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Cited by 84Genetic Neurodegenerative DiseasesNeurogenesis and neuroplasticity mechanismsGenetics and Neurodevelopmental Disorders

Huntington disease oligodendrocyte maturation deficits revealed by single-nucleus RNAseq are rescued by thiamine-biotin supplementation

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Lim, Ryan G., Al-Dalahmah, Osama, Wu, Jie, Gold, Maxwell P., Reidling, Jack C., Tang, Guomei, Adam, Miriam, Dansu, David K. +21 more·2022·Nature Communications
DOI 10.1038/s41467-022-35388-xPMC PMC9772349
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Abstract

The complexity of affected brain regions and cell types is a challenge for Huntington’s disease (HD) treatment. Here we use single nucleus RNA sequencing to investigate molecular pathology in the cortex and striatum from R6/2 mice and human HD post-mortem tissue. We identify cell type-specific and -agnostic signatures suggesting oligodendrocytes (OLs) and oligodendrocyte precursors (OPCs) are arrested in intermediate maturation states. OL-lineage regulators OLIG1 and OLIG2 are negatively correlated with CAG length in human OPCs, and ATACseq analysis of HD mouse NeuN-negative cells shows decreased accessibility regulated by OL maturation genes. The data implicates glucose and lipid metabolism in abnormal cell maturation and identify PRKCE and Thiamine Pyrophosphokinase 1 ( TPK1 ) as central genes. Thiamine/biotin treatment of R6/1 HD mice to compensate for TPK1 dysregulation restores OL maturation and rescues neuronal pathology. Our insights into HD OL pathology spans multiple brain regions and link OL maturation deficits to abnormal thiamine metabolism.

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  • In the cerebral cortex of Huntington's disease patients, SGK1 protein expression is downregulated.

    “However, a more specific study has revealed that in the cerebral cortex of HD patients, SGK1 protein expression is downregulated”

    From Serum/glucocorticoid regulated kinase 1 (SGK1) in neurological disorders: pain or gain (2024) · cites this paper as Lim et al., 2022

    The Results section on causal network modeling reports that Sgk1 is downregulated in R6/2 mice. The Results section on human HD validation reports decreased cortical protein levels for PRKCE, not SGK1. Thus, the cited paper supports SGK1 downregulation in a mouse model but not the claimed decrease in SGK1 protein in patients’ cerebral cortex. (LLM, conf=0.85)

    Experimental — accuracy not yet measured

  • Detailed single-cell sequencing shows downregulation of SGK1 protein expression in oligodendrocytes in Huntington's disease.

    “Detailed single-cell sequencing has also shown a downregulation of SGK1 protein expression in oligodendrocytes”

    From Serum/glucocorticoid regulated kinase 1 (SGK1) in neurological disorders: pain or gain (2024) · cites this paper as Lim et al., 2022

    The Results section on causal network modeling reports that Sgk1 is downregulated in the oligodendrocyte/precursor-cell network of R6/2 Huntington disease mice. But single-nucleus RNA sequencing measures gene expression, not protein expression; the cited passage does not establish that SGK1 protein is reduced specifically in oligodendrocytes. (LLM, conf=0.88)

    Experimental — accuracy not yet measured

  • In Huntington's disease models, oligodendrocytes exhibit maturation and functional impairments associated with changes in SGK1 expression, particularly in myelination and maintenance.

    “In HD models, oligodendrocytes exhibit maturation and functional impairments (Wilton and Stevens, 2020), associated with changes in SGK1 expression, particularly in myelination and maintenance”

    From Serum/glucocorticoid regulated kinase 1 (SGK1) in neurological disorders: pain or gain (2024) · cites this paper as Lim et al., 2022

    The Results sections on R6/2 oligodendrocytes and causal network modeling report maturation and myelination-related impairments, and identify Sgk1 as downregulated in R6/2 mice. The Discussion describes a possible inability to turn over myelin components, but does not establish that SGK1 changes specifically impair myelination or myelin maintenance. The indexed passages have no page numbers. (LLM, conf=0.85)

    Experimental — accuracy not yet measured

  • The role of SGK1 in metabolic pathways and glucose uptake may be disrupted and potentially exacerbate the pathological process of Huntington's disease.

