroid dehydrogenase activity on androsterone and 5-alpha-androstane-3-alpha,17-beta-diol). A genuine but extra-ocular activity; steroid metabolism is a secondary, non-core process for RDH5.
Supporting Evidence:
PMID:9931293
recognizes 5alpha-androstan-3alpha,17beta-diol and androsterone as substrates
GO:0042803 protein homodimerization activity
IDA
PMID:11675386
Biochemical defects in 11-cis-retinol dehydrogenase mutants ...
KEEP AS NON CORE
Summary: Cross-linking and molecular-modeling evidence that RDH5 is a homodimer. Correct; supports quaternary structure. Not the core catalytic function but a real, direct biochemical property.
Supporting Evidence:
PMID:11675386
molecular modeling showed that RDH5 is
GO:0047023 androsterone dehydrogenase [NAD(P)+] activity
IDA
PMID:9931293
Activity of human 11-cis-retinol dehydrogenase (Rdh5) with s...
KEEP AS NON CORE
Summary: Direct-assay androsterone dehydrogenase activity (3-alpha-HSD). Genuine secondary activity, extra-ocular, non-core. Redundant with the EXP annotation from PMID:29541409.
Supporting Evidence:
PMID:9931293
recognizes 5alpha-androstan-3alpha,17beta-diol and androsterone as substrates
GO:0047044 androstan-3-alpha,17-beta-diol dehydrogenase (NAD+) activity
IDA
PMID:9931293
Activity of human 11-cis-retinol dehydrogenase (Rdh5) with s...
KEEP AS NON CORE
Summary: Direct-assay 5-alpha-androstane-3-alpha,17-beta-diol dehydrogenase activity. Genuine secondary 3-alpha-HSD activity, extra-ocular and non-core.
Supporting Evidence:
PMID:9931293
recognizes 5alpha-androstan-3alpha,17beta-diol and androsterone as substrates
GO:0005789 endoplasmic reticulum membrane
TAS
Reactome:R-HSA-2466832
ACCEPT
Summary: Reactome localization (defective-RDH5 reaction) to the ER membrane. Correct; redundant with other ER membrane annotations. Core localization.
Supporting Evidence:
file:human/RDH5/RDH5-uniprot.txt
SUBCELLULAR LOCATION: Endoplasmic reticulum membrane
GO:0004745 all-trans-retinol dehydrogenase (NAD+) activity
TAS
PMID:9115228
Identification and characterization of a stereospecific huma...
MODIFY
Summary: Older traceable assignment (PINC) mapping RDH5 to all-trans-retinol dehydrogenase activity. PMID:9115228 in fact showed RDH5 oxidizes 9-cis- (and 13-cis-) retinol but "does not catalyze all-trans-retinol oxidation". The all-trans label is contradicted by the very reference cited; modify to the substrate-correct cis-retinol dehydrogenase term.
Supporting Evidence:
PMID:9115228
does not catalyze all-trans-retinol oxidation
GO:0007601 visual perception
TAS
PMID:10369264
Mutations in the gene encoding 11-cis retinol dehydrogenase ...
ACCEPT
Summary: Traceable assignment of a role in visual perception. RDH5 supplies 11-cis-retinal for regeneration of rod and cone visual pigments; loss of function causes fundus albipunctatus with delayed dark adaptation. This is the core physiological process.
Supporting Evidence:
PMID:10369264
this retinoid is the chromophore residing in rhodopsin and the cone opsins

Core Functions

NAD(H)-dependent oxidation of 11-cis-retinol to 11-cis-retinal in the endoplasmic reticulum of the retinal pigment epithelium, providing the 11-cis-retinal chromophore for regeneration of rod and cone visual pigments (the step of the visual/retinoid cycle following the isomerase RPE65).

Supporting Evidence:
  • file:human/RDH5/RDH5-uniprot.txt
    Plays a
  • file:human/RDH5/RDH5-uniprot.txt
    significant role in 11-cis retinol oxidation in the retinal pigment
  • PMID:10369264
    this retinoid is the chromophore residing in rhodopsin and the cone opsins

NAD(H)-dependent oxidation of 9-cis-retinol (and, less efficiently, 13-cis-retinol) to the corresponding retinaldehydes, a cis-retinol dehydrogenase activity contributing to retinoid metabolism outside the eye.

Supporting Evidence:
  • PMID:9115228
    avidly catalyzes oxidation of 9-cis-retinol to 9-cis-retinaldehyde
  • PMID:9931293
    Rdh5 catalyses 9-cis-retinol metabolism equally efficiently as 11-cis-retinol metabolism

Secondary NAD(H)-dependent 3-alpha-hydroxysteroid dehydrogenase activity oxidizing androsterone and 5-alpha-androstane-3-alpha,17-beta-diol, contributing to extra-ocular androgen (steroid) metabolism.

Supporting Evidence:
  • PMID:9931293
    recognizes 5alpha-androstan-3alpha,17beta-diol and androsterone as substrates
  • PMID:29541409
    metabolism of 5Ξ±-androstan-3Ξ±-ol-17-one (androsterone; AND) to 5Ξ±-dione

References

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Notes

(RDH5-notes.md)

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