Article Text

Download PDFPDF
Original research
Pediatric brain arteriovenous malformation recurrence: a cohort study, systematic review and meta-analysis
  1. Jean-Francois Hak1,2,
  2. Gregoire Boulouis1,2,
  3. Basile Kerleroux1,2,
  4. Sandro Benichi3,
  5. Sarah Stricker3,
  6. Florent Gariel1,4,
  7. Lorenzo Garzelli1,
  8. Philippe Meyer5,
  9. Manoelle Kossorotoff6,7,
  10. Nathalie Boddaert1,
  11. Vincent Vidal8,
  12. Nadine Girard9,
  13. Volodia Dangouloff-Ros1,
  14. Francis Brunelle1,
  15. Heather Fullerton10,11,
  16. Steven W Hetts12,
  17. Thomas Blauwblomme3,7,
  18. Olivier Naggara1,2,7
  1. 1Department of Pediatric Radiology UMR 1163, Institut Imagine, INSERM U1000, APHP, Necker Sick Children Hospital, Paris, Paris, France
  2. 2Department of Neuroradiology, INSERM UMR 1266 IMA-BRAIN, GHU Paris, Paris, France
  3. 3Department of Pediatric Neurosurgery, Institut Imagine, INSERM UMR 1163, APHP, Necker Sick Children Hospital, Paris, France
  4. 4Department of Neuroradiology, CHU Bordeaux GH Pellegrin, Bordeaux, France
  5. 5Department of Pediatric Neuro ICU, APHP, Necker Sick Children Hospital, Paris, France
  6. 6Department of Pediatric Neurology, APHP University Necker Children Hospital, Paris, France
  7. 7French Center for Pediatric Stroke, INSERM U894, APHP, Necker Sick Children Hospital, Paris, France
  8. 8Department of Radiology, University Hospital La Timone, AP-HM, Marseille, France
  9. 9Department of Neuroradiology, University Hospital La Timone, AP-HM, Marseille, France
  10. 10Department of Neurology, University of California San Francisco, San Francisco, California, USA
  11. 11Department of Pediatrics, University of California San Francisco, San Francisco, California, USA
  12. 12Department of Radiology and Biomedical Imaging, University of California San Francisco, San Francisco, California, USA
  1. Correspondence to Dr Jean-Francois Hak, Department of Radiology, Necker-Enfants Malades Hospitals, Paris, Île-de-France, France; jeanfrancois.hak{at}gmail.com

Abstract

Background Recurrence following obliteration of brain arteriovenous malformations (AVMs) is common in children surgically treated, but recurrences following endovascular (EVT) and radiosurgical approaches are scantily reported.

Objective To analyze the rates and risk factors for AVM recurrence after obliteration in a single-center cohort of children with ruptured AVMs treated with multimodal approaches, and to carry out a comprehensive review and meta-analysis of current data.

Methods Children with ruptured AVMs between 2000 and 2019 enrolled in a prospective registry were retrospectively screened and included after angiographically determined obliteration to differentiate children with/without recurrence. A complementary systematic review and meta-analysis of studies investigating AVM recurrence in children between 2000 and 2020 was aggregated to explore the overall recurrence rates across treatment modalities by analyzing surgery versus other treatments.

Results Seventy children with obliterated AVMs were included. AVM recurrences (n=10) were more commonly treated with EVT as final treatment (60% in the recurrence vs 13.3% in the no-recurrence group, p=0.018). Infratentorial locations were associated with earlier and more frequent recurrences (adjusted relative risk=4.62, 95% CI 1.08 to 19.04; p=0.04).

In the aggregate analysis, the pooled rate of AVM recurrence was 10.9% (95% CI 8.7% to 13.5%). Younger age at presentation was associated with more frequent recurrences (RR per year increase, 0.97, 95% CI 0.93 to 0.99; p=0.046).

Conclusion Location of infratentorial AVMs and younger age at presentation may be associated with earlier and more frequent recurrences. The higher rates of recurrence in patients with AVMs obliterated with EVT questions its role in an intent-to-cure approach and reinforces its position as an adjunct to surgery and/or radiosurgery.

  • arteriovenous malformation
  • brain
  • pediatrics
  • stroke
  • hemorrhage

Data availability statement

Data are available upon reasonable request.

Statistics from Altmetric.com

Data availability statement

Data are available upon reasonable request.

View Full Text

Footnotes

  • Twitter @gboulouis, @gboulouis

  • J-FH and GB contributed equally.

  • Contributors J-FH, ON, GB, TB were responsible for the conception of the study; J-FH, ON, GB, TB, BK, SB, SS, FG, LG, PM, MK, NB collected the data, had full access to data, and take responsibility for accuracy of data analysis. J-FH, GB, ON drafted the initial version of the manuscript. All authors, contributed to data acquisition, analysis and interpretation, revised and approved the final version of this manuscript.

  • Funding J-FH was supported by a grant provided by the Société Française de Radiologie - French Society of Radiology - (SFR) together with the Collège des Enseignants en Radiologie de France - French Academic College of Radiology (CERF)

  • Competing interests None declared.

  • Provenance and peer review Not commissioned; externally peer reviewed.

  • Supplemental material This content has been supplied by the author(s). It has not been vetted by BMJ Publishing Group Limited (BMJ) and may not have been peer-reviewed. Any opinions or recommendations discussed are solely those of the author(s) and are not endorsed by BMJ. BMJ disclaims all liability and responsibility arising from any reliance placed on the content. Where the content includes any translated material, BMJ does not warrant the accuracy and reliability of the translations (including but not limited to local regulations, clinical guidelines, terminology, drug names and drug dosages), and is not responsible for any error and/or omissions arising from translation and adaptation or otherwise.

Request Permissions

If you wish to reuse any or all of this article please use the link below which will take you to the Copyright Clearance Center’s RightsLink service. You will be able to get a quick price and instant permission to reuse the content in many different ways.