Highly conserved brain vascular receptor ALPL mediates transport of engineered AAV vectors across the blood-brain barrier

Mol Ther. 2025 Aug 6;33(8):3902-3916. doi: 10.1016/j.ymthe.2025.04.046. Epub 2025 May 8.

Abstract

Delivery of systemically administered therapeutics to the central nervous system (CNS) is restricted by the blood-brain barrier (BBB). Bioengineered adeno-associated virus (AAV) capsids have been shown to penetrate the BBB with great efficacy in mouse and non-human primate models, but their translational potential is often limited by species selectivity and undefined mechanisms of action. Here, we apply our RNA-guided TRACER AAV capsid evolution platform to generate VCAP-102, an AAV9 variant with markedly increased brain tropism following intravenous delivery in both rodents and primates. Relative to AAV9, VCAP-102 demonstrates 20- to 400-fold increased gene transfer across multiple brain regions. We identify alkaline phosphatase (ALPL) as the primary receptor used by VCAP-102 to cross the BBB and demonstrate that direct binding of VCAP-102 to human ALPL can initiate receptor-mediated transcytosis in a cell barrier model. Our work identifies VCAP-102 as a cross-species CNS gene delivery vector with a strong potential for clinical translation and establishes ALPL as a brain delivery shuttle capable of efficient BBB transport to maximize CNS delivery of biotherapeutics.

Keywords: AAV; ALPL; blood-brain barrier; brain; directed evolution; non-human primate.

MeSH terms

  • Alkaline Phosphatase* / genetics
  • Alkaline Phosphatase* / metabolism
  • Animals
  • Bioengineering
  • Blood-Brain Barrier* / metabolism
  • Capsid
  • Dependovirus / genetics
  • Directed Molecular Evolution
  • Genetic Vectors* / administration & dosage
  • Genetic Vectors* / pharmacokinetics
  • HEK293 Cells
  • Humans
  • Injections, Intravenous
  • Mice
  • Mice, Inbred BALB C
  • Mice, Inbred C57BL
  • Transduction, Genetic

Substances

  • ALPL protein, human
  • ALPL protein, mouse
  • Alkaline Phosphatase