articleNature MedicineJan 16, 2026HYBRID OA

Contaminating plasmid sequences and disrupted vector genomes in the liver following adeno-associated virus gene therapy

Great Ormond Street Hospital · University College London · +13 more institutions

PubMed
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Abstract

Adeno-associated viruses (AAVs) are common vectors in gene therapy but can frequently cause liver complications in patients. The mechanisms underlying AAV-related liver toxicity remain poorly understood, posing challenges for effective prevention and intervention. Here we conducted a case study of a child with spinal muscular atrophy type 1 experiencing substantial hepatitis after receiving onasemnogene abeparvovec, undertaking long- and short-read metagenomic sequencing of liver tissue. We identified manufacturing plasmid sequences with complex structures and recombination. Vector genomes had extensive disruption and concatemerization as well as numerous vector-human fusion junctions. We also identified human…

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