Search bioRxiv⌕ Search

Biology subjects

Prevot, V.

Publications and source records attributed to Prevot, V..

7 recordsLinked to original sources

Ciliary sensing in tanycytes couples nutrient availability to metabolic regulation

Body homeostasis relies on accurate communication between the brain and the periphery. Disruption of this communication can contribute to disease. Tanycytes are located at the interface of the cerebrospinal fluid (CSF), bloodstream, and hypothalamus, where they sense circulating nutrients and regulate neuroendocrine axes and metabolism. However, the mechanisms by which they sense CSF signals remain largely unclear. Recent evidence that tanycytes possess primary cilia -- key sensory organelles -- led us to hypothesize that tanycytic cilia function as sensory antennae that detect metabolic cues in the CSF. Here, we demonstrate that tanycytic cilia exhibit distinct morphologies across subtypes and physiological states. They respond dynamically to hormonal and nutrient availability; notably, excess oleic acid shortens cilia, promotes lipid droplet accumulation, and reduces Ca{superscript 2} responses to ATP and glucose. Disrupting cilia via knockdown of intraflagellar transport (IFT) genes produced similar defects and impaired autophagy. Finally, selective Ift88 knockout in tanycytes increased body weight and reduced thermogenic activity in female mice. These findings identify tanycytic cilia as key sensors regulating energy balance.

neuroscience↗

Gαq/11 signaling counteracts endothelial dysfunction in the brain and protects cognition in aged mice

BackgroundCerebral small vessel disease (cSVD) is a major cause of stroke and dementia, and is associated with increased blood-brain barrier permeability, neuroinflammation, and endothelial dysfunction. Endothelial Gq/11 proteins are involved in vascular tone regulation and have been shown to affect capillary blood flow in the brain. Since factors downstream of activated Gq/11 proteins, such as endothelial NO synthase (eNOS) activity are discussed in cSVD, we wondered whether the brain endothelial Gq/11 signalling pathway might influence cSVD-related pathology. MethodsHere, we generated mice carrying a brain endothelial-specific deletion of the Gq/11 signalling and characterised these mice using different imaging and staining techniques, as well as behaviour tests measuring cognition in adult and aged mice. Immunoblots, electrophysiology, perfusion measurements, and in vitro experiments complemented those techniques. FindingsThe brain endothelial Gq/11 signalling pathway preserves normal vascular reactivity, and its loss resembles mild endothelial dysfunction in the brain. While the vessel structure was maintained in adult mice, a deletion of the Gq/11 signalling led to capillary rarefaction and blood-brain barrier disruption in aged mice. These effects were accompanied by disturbed VEGF signalling and an increase in senescence markers and oxidative stress in the vasculature, culminating in cognitive impairment with increased tau phosphorylation in the cortex and hippocampus, and decreased myelination in the white matter. InterpretationThese findings reflect the main hallmarks of cSVD and demonstrate a protective role of Gq/11 in endothelial cells in ageing. Furthermore, our results show that the combination of cerebral endothelial dysfunction and ageing accelerates cognitive impairment. FundingResearch was supported by grants from the European Research Council (2019-WATCH-810331 to R.N., V.P., and M.S.), the Deutsche Forschungsgemeinschaft (SCHW 416/12-1 to M.S.; WE 6456/1-1 to J.W.; GRK1957 to M.S., H.M.-F., and J.W.), the institutional priority program MI-VascAD of the University of Lubeck (to H.M.-F. and J.W.), and the Marie-Sklodowska-Curie European Unions Horizon 2020 research program (ENTRAIN-813294 to M.S. and J.W.). RESEARCH IN CONTEXTO_ST_ABSEvidence before this studyC_ST_ABSCerebral small vessel disease (cSVD) is associated with advanced age, hypertension, and metabolic diseases. It is considered causal for a substantial part of stroke and dementia cases. Endothelial dysfunction is described as being present in all of the diseases mentioned, and it is discussed to play a role in the development of cSVD. However, most of the evidence is correlational rather than causal, and studies are usually carried out on subjects with an underlying disease for which endothelial dysfunction is a side effect. Added value of this studyHere, we describe a novel brain endothelial signalling pathway that protects against endothelial dysfunction and loss of cerebrovascular reactivity. Without Gq/11 signalling exclusively in the brain endothelium, mice show an impaired ability to increase brain perfusion in response to vasodilatory stimuli, such as neuronal activity. These mice, combined with an older age, develop vessel rarefaction and cognitive dysfunction, which can be explained by increased tau phosphorylation and white matter alteration. Implications of all the available evidenceThese findings, in the context of the existing studies, suggest that endothelial dysfunction per se is sufficient to induce cognitive impairment. They imply that the diagnoses and treatments for diseases associated with cognitive impairment should focus more on a dysfunctional endothelium. The Gq/11 signalling pathway offers a new approach to address treatment endeavours in cSVD and, more broadly, in all diseases associated with a higher risk of vascular dementia and stroke.

neuroscience↗

Investigation of a Novel Mouse Model of Prader-Willi Syndrome with Invalidation of Necdin and Magel2