    “The role of SGK1 in metabolic pathways and glucose uptake may be disrupted and potentially exacerbate the pathological process of HD”

    From Serum/glucocorticoid regulated kinase 1 (SGK1) in neurological disorders: pain or gain (2024) · cites this paper as Lim et al., 2022

    In Results, under causal network modeling, the paper says Sgk1 regulates transporters involved in metabolic pathways and glucose uptake and is downregulated in R6/2 mice, indicating a potential loss of function in HD. It does not establish that this loss exacerbates HD pathology. (LLM, conf=0.82)

    Experimental — accuracy not yet measured

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Cited by

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Authors
Lim, R. G., Al-Dalahmah, O., Wu, J., Gold, M. P., Reidling, J. C., Tang, G., Adam, M., Dansu, D. K., Park, H.-J., Casaccia, P., Miramontes, R., Reyes-Ortiz, A. M., Lau, A., Hickman, R. A., Khan, F., Paryani, F., Tang, A., Ofori, K., Miyoshi, E., Michael, N., McClure, N., Flowers, X. E., Vonsattel, J. P., Davidson, S., Menon, V., Swarup, V., Fraenkel, E., Goldman, J. E., & Thompson, L. M.
Affiliations
  • University of California, Irvine (US)
  • Massachusetts Institute of Technology (US)
  • Columbia University Irving Medical Center (US)
  • City University of New York (US)
  • Princeton University (US)
Corresponding author
  • Goldman, James E.
  • Thompson, Leslie M.
ORCID
Venue
Nature Communications
Publisher
Springer Science and Business Media LLC
Published
2022-12-21
Volume
13
Issue
1
Pages
7791-7791
ISSN
2041-1723 (ISSN-L)
DOI
10.1038/s41467-022-35388-x
PMC
PMC9772349
OpenAlex
W4313340421
Cited by
84
References
15
Citations by year
2026: 28 · 2025: 27 · 2024: 17 · 2023: 11 · 2022: 1
FWCI
6.19
Type
journal article
Language
en
Open access
Licence
Locations
  • Nature Communications (publishedVersion, cc-by)
  • PubMed Central (publishedVersion, cc-by)
  • PubMed (publishedVersion)
  • DSpace@MIT (Massachusetts Institute of Technology) (submittedVersion, cc-by-nc)
  • eScholarship (California Digital Library) (submittedVersion, cc-by)
  • RePEc: Research Papers in Economics (submittedVersion)
  • DOAJ (DOAJ: Directory of Open Access Journals) (submittedVersion, cc-by-sa)
Funders
U.S. Department of Health & Human Services | NIH | National Institute of Neurological Disorders and Stroke (R01 NS089076, R35 NS116872)
Topics
  • Life Sciences › Neuroscience › Cellular and Molecular Neuroscience › Genetic Neurodegenerative Diseases
  • Life Sciences › Neuroscience › Developmental Neuroscience › Neurogenesis and neuroplasticity mechanisms
  • Life Sciences › Biochemistry, Genetics and Molecular Biology › Genetics › Genetics and Neurodevelopmental Disorders
Keywords
Huntington's disease, oligodendrocyte maturation, oligodendrocyte precursor cells, protein kinase C epsilon, Olig1, Olig2, CAG repeat length, thiamine metabolism, glucose metabolism, lipid metabolism, chromatin accessibility, R6/2 mice, cortical pathology, neuronal pathology
MeSH terms
Animals, Biotin, Disease Models, Animal, Humans, Huntington Disease, Mice, Transgenic, Nerve Tissue Proteins, Oligodendroglia, Thiamine, Solitary Nucleus, Dietary Supplements, Mice

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