Evidence map›Paper›PMID 36802322›Full record

ArticleMolecular neurobiology2023

HPRT1 Deficiency Induces Alteration of Mitochondrial Energy Metabolism in the Brain.

Andrey Y Vinokurov, Vladislav O Soldatov, Evgenia S Seregina, Angelina I Dolgikh, Pavel A Tagunov, Andrey V Dunaev, Marina Y Skorkina, Alexey V Deykin, Andrey Y Abramov

Open access · hybridAbstract read
In one paragraph

Article in Molecular neurobiology, 2023. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 13 papers.

0numbers the graph read from it
0cells of the map it votes in
13citing papers in PubMed
4.5field-weighted citation impact, top 5% of its field
1 · What the graph read from it

What it found

Each row is one number read from the abstract, on the scale the paper reported it, with its interval. Left of the dashed line favours the treatment, right favours the comparator. Under each row is the sentence it came from. New to these charts? A ten-minute tutorial.

The abstract states no effect estimate the extractor could read, or names no intervention and outcome on the map, so this paper lights no cell and moves no belief. It is still indexed, cited and linked below.

2 · The registry

The trial behind it

Trials whose registry record cites this paper, or whose number appears in the abstract. A trial that started after this paper was published is citing it as background, not reporting it.

Neither the registry nor the abstract names a trial number. If this is a trial report, that itself is worth knowing.

3 · Its place in the literature

Who cites it

13 citing papers in PubMed, 29 citations in OpenAlex.

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  8. Cytoprotective Action of Sodium Fumarate in anSovremennye tekhnologii v meditsine · 2025
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4 · The record

Corrections and comments

PubMed lists nothing against this paper. Absence here is not a guarantee, only a check that was made.

5 · Who and what money

Authors and funding

9 authors at 3 institutions in 2 countries.

Andrey Y VinokurovCell Physiology & Pathology Laboratory of R&D Center of Biomedical Photonics, Orel State University, Orel, 302026, Russia. vinokurovayu@oreluniver.ru.
Vladislav O SoldatovLaboratory of Genome Editing for Biomedicine and Animal Health, Belgorod State National Research University, Belgorod, 308015, Russia.
Evgenia S SereginaCell Physiology & Pathology Laboratory of R&D Center of Biomedical Photonics, Orel State University, Orel, 302026, Russia.
Angelina I DolgikhCell Physiology & Pathology Laboratory of R&D Center of Biomedical Photonics, Orel State University, Orel, 302026, Russia.
Pavel A TagunovCell Physiology & Pathology Laboratory of R&D Center of Biomedical Photonics, Orel State University, Orel, 302026, Russia.
Andrey V DunaevCell Physiology & Pathology Laboratory of R&D Center of Biomedical Photonics, Orel State University, Orel, 302026, Russia.
Marina Y SkorkinaLaboratory of Genome Editing for Biomedicine and Animal Health, Belgorod State National Research University, Belgorod, 308015, Russia.
Alexey V DeykinLaboratory of Genome Editing for Biomedicine and Animal Health, Belgorod State National Research University, Belgorod, 308015, Russia.
Andrey Y AbramovCell Physiology & Pathology Laboratory of R&D Center of Biomedical Photonics, Orel State University, Orel, 302026, Russia. a.abramov@ucl.ac.uk.ORCID http://orcid.org/0000-0002-7646-7235
Orel State University named after I.S. Turgenev · RUBelgorod National Research University · RUQueen Mary University of London · GB

Funding

Russian Federation Government 075-15-2022-1095
6 · The paper itself

Abstract

Alterations in function of hypoxanthine guanine phosphoribosyl transferase (HPRT), one of the major enzymes involved in purine nucleotide exchange, lead to overproduction of uric acid and produce various symptoms of Lesch-Nyhan syndrome (LNS). One of the hallmarks of LNS is maximal expression of HPRT in the central nervous system with the highest activity of this enzyme in the midbrain and basal ganglia. However, the nature of neurological symptoms has yet to be clarified in details. Here, we studied whether HPRT1 deficiency changes mitochondrial energy metabolism and redox balance in murine neurons from the cortex and midbrain. We found that HPRT1 deficiency inhibits complex I-dependent mitochondrial respiration resulting in increased levels of mitochondrial NADH, reduction of the mitochondrial membrane potential, and increased rate of reactive oxygen species (ROS) production in mitochondria and cytosol. However, increased ROS production did not induce oxidative stress and did not decrease the level of endogenous antioxidant glutathione (GSH). Thus, disruption of mitochondrial energy metabolism but not oxidative stress could play a role of potential trigger of brain pathology in LNS.

Indexed as

Lesch-Nyhan SyndromeAnimalsBrainEnergy MetabolismHypoxanthine PhosphoribosyltransferaseMiceReactive Oxygen SpeciesHypoxanthine PhosphoribosyltransferaseReactive Oxygen SpeciesComplex I inhibitionHPRT deficiencyMitochondrial dysfunctionReactive oxygen species

Identifiers

PMID36802322
PMCPMC10122629
OpenAlexW4321437549

What OpenQuestion holds

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LicenceCC BY
Read underepoch 390

Registered trials

None linked

Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the OpenQuestion graph.