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

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...

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Autores principales: Savage, Cody, Hale, Andrew T., Parr, Matthew S., Hedaya, Alexander, Saccomano, Benjamin W., Tsemo, Georges Bouobda, Hafeez, Muhammad U., Tanweer, Omar, Kan, Peter, Solomon, Laurent J., Meila, Dan, Dirks, Peter B., Blount, Jeffrey P., Johnston, James M., Rocque, Brandon G., Rozzelle, Curtis J., Bhatia, Kartik, Muthusami, Prakash, Krings, Timo, Jones, Jesse
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Frontiers Media S.A. 2022
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9561813/
https://www.ncbi.nlm.nih.gov/pubmed/36245731
http://dx.doi.org/10.3389/fped.2022.976060
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author Savage, Cody
Hale, Andrew T.
Parr, Matthew S.
Hedaya, Alexander
Saccomano, Benjamin W.
Tsemo, Georges Bouobda
Hafeez, Muhammad U.
Tanweer, Omar
Kan, Peter
Solomon, Laurent J.
Meila, Dan
Dirks, Peter B.
Blount, Jeffrey P.
Johnston, James M.
Rocque, Brandon G.
Rozzelle, Curtis J.
Bhatia, Kartik
Muthusami, Prakash
Krings, Timo
Jones, Jesse
author_facet Savage, Cody
Hale, Andrew T.
Parr, Matthew S.
Hedaya, Alexander
Saccomano, Benjamin W.
Tsemo, Georges Bouobda
Hafeez, Muhammad U.
Tanweer, Omar
Kan, Peter
Solomon, Laurent J.
Meila, Dan
Dirks, Peter B.
Blount, Jeffrey P.
Johnston, James M.
Rocque, Brandon G.
Rozzelle, Curtis J.
Bhatia, Kartik
Muthusami, Prakash
Krings, Timo
Jones, Jesse
author_sort Savage, Cody
collection PubMed
description 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. 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(2), p-value) 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(2) < 0.01; p < 0.001] and incomplete embolization [OR = 10.87; 95% CI (1.86–63.55); I(2) < 0.01; p < 0.001] were associated with mortality. First embolization as a neonate [OR = 3.24; 95% CI (1.47–7.15); I(2) < 0.01; p < 0.001], incomplete embolization [OR = 5.26; 95% CI (2.06–13.43); I(2) < 0.01; p < 0.001], and heart failure at presentation [OR = 3.10; 95% CI (1.03–9.33); I(2) < 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. 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.
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spelling pubmed-95618132022-10-15 Outcomes of endovascular embolization for Vein of Galen malformations: An individual participant data meta-analysis Savage, Cody Hale, Andrew T. Parr, Matthew S. Hedaya, Alexander Saccomano, Benjamin W. Tsemo, Georges Bouobda Hafeez, Muhammad U. Tanweer, Omar Kan, Peter Solomon, Laurent J. Meila, Dan Dirks, Peter B. Blount, Jeffrey P. Johnston, James M. Rocque, Brandon G. Rozzelle, Curtis J. Bhatia, Kartik Muthusami, Prakash Krings, Timo Jones, Jesse Front Pediatr Pediatrics 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. 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(2), p-value) 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(2) < 0.01; p < 0.001] and incomplete embolization [OR = 10.87; 95% CI (1.86–63.55); I(2) < 0.01; p < 0.001] were associated with mortality. First embolization as a neonate [OR = 3.24; 95% CI (1.47–7.15); I(2) < 0.01; p < 0.001], incomplete embolization [OR = 5.26; 95% CI (2.06–13.43); I(2) < 0.01; p < 0.001], and heart failure at presentation [OR = 3.10; 95% CI (1.03–9.33); I(2) < 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. 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. Frontiers Media S.A. 2022-09-30 /pmc/articles/PMC9561813/ /pubmed/36245731 http://dx.doi.org/10.3389/fped.2022.976060 Text en © 2022 Savage, Hale, Parr, Hedaya, Saccomano, Tsemo, Hafeez, Tanweer, Kan, Solomon, Meila, Dirks, Blount, Johnston, Rocque, Rozzelle, Bhatia, Muthusami, Krings and Jones. https://creativecommons.org/licenses/by/4.0/This is an open-access article distributed under the terms of the Creative Commons Attribution License (CC BY) (https://creativecommons.org/licenses/by/4.0/) . The use, distribution or reproduction in other forums is permitted, provided the original author(s) and the copyright owner(s) are credited and that the original publication in this journal is cited, in accordance with accepted academic practice. No use, distribution or reproduction is permitted which does not comply with these terms.
spellingShingle Pediatrics
Savage, Cody
Hale, Andrew T.
Parr, Matthew S.
Hedaya, Alexander
Saccomano, Benjamin W.
Tsemo, Georges Bouobda
Hafeez, Muhammad U.
Tanweer, Omar
Kan, Peter
Solomon, Laurent J.
Meila, Dan
Dirks, Peter B.
Blount, Jeffrey P.
Johnston, James M.
Rocque, Brandon G.
Rozzelle, Curtis J.
Bhatia, Kartik
Muthusami, Prakash
Krings, Timo
Jones, Jesse
Outcomes of endovascular embolization for Vein of Galen malformations: An individual participant data meta-analysis
title Outcomes of endovascular embolization for Vein of Galen malformations: An individual participant data meta-analysis
title_full Outcomes of endovascular embolization for Vein of Galen malformations: An individual participant data meta-analysis
title_fullStr Outcomes of endovascular embolization for Vein of Galen malformations: An individual participant data meta-analysis
title_full_unstemmed Outcomes of endovascular embolization for Vein of Galen malformations: An individual participant data meta-analysis
title_short Outcomes of endovascular embolization for Vein of Galen malformations: An individual participant data meta-analysis
title_sort outcomes of endovascular embolization for vein of galen malformations: an individual participant data meta-analysis
topic Pediatrics
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9561813/
https://www.ncbi.nlm.nih.gov/pubmed/36245731
http://dx.doi.org/10.3389/fped.2022.976060
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