HSD17B10

UniProt ID: Q99714
Organism: Homo sapiens
Review Status: INITIALIZED
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Gene Description

HSD17B10 (also known as HADH2, SCHAD, SDR5C1, MRPP2, and historically ERAB/ABAD) is a mitochondrial-matrix protein of the short-chain dehydrogenase/reductase (SDR) family that acts as a functional homotetramer and is a genuinely moonlighting, multifunctional enzyme. In its catalytic role it is an NAD+-dependent dehydrogenase whose physiologically important activity is (S)-2-methyl-3-hydroxybutyryl-CoA dehydrogenase (MHBD) in L-isoleucine catabolism, oxidizing (2S,3S)-3-hydroxy-2-methylbutanoyl-CoA to 2-methyl-3-oxobutanoyl-CoA. It has broad (S)-3-hydroxyacyl-CoA dehydrogenase activity on short/medium straight-chain acyl-CoAs and weaker intrinsic 17-beta-/3-alpha-hydroxysteroid and alcohol dehydrogenase activities toward steroid hormones (including the neurosteroid allopregnanolone/brexanolone), bile acids and other substrates. Independently of its enzymatic activity, it moonlights as MRPP2, an essential non-catalytic subunit of mitochondrial RNase P (together with the methyltransferase TRMT10C/MRPP1 and the nuclease PRORP/MRPP3), which performs 5'-end processing of mitochondrial tRNAs; the TRMT10C-HSD17B10 subcomplex additionally catalyzes N1-methylation of purine-9 (m1A9/m1G9) of mt-tRNAs and acts as a tRNA-maturation platform that supports downstream 3'-processing by ELAC2 and 3'-CCA addition by TRNT1. It localizes to the mitochondrial matrix and associates with the mitochondrial nucleoid. Loss-of-function mutations in the X-linked HSD17B10 gene cause HSD10 mitochondrial disease (a progressive infantile/childhood neurodegeneration with cardiomyopathy), and disease severity tracks with the RNase P/tRNA-processing and mitochondrial-integrity defect rather than with residual dehydrogenase activity.

