A brain microvasculature endothelial cell‐specific viral vector with the potential to treat neurovascular and neurological diseases

Abstract Gene therapy critically relies on vectors that combine high transduction efficiency with a high degree of target specificity and that can be administered through a safe intravenous route. The lack of suitable vectors, especially for gene therapy of brain disorders, represents a major obstac...

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Main Authors: Jakob Körbelin, Godwin Dogbevia, Stefan Michelfelder, Dirk A Ridder, Agnes Hunger, Jan Wenzel, Henning Seismann, Melanie Lampe, Jacqueline Bannach, Manolis Pasparakis, Jürgen A Kleinschmidt, Markus Schwaninger, Martin Trepel
Format: Article
Language:English
Published: Springer Nature 2016-04-01
Series:EMBO Molecular Medicine
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Online Access:https://doi.org/10.15252/emmm.201506078
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author Jakob Körbelin
Godwin Dogbevia
Stefan Michelfelder
Dirk A Ridder
Agnes Hunger
Jan Wenzel
Henning Seismann
Melanie Lampe
Jacqueline Bannach
Manolis Pasparakis
Jürgen A Kleinschmidt
Markus Schwaninger
Martin Trepel
author_facet Jakob Körbelin
Godwin Dogbevia
Stefan Michelfelder
Dirk A Ridder
Agnes Hunger
Jan Wenzel
Henning Seismann
Melanie Lampe
Jacqueline Bannach
Manolis Pasparakis
Jürgen A Kleinschmidt
Markus Schwaninger
Martin Trepel
author_sort Jakob Körbelin
collection DOAJ
description Abstract Gene therapy critically relies on vectors that combine high transduction efficiency with a high degree of target specificity and that can be administered through a safe intravenous route. The lack of suitable vectors, especially for gene therapy of brain disorders, represents a major obstacle. Therefore, we applied an in vivo screening system of random ligand libraries displayed on adeno‐associated viral capsids to select brain‐targeted vectors for the treatment of neurovascular diseases. We identified a capsid variant showing an unprecedented degree of specificity and long‐lasting transduction efficiency for brain microvasculature endothelial cells as the primary target of selection. A therapeutic vector based on this selected viral capsid was used to markedly attenuate the severe cerebrovascular pathology of mice with incontinentia pigmenti after a single intravenous injection. Furthermore, the versatility of this selection system will make it possible to select ligands for additional in vivo targets without requiring previous identification of potential target‐specific receptors.
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spelling doaj-art-b52f1692373e499a82e3aeb7fb5c9aef2025-08-20T03:06:00ZengSpringer NatureEMBO Molecular Medicine1757-46761757-46842016-04-018660962510.15252/emmm.201506078A brain microvasculature endothelial cell‐specific viral vector with the potential to treat neurovascular and neurological diseasesJakob Körbelin0Godwin Dogbevia1Stefan Michelfelder2Dirk A Ridder3Agnes Hunger4Jan Wenzel5Henning Seismann6Melanie Lampe7Jacqueline Bannach8Manolis Pasparakis9Jürgen A Kleinschmidt10Markus Schwaninger11Martin Trepel12Hubertus Wald Cancer Center, Department of Oncology and Hematology, University Medical Center Hamburg‐EppendorfInstitute of Experimental and Clinical Pharmacology and Toxicology, University of LübeckHubertus Wald Cancer Center, Department of Oncology and Hematology, University Medical Center Hamburg‐EppendorfInstitute of Experimental and Clinical Pharmacology and Toxicology, University of LübeckHubertus Wald Cancer Center, Department of Oncology and Hematology, University Medical Center Hamburg‐EppendorfInstitute of Experimental and Clinical Pharmacology and Toxicology, University of LübeckHubertus Wald Cancer Center, Department of Oncology and Hematology, University Medical Center Hamburg‐EppendorfHubertus Wald Cancer Center, Department of Oncology and Hematology, University Medical Center Hamburg‐EppendorfInstitute of Experimental and Clinical Pharmacology and Toxicology, University of LübeckCologne Excellence Cluster on Cellular Stress Responses in Aging‐Associated Diseases (CECAD) and Centre for Molecular Medicine (CMMC), Institute for Genetics, University of CologneDepartment of Tumor Virology, German Cancer Research CenterInstitute of Experimental and Clinical Pharmacology and Toxicology, University of LübeckHubertus Wald Cancer Center, Department of Oncology and Hematology, University Medical Center Hamburg‐EppendorfAbstract Gene therapy critically relies on vectors that combine high transduction efficiency with a high degree of target specificity and that can be administered through a safe intravenous route. The lack of suitable vectors, especially for gene therapy of brain disorders, represents a major obstacle. Therefore, we applied an in vivo screening system of random ligand libraries displayed on adeno‐associated viral capsids to select brain‐targeted vectors for the treatment of neurovascular diseases. We identified a capsid variant showing an unprecedented degree of specificity and long‐lasting transduction efficiency for brain microvasculature endothelial cells as the primary target of selection. A therapeutic vector based on this selected viral capsid was used to markedly attenuate the severe cerebrovascular pathology of mice with incontinentia pigmenti after a single intravenous injection. Furthermore, the versatility of this selection system will make it possible to select ligands for additional in vivo targets without requiring previous identification of potential target‐specific receptors.https://doi.org/10.15252/emmm.201506078adeno‐associated virusbrain microvascular endothelial cellsgene therapyneurovascular diseases
spellingShingle Jakob Körbelin
Godwin Dogbevia
Stefan Michelfelder
Dirk A Ridder
Agnes Hunger
Jan Wenzel
Henning Seismann
Melanie Lampe
Jacqueline Bannach
Manolis Pasparakis
Jürgen A Kleinschmidt
Markus Schwaninger
Martin Trepel
A brain microvasculature endothelial cell‐specific viral vector with the potential to treat neurovascular and neurological diseases
EMBO Molecular Medicine
adeno‐associated virus
brain microvascular endothelial cells
gene therapy
neurovascular diseases
title A brain microvasculature endothelial cell‐specific viral vector with the potential to treat neurovascular and neurological diseases
title_full A brain microvasculature endothelial cell‐specific viral vector with the potential to treat neurovascular and neurological diseases
title_fullStr A brain microvasculature endothelial cell‐specific viral vector with the potential to treat neurovascular and neurological diseases
title_full_unstemmed A brain microvasculature endothelial cell‐specific viral vector with the potential to treat neurovascular and neurological diseases
title_short A brain microvasculature endothelial cell‐specific viral vector with the potential to treat neurovascular and neurological diseases
title_sort brain microvasculature endothelial cell specific viral vector with the potential to treat neurovascular and neurological diseases
topic adeno‐associated virus
brain microvascular endothelial cells
gene therapy
neurovascular diseases
url https://doi.org/10.15252/emmm.201506078
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