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Surgical revascularizations for pediatric moyamoya: a systematic review, meta-analysis, and meta-regression analysis
Keng Siang Lee1,2,3,4,5, John J Y Zhang6, Sanjay Bhate7,8
1Department of Neurosurgery, Great Ormond Street Hospital for Children, London, UK. mrkengsianglee@gmail.com.
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.
Insights
Surgical revascularization techniques for pediatric moyamoya disease and syndrome (MMD/MMS) were compared. Direct or combined bypasses showed better angiographic outcomes than indirect bypasses, but no significant differences in stroke or mortality were observed.
Area of Science:
- Neurosurgery
- Pediatric Neurology
- Vascular Surgery
Background:
- Moyamoya disease and syndrome (MMD/MMS) in children lacks consensus on optimal surgical revascularization techniques.
- Previous meta-analyses had methodological limitations impacting reliability.
- This study provides a transparent comparison of indirect (IB), direct (DB), and combined bypasses (CB).
Purpose of the Study:
- To compare the efficacy and safety of indirect, direct, and combined bypass surgical techniques for pediatric moyamoya disease and syndrome.
- To provide an accurate and transparent comparison between different revascularization strategies.
Main Methods:
- Systematic literature search of Medline, Embase, and Cochrane Central up to October 7, 2022, following PRISMA guidelines.
- Meta-analysis of 37 studies including 2460 pediatric patients (4432 hemispheres).
- Primary outcome: perioperative adverse events. Secondary outcomes: revascularization rates, stroke recurrence, morbidity, and mortality.
Main Results:
- Pooled analysis of 2460 pediatric patients revealed similar perioperative adverse events between direct/combined bypasses (DB/CB) and indirect bypasses (IB), except for a higher wound complication rate in DB/CB.
- Angiographic revascularization (Matsushima Grade A/B) favored DB/CB over IB.
- No significant differences were found in long-term stroke recurrence, morbidity, or mortality between the groups.
Conclusions:
- Indirect bypass (IB) and direct/combined bypasses (DB/CB) are safe and effective for pediatric MMD/MMS.
- Low-quality evidence suggests DB/CB offers superior long-term angiographic revascularization compared to IB.
- These angiographic benefits did not translate into significant improvements in long-term stroke or mortality outcomes.
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