Understanding and Targeting Pulmonary Arteriovenous Malformations Using Repurposed Drugs

Abstract

Hereditary hemorrhagic telangiectasia (HHT) is a genetic disease characterized by multiple arteriovenous malformations (AVMs) which are direct connections between arteries and veins, bypassing the capillary bed. Severe epistaxis (nosebleeds) is the most common symptom, yet visceral AVMs in the brain (1-10 percent), lung (15-45 percent), liver and gastrointestinal tract cause significant morbidity and mortality due to embolic stroke, cerebral abscess, migraines, hemorrhagic stroke, seizures and life-threatening bleeding complications. In order to reduce the morbidity and mortality associated with HHT, we need a better understanding of HHT development and novel treatment approaches. Our aims are: Aim 1: To understand the cellular and molecular mechanisms of PAVM development in mice and to identify the cell behaviors and populations that give rise to PAVMs. Aim 2: To identify and target pathological downstream signaling in endothelial cells derived from iPSCs (iPSC-ECs) from HHT patients with visceral AVMs. Aim 3: To target pathological downstream signaling with repurposed drugs to prevent and reverse PAVMs in the mouse model. The short-term impact will be a better understanding of how AVMs form in the lung and potentially in other organs. The long-term impact will be the identification of potential novel treatments for AVMs.

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Document Details

Document Type
Technical Report
Publication Date
Aug 01, 2021
Accession Number
AD1148040

Entities

People

  • Astrid Gillich
  • David Stevenson
  • Edda Spiekerkoetter
  • Ross Metzger

Organizations

  • Stanford University

Tags

DTIC Thesaurus Topics

  • Arteries
  • Biomedical Research
  • Blood
  • Cells
  • Confocal Microscopy
  • Covid-19
  • Culture Techniques
  • Department Of Defense
  • Diseases And Disorders
  • Electronic Mail
  • Endothelial Cells
  • Gastrointestinal Tract
  • Genetic Diseases
  • Genetics
  • Identification
  • Imaging Techniques
  • Institutional Review Board
  • Medical Personnel
  • Microvessels
  • Rna Sequence Analysis
  • Stem Cells
  • Three Dimensional
  • Vascular Diseases
  • Vascular Malformations

Fields of Study

  • Medicine

Readers

  • Molecular and Cellular Biology
  • Trauma Surgery or Emergency Medicine.

Technology Areas

  • Biotechnology