Existing Annotations Review

GO Term Evidence Action Reason
GO:0004303 estradiol 17-beta-dehydrogenase [NAD(P)+] activity
IBA
GO_REF:0000033
KEEP AS NON CORE
Summary: Phylogenetically inferred 17-beta-estradiol dehydrogenase activity. This is a real but weak intrinsic in-vitro activity of HSD17B10 (its k_cat for 17-beta-oestradiol is orders of magnitude below the 3-hydroxyacyl-CoA activity), not its physiological core function. Kept as a valid but non-core side activity.
Reason: Confirmed intrinsic activity but of low catalytic efficiency and not the evolved core function; the steroid activities are minor relative to 3-hydroxyacyl-CoA/MHBD chemistry.
Supporting Evidence:
PMID:10600649
The k(cat) of the ADH activity was three orders of magnitude less than the l-3-hydroxyacyl-CoA dehydrogenase activity but was comparable with that of the enzyme's hydroxysteroid dehydrogenase (HSD) activity for oxidizing 17beta-oestradiol
GO:0005739 mitochondrion
IBA
GO_REF:0000033
ACCEPT
Summary: Phylogenetic inference of mitochondrial localization, consistent with all experimental data placing HSD17B10 in the mitochondrion (matrix/nucleoid).
Supporting Evidence:
file:human/HSD17B10/HSD17B10-uniprot.txt
SUBCELLULAR LOCATION: Mitochondrion
GO:0006631 fatty acid metabolic process
IBA
GO_REF:0000033
KEEP AS NON CORE
Summary: Broad fatty-acid metabolic process, phylogenetically inferred. HSD17B10 does act as a (S)-3-hydroxyacyl-CoA dehydrogenase, but the more specific and better supported process annotations are fatty acid beta-oxidation and isoleucine catabolism. Kept as a correct but general, non-core parent.
Reason: Correct but too general; more specific child processes (beta-oxidation, isoleucine catabolism) are separately annotated.
GO:0008209 androgen metabolic process
IBA
GO_REF:0000033
KEEP AS NON CORE
Summary: Androgen metabolic process inferred phylogenetically. Reflects the weak in-vitro testosterone/androstanediol dehydrogenase side activities; not a demonstrated physiological role. Kept as non-core.
Reason: Minor in-vitro steroid side activity, not the evolved core function.
GO:0008210 estrogen metabolic process
IBA
GO_REF:0000033
KEEP AS NON CORE
Summary: Estrogen metabolic process inferred phylogenetically; reflects weak in-vitro 17-beta-estradiol dehydrogenase side activity. Kept as non-core.
Reason: Minor in-vitro steroid side activity, not the evolved core function.
GO:0003857 (3S)-3-hydroxyacyl-CoA dehydrogenase (NAD+) activity
IEA
GO_REF:0000120
ACCEPT
Summary: Electronic (Rhea/EC 1.1.1.35) assignment of (3S)-3-hydroxyacyl-CoA dehydrogenase activity. This is a genuine, well-established core catalytic activity of HSD17B10, independently supported by multiple experimental annotations below. Accept.
Supporting Evidence:
PMID:9553139
This NAD+-dependent dehydrogenase catalyzes the reversible oxidation of L-3-hydroxyacyl-CoAs to form 3-ketoacyl-CoAs, but it does not act on the D-isomers.
GO:0004022 alcohol dehydrogenase (NAD+) activity
IEA
GO_REF:0000116
MARK AS OVER ANNOTATED
Summary: Generic alcohol dehydrogenase (NAD+) activity assigned electronically from Rhea corticosteroid/steroid reactions. HSD17B10 does have a very weak intrinsic alcohol dehydrogenase activity (e.g. on 2-propanol), but this broad term is an over-annotation of what is really a hydroxysteroid/hydroxyacyl-CoA dehydrogenase; the generic parent is uninformative for this gene.
Reason: Over-broad electronic term; the real chemistry is hydroxysteroid/hydroxyacyl-CoA dehydrogenation and the generic ADH activity is trivial and misleading.
Supporting Evidence:
PMID:10600649
The k(cat) of the ADH activity was three orders of magnitude less than the l-3-hydroxyacyl-CoA dehydrogenase activity
GO:0004303 estradiol 17-beta-dehydrogenase [NAD(P)+] activity
IEA
GO_REF:0000120
KEEP AS NON CORE
Summary: Electronic (Rhea/EC 1.1.1.62) assignment of 17-beta-estradiol dehydrogenase activity. Real but weak intrinsic side activity; kept as non-core (see the corresponding experimental annotations).
Reason: Minor in-vitro steroid side activity, not the evolved core function.
GO:0005739 mitochondrion
IEA
GO_REF:0000120
ACCEPT
Summary: Electronic mitochondrial localization, consistent with all experimental data.
Supporting Evidence:
file:human/HSD17B10/HSD17B10-uniprot.txt
SUBCELLULAR LOCATION: Mitochondrion
GO:0008709 cholate 7-alpha-dehydrogenase (NAD+) activity
IEA
GO_REF:0000120
KEEP AS NON CORE
Summary: Electronic (Rhea/EC 1.1.1.159) assignment of cholate 7-alpha-dehydrogenase activity, matching the in-vitro bile-acid activity characterized for the human enzyme. Real but non-core (in-vitro bile-acid metabolism).
Reason: In-vitro bile-acid side activity, not the evolved core function.
Supporting Evidence:
PMID:12917011
the human orthologue oxidizes the 7alpha-OH of chenodeoxycholic acid (5beta-cholanic acid, 3alpha,7alpha-diol) and cholic acid
GO:0016509 long-chain (3S)-3-hydroxyacyl-CoA dehydrogenase (NAD+) activity
IEA
GO_REF:0000116
MARK AS OVER ANNOTATED
Summary: Electronic (Rhea:31159, hydroxyhexadecanoyl-CoA) assignment of LONG-chain 3-hydroxyacyl-CoA dehydrogenase activity. HSD17B10 actually PREFERS short- and medium-chain substrates and is a poor long-chain enzyme; this specific long-chain child term over-states its chain-length specificity.
Reason: HSD17B10 preferentially accepts straight medium- and short-chain acyl-CoA; the long-chain-specific term misrepresents its substrate preference.
Supporting Evidence:
file:human/HSD17B10/HSD17B10-uniprot.txt
Preferentially accepts straight medium- and short-chain acyl-CoA substrates
GO:0042645 mitochondrial nucleoid
IEA
GO_REF:0000044
ACCEPT
Summary: Electronic (UniProt SubCell) mitochondrial-nucleoid localization, supported experimentally: HSD17B10 (as part of the RNase P machinery) associates with mtDNA nucleoids to initiate RNA processing.
Supporting Evidence:
PMID:24703694
mitochondrial RNA processing enzymes involved in tRNA excision, ribonuclease P (RNase P) and ELAC2, as well as a subset of nascent mitochondrial ribosomal proteins (MRPs) associate with nucleoids to initiate RNA processing and ribosome assembly
GO:0044594 17-beta-hydroxysteroid dehydrogenase (NAD+) activity
IEA
GO_REF:0000116
KEEP AS NON CORE
Summary: Electronic (Rhea:41992) assignment of 17-beta-hydroxysteroid dehydrogenase activity; corresponds to the enzyme's name-giving but catalytically weak steroid side activity. Kept as non-core.
Reason: Minor in-vitro steroid side activity, not the evolved core function.
GO:0047015 3-hydroxy-2-methylbutyryl-CoA dehydrogenase activity
IEA
GO_REF:0000120
ACCEPT
Summary: Electronic (Rhea:13281/EC 1.1.1.178) assignment of 2-methyl-3-hydroxybutyryl-CoA (MHBD) dehydrogenase activity. This is the physiologically most important catalytic activity of HSD17B10 (isoleucine catabolism) and is strongly supported experimentally. Accept as a core molecular function.
Supporting Evidence:
file:human/HSD17B10/HSD17B10-uniprot.txt
Reaction=(2S,3S)-3-hydroxy-2-methylbutanoyl-CoA + NAD(+) = 2-methyl-3-
GO:0047035 testosterone dehydrogenase (NAD+) activity
IEA
GO_REF:0000120
KEEP AS NON CORE
Summary: Electronic (Rhea:14929/EC 1.1.1.239) assignment of testosterone dehydrogenase activity; a demonstrated in-vitro steroid side activity (see IDA below). Kept as non-core.
Reason: In-vitro steroid side activity, not the evolved core function.
GO:0047044 androstan-3-alpha,17-beta-diol dehydrogenase (NAD+) activity
IEA
GO_REF:0000120
KEEP AS NON CORE
