ArticleMolecular psychiatry2019
Familial t(1;11) translocation is associated with disruption of white matter structural integrity and oligodendrocyte-myelin dysfunction.
Article in Molecular psychiatry, 2019. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 19 papers.
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Who cites it
19 citing papers in PubMed, 24 citations in OpenAlex.
- Myelin sheath lengths in the central nervous system scale to axon diameter via oligodendroglial Piezo1.PLoS biology · 2026Article
- The EH-binding protein EHBP1 operates in a ciliary functional module affected by INPP5E dysfunction.Journal of cell science · 2026Article
- Psychiatric Implications of Genetic Variations in Oligodendrocytes: Insights from hiPSC Models.Life (Basel, Switzerland) · 2025Review
- Canonical and non-canonical roles of oligodendrocyte precursor cells in mental disorders.Npj mental health research · 2025Review
- Selective suppression of oligodendrocyte-derived amyloid beta rescues neuronal dysfunction in Alzheimer's disease.PLoS biology · 2024Article
- Pathological oligodendrocyte precursor cells revealed in human schizophrenic brains and trigger schizophrenia-like behaviors and synaptic defects in genetic animal model.Molecular psychiatry · 2022Article
- Abnormalities in the migration of neural precursor cells in familial bipolar disorder.Disease models & mechanisms · 2022Article
- The ciliary gene INPP5E confers dorsal telencephalic identity to human cortical organoids by negatively regulating Sonic hedgehog signaling.Cell reports · 2022Article
- Heterogeneity and regulation of oligodendrocyte morphology.Frontiers in cell and developmental biology · 2022Review
- Protocol for magnetic resonance imaging acquisition, quality assurance, and quality check for the Accelerator program for Discovery in Brain disorders using Stem cells.International journal of methods in psychiatric research · 2021Article
- iPSC-derived myelinoids to study myelin biology of humans.Developmental cell · 2021Article
- Functional brain defects in a mouse model of a chromosomal t(1;11) translocation that disrupts DISC1 and confers increased risk of psychiatric illness.Translational psychiatry · 2021Article
- Mitochondrial bioenergetic deficits in C9orf72 amyotrophic lateral sclerosis motor neurons cause dysfunctional axonal homeostasis.Acta neuropathologica · 2021Article
- Transactive response DNA-binding protein-43 proteinopathy in oligodendrocytes revealed using an induced pluripotent stem cell model.Brain communications · 2021Article
- Altered Expression of Ion Channels in White Matter Lesions of Progressive Multiple Sclerosis: What Do We Know About Their Function?Frontiers in cellular neuroscience · 2021Review
- Development of the Entorhinal Cortex Occurs via Parallel Lamination During Neurogenesis.Frontiers in neuroanatomy · 2021Article
- Staining and Quantitative Analysis of Myelinating Oligodendrocytes in the Mouse Grey Matter.Bio-protocol · 2020Article
- What neuroscience has already done for us.BJPsych bulletin · 2020Article
- Altered White Matter and Layer VIb Neurons in HeterozygousFrontiers in neuroanatomy · 2020Article
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Authors and funding
24 authors at 8 institutions in 4 countries.
Funding
Abstract
Although the underlying neurobiology of major mental illness (MMI) remains unknown, emerging evidence implicates a role for oligodendrocyte-myelin abnormalities. Here, we took advantage of a large family carrying a balanced t(1;11) translocation, which substantially increases risk of MMI, to undertake both diffusion tensor imaging and cellular studies to evaluate the consequences of the t(1;11) translocation on white matter structural integrity and oligodendrocyte-myelin biology. This translocation disrupts among others the DISC1 gene which plays a crucial role in brain development. We show that translocation-carrying patients display significant disruption of white matter integrity compared with familial controls. At a cellular level, we observe dysregulation of key pathways controlling oligodendrocyte development and morphogenesis in induced pluripotent stem cell (iPSC) derived case oligodendrocytes. This is associated with reduced proliferation and a stunted morphology in vitro. Further, myelin internodes in a humanized mouse model that recapitulates the human translocation as well as after transplantation of t(1;11) oligodendrocyte progenitors were significantly reduced when compared with controls. Thus we provide evidence that the t(1;11) translocation has biological effects at both the systems and cellular level that together suggest oligodendrocyte-myelin dysfunction.
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