ArticleJournal of inherited metabolic disease2019
Desmosterolosis and desmosterol homeostasis in the developing mouse brain.
Article in Journal of inherited metabolic disease, 2019. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 18 papers.
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Who cites it
18 citing papers in PubMed, 32 citations in OpenAlex.
- Physiological Functions of Side-Chain-Retaining Sterols in the Brain and Their Roles in Neurodegenerative Diseases.Metabolites · 2026Review
- Dysregulated lipid metabolism and hypomyelination in postnatal peroxisome-deficientFrontiers in molecular neuroscience · 2026Article
- Dishevelled localization and function are differentially regulated by structurally distinct sterols.iScience · 2025Article
- Interaction Between DHCR24 and hsa_circ_0015335 Facilitates Cognitive Impairment in Cerebral Small Vessel Disease Patients.CNS neuroscience & therapeutics · 2024Article
- Behavioral and Metabolic Effects of ABCG4 KO in the APPJournal of molecular neuroscience : MN · 2024Article
- Aziridination-Assisted Mass Spectrometry of Nonpolar Sterol Lipids with Isomeric Resolution.Journal of the American Society for Mass Spectrometry · 2023Article
- An iPSC-derived astrocyte model of fragile X syndrome exhibits dysregulated cholesterol homeostasis.Communications biology · 2023Article
- DHCR24 reverses Alzheimer's disease-related pathology and cognitive impairment via increasing hippocampal cholesterol levels in 5xFAD mice.Acta neuropathologica communications · 2023Article
- Individual and simultaneous treatment with antipsychotic aripiprazole and antidepressant trazodone inhibit sterol biosynthesis in the adult brain.Journal of lipid research · 2022Article
- The role of DHCR24 in the pathogenesis of AD: re-cognition of the relationship between cholesterol and AD pathogenesis.Acta neuropathologica communications · 2022Review
- Cholesterol in Brain Development and Perinatal Brain Injury: More than a Building Block.Current neuropharmacology · 2022Review
- Sterol Biosynthesis Inhibition in Pregnant Women Taking Prescription Medications.ACS pharmacology & translational science · 2021Article
- Prescription Medications Alter Neuronal and Glial Cholesterol Synthesis.ACS chemical neuroscience · 2021Article
- Maternal cariprazine exposure inhibits embryonic and postnatal brain cholesterol biosynthesis.Molecular psychiatry · 2020Article
- The metabolic changes in the hippocampus of an atherosclerotic rat model and the regulation of aerobic training.Metabolic brain disease · 2020Article
- Amiodarone Alters Cholesterol Biosynthesis through Tissue-Dependent Inhibition of Emopamil Binding Protein and Dehydrocholesterol Reductase 24.ACS chemical neuroscience · 2020Article
- Article
- Cholesterol Biosynthesis and Uptake in Developing Neurons.ACS chemical neuroscience · 2019Article
Corrections and comments
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Authors and funding
5 authors at 3 institutions in 1 country.
Funding
Abstract
Cholesterol serves as a building material for cellular membranes and plays an important role in cellular metabolism. The brain relies on its own cholesterol biosynthesis, which starts during embryonic development. Cholesterol is synthesized from two immediate precursors, desmosterol and 7-dehydrocholesterol (7-DHC). Mutations in the DHCR24 enzyme, which converts desmosterol into cholesterol, lead to desmosterolosis, an autosomal recessive developmental disorder. In this study, we assessed the brain content of desmosterol, 7-DHC, and cholesterol from development to adulthood, and analyzed the biochemical, molecular, and anatomical consequences of Dhcr24 mutations on the sterol profile in a mouse model of desmosterolosis and heterozygous Dhcr24
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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.