Summary: Electronic (Rhea:42004/EC 1.1.1.53) assignment of androstanediol dehydrogenase activity, matching the in-vitro 3-alpha-hydroxysteroid activity. Non-core.
Reason: In-vitro steroid side activity, not the evolved core function.
GO:0106281 chenodeoxycholate 7-alpha-dehydrogenase (NAD+) activity
IEA
GO_REF:0000116
KEEP AS NON CORE
Summary: Electronic (Rhea:42036) bile-acid 7-alpha-dehydrogenase activity, also supported by IDA (PMID:12917011). Real but non-core in-vitro bile-acid activity.
Reason: In-vitro bile-acid side activity, not the evolved core function.
GO:0106282 isoursodeoxycholate 7-beta-dehydrogenase (NAD+) activity
IEA
GO_REF:0000116
KEEP AS NON CORE
Summary: Electronic (Rhea:42024) bile-acid 7-beta-dehydrogenase activity, supported by IDA (PMID:12917011). Non-core in-vitro bile-acid activity.
Reason: In-vitro bile-acid side activity, not the evolved core function.
GO:0106283 ursodeoxycholate 7-beta-dehydrogenase (NAD+) activity
IEA
GO_REF:0000116
KEEP AS NON CORE
Summary: Electronic (Rhea:42028) bile-acid 7-beta-dehydrogenase activity, supported by IDA (PMID:12917011). Non-core in-vitro bile-acid activity.
Reason: In-vitro bile-acid side activity, not the evolved core function.
GO:0005515 protein binding
IPI
PMID:18984158
RNase P without RNA: identification and functional reconstit...
MARK AS OVER ANNOTATED
Summary: IPI protein-binding to TRMT10C (Q7L0Y3), from the study that identified HSD17B10/MRPP2 as a mitochondrial RNase P subunit. The interaction is real and biologically central (MRPP1-MRPP2 subcomplex), but the bare "protein binding" term is uninformative; the functional consequence is captured by the RNase P complex / tRNA-processing annotations.
Reason: Uninformative generic protein-binding term; the meaningful TRMT10C interaction is captured by the mitochondrial RNase P complex and tRNA-processing annotations.
Supporting Evidence:
PMID:18984158
composed of a tRNA methyltransferase, a short-chain dehydrogenase/reductase- family member, and a protein of hitherto unknown functional and evolutionary origin
GO:0005515 protein binding
IPI
PMID:28514442
Architecture of the human interactome defines protein commun...
MARK AS OVER ANNOTATED
Summary: IPI protein-binding to TRMT10C (Q7L0Y3) from a large-scale interactome study. Bare "protein binding" is uninformative; functional meaning captured elsewhere.
Reason: Uninformative generic protein-binding term.
GO:0005515 protein binding
IPI
PMID:29128334
A Map of Human Mitochondrial Protein Interactions Linked to ...
MARK AS OVER ANNOTATED
Summary: IPI protein-binding to EEF2 (P13639) from a mitochondrial neurodegeneration interactome map. Bare "protein binding" is uninformative and this interaction has no established functional role for HSD17B10.
Reason: Uninformative generic protein-binding term; interaction of unclear significance.
GO:0005515 protein binding
IPI
PMID:29128334
A Map of Human Mitochondrial Protein Interactions Linked to ...
MARK AS OVER ANNOTATED
Summary: IPI protein-binding to TRAP1 (Q12931) from a mitochondrial interactome map. Bare "protein binding" is uninformative; no established functional role.
Reason: Uninformative generic protein-binding term; interaction of unclear significance.
GO:0005515 protein binding
IPI
PMID:29880640
Structural insight into the human mitochondrial tRNA purine ...
MARK AS OVER ANNOTATED
Summary: IPI protein-binding to TRMT10C (Q7L0Y3) from the structural study of the MRPP1/MRPP2 complex. Real and central, but bare "protein binding" is uninformative; captured by the RNase P/methyltransferase complex annotations.
Reason: Uninformative generic protein-binding term; the TRMT10C interaction is captured by the complex/process annotations.
GO:0005515 protein binding
IPI
PMID:32814053
Interactome Mapping Provides a Network of Neurodegenerative ...
MARK AS OVER ANNOTATED
Summary: IPI protein-binding to APP/amyloid-beta (P05067) from a neurodegenerative-disease interactome study. The AΞ² interaction (ABAD/ERAB) is a real disease-association but is non-core; the bare term is uninformative.
Reason: Uninformative generic protein-binding term; AΞ² interaction is a disease association, not a core function.
GO:0005515 protein binding
IPI
PMID:32825572
Study of Biomolecular Interactions of Mitochondrial Proteins...
MARK AS OVER ANNOTATED
Summary: IPI protein-binding to APP/amyloid-beta processed peptide (P05067-PRO_0000000092) from a study of Alzheimer-related mitochondrial protein interactions. Non-core disease association; bare term uninformative.
Reason: Uninformative generic protein-binding term; AΞ² interaction is a disease association, not a core function.
GO:0005515 protein binding
IPI
PMID:33961781
Dual proteome-scale networks reveal cell-specific remodeling...
MARK AS OVER ANNOTATED
Summary: IPI protein-binding to TRMT10C (Q7L0Y3) from a proteome-scale interactome study. Bare "protein binding" uninformative; functional meaning captured elsewhere.
Reason: Uninformative generic protein-binding term.
GO:0005515 protein binding
IPI
PMID:9338779
An intracellular protein that binds amyloid-beta peptide and...
MARK AS OVER ANNOTATED
Summary: IPI protein-binding to APP/amyloid-beta (P05067) from the original ERAB/AΞ² discovery paper. Real disease-association interaction but non-core; the bare term is uninformative.
Reason: Uninformative generic protein-binding term; AΞ² interaction is a disease association, not a core function.
GO:0006550 L-isoleucine catabolic process
IEA
GO_REF:0000041
ACCEPT
Summary: Electronic (UniPathway) assignment of L-isoleucine catabolism, the pathway in which HSD17B10's MHBD activity acts. Strongly supported experimentally (see IDA annotations). Core biological process.
Supporting Evidence:
PMID:20077426
the mitochondrial enzyme 2-methyl-3-hydroxybutyryl-CoA dehydrogenase (MHBD) involved in isoleucine metabolism
GO:0006635 fatty acid beta-oxidation
IEA
GO_REF:0000041
ACCEPT
Summary: Electronic (UniPathway) assignment of fatty acid beta-oxidation, where HSD17B10 acts as a short/medium-chain (S)-3-hydroxyacyl-CoA dehydrogenase (third step of the cycle). Supported by IDA (PMID:12917011). Accept.
Supporting Evidence:
file:human/HSD17B10/HSD17B10-uniprot.txt
Acts as (S)-3-hydroxyacyl-CoA dehydrogenase in mitochondrial fatty acid
GO:0006699 bile acid biosynthetic process
IEA
GO_REF:0000041
KEEP AS NON CORE
Summary: Electronic (UniPathway) assignment of bile-acid metabolism, reflecting the in-vitro bile-acid (7-alpha/7-beta-hydroxy) dehydrogenase activities. A real but in-vitro, non-core role. Kept as non-core.
Reason: Based on in-vitro bile-acid dehydrogenase activities; physiological contribution to bile-acid biosynthesis is not established.
GO:0160241 cardiolipin dehydrogenase (NAD+) activity
IDA
PMID:26338420
Myxococcus CsgA, Drosophila Sniffer, and human HSD10 are car...
KEEP AS NON CORE
Summary: IDA for a phospholipase C-like / cardiolipin-oxidizing activity, with a preference for cardiolipin bearing oxidized fatty acids, proposed to protect against reactive oxygen species. Experimentally supported in vitro but a distinct, non-core moonlighting activity. Keep as non-core.
Reason: Experimentally supported in-vitro activity but a separate moonlighting role, not the evolved core (isoleucine/tRNA) functions.
Supporting Evidence:
PMID:26338420
HSD10 exhibits a strong preference for cardiolipin with oxidized fatty acids.
GO:0047035 testosterone dehydrogenase (NAD+) activity
IDA
PMID:28888424
Novel patient missense mutations in the HSD17B10 gene affect...
KEEP AS NON CORE
