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The GPI-Linked Protein LY6A Drives AAV-PHP.B Transport across the Blood-Brain Barrier
Journal article   Open access   Peer reviewed

The GPI-Linked Protein LY6A Drives AAV-PHP.B Transport across the Blood-Brain Barrier

Juliette Hordeaux, Yuan Yuan, Peter M Clark, Qiang Wang, R Alexander Martino, Joshua J Sims, Peter Bell, Angela Raymond, William L Stanford and James M Wilson
Molecular therapy, v 27(5), pp 912-921
08 May 2019
PMID: 30819613
url
http://www.cell.com/article/S1525001619300541/pdfView
Published, Version of Record (VoR) Open
url
https://doi.org/10.1016/j.ymthe.2019.02.013View
Published, Version of Record (VoR) Open

Abstract

Animals Antigens, Ly - genetics Antigens, Ly - pharmacology Biological Transport - genetics Blood-Brain Barrier - metabolism Brain - drug effects Brain - pathology Dependovirus - genetics Gene Transfer Techniques Genetic Therapy Genetic Vectors - genetics Genetic Vectors - therapeutic use Glycosylphosphatidylinositols - genetics Hematopoiesis - genetics Humans Membrane Proteins - genetics Membrane Proteins - pharmacology Mice Neurons - drug effects Neurons - pathology Whole Exome Sequencing
Efficient delivery of gene therapy vectors across the blood-brain barrier (BBB) is the holy grail of neurological disease therapies. A variant of the neurotropic vector adeno-associated virus (AAV) serotype 9, called AAV-PHP.B, was shown to very efficiently deliver transgenes across the BBB in C57BL/6J mice. Based on our recent observation that this phenotype is mouse strain dependent, we used whole-exome sequencing-based genetics to map this phenotype to a specific haplotype of lymphocyte antigen 6 complex, locus A (Ly6a) (stem cell antigen-1 [Sca-1]), which encodes a glycosylphosphatidylinositol (GPI)-anchored protein whose function had been thought to be limited to the biology of hematopoiesis. Additional biochemical and genetic studies definitively linked high BBB transport to the binding of AAV-PHP.B with LY6A (SCA-1). These studies identify, for the first time, a ligand for this GPI-anchored protein and suggest a role for it in BBB transport that could be hijacked by viruses in natural infections or by gene therapy vectors to treat neurological diseases.

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Collaboration types
Domestic collaboration
International collaboration
Web of Science research areas
Biotechnology & Applied Microbiology
Genetics & Heredity
Medicine, Research & Experimental
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