Surgical revascularizations for pediatric moyamoya: a systematic review, meta-analysis, and meta-regression analysis

Childs Nerv Syst. 2023 May;39(5):1225-1243. doi: 10.1007/s00381-023-05868-6. Epub 2023 Feb 8.

Abstract

Introduction: There is no clear consensus regarding the technique of surgical revascularization for moyamoya disease and syndrome (MMD/MMS) in the pediatric population. Previous meta-analyses have attempted to address this gap in literature but with methodological limitations that affect the reliability of their pooled estimates. This meta-analysis aimed to report an accurate and transparent comparison between studies of indirect (IB), direct (DB), and combined bypasses (CB) in pediatric patients with MMD/MMS.

Methods: In accordance with PRISMA guidelines, systematic searches of Medline, Embase, and Cochrane Central were undertaken from database inception to 7 October 2022. Perioperative adverse events were the primary outcome measure. Secondary outcomes were rates of long-term revascularization, stroke recurrence, morbidity, and mortality.

Results: Thirty-seven studies reporting 2460 patients and 4432 hemispheres were included in the meta-analysis. The overall pooled mean age was 8.6 years (95% CI: 7.7; 9.5), and 45.0% were male. Pooled proportions of perioperative adverse events were similar between the DB/CB and IB groups except for wound complication which was higher in the former group (RR = 2.54 (95% CI: 1.82; 3.55)). Proportions of post-surgical Matsushima Grade A/B revascularization favored DB/CB over IB (RR = 1.12 (95% CI 1.02; 1.24)). There was no significant difference in stroke recurrence, morbidity, and mortality. After meta-regression analysis, year of publication and age were significant predictors of outcomes.

Conclusions: IB, DB/CB are relatively effective and safe revascularization options for pediatric MMD/MMS. Low-quality GRADE evidence suggests that DB/CB was associated with better long-term angiographic revascularization outcomes when compared with IB, although this did not translate to long-term stroke and mortality benefits.

Keywords: Bypass; Moyamoya disease; Moyamoya syndrome; Neurosurgery; Pediatric; Revascularization.

Publication types

  • Meta-Analysis
  • Systematic Review

MeSH terms

  • Cerebral Revascularization* / methods
  • Child
  • Female
  • Humans
  • Male
  • Moyamoya Disease* / complications
  • Moyamoya Disease* / diagnostic imaging
  • Moyamoya Disease* / surgery
  • Reproducibility of Results
  • Stroke* / epidemiology
  • Stroke* / etiology
  • Stroke* / surgery
  • Treatment Outcome
  • Vascular Surgical Procedures