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Original research
Clarifying the clinical landscape of pediatric spinal arteriovenous shunts: an institutional experience and individual patient-data meta-analysis
  1. Conner D Reynolds1,
  2. M Travis Caton2,3,
  3. Amanda Baker3,
  4. Eric R Smith4,
  5. Matthew R Amans3,4,5,
  6. Daniel L Cooke3,4,5,
  7. Christopher F Dowd3,4,5,
  8. Randall T Higashida3,4,5,
  9. Nalin Gupta5,6,
  10. Adib A Abla3,4,5,
  11. Kurtis Auguste5,6,
  12. Christine H Fox6,7,
  13. Heather Fullerton6,7,
  14. Steven W Hetts3,4,5
  1. 1 Department of Medical Imaging, The University of Arizona College of Medicine Tucson, Tucson, Arizona, USA
  2. 2 Department of Neurosurgery, Mount Sinai Health System, New York, New York, USA
  3. 3 Department of Radiology and Biomedical Imaging, University of California San Francisco, San Francisco, California, USA
  4. 4 Department of Neurointerventional Radiology, University of California San Francisco, San Francisco, California, USA
  5. 5 Department of Neurological Surgery, University of California San Francisco, San Francisco, California, USA
  6. 6 Department of Pediatrics, University of California San Francisco, San Francisco, California, USA
  7. 7 Department of Neurology, University of California San Francisco, San Francisco, California, USA
  1. Correspondence to Dr Steven W Hetts, Department of Radiology and Biomedical Imaging, University of California San Francisco, San Francisco, California, USA; steven.hetts{at}ucsf.edu

Abstract

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.

  • Pediatrics
  • Hemorrhage
  • Intervention
  • Spinal cord
  • Vascular Malformation

Data availability statement

Data are available upon reasonable request. Data are available upon reasonable request to the corresponding author.

https://doi.org/10.1136/jnis-2022-019797

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    Data availability statement

    Data are available upon reasonable request. Data are available upon reasonable request to the corresponding author.

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    Footnotes

    • Twitter @RadDocReynolds, @amandaebaker, @EricRSmithMD

    • CDR and MTC contributed equally.

    • Contributors CDR, MTC: conceptualization, data collection, analysis, drafting original manuscript. AB, ERS, MRA, DLC, CFD, RTH, NG, AAA, KA, CHF, HF, SWH: conceptualization, oversight, revising original manuscript. SWH is the guarantor for this work and accepts responsibility for the data presented and controlled the decision to publish.

    • Funding The authors have not declared a specific grant for this research from any funding agency in the public, commercial or not-for-profit sectors.

    • Competing interests None declared.

    • Provenance and peer review Not commissioned; externally peer reviewed.

    • Supplemental material This content has been supplied by the author(s). It has not been vetted by BMJ Publishing Group Limited (BMJ) and may not have been peer-reviewed. Any opinions or recommendations discussed are solely those of the author(s) and are not endorsed by BMJ. BMJ disclaims all liability and responsibility arising from any reliance placed on the content. Where the content includes any translated material, BMJ does not warrant the accuracy and reliability of the translations (including but not limited to local regulations, clinical guidelines, terminology, drug names and drug dosages), and is not responsible for any error and/or omissions arising from translation and adaptation or otherwise.