Summary: IDA testosterone dehydrogenase activity from biochemical characterization of the recombinant enzyme. A demonstrated in-vitro steroid side activity; kept as non-core.
Reason: In-vitro steroid side activity, not the evolved core function.
GO:0001680 tRNA 3'-terminal CCA addition
IDA
PMID:38824131
Structural basis for human mitochondrial tRNA maturation.
KEEP AS NON CORE
Summary: IDA (ComplexPortal) involvement in 3'-CCA addition: cryo-EM shows the TRMT10C/SDR5C1 maturation platform retains the tRNA and presents it to the CCA-adding enzyme TRNT1. HSD17B10 participates as part of the platform rather than catalyzing CCA addition itself. Accept as a valid (non-core) process role.
Reason: Real platform role in CCA addition, but ancillary to the core 5'-processing/ RNase P function; HSD17B10 does not itself add CCA.
Supporting Evidence:
PMID:29040705
presents the nascent tRNA to the mitochondrial CCA-adding enzyme
GO:0005739 mitochondrion
NAS
PMID:21593607
Involvement of human ELAC2 gene product in 3' end processing...
ACCEPT
Summary: NAS mitochondrial localization; consistent with all experimental evidence.
GO:0042780 tRNA 3'-end processing
IDA
PMID:39516281
Structural basis of 3'-tRNA maturation by the human mitochon...
KEEP AS NON CORE
Summary: IDA (ComplexPortal) involvement in tRNA 3'-end processing: cryo-EM of the mitochondrial RNase Z complex (ELAC2/SDR5C1/TRMT10C) shows the SDR5C1/TRMT10C platform supports ELAC2-catalyzed 3'-processing. A real, platform-associated role; kept as non-core relative to the 5'-processing core.
Reason: Platform-supported 3'-processing role; ancillary to the core RNase P 5'-processing function.
Supporting Evidence:
PMID:29040705
significantly enhances the efficiency of ELAC2-catalyzed 3'-processing for 17 of the 22 tRNAs
GO:1902494 catalytic complex
IPI
PMID:38824131
Structural basis for human mitochondrial tRNA maturation.
KEEP AS NON CORE
Summary: Part_of a catalytic complex (ComplexPortal), a generic parent of the mitochondrial RNase P / tRNA-maturation complexes HSD17B10 belongs to. Correct but very general; more specific complex terms (mitochondrial ribonuclease P complex, endoribonuclease complex) are separately annotated.
Reason: Correct but over-general complex term; superseded by specific mitochondrial RNase P complex annotations.
GO:1902555 endoribonuclease complex
IDA
PMID:39516281
Structural basis of 3'-tRNA maturation by the human mitochon...
KEEP AS NON CORE
Summary: IDA part_of an endoribonuclease complex (the mitochondrial RNase Z / ELAC2-SDR5C1-TRMT10C complex from cryo-EM). Correct but general; the specific mitochondrial RNase P complex term is the core CC.
Reason: Correct but general complex term; the specific mitochondrial RNase P complex is the core cellular component.
Supporting Evidence:
PMID:39516281
cryo-EM structures of the mitochondrial RNase Z complex (ELAC2/SDR5C1/TRMT10C)
GO:0005739 mitochondrion
IDA
PMID:29880640
Structural insight into the human mitochondrial tRNA purine ...
ACCEPT
Summary: IDA mitochondrial localization from the RNase P structural study. Accept.
GO:0030678 mitochondrial ribonuclease P complex
IPI
PMID:29880640
Structural insight into the human mitochondrial tRNA purine ...
ACCEPT
Summary: Part_of the mitochondrial RNase P complex, from structural characterization of the MRPP1/MRPP2 (and RNase P) complexes. This is a core cellular component for HSD17B10/MRPP2. Accept.
Supporting Evidence:
PMID:29040705
The human mitochondrial RNase P is composed of three protein subunits: mitochondrial RNase P proteins 1, 2 and 3 (MRPP1, MRPP2 and MRPP3, respectively)
GO:0097745 mitochondrial tRNA 5'-end processing
IDA
PMID:29880640
Structural insight into the human mitochondrial tRNA purine ...
ACCEPT
Summary: IDA involvement in mitochondrial tRNA 5'-end processing, the core RNase P function of HSD17B10/MRPP2. Accept as a core biological process.
Supporting Evidence:
PMID:18984158
extra nucleotides at their 5' ends are removed by an endonuclease called RNase P
GO:0003857 (3S)-3-hydroxyacyl-CoA dehydrogenase (NAD+) activity
EXP
PMID:10600649
Intrinsic alcohol dehydrogenase and hydroxysteroid dehydroge...
ACCEPT
Summary: EXP (3S)-3-hydroxyacyl-CoA dehydrogenase activity, kinetically characterized on the purified enzyme. Core catalytic activity. Accept.
Supporting Evidence:
PMID:10600649
The k(cat) of the ADH activity was three orders of magnitude less than the l-3-hydroxyacyl-CoA dehydrogenase activity
GO:0003857 (3S)-3-hydroxyacyl-CoA dehydrogenase (NAD+) activity
EXP
PMID:26950678
A novel HSD17B10 mutation impairing the activities of the mi...
ACCEPT
Summary: EXP 3-hydroxyacyl-CoA dehydrogenase activity, assayed together with RNase P activities for the K212E disease mutant. Core catalytic activity. Accept.
Supporting Evidence:
PMID:26950678
the p.K212E mutation impairs the SDR5C1-dependent mitochondrial RNase P activities
GO:0003857 (3S)-3-hydroxyacyl-CoA dehydrogenase (NAD+) activity
EXP
PMID:9553139
A human brain L-3-hydroxyacyl-coenzyme A dehydrogenase is id...
ACCEPT
Summary: EXP 3-hydroxyacyl-CoA dehydrogenase activity from cloning/characterization of the purified human brain enzyme. Core catalytic activity. Accept.
Supporting Evidence:
PMID:9553139
This NAD+-dependent dehydrogenase catalyzes the reversible oxidation of L-3-hydroxyacyl-CoAs to form 3-ketoacyl-CoAs, but it does not act on the D-isomers.
GO:0004303 estradiol 17-beta-dehydrogenase [NAD(P)+] activity
EXP
PMID:10600649
Intrinsic alcohol dehydrogenase and hydroxysteroid dehydroge...
KEEP AS NON CORE
Summary: EXP 17-beta-estradiol dehydrogenase activity, kinetically characterized. Real but weak intrinsic steroid activity; kept as non-core.
Reason: Weak in-vitro steroid side activity, not the evolved core function.
Supporting Evidence:
PMID:10600649
comparable with that of the enzyme's hydroxysteroid dehydrogenase (HSD) activity for oxidizing 17beta-oestradiol
GO:0004303 estradiol 17-beta-dehydrogenase [NAD(P)+] activity
EXP
PMID:12917011
Expanded substrate screenings of human and Drosophila type 1...
KEEP AS NON CORE
Summary: EXP 17-beta-estradiol/hydroxysteroid dehydrogenase activity from the expanded substrate screen. Weak in-vitro steroid activity; non-core.
Reason: Weak in-vitro steroid side activity, not the evolved core function.
Supporting Evidence:
PMID:12917011
3alpha-OH and 17beta-OH activities with sex steroids
GO:0005739 mitochondrion
EXP
PMID:12917011
Expanded substrate screenings of human and Drosophila type 1...
ACCEPT
Summary: EXP mitochondrial localization by confocal/electron microscopy; the human enzyme carries an N-terminal mitochondrial targeting signal. Accept.
Supporting Evidence:
PMID:12917011
the human form is localized to mitochondria
GO:0005739 mitochondrion
EXP
PMID:18984158
RNase P without RNA: identification and functional reconstit...
ACCEPT
Summary: EXP mitochondrial localization from the RNase P identification study. Accept.
GO:0047015 3-hydroxy-2-methylbutyryl-CoA dehydrogenase activity
EXP
PMID:20077426
A non-enzymatic function of 17beta-hydroxysteroid dehydrogen...
ACCEPT
Summary: EXP 2-methyl-3-hydroxybutyryl-CoA (MHBD) dehydrogenase activity, measured with the substrate 2-methyl-3-hydroxybutyryl-CoA on WT and disease-mutant enzymes. Core physiological catalytic activity (isoleucine catabolism). Accept.
Supporting Evidence:
PMID:20077426
substrate 2-methyl-3-hydroxybutyryl-CoA
GO:0005739 mitochondrion
IDA
PMID:23042678
A subcomplex of human mitochondrial RNase P is a bifunctiona...
ACCEPT
Summary: IDA mitochondrial localization. Accept.
