The Evolutionarily Conserved Tre2/Bub2/Cdc16 (TBC), Lysin Motif (LysM), Domain Catalytic (TLDc) Domain Is Neuroprotective against Oxidative Stress
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Oxidative stress is a pathological feature of many neurological disorders; therefore, utilizing proteins that are protective against such cellular insults is a potentially valuable therapeutic approach. Oxidation resistance 1 (OXR1) has been shown previously to be critical for oxidative stress resistance in neuronal cells; deletion of this gene causes neurodegeneration in mice, yet conversely, overexpression of OXR1 is protective in cellular and mouse models of amyotrophic lateral sclerosis. However, the molecular mechanisms involved are unclear. OXR1 contains the Tre2/Bub2/Cdc16 (TBC), lysin motif (LysM), domain catalytic (TLDc) domain, a motif present in a family of proteins including TBC1 domain family member 24 (TBC1D24), a protein mutated in a range of disorders characterized by seizures, hearing loss, and neurodegeneration. The TLDc domain is highly conserved across species, although the structure-function relationship is unknown. To understand the role of this domain in the stress response, we carried out systematic analysis of all mammalian TLDc domain-containing proteins, investigating their expression and neuroprotective properties in parallel. In addition, we performed a detailed structural and functional study of this domain in which we identified key residues required for its activity. Finally, we present a new mouse insertional mutant of Oxr1, confirming that specific disruption of the TLDc domain in vivo is sufficient to cause neurodegeneration. Our data demonstrate that the integrity of the TLDc domain is essential for conferring neuroprotection, an important step in understanding the functional significance of all TLDc domain-containing proteins in the cellular stress response and disease.
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The TLDc domain is a highly conserved c-terminal domain motif shared by a small family of proteins that protect against oxidative stress in the CNS, such as OXR1 and NCOA7.
“The TLDc domain is a highly conserved c-terminal domain motif shared by a small family of proteins that protect against oxidative stress in the CNS, such as oxidation resistance 1 (OXR1) and nuclear receptor coactivator 7 (NCOA7) [182].”
From ROS Generation in Microglia: Understanding Oxidative Stress and Inflammation in Neurodegenerative Disease (2020) · cites this paper as [182]
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- Authors
- Finelli, M. J., Sanchez-Pulido, L., Liu, K. X., Davies, K. E., & Oliver, P. L.
- Affiliations
- University of Oxford (GB)
- Corresponding author
- Oliver, Peter L.
- Venue
- Journal of Biological Chemistry
- Publisher
- Elsevier BV
- Published
- 2016-02
- Volume
- 291
- Issue
- 6
- Pages
- 2751-2763
- ISSN
- 0021-9258 (ISSN-L)
- PMC
- PMC4742741
- OpenAlex
- W2202850844
- Cited by
- 76
- References
- 59
- Citations by year
- 2026: 7 · 2025: 10 · 2024: 14 · 2023: 6 · 2022: 5 · 2021: 7 · 2020: 8 · 2019: 6 · 2018: 6 · 2017: 4 · 2016: 3
- FWCI
- 1.88
- Type
- journal article
- Language
- en
- Open access
- hybrid
- doi.org
- Licence
- Locations
- Journal of Biological Chemistry (publishedVersion, cc-by)
- PubMed Central (publishedVersion, cc-by)
- PubMed (publishedVersion)
- Repository@Nottingham (University of Nottingham) (publishedVersion, other-oa)
- Repository@Nottingham (University of Nottingham) (submittedVersion, cc-by)
- Funders
- Medical Research Council (MRC Functional Genomics Unit Programme Grant)
- European Research Council (311394)
- Topics
- Life Sciences › Biochemistry, Genetics and Molecular Biology › Molecular Biology › Redox biology and oxidative stress
- Health Sciences › Medicine › Neurology › Amyotrophic Lateral Sclerosis Research
- Health Sciences › Medicine › Epidemiology › Autophagy in Disease and Therapy
- Keywords
- OXR1, oxidative stress, neuroprotection, neurodegeneration, TBC1D24, amyotrophic lateral sclerosis, stress response
- MeSH terms
- Amyotrophic Lateral Sclerosis, Animals, Carrier Proteins, Cell Line, Disease Models, Animal, Nuclear Proteins, Protein Structure, Tertiary, Oxidative Stress, Neuroprotective Agents, Evolution, Molecular, GTPase-Activating Proteins, Amino Acid Motifs, Mitochondrial Proteins, Mice, INDEL Mutation
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