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Outcomes of endovascular embolization for Vein of Galen malformations: An individual participant data meta-analysis.

Authors :
Savage C
Hale AT
Parr MS
Hedaya A
Saccomano BW
Tsemo GB
Hafeez MU
Tanweer O
Kan P
Solomon LJ
Meila D
Dirks PB
Blount JP
Johnston JM
Rocque BG
Rozzelle CJ
Bhatia K
Muthusami P
Krings T
Jones J
Source :
Frontiers in pediatrics [Front Pediatr] 2022 Sep 30; Vol. 10, pp. 976060. Date of Electronic Publication: 2022 Sep 30 (Print Publication: 2022).
Publication Year :
2022

Abstract

Introduction: Understanding outcomes after Vein of Galen malformation (VOGM) embolization has been limited by small sample size in reported series and predominantly single center studies. To address these limitations, we perform an individual-participant meta-analysis (IPMA) to identify risk factors associated with all-cause mortality and clinical outcome after VOGM endovascular embolization.<br />Methods: We performed a systematic review and IPMA of VOGM endovascular outcomes according to PRISMA guidelines. Individual patient characteristics including demographic, intra/post-operative adverse events, treatment efficacy (partial or complete occlusion), and clinical outcome were collected. Mixed-effects logistic regression with random effects modeling and Bonferroni correction was used ( p  ≤ 0.003 threshold for statistical significance). The primary and secondary outcomes were all-cause mortality and poor clinical outcome (moderate/severe developmental delay or permanent disabling injury), respectively. Data are expressed as (mean ± standard deviation (SD)) or (odds ratio (OR), 95% confidence interval (CI), I <superscript>2</superscript> , p -value).<br />Results: Thirty-five studies totaling 307 participants quantifying outcomes after endovascular embolization for VOGM were included. Follow up time was 42 (±57) months. Our analysis contained 42% neonates (<1 month) at first embolization, 45% infants (1 month ≤2 years), and 13% children (>2 years). Complete occlusion was reported in 48% of participants. Overall all-cause mortality was 16%. Overall, good clinical outcome was achieved in 68% of participants. First embolization as a neonate [OR = 6.93; 95% CI (1.99-24.08); I <superscript>2</superscript>  < 0.01; p  < 0.001] and incomplete embolization [OR = 10.87; 95% CI (1.86-63.55); I <superscript>2</superscript>  < 0.01; p  < 0.001] were associated with mortality. First embolization as a neonate [OR = 3.24; 95% CI (1.47-7.15); I <superscript>2</superscript>  < 0.01; p  < 0.001], incomplete embolization [OR = 5.26; 95% CI (2.06-13.43); I <superscript>2</superscript>  < 0.01; p  < 0.001], and heart failure at presentation [OR = 3.10; 95% CI (1.03-9.33); I <superscript>2</superscript>  < 0.01; p  = 0.002] were associated with poor clinical outcomes. Sex, angioarchitecture of lesion, embolization approach (transvenous vs. transarterial), and single or multistage embolization were not associated with mortality or clinical outcome.<br />Conclusions: We identify incomplete VOGM embolization independently associated with mortality and poor clinical outcome. While this study provides the highest level of evidence for VOGM embolization to date, prospective multicenter studies are needed to understand the optimal treatment strategies, outcomes, and natural history after VOGM embolization.<br />Competing Interests: The authors declare that the research was conducted in the absence of any commercial or financial relationships that could be construed as a potential conflict of interest.<br /> (© 2022 Savage, Hale, Parr, Hedaya, Saccomano, Tsemo, Hafeez, Tanweer, Kan, Solomon, Meila, Dirks, Blount, Johnston, Rocque, Rozzelle, Bhatia, Muthusami, Krings and Jones.)

Details

Language :
English
ISSN :
2296-2360
Volume :
10
Database :
MEDLINE
Journal :
Frontiers in pediatrics
Publication Type :
Academic Journal
Accession number :
36245731
Full Text :
https://doi.org/10.3389/fped.2022.976060