GO:0043527 tRNA methyltransferase complex
IPI
PMID:23042678
A subcomplex of human mitochondrial RNase P is a bifunctiona...
ACCEPT
Summary: Part_of a tRNA methyltransferase complex: the MRPP1-MRPP2 (TRMT10C-SDR5C1) subcomplex is the m1A9/m1G9 methyltransferase, with HSD17B10 as the required non-catalytic partner. Accept as a valid cellular component (methyltransferase subcomplex of RNase P).
Supporting Evidence:
PMID:23042678
the human mitochondrial enzyme, moreover, requires a short-chain dehydrogenase as a partner protein
GO:0070901 mitochondrial tRNA methylation
IDA
PMID:23042678
A subcomplex of human mitochondrial RNase P is a bifunctiona...
ACCEPT
Summary: IDA involvement in mitochondrial tRNA methylation (m1A9/m1G9 formation by the MRPP1-MRPP2 subcomplex). A genuine, experimentally supported role of the RNase P machinery HSD17B10 is part of. Accept.
Supporting Evidence:
PMID:23042678
is the methyltransferase responsible for m(1)G9 and m(1)A9 formation
GO:0005739 mitochondrion
HTP
PMID:34800366
Quantitative high-confidence human mitochondrial proteome an...
ACCEPT
Summary: HTP mitochondrial localization from a high-confidence mitochondrial proteome study. Consistent with all evidence. Accept.
GO:0106281 chenodeoxycholate 7-alpha-dehydrogenase (NAD+) activity
IDA
PMID:12917011
Expanded substrate screenings of human and Drosophila type 1...
KEEP AS NON CORE
Summary: IDA bile-acid 7-alpha-dehydrogenase activity (chenodeoxycholate/cholate) from the expanded substrate screen. Real in-vitro bile-acid activity; non-core.
Reason: In-vitro bile-acid side activity, not the evolved core function.
Supporting Evidence:
PMID:12917011
the human orthologue oxidizes the 7alpha-OH of chenodeoxycholic acid
GO:0106282 isoursodeoxycholate 7-beta-dehydrogenase (NAD+) activity
IDA
PMID:12917011
Expanded substrate screenings of human and Drosophila type 1...
KEEP AS NON CORE
Summary: IDA bile-acid 7-beta-dehydrogenase activity on (iso)ursodeoxycholic acid from the substrate screen. Real in-vitro bile-acid activity; non-core.
Reason: In-vitro bile-acid side activity, not the evolved core function.
Supporting Evidence:
PMID:12917011
act as 7beta-OH dehydrogenases of ursodeoxycholic or isoursodeoxycholic acid
GO:0106283 ursodeoxycholate 7-beta-dehydrogenase (NAD+) activity
IDA
PMID:12917011
Expanded substrate screenings of human and Drosophila type 1...
KEEP AS NON CORE
Summary: IDA bile-acid 7-beta-dehydrogenase activity on ursodeoxycholic acid. Real in-vitro bile-acid activity; non-core.
Reason: In-vitro bile-acid side activity, not the evolved core function.
Supporting Evidence:
PMID:12917011
act as 7beta-OH dehydrogenases of ursodeoxycholic or isoursodeoxycholic acid
GO:0006550 L-isoleucine catabolic process
IDA
PMID:18996107
Study of patients and carriers with 2-methyl-3-hydroxybutyry...
ACCEPT
Summary: IDA involvement in L-isoleucine catabolism from clinical/biochemical study of MHBD-deficient patients (deficient MHBD activity, isoleucine-pathway metabolites). Core biological process. Accept.
Supporting Evidence:
PMID:18996107
2-Methyl-3-hydroxybutyryl-CoA dehydrogenase (MHBD) deficiency
GO:0006550 L-isoleucine catabolic process
IDA
PMID:19706438
Mental retardation linked to mutations in the HSD17B10 gene ...
ACCEPT
Summary: IDA involvement in isoleucine catabolism; the disease mutant fails to dehydrogenate 2-methyl-3-hydroxybutyryl-CoA. Core biological process. Accept.
Supporting Evidence:
PMID:19706438
HSD10(E249Q) was unable to catalyze the dehydrogenation of 2-methyl-3-hydroxybutyryl-CoA
GO:0044594 17-beta-hydroxysteroid dehydrogenase (NAD+) activity
IDA
PMID:12917011
Expanded substrate screenings of human and Drosophila type 1...
KEEP AS NON CORE
Summary: IDA 17-beta-hydroxysteroid dehydrogenase activity from the expanded substrate screen. Real but weak intrinsic steroid activity; non-core.
Reason: Weak in-vitro steroid side activity, not the evolved core function.
Supporting Evidence:
PMID:12917011
3alpha-OH and 17beta-OH activities with sex steroids
GO:0047015 3-hydroxy-2-methylbutyryl-CoA dehydrogenase activity
IDA
PMID:18996107
Study of patients and carriers with 2-methyl-3-hydroxybutyry...
ACCEPT
Summary: IDA MHBD activity measured in patient fibroblasts. Core physiological catalytic activity (isoleucine catabolism). Accept.
Supporting Evidence:
PMID:18996107
MHBD activity was clearly deficient in males
GO:0003857 (3S)-3-hydroxyacyl-CoA dehydrogenase (NAD+) activity
IDA
PMID:12917011
Expanded substrate screenings of human and Drosophila type 1...
ACCEPT
Summary: IDA (3S)-3-hydroxyacyl-CoA dehydrogenase activity from the substrate screen. Core catalytic activity. Accept.
Supporting Evidence:
PMID:12917011
In addition to the known hydroxyacyl-CoA dehydrogenase
GO:0006635 fatty acid beta-oxidation
IDA
PMID:12917011
Expanded substrate screenings of human and Drosophila type 1...
ACCEPT
Summary: IDA involvement in fatty acid beta-oxidation (short/medium-chain 3-hydroxyacyl-CoA dehydrogenase step). Accept.
Supporting Evidence:
file:human/HSD17B10/HSD17B10-uniprot.txt
Acts as (S)-3-hydroxyacyl-CoA dehydrogenase in mitochondrial fatty acid
GO:0006699 bile acid biosynthetic process
IDA
PMID:12917011
Expanded substrate screenings of human and Drosophila type 1...
KEEP AS NON CORE
Summary: IDA involvement in bile-acid metabolism, from the in-vitro bile-acid dehydrogenase activities and bile-acid isomerization proposal. Real but in-vitro; physiological contribution unestablished. Non-core.
Reason: Based on in-vitro bile-acid activities; physiological role in bile-acid biosynthesis not established.
Supporting Evidence:
PMID:12917011
participates in bile acid isomerization
GO:0008207 C21-steroid hormone metabolic process
IDA
PMID:12917011
Expanded substrate screenings of human and Drosophila type 1...
KEEP AS NON CORE
Summary: IDA involvement in C21-steroid (glucocorticoid/gestagen) metabolism, from the 20-beta/21-OH oxidation activities. In-vitro steroid metabolism; non-core.
Reason: In-vitro steroid metabolism, not the evolved core function.
Supporting Evidence:
PMID:12917011
oxidize the 20beta-OH and 21-OH groups in C21 steroids
GO:0008209 androgen metabolic process
IDA
PMID:12917011
Expanded substrate screenings of human and Drosophila type 1...
KEEP AS NON CORE
Summary: IDA involvement in androgen metabolism, from testosterone/androstanediol dehydrogenase activities. In-vitro steroid metabolism; non-core.
Reason: In-vitro steroid side activity, not the evolved core function.
Supporting Evidence:
PMID:12917011
3alpha-OH and 17beta-OH activities with sex steroids
GO:0008210 estrogen metabolic process
IDA
PMID:12917011
Expanded substrate screenings of human and Drosophila type 1...
KEEP AS NON CORE
Summary: IDA involvement in estrogen metabolism, from 17-beta-estradiol dehydrogenase activity. In-vitro steroid metabolism; non-core.
Reason: In-vitro steroid side activity, not the evolved core function.
Supporting Evidence:
PMID:12917011
17beta-OH activities with sex steroids
GO:0008709 cholate 7-alpha-dehydrogenase (NAD+) activity
IDA
PMID:12917011
Expanded substrate screenings of human and Drosophila type 1...
KEEP AS NON CORE
Summary: IDA cholate 7-alpha-dehydrogenase activity from the substrate screen. Real in-vitro bile-acid activity; non-core.
Reason: In-vitro bile-acid side activity, not the evolved core function.
Supporting Evidence:
PMID:12917011
the human orthologue oxidizes the 7alpha-OH of chenodeoxycholic acid (5beta-cholanic acid, 3alpha,7alpha-diol) and cholic acid
GO:0042645 mitochondrial nucleoid
IDA
