RT Journal Article
SR Electronic
T1 Clarifying the clinical landscape of pediatric spinal arteriovenous shunts: an institutional experience and individual patient-data meta-analysis
JF Journal of NeuroInterventional Surgery
JO J NeuroIntervent Surg
FD BMJ Publishing Group Ltd.
SP jnis-2022-019797
DO 10.1136/jnis-2022-019797
A1 Conner D Reynolds
A1 M Travis Caton
A1 Amanda Baker
A1 Eric R Smith
A1 Matthew R Amans
A1 Daniel L Cooke
A1 Christopher F Dowd
A1 Randall T Higashida
A1 Nalin Gupta
A1 Adib A Abla
A1 Kurtis Auguste
A1 Christine H Fox
A1 Heather Fullerton
A1 Steven W Hetts
YR 2023
UL http://jnis.bmj.com/content/early/2023/03/14/jnis-2022-019797.abstract
AB Background Pediatric spinal arteriovenous shunts (SAVS) are rare lesions with heterogeneous pathogenesis and clinical manifestations.Objective To evaluate the clinical characteristics, angioarchitecture, and technical/clinical outcomes in SAVS through a large single-center cohort analysis and meta-analysis of individual patient data.Methods A retrospective institutional database identified children (aged 0–21 years) who underwent digital subtraction spinal angiography (DSA) for SAVS between January 1996 and July 2021. Clinical data were recorded to evaluate angioarchitecture, generate modified Aminoff-Logue gait disturbance scores (AL) and McCormick grades (MC), and assess outcomes. We then performed a systematic literature review following PRISMA-IPD (Preferred Reporting Items for Systematic Reviews and Meta-Analyses for individual patient data) guidelines, extracting similar data on individual patients for meta-analysis.Results The cohort consisted of 28 children (M:F=11:17) with 32 SAVS lesions, with a mean age of 12.8±1.1 years at diagnosis. At presentation, SAVS were most highly concentrated in the cervical region (40.6%). Children had a median AL=2 and MC=2, with thoracolumbar AVS carrying the greatest disability. Among treated cases, complete obliteration was achieved in 48% of cases and median AL scores and MC grades both improved by one point. Systematic literature review identified 161 children (M:F=96:65) with 166 SAVS lesions with a mean age of 8.7±0.4 years. Among studies describing symptom chronicity, 37/51 (72.5%) of children presented acutely. At presentation, children had a median AL=4 and MC=3, with thoracolumbar AVS carrying the highest MC grades. After intervention, median AL and MC both improved by one point.Conclusions This study provides epidemiologic information on the location, onset, and presentation of the full spectrum of pediatric SAVS, highlighting the role of targeted treatment of high-risk features.Data are available upon reasonable request. Data are available upon reasonable request to the corresponding author.