Evidence map›Paper›PMID 39653737›Full record

ArticleGene therapy2025

Intracisternal vs intraventricular injection of AAV1 result in comparable, widespread transduction of the dog brain.

Jacqueline E Hunter, Charles H Vite, Caitlyn M Molony, Patricia A O'Donnell, John H Wolfe

Abstract readComparative Study
In one paragraph

Article in Gene therapy, 2025. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 4 papers.

0numbers the graph read from it
0cells of the map it votes in
4citing papers in PubMed
–field-weighted citation impact
1 · What the graph read from it

What it found

Each row is one number read from the abstract, on the scale the paper reported it, with its interval. Left of the dashed line favours the treatment, right favours the comparator. Under each row is the sentence it came from. New to these charts? A ten-minute tutorial.

The abstract states no effect estimate the extractor could read, or names no intervention and outcome on the map, so this paper lights no cell and moves no belief. It is still indexed, cited and linked below.

2 · The registry

The trial behind it

Trials whose registry record cites this paper, or whose number appears in the abstract. A trial that started after this paper was published is citing it as background, not reporting it.

Neither the registry nor the abstract names a trial number. If this is a trial report, that itself is worth knowing.

3 · Its place in the literature

Who cites it

4 citing papers in PubMed.

  1. Review
  2. Current trends in gene therapy to treat inherited disorders of the brain.Molecular therapy : the journal of the American Society of Gene Therapy · 2025
    Review
  3. Review
  4. Review
4 · The record

Corrections and comments

PubMed lists nothing against this paper. Absence here is not a guarantee, only a check that was made.

5 · Who and what money

Authors and funding

5 authors.

Jacqueline E HunterResearch Institute of Children's Hospital of Philadelphia, Philadelphia, PA, USA.
Charles H ViteW.F. Goodman Center for Comparative Medical Genetics, School of Veterinary Medicine, University of Pennsylvania, Philadelphia, PA, 19104, USA.
Caitlyn M MolonyW.F. Goodman Center for Comparative Medical Genetics, School of Veterinary Medicine, University of Pennsylvania, Philadelphia, PA, 19104, USA.
Patricia A O'DonnellW.F. Goodman Center for Comparative Medical Genetics, School of Veterinary Medicine, University of Pennsylvania, Philadelphia, PA, 19104, USA.
John H WolfeResearch Institute of Children's Hospital of Philadelphia, Philadelphia, PA, USA. jhwolfe@vet.upenn.edu.ORCID 0000-0002-7497-3943

Funding

Gene transfer &NMR studies in alpha-mannosidosis brainR01DK063973 · NIDDK · CHILDREN'S HOSP OF PHILADELPHIA · PI WOLFE, JOHN H · 2002 to 2025
$11.0M
Translational studies on cerebrospinal fluid (CSF)-directed gene therapy for global neurometabolic brain diseaseR01NS110349 · NINDS · CHILDREN'S HOSP OF PHILADELPHIA · PI WOLFE, JOHN H · 2019 to 2023
$3.3M
NIDDK NIH HHS R01 DK063973NINDS NIH HHS R01 NS110349U.S. Department of Health & Human Services | NIH | National Institute of Neurological Disorders and Stroke (NINDS) R01-NS110349
6 · The paper itself

Abstract

Widespread distribution of transduced brain cells following delivery of AAV vectors into the cerebrospinal fluid (CSF) of the cisterna magna (CM) has been demonstrated in large animal brains. In humans, intraventricular injection is preferred to intracisternal injection for CSF delivery due to the risk of brain stem injury. One study in the dog reported adverse reactions to AAV vectors expressing GFP injected into the lateral ventricle but not when injected into the CM. In contrast, AAV expressing mammalian genes in diseased animals have not triggered adverse responses since many genetic diseases also have compromised immune systems. Differences in circulation of CSF from each site could potentially affect vector spread within the brain, but a direct comparison has not been made using both a mammalian gene and immunologically normal animals. In this study we evaluated the dopamine-2-receptor (D2R) variant D2R80A, which is inactivated for intracellular signaling and has been used as a reporter gene in large animal brains. No adverse reactions to the D2R80A gene were observed from either injection route in normal dogs and both routes resulted in comparable distribution of D2R80A within the brain.

Indexed as

BrainDependovirusGenetic VectorsTransduction, GeneticAnimalsCisterna MagnaDogsGenetic TherapyGene Transfer TechniquesInjections, IntraventricularReceptors, Dopamine D2Receptors, Dopamine D2

Identifiers

PMID39653737
PMCPMC12106067

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Registered trials

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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.