ArticleHuman molecular genetics2015
Reduced PLP2 expression increases ER-stress-induced neuronal apoptosis and risk for adverse neurological outcomes after hypoxia ischemia injury.
Article in Human molecular genetics, 2015. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 14 papers.
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Who cites it
14 citing papers in PubMed, 21 citations in OpenAlex.
- Therapeutic potential of TMSC-Exo for non-alcoholic fatty liver disease using the liver-on-a-chip model.Journal of translational internal medicine · 2026Article
- Nanoparticle-mediated sodium butyrate delivery for repairing hypoxic-ischemic brain injury in premature infants.Materials today. Bio · 2025Article
- ST8Sia2 polysialyltransferase protects against infection by Trypanosoma cruzi.PLoS neglected tropical diseases · 2024Article
- Label-free quantitative proteomic profiling reveals differential plasma protein expression in patients with obesity after treatment with liraglutide.Frontiers in molecular biosciences · 2024Article
- Reduced Proteolipid Protein 2 promotes endoplasmic reticulum stress-related apoptosis and increases drug sensitivity in acute myeloid leukemia.Molecular biology reports · 2023Article
- MicroRNA-765 is upregulated in myelodysplastic syndromes and induces apoptosis via PLP2 inhibition in leukemia cells.Blood research · 2023Article
- Celecoxib-Dependent Neuroprotection in a Rat Model of Transient Middle Cerebral Artery Occlusion (tMCAO) Involves Modifications in Unfolded Protein Response (UPR) and Proteasome.Molecular neurobiology · 2021Article
- Reduced expression of proteolipid protein 2 increases ER stress-induced apoptosis and autophagy in glioblastoma.Journal of cellular and molecular medicine · 2020Article
- PLP2 Expression as a Prognostic and Therapeutic Indicator in High-Risk Multiple Myeloma.BioMed research international · 2020Article
- Biliverdin administration regulates the microRNA-mRNA expressional network associated with neuroprotection in cerebral ischemia reperfusion injury in rats.International journal of molecular medicine · 2019Article
- miR-124-3p is a chronic regulator of gene expression after brain injury.Cellular and molecular life sciences : CMLS · 2018Article
- Proteolipid Protein 2 Overexpression Indicates Aggressive Tumor Behavior and Adverse Prognosis in Human Gliomas.International journal of molecular sciences · 2018Article
- Article
- CircRNA expression pattern and circRNA-miRNA-mRNA network in the pathogenesis of nonalcoholic steatohepatitis.Oncotarget · 2016Article
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3 authors at 2 institutions in 1 country.
Funding
Abstract
Both genetic and environmental factors contribute to the development of intellectual disability (ID). Previously, we identified a promoter variant (-113C>A) in PLP2 (proteolipid protein 2) that results in an ∼4-fold reduction of transcript and protein and is overly represented in males with X-linked ID (XLID). The functional connection between reduced PLP2 expression and increased risk to XLID is unknown. To investigate the pathophysiological mechanisms, we studied a Plp2-loss-of-function murine model and fibroblasts from XLID patients hemizygous for PLP2-(-113C>A). We found that Plp2-deficient mouse embryonic fibroblast and human fibroblasts carrying PLP2-(-113C>A) have similarly defective endoplasmic reticulum (ER) trafficking, increased basal ER stress and exaggerated susceptibility to inducers of ER stress. Plp2-deficient mice show increased neuronal death to ER stress and hypoxia in vitro and in a neonatal hypoxia-ischemia model in vivo. Finally, we provide evidence that up-regulation of PLP2 directly promotes resistance to ER stressors. Results of our studies support the hypothesis that reduced PLP2 expression increase susceptibility of neurons to environmental ER stressors such as hypoxia and ischemia and that increased apoptosis and neuronal death contribute to the risks to ID in humans.
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