ArticleJCI insight2020
Deficiency of Notch signaling in pericytes results in arteriovenous malformations.
Article in JCI insight, 2020. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 43 papers.
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Who cites it
43 citing papers in PubMed, 64 citations in OpenAlex.
- Friend or foe? Glial-vascular interactions in health and neurodegenerative disease.Pharmacological reviews · 2026Review
- Towards precision medicine for brain arteriovenous malformations.The Journal of clinical investigation · 2026Review
- Pulsatile flow dynamics maintain pulmonary artery architecture.JCI insight · 2026Article
- Bmp9 regulates Notch signaling and the temporal dynamics of angiogenesis via Lunatic Fringe.Developmental cell · 2026Article
- Development of the blood-brain barrier.Development (Cambridge, England) · 2026Review
- A Transcriptional Atlas of Endothelial Cell Zonation Along the Pulmonary Vascular Tree.bioRxiv : the preprint server for biology · 2026Article
- Flow-induced Klf4-Akt signaling links EC cycling to mural cell defects in arterial-venous malformations.Theranostics · 2026Article
- Multifaceted Role of Notch Signaling in Vascular Health and Diseases.Biomedicines · 2025Review
- The Triad of Blood-Brain Barrier Integrity: Endothelial Cells, Astrocytes, and Pericytes in Perinatal Stroke Pathophysiology.International journal of molecular sciences · 2025Review
- Article
- Inhibition of Angiopoietin-2 rescues sporadic brain arteriovenous malformations by reducing pericyte loss.Angiogenesis · 2024Article
- A non-genetic model of vascular shunts informs on the cellular mechanisms of formation and resolution of arteriovenous malformations.Cardiovascular research · 2024Article
- Review
- Similarities and differences between brain and skin GNAQ p.R183Q driven capillary malformations.Angiogenesis · 2024Article
- Similarities and differences between brain and skinbioRxiv : the preprint server for biology · 2024Article
- Notch signaling regulates UNC5B to suppress endothelial proliferation, migration, junction activity, and retinal plexus branching.Scientific reports · 2024Article
- Endothelial PDGF-D contributes to neurovascular protection after ischemic stroke by rescuing pericyte functions.Cellular and molecular life sciences : CMLS · 2024Article
- The Notch-PDGFRβ axis suppresses brown adipocyte progenitor differentiation in early post-natal mice.Developmental cell · 2024Article
- Villus myofibroblasts are developmental and adult progenitors of mammalian gut lymphatic musculature.Developmental cell · 2024Article
- Role of endothelial PDGFB in arterio-venous malformations pathogenesis.Angiogenesis · 2024Article
Corrections and comments
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Authors and funding
4 authors.
Funding
Abstract
Arteriovenous malformations (AVMs) are high-flow lesions directly connecting arteries and veins. In the brain, AVM rupture can cause seizures, stroke, and death. Patients with AVMs exhibit reduced coverage of the vessels by pericytes, the mural cells of microvascular capillaries; however, the mechanism underlying this pericyte reduction and its association with AVM pathogenesis remains unknown. Notch signaling has been proposed to regulate critical pericyte functions. We hypothesized that Notch signaling in pericytes is crucial to maintain pericyte homeostasis and prevent AVM formation. We inhibited Notch signaling specifically in perivascular cells and analyzed the vasculature of these mice. The retinal vessels of mice with deficient perivascular Notch signaling developed severe AVMs, together with a significant reduction in pericytes and vascular smooth muscle cells (vSMC) in the arteries, while vSMCs were increased in the veins. Vascular malformations and pericyte loss were also observed in the forebrain of embryonic mice deficient for perivascular Notch signaling. Moreover, the loss of Notch signaling in pericytes downregulated Pdgfrb levels and increased pericyte apoptosis, pointing to a critical role for Notch in pericyte survival. Overall, our findings reveal a mechanism of AVM formation and highlight the Notch signaling pathway as an essential mediator in this process.
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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.