Prader-Willi syndrome (PWS) is a multigenic disorder caused by the loss of seven contiguous paternally expressed genes. Mouse models with inactivation of all PWS genes are lethal. Knockout (KO) mouse models for each candidate gene were generated, but they lack the functional interactions between PWS genes. Here, we revealed an interplay between Necdin and Magel2 "PWS" genes and generated a novel mouse model (named "Madin") with a deletion including both genes. A subset of Madin KO mice showed neonatal lethality. Behaviorally, surviving mutant mice exhibited sensory delays during infancy and alterations in social exploration at adulthood. Madin KO mice had a lower body weight before weaning, persisting after weaning in males only, with reduced fat mass and improved glucose tolerance. Delayed sexual maturation and altered timing of puberty onset were observed in mutant mice. Adult Madin KO mice displayed increased ventilation and a persistent increase in apneas following a hypercapnic challenge. Transcriptomics analyses revealed a dysregulation of key circadian genes and alterations of genes associated with axonal function that were also found in the hypothalamus of patients with PWS. At neuroanatomical levels, we report an impaired maturation of oxytocin neurons and a disrupted development of melanocortin circuits. Together, these data indicate that the Madin KO mouse is a reliable and more genetically relevant model for the study of PWS.

systems biology↗

LRP1 mediates leptin transport by coupling with the short-form leptin receptor in the choroid plexus

Adipocyte-derived leptin enters the brain to exert its anorexigenic action, yet its transport mechanism is poorly understood. Here we report that LRP1 (low-density lipoprotein receptor-related protein-1) mediates the transport of leptin across the blood-CSF barrier in Foxj1 expressing cells highly enriched at the choroid plexus (ChP), coupled with the short-form leptin receptor, and LRP1 deletion from ependymocytes and ChP cells leads to leptin resistance and hyperphagia, causing obesity. Thus, LRP1 in epithelial cells is a principal regulator of leptin transport in the brain.

neuroscience↗

Gene therapy targeting the blood-brain barrier improves neurological symptoms in a model of genetic MCT8 deficiency

The solute carrier monocarboxylate transporter 8 (MCT8) transports the thyroid hormones thyroxine and tri-iodothyronine (T3) across cell membranes. MCT8 gene deficiency, termed Allan-Herndon-Dudley syndrome, is an important cause of X-linked intellectual and motor disability. As no treatment of the neurological symptoms is available yet, we tested a gene replacement therapy in Mct8- and Oatp1c1-deficient mice as a well-established model of the disease. Here, we report that targeting brain endothelial cells for Mct8 expression by intravenously injecting the vector AAV-BR1-Mct8 increased T3 levels in the brain and ameliorated morphological and functional parameters associated with the disease. Importantly, the therapy resulted in a long-lasting improvement in motor coordination. Thus, the data support the concept that MCT8 mediates the transport of thyroid hormones into the brain and indicate that a readily accessible vascular target can help overcome the consequences of the severe disability associated with MCT8 deficiency.

neuroscience↗

The cryptic gonadotropin-releasing hormone neuronal system of human basal ganglia

Human reproduction is controlled by [~]2,000 hypothalamic gonadotropin-releasing hormone (GnRH) neurons. Here we report the discovery and characterization of additional 150-200,000 GnRH-synthesizing cells in the human basal ganglia and basal forebrain. Extrahypothalamic GnRH neurons were cholinergic. Though undetectable in adult rodents, the GnRH-GFP transgene was expressed transiently by caudate-putamen cholinergic interneurons in newborn transgenic mice. In slice electrophysiological studies, GnRH inhibited these interneurons via GnRHR1 autoreceptors. Whole-transcriptome analysis of cholinergic interneurons and medium spiny projection neurons laser-microdissected from the human putamen confirmed selective expression of GNRH1 and GNRHR1 autoreceptors in cholinergic cells and uncovered the detailed transcriptome profile and molecular connectome of these two cell types. Higher- order non-reproductive functions regulated by GnRH under physiological conditions in the human basal ganglia and basal forebrain require clarification. GnRH/GnRHR1 signaling as a potential therapeutic target in the treatment of neurodegenerative disorders affecting cholinergic neurocircuitries, including Parkinsons and Alzheimers diseases, needs to be explored.

cell biology↗

The hypothalamus as a hub for putative SARS-CoV-2 brain infection

Most patients with COVID-19, caused by severe acute respiratory syndrome coronavirus 2 (SARS-CoV-2), display neurological symptoms, and respiratory failure in certain cases could be of extra-pulmonary origin. Hypothalamic neural circuits play key roles in sex differences, diabetes, hypertension, obesity and aging, all risk factors for severe COVID-19, besides being connected to olfactory/gustative and brainstem cardiorespiratory centers. Here, human brain gene-expression analyses and immunohistochemistry reveal that the hypothalamus and associated regions express angiotensin-converting enzyme 2 and transmembrane proteinase, serine 2, which mediate SARS-CoV-2 cellular entry, in correlation with genes or pathways involved in physiological functions or viral pathogenesis. A post-mortem patient brain shows viral invasion and replication in both the olfactory bulb and the hypothalamus, while animal studies indicate that sex hormones and metabolic diseases influence this susceptibility.

neuroscience↗