Evidence map›Paper›PMID 30027761›Full record

ArticleHuman gene therapy2019

Age-Related Seroprevalence of Antibodies Against AAV-LK03 in a UK Population Cohort.

Dany P Perocheau, Sharon Cunningham, Juhee Lee, Juan Antinao Diaz, Simon N Waddington, Kimberly Gilmour, Simon Eaglestone, Leszek Lisowski, Adrian J Thrasher, Ian E Alexander and 2 more

Open access · hybridAbstract read
In one paragraph

Article in Human gene therapy, 2019. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 55 papers, 2 of them syntheses that pooled it.

0numbers the graph read from it
0cells of the map it votes in
55citing papers in PubMed, 2 pooled it
4.3field-weighted citation impact, top 5% of its field
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

55 citing papers in PubMed, 2 syntheses or guidelines pooled it, 76 citations in OpenAlex.

  1. A systematic review of immunosuppressive protocols used in AAV gene therapy for monogenic disorders.Molecular therapy : the journal of the American Society of Gene Therapy · 2024
    Pooled it
  2. Pooled it
  3. Article
  4. Consequences of human genetic variations inMolecular therapy. Advances · 2026
    Article
  5. Article
  6. Article
  7. Genetic therapies for neurological diseases.Pharmacological reviews · 2026
    Review
  8. Review
  9. Review
  10. Review
  11. Review
  12. Review
  13. Article
  14. Article
  15. Gene Therapy for Inherited Liver Disease: To Add or to Edit.International journal of molecular sciences · 2024
    Review
  16. Article
  17. Review
  18. Article
  19. Article
  20. Article
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

12 authors at 6 institutions in 5 countries.

Dany P Perocheau1 Genetics and Genomic Medicine Programme, Great Ormond Street Institute of Child Health, University College London, London, United Kingdom.
Sharon Cunningham3 Gene Therapy Research Unit, Children's Medical Research Institute, Faculty of Medicine and Health, University of Sydney and Sydney Children's Hospital Network, Westmead, Australia.
Juhee Lee1 Genetics and Genomic Medicine Programme, Great Ormond Street Institute of Child Health, University College London, London, United Kingdom.
Juan Antinao Diaz2 Gene Transfer Technology Group, Institute for Women's Health, University College London, London, United Kingdom.
Simon N Waddington2 Gene Transfer Technology Group, Institute for Women's Health, University College London, London, United Kingdom.
Kimberly Gilmour5 Clinical Immunology Department, Great Ormond Street Hospital for Children NHS Foundation Trust, London, United Kingdom.
Simon Eaglestone6 Translational Research Office, University College London, London, United Kingdom.
Leszek Lisowski3 Gene Therapy Research Unit, Children's Medical Research Institute, Faculty of Medicine and Health, University of Sydney and Sydney Children's Hospital Network, Westmead, Australia.
Adrian J Thrasher5 Clinical Immunology Department, Great Ormond Street Hospital for Children NHS Foundation Trust, London, United Kingdom.
Ian E Alexander3 Gene Therapy Research Unit, Children's Medical Research Institute, Faculty of Medicine and Health, University of Sydney and Sydney Children's Hospital Network, Westmead, Australia.
Paul Gissen1 Genetics and Genomic Medicine Programme, Great Ormond Street Institute of Child Health, University College London, London, United Kingdom.
Julien Baruteau1 Genetics and Genomic Medicine Programme, Great Ormond Street Institute of Child Health, University College London, London, United Kingdom.
Great Ormond Street Hospital · GBChildren's Medical Research Institute · AUGreat Ormond Street Hospital for Children NHS Foundation Trust · GBUniversity College London · GBThe University of Sydney · AUUniversity of the Witwatersrand · ZA

Funding

Medical Research Council MR/N019075/1Medical Research Council MR/P026494/1Medical Research Council MR/R015325/1Medical Research Council MR/S019111/1
6 · The paper itself

Abstract

Recombinant adeno-associated virus (rAAV) vectors are a promising platform for in vivo gene therapy. The presence of neutralizing antibodies (Nab) against AAV capsids decreases cell transduction efficiency and is a common exclusion criterion for participation in clinical trials. Novel engineered capsids are being generated to improve gene delivery to the target cells and facilitate success of clinical trials; however, the prevalence of antibodies against such capsids remains largely unknown. We therefore assessed the seroprevalence of antibodies against a novel synthetic liver-tropic capsid AAV-LK03. We measured seroprevalence of immunoglobulin (Ig)G (i.e., neutralizing and nonneutralizing) antibodies and Nab to AAV-LK03 in a cohort of 323 UK patients (including 260 pediatric) and 52 juvenile rhesus macaques. We also performed comparative analysis of seroprevalence of Nab against wild-type AAV8 and AAV3B capsids. Overall IgG seroprevalence for AAV-LK03 was 39% in human samples. The titer increased with age. Prevalence of Nab was 23%, 35%, and 18% for AAV-LK03, AAV3B, and AAV8, respectively, with the lowest seroprevalence between 3 and 17 years of age for all serotypes. Presence of Nab against AAV-LK03 decreased from 36% in the youngest cohort (birth to 6 months) to 7% in older primary school-age children (9-11 years) and then progressively increased to 54% in late adulthood. Cross-reactivity between serotypes was >60%. Nab seroprevalence in macaques was 62%, 85%, and 40% for AAV-LK03, AAV3B, and AAV8, respectively. When planning for AAV gene therapy clinical trials, knowing the seropositivity of the target population is critical. In the population studied, AAV seroprevalence for AAV serotypes tested was low. However, high cross-reactivity between AAV serotypes remains a barrier for re-injection. Shifts in Nab seroprevalence during the first decade need to be confirmed by longitudinal studies. This possibility suggests that pediatric patients could respond differently to AAV therapy according to age. If late childhood is an ideal age window, intervention at an early age when maternal Nab levels are high may be challenging. Nab-positive children excluded from trials could be rescreened for eligibility at regular intervals because this status may change.

Indexed as

Seroepidemiologic StudiesAdolescentAntibodies, NeutralizingAntibodies, ViralCapsidChildChild, PreschoolCross ReactionsDependovirusFemaleGenetic VectorsGene Transfer TechniquesHumansInfantInfant, NewbornMaleAntibodies, NeutralizingAntibodies, ViralAAVAAV-LK03adeno-associated virusgene therapyliverneutralizing antibodiesseroprevalence

Identifiers

PMID30027761
PMCPMC6343184
OpenAlexW2883141789

What OpenQuestion holds

Textmetadata
LicenceCC BY
Read underepoch 390

Registered trials

None linked

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.