PMID:24703694
Initial steps in RNA processing and ribosome assembly occur ...
ACCEPT
Summary: IDA localization to the mitochondrial nucleoid, where RNase P/ELAC2 initiate tRNA processing and ribosome assembly. Accept.
Supporting Evidence:
PMID:24703694
associate with nucleoids to initiate RNA processing and ribosome assembly
GO:0047015 3-hydroxy-2-methylbutyryl-CoA dehydrogenase activity
IDA
PMID:19706438
Mental retardation linked to mutations in the HSD17B10 gene ...
ACCEPT
Summary: IDA MHBD activity; disease mutant loses dehydrogenation of 2-methyl-3-hydroxybutyryl-CoA. Core physiological catalytic activity. Accept.
Supporting Evidence:
PMID:19706438
HSD10(E249Q) was unable to catalyze the dehydrogenation of 2-methyl-3-hydroxybutyryl-CoA
GO:0047035 testosterone dehydrogenase (NAD+) activity
IDA
PMID:12917011
Expanded substrate screenings of human and Drosophila type 1...
KEEP AS NON CORE
Summary: IDA testosterone dehydrogenase activity from the substrate screen. In-vitro steroid side activity; non-core.
Reason: In-vitro steroid side activity, not the evolved core function.
Supporting Evidence:
PMID:12917011
3alpha-OH and 17beta-OH activities with sex steroids
GO:0062173 brexanolone metabolic process
IDA
PMID:19706438
Mental retardation linked to mutations in the HSD17B10 gene ...
KEEP AS NON CORE
Summary: IDA involvement in brexanolone (allopregnanolone) metabolism: HSD17B10 oxidizes the 3-alpha-OH of this neurosteroid GABA-A modulator, and disease mutants lose this activity. A genuine, potentially neurologically relevant side activity; kept as non-core relative to the isoleucine/tRNA core roles.
Reason: Real neurosteroid activity of possible pathophysiological relevance, but a side activity rather than the evolved core function.
Supporting Evidence:
PMID:19706438
the oxidation of allopregnanolone, a positive modulator of the gamma-aminobutyric acid type A receptor
GO:0000049 tRNA binding
IDA
PMID:29040705
The MRPP1/MRPP2 complex is a tRNA-maturation platform in hum...
ACCEPT
Summary: IDA tRNA binding: the MRPP1/2 (TRMT10C/SDR5C1) complex binds and retains mitochondrial pre-tRNA throughout maturation. tRNA binding is the subunit-level molecular activity underlying HSD17B10/MRPP2's structural role in RNase P/tRNA processing. Accept as a core molecular function.
Supporting Evidence:
PMID:29040705
it retains the tRNA product from the 5'-processing step
GO:0003857 (3S)-3-hydroxyacyl-CoA dehydrogenase (NAD+) activity
IDA
PMID:25925575
Molecular insights into HSD10 disease: impact of SDR5C1 muta...
ACCEPT
Summary: IDA 3-hydroxyacyl-CoA dehydrogenase activity assayed for disease mutants alongside RNase P/tRNA functions. Core catalytic activity. Accept.
Supporting Evidence:
PMID:25925575
pathogenic mutations impair SDR5C1-dependent dehydrogenation, tRNA processing and methylation
GO:0003857 (3S)-3-hydroxyacyl-CoA dehydrogenase (NAD+) activity
IDA
PMID:28888424
Novel patient missense mutations in the HSD17B10 gene affect...
ACCEPT
Summary: IDA 3-hydroxyacyl-CoA dehydrogenase activity for the V12L/V176M disease mutants (reduced dehydrogenase activity). Core catalytic activity. Accept.
Supporting Evidence:
PMID:28888424
Both mutant proteins showed significant reduction in the dehydrogenase, methyltransferase and tRNA processing activities compared to wildtype
GO:0005515 protein binding
IPI
PMID:29040705
The MRPP1/MRPP2 complex is a tRNA-maturation platform in hum...
MARK AS OVER ANNOTATED
Summary: IPI protein-binding to TRMT10C (Q7L0Y3) from the MRPP1/2 tRNA-maturation platform study. Real and central, but bare "protein binding" is uninformative; functional meaning captured by the RNase P complex/process annotations.
Reason: Uninformative generic protein-binding term; TRMT10C interaction captured by complex/process annotations.
GO:0030678 mitochondrial ribonuclease P complex
IDA
PMID:25925575
Molecular insights into HSD10 disease: impact of SDR5C1 muta...
ACCEPT
Summary: IDA part_of the mitochondrial RNase P complex; disease mutations disrupt complex assembly/interaction with TRMT10C. Core cellular component. Accept.
Supporting Evidence:
PMID:25925575
moonlighting as a component of human mitochondrial RNase P
GO:0030678 mitochondrial ribonuclease P complex
IDA
PMID:28888424
Novel patient missense mutations in the HSD17B10 gene affect...
ACCEPT
Summary: IDA part_of the mitochondrial RNase P complex (MRPP1/MRPP2/MRPP3). Core cellular component. Accept.
Supporting Evidence:
PMID:28888424
in a complex with MRPP1 and MRPP3 (also known as PRORP) proteins for 5'-end processing of mitochondrial precursor tRNA
GO:0051289 protein homotetramerization
IDA
PMID:25925575
Molecular insights into HSD10 disease: impact of SDR5C1 muta...
ACCEPT
Summary: IDA protein homotetramerization; HSD17B10 functions as a homotetramer and some disease mutations disrupt tetramerization. A genuine, structurally important property. Accept.
Supporting Evidence:
PMID:25925575
Some mutations disrupt the homotetramerization of SDR5C1
GO:0070901 mitochondrial tRNA methylation
IDA
PMID:25925575
Molecular insights into HSD10 disease: impact of SDR5C1 muta...
ACCEPT
Summary: IDA involvement in mitochondrial tRNA methylation (m1A9/m1G9); disease mutations impair methylation. Genuine RNase P subcomplex role. Accept.
Supporting Evidence:
PMID:25925575
pathogenic mutations impair SDR5C1-dependent dehydrogenation, tRNA processing and methylation
GO:0070901 mitochondrial tRNA methylation
IDA
PMID:28888424
Novel patient missense mutations in the HSD17B10 gene affect...
ACCEPT
Summary: IDA involvement in mitochondrial tRNA methylation; V12L/V176M mutants show reduced methyltransferase activity. Accept.
Supporting Evidence:
PMID:28888424
N1-methylation of purines at position 9 of mitochondrial tRNA
GO:0097745 mitochondrial tRNA 5'-end processing
IDA
PMID:24549042
Mutation or knock-down of 17Ξ²-hydroxysteroid dehydrogenase t...
ACCEPT
Summary: IDA involvement in mitochondrial tRNA 5'-end processing; knock-down / patient mutation impairs heavy-strand transcript processing. Core biological process. Accept.
Supporting Evidence:
PMID:24549042
evidence of impaired processing of precursor tRNA transcripts of the mitochondrial heavy strand
GO:0097745 mitochondrial tRNA 5'-end processing
IDA
PMID:25925575
Molecular insights into HSD10 disease: impact of SDR5C1 muta...
ACCEPT
Summary: IDA involvement in mitochondrial tRNA 5'-end processing; disease mutations impair RNase P tRNA processing. Core biological process. Accept.
Supporting Evidence:
PMID:25925575
the enzyme removing 5'-extensions of tRNAs, an early and crucial step in tRNA maturation
GO:0097745 mitochondrial tRNA 5'-end processing
IDA
PMID:28888424
Novel patient missense mutations in the HSD17B10 gene affect...
ACCEPT
Summary: IDA involvement in mitochondrial tRNA 5'-end processing; disease mutants show reduced tRNA processing. Core biological process. Accept.
Supporting Evidence:
PMID:28888424
5'-end processing of mitochondrial precursor tRNA
GO:0097745 mitochondrial tRNA 5'-end processing
IDA
PMID:29040705
The MRPP1/MRPP2 complex is a tRNA-maturation platform in hum...
ACCEPT
Summary: IDA involvement in mitochondrial tRNA 5'-end processing; MRPP1/2 mediates RNase P 5'-leader removal. Core biological process. Accept.
Supporting Evidence:
PMID:29040705
MRPP1/2 is not only responsible for mitochondrial RNase P-based 5'-leader removal
GO:1990180 mitochondrial tRNA 3'-end processing
IDA
PMID:29040705
The MRPP1/MRPP2 complex is a tRNA-maturation platform in hum...
KEEP AS NON CORE
