Return of the lesion: a meta-analysis of 1134 angiographically cured pediatric arteriovenous malformations

Abstract

OBJECTIVE Brain arteriovenous malformations (AVMs) carry a risk of rupture and subsequent morbidity or mortality unless fully treated. AVMs in pediatric patients are known to occasionally recur after obliteration. The objective of this study was to characterize the risk of AVM recurrence following angiographically confirmed obliteration in children. METHODS Consecutive pediatric AVMs treated at a single center were identified from a prospective database. Patients with angiographically confirmed AVM obliteration following treatment were included in this study. Associations between AVM recurrence and patient or procedural factors were characterized using the two-tailed Fisher exact test or Mann-Whitney U-test. A literature search was conducted using PubMed, Scopus, Embase, and the Clarivate Web of Science with defined search criteria, and eligible studies were included alongside this study cohort in a meta-analysis. Rates of AVM recurrence following obliteration were pooled across studies with a random-effects model and reported with 95% confidence intervals (CIs). RESULTS Recurrence after angiographic confirmation of AVM obliteration was observed in 10.4% (7/67) of pediatric AVMs treated at the authors’ center. Patients with recurrent AVMs were significantly younger than those without recurrence (p = 0.002). In the meta-analysis, which included 1134 patients across 24 studies, the rate of recurrence was 4.8% (95% CI 3.0%–6.7%). The rate of AVM recurrence following radiosurgery was 0.7% (95% CI 0%–1.6%), which was significantly lower than the 8.5% rate (95% CI 5.0%–12.0%) following microsurgery. CONCLUSIONS Recurrence of obliterated brain AVMs is common in children. Recurrence is more common in young children and following microsurgery.

The clear need for a prospective pediatric arteriovenous malformation trial

“…Their study describes an eye-opening 15% recurrence rate in a retrospectively analyzed cohort of 70 children with AVMs and an 11% recurrence rate in an accompanying meta-analysis. These results are in agreement with the findings of two other studies from the past 18 months that each report a 10.4% recurrence rate, one in a cohort of 67 children2 and another in a cohort of 115 children 3. All three papers established younger age as a risk factor for recurrence.…”

“…All three papers established younger age as a risk factor for recurrence. Other factors that may portend a higher risk of recurrence include hemorrhagic presentation and treatment modality 2 3…”

“…The ARUBA trial, which has important flaws that have been discussed elsewhere but is nonetheless the most well-known source of prospective data for AVM treatment, excluded pediatric patients entirely 7 8. The currently active Treatment of Brain AVMs Study (TOBAS) will recruit adult patients and children, but as yet includes few children, with the youngest patient currently enrolled being 11 years of age,9 significantly older than the 8-year median age of children with brain AVM recurrence 1 2. Appropriate long-term follow-up in prospective AVM trials focused on adults is also likely to be different than in children based on the fivefold higher recurrence rate in children than in adults 10…”

The Hybrid Operative Suite with Intraoperative Biplane Rotational Angiography in Pediatric Cerebrovascular Neurosurgery: Utility and Lessons Learned

“…Our sample size is modest, with a predominance of ruptured, first-time presentations of AVMs. Prior large pediatric series have shown a 5–10% risk of overall recurrence in angiographically cured AVM cases [23, 24], with an elevated 5-year rate of 18% in ruptured cases [24]. As such, the utility of the hybrid neuroangiography surgical suite may benefit from further validation in ruptured versus unruptured and initial versus recurrent cases.…”

Endovascular treatment in the multimodality management of brain arteriovenous malformations: report of the Society of NeuroInterventional Surgery Standards and Guidelines Committee

“…Once an AVM appears to be obliterated on a post-treatment DSA, it is unclear what the optimal imaging follow-up strategy should be. Pediatric patients, in particular, have been reported to have recurrence of apparently cured brain AVMs; such recurrences may be more common in younger patients who have presented with ruptured AVMs as opposed to unruptured AVMs 146–150…”