Summary: IDA involvement in mitochondrial tRNA 3'-end processing: the MRPP1/2 platform retains the tRNA after 5'-cleavage and enhances ELAC2-catalyzed 3'-processing. Real platform-associated role; kept as non-core relative to the 5'-processing core (HSD17B10 does not itself perform 3'-cleavage).
Reason: Platform-supported 3'-processing role; ancillary to the core RNase P 5'-processing function.
Supporting Evidence:
PMID:29040705
significantly enhances the efficiency of ELAC2-catalyzed 3'-processing for 17 of the 22 tRNAs
GO:0007005 mitochondrion organization
IMP
PMID:20077426
A non-enzymatic function of 17beta-hydroxysteroid dehydrogen...
KEEP AS NON CORE
Summary: IMP involvement in mitochondrion organization: knock-down/knock-out and disease mutations disrupt mitochondrial structural integrity (cristae) independently of dehydrogenase activity. This reflects the essential non-enzymatic (RNase P/tRNA-processing) role. A genuine but downstream/pleiotropic process; kept as non-core.
Reason: Genuine IMP phenotype but a downstream consequence of the RNase P/tRNA-processing role; more specific molecular roles capture the core function.
Supporting Evidence:
PMID:20077426
a property of HSD10 independent of its enzymatic activity is essential for structural and functional integrity of mitochondria
GO:0003857 (3S)-3-hydroxyacyl-CoA dehydrogenase (NAD+) activity
IDA
PMID:23042678
A subcomplex of human mitochondrial RNase P is a bifunctiona...
ACCEPT
Summary: IDA 3-hydroxyacyl-CoA dehydrogenase activity (CAFA-assigned) associated with the methyltransferase-subcomplex study. Core catalytic activity. Accept.
Supporting Evidence:
PMID:23042678
a fatty and amino acid degradation enzyme in tRNA methylation
GO:0005515 protein binding
IPI
PMID:23042678
A subcomplex of human mitochondrial RNase P is a bifunctiona...
MARK AS OVER ANNOTATED
Summary: IPI protein-binding to TRMT10C (Q7L0Y3), the methyltransferase partner. Real and central, but bare "protein binding" is uninformative; captured by the RNase P / methyltransferase complex annotations.
Reason: Uninformative generic protein-binding term; TRMT10C interaction captured by complex annotations.
GO:0030678 mitochondrial ribonuclease P complex
TAS
PMID:23042678
A subcomplex of human mitochondrial RNase P is a bifunctiona...
ACCEPT
Summary: TAS part_of the mitochondrial RNase P complex. Core cellular component, independently supported by IDA. Accept.
Supporting Evidence:
PMID:23042678
a subcomplex of human mitochondrial RNase P
GO:0003723 RNA binding
HDA
PMID:22681889
The mRNA-bound proteome and its global occupancy profile on ...
MARK AS OVER ANNOTATED
Summary: HDA RNA binding from a global mRNA-interactome capture screen. HSD17B10 does bind tRNA (specifically captured by GO:0000049), but this high-throughput generic "RNA binding" from a poly(A)-mRNA capture experiment is a non-specific over-annotation for a protein whose relevant RNA ligand is tRNA.
Reason: Non-specific high-throughput RNA-binding capture; the specific and relevant activity (tRNA binding) is separately and better annotated.
GO:0005759 mitochondrial matrix
TAS
Reactome:R-HSA-508369
ACCEPT
Summary: TAS (Reactome) mitochondrial-matrix localization for the isoleucine-catabolism reaction. Consistent with the authoritative matrix localization. Accept.
Supporting Evidence:
file:human/HSD17B10/HSD17B10-uniprot.txt
Mitochondrion matrix
GO:0005759 mitochondrial matrix
TAS
Reactome:R-HSA-6787591
ACCEPT
Summary: TAS (Reactome) mitochondrial-matrix localization for the tRNA m1G9 methylation reaction. Consistent with authoritative matrix localization. Accept.
Supporting Evidence:
file:human/HSD17B10/HSD17B10-uniprot.txt
Mitochondrion matrix
GO:0005759 mitochondrial matrix
TAS
Reactome:R-HSA-6787594
ACCEPT
Summary: TAS (Reactome) mitochondrial-matrix localization for the tRNA m1A9 methylation reaction. Accept.
Supporting Evidence:
file:human/HSD17B10/HSD17B10-uniprot.txt
Mitochondrion matrix
GO:0005759 mitochondrial matrix
TAS
Reactome:R-HSA-70837
ACCEPT
Summary: TAS (Reactome) mitochondrial-matrix localization for the isoleucine-catabolism (MHBD) reaction. Accept.
Supporting Evidence:
file:human/HSD17B10/HSD17B10-uniprot.txt
Mitochondrion matrix
GO:0005759 mitochondrial matrix
TAS
Reactome:R-HSA-9838081
ACCEPT
Summary: TAS (Reactome) mitochondrial-matrix localization (LONP1 matrix-protein degradation context). Localization consistent with authoritative matrix localization. Accept.
Supporting Evidence:
file:human/HSD17B10/HSD17B10-uniprot.txt
Mitochondrion matrix
GO:0005759 mitochondrial matrix
TAS
Reactome:R-HSA-9838093
ACCEPT
Summary: TAS (Reactome) mitochondrial-matrix localization (LONP1 matrix-protein binding context). Accept.
Supporting Evidence:
file:human/HSD17B10/HSD17B10-uniprot.txt
Mitochondrion matrix
GO:0005739 mitochondrion
ISS
GO_REF:0000024
ACCEPT
Summary: ISS mitochondrial localization by orthology to mouse (O08756). Consistent with all experimental data. Accept.
GO:0005737 cytoplasm
TAS
PMID:9338779
An intracellular protein that binds amyloid-beta peptide and...
MARK AS OVER ANNOTATED
Summary: TAS cytoplasm from the 1997 ERAB discovery paper, which reported an ER/plasma-membrane-associated AΞ²-binding protein. This localization is a legacy mis-assignment superseded by all subsequent work establishing the protein as mitochondrial (matrix/nucleoid). Over-annotated.
Reason: Legacy mislocalization from the original ERAB paper; superseded by mitochondrial matrix/nucleoid localization in all later studies.
Supporting Evidence:
PMID:12917011
the human form is localized to mitochondria
GO:0005886 plasma membrane
TAS
PMID:9338779
An intracellular protein that binds amyloid-beta peptide and...
MARK AS OVER ANNOTATED
Summary: TAS plasma membrane from the 1997 ERAB discovery paper. A legacy mis-assignment; the protein is a mitochondrial-matrix enzyme with no established plasma-membrane role. Over-annotated.
Reason: Legacy mislocalization from the original ERAB paper; superseded by mitochondrial localization.
Supporting Evidence:
PMID:12917011
the human form is localized to mitochondria
GO:0006629 lipid metabolic process
TAS
PMID:9338779
An intracellular protein that binds amyloid-beta peptide and...
KEEP AS NON CORE
Summary: TAS broad "lipid metabolic process" from the 1997 ERAB paper. HSD17B10 does have fatty-acid/cardiolipin/steroid-lipid activities, so a lipid-metabolism parent is not wrong, but this is a very general term captured better by fatty acid beta-oxidation. Kept as non-core.
Reason: Over-general parent; more specific lipid processes are separately annotated.
GO:0008709 cholate 7-alpha-dehydrogenase (NAD+) activity
TAS
PMID:9338779
An intracellular protein that binds amyloid-beta peptide and...
KEEP AS NON CORE
Summary: TAS cholate 7-alpha-dehydrogenase activity attributed to the 1997 ERAB paper, which did NOT establish bile-acid activity (it described an AΞ²-binding dehydrogenase); the bile-acid activity was actually characterized later (PMID:12917011, separately annotated by IDA). The activity itself is a real but non-core in-vitro side activity, so this is kept as non-core; note that the 1997 citation is a legacy mis-attribution and the true support is PMID:12917011.
Reason: Real but non-core in-vitro bile-acid side activity; the cited 1997 paper does not actually support it (true support is the later IDA, PMID:12917011).
Supporting Evidence:
PMID:12917011
the human orthologue oxidizes the 7alpha-OH of chenodeoxycholic acid (5beta-cholanic acid, 3alpha,7alpha-diol) and cholic acid

Core Functions

NAD+-dependent (S)-2-methyl-3-hydroxybutyryl-CoA dehydrogenase (MHBD) acting in the mitochondrial matrix in L-isoleucine catabolism, oxidizing (2S,3S)-3-hydroxy-2-methylbutanoyl-CoA to 2-methyl-3-oxobutanoyl-CoA; the enzyme also has broad short/medium-chain (S)-3-hydroxyacyl-CoA dehydrogenase activity in fatty acid beta-oxidation. Deficiency of this activity causes the MHBD organic aciduria.

Supporting Evidence:
  • PMID:19706438
    HSD10(E249Q) was unable to catalyze the dehydrogenation of 2-methyl-3-hydroxybutyryl-CoA
  • PMID:20077426
    the mitochondrial enzyme 2-methyl-3-hydroxybutyryl-CoA dehydrogenase (MHBD) involved in isoleucine metabolism
  • PMID:9553139
    This NAD+-dependent dehydrogenase catalyzes the reversible oxidation of L-3-hydroxyacyl-CoAs to form 3-ketoacyl-CoAs, but it does not act on the D-isomers.

Broad-substrate NAD+-dependent (S)-3-hydroxyacyl-CoA dehydrogenase catalyzing the third step of mitochondrial fatty acid beta-oxidation, with NAD+ as the required cofactor bound in a Rossmann fold. This is the family-defining SDR catalytic activity underlying the MHBD and other hydroxyacyl-CoA/hydroxysteroid activities.

Supporting Evidence:
  • PMID:9553139
    This NAD+-dependent dehydrogenase catalyzes the reversible oxidation of L-3-hydroxyacyl-CoAs to form 3-ketoacyl-CoAs, but it does not act on the D-isomers.
  • PMID:12917011
    In addition to the known hydroxyacyl-CoA dehydrogenase

Essential non-catalytic (moonlighting) structural subunit MRPP2 of mitochondrial RNase P. As a homotetramer it binds mitochondrial pre-tRNA and, together with the methyltransferase TRMT10C/MRPP1 and the nuclease PRORP/MRPP3, carries out 5'-end processing of mitochondrial tRNAs; the TRMT10C-HSD17B10 subcomplex also acts as a tRNA-maturation platform and enables N1-methylation of purine-9 (m1A9/m1G9). This role is independent of the enzyme's dehydrogenase activity, and its loss underlies the severity of HSD10 mitochondrial disease.

Supporting Evidence:
  • PMID:18984158
    human mitochondrial RNase P is a protein enzyme that does not require a trans-acting RNA component for catalysis
  • PMID:29040705
    it retains the tRNA product from the 5'-processing step
  • PMID:23042678
    the human mitochondrial enzyme, moreover, requires a short-chain dehydrogenase as a partner protein
  • PMID:20077426
    a property of HSD10 independent of its enzymatic activity is essential for structural and functional integrity of mitochondria

References

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Suggested Questions for Experts

Q: Which of HSD17B10's catalytic activities (isoleucine-pathway MHBD, general 3-hydroxyacyl-CoA, neurosteroid/allopregnanolone, cardiolipin phospholipase) are physiologically significant in vivo versus in-vitro artifacts of a broad-specificity SDR active site?

Q: How does the non-catalytic RNase P/tRNA-processing role mechanistically account for HSD10 disease severity, and can it be uncoupled therapeutically from the metabolic (isoleucine) role?

Q: Does the neurosteroid (allopregnanolone/brexanolone) oxidation activity contribute to the neurological phenotype of HSD10 disease independently of tRNA processing?

Suggested Experiments

Experiment: Separation-of-function mutants that selectively abolish either dehydrogenase activity or RNase P/TRMT10C-binding, tested in patient-derived and knockout cells for mitochondrial tRNA processing, respiratory chain assembly, and viability.

Experiment: Quantitative in-vivo metabolic flux analysis of isoleucine catabolism and neurosteroid (allopregnanolone) levels in HSD10-deficient models to define which metabolic defects contribute to neuropathology.

Experiment: Structural and biochemical dissection of how the HSD17B10 homotetramer scaffolds TRMT10C and pre-tRNA across the sequential 5'-processing, methylation, 3'-processing and CCA-addition steps.

πŸ“š Additional Documentation

Notes

(HSD17B10-notes.md)

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