Clarifying the clinical landscape of pediatric spinal arteriovenous shunts: an institutional experience and individual patient-data meta-analysis.

Hemorrhage Intervention Pediatrics Spinal cord Vascular Malformation

Journal

Journal of neurointerventional surgery
ISSN: 1759-8486
Titre abrégé: J Neurointerv Surg
Pays: England
ID NLM: 101517079

Informations de publication

Date de publication:
15 Mar 2023
Historique:
received: 16 11 2022
accepted: 07 03 2023
entrez: 15 3 2023
pubmed: 16 3 2023
medline: 16 3 2023
Statut: aheadofprint

Résumé

Pediatric spinal arteriovenous shunts (SAVS) are rare lesions with heterogeneous pathogenesis and clinical manifestations. 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. 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. 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. 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.

Sections du résumé

BACKGROUND BACKGROUND
Pediatric spinal arteriovenous shunts (SAVS) are rare lesions with heterogeneous pathogenesis and clinical manifestations.
OBJECTIVE 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 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 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 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.

Identifiants

DOI: 10.1136/jnis-2022-019797 PMID: 36922033
pubmed: 36922033
pii: jnis-2022-019797
doi: 10.1136/jnis-2022-019797
pii:
doi:

Types de publication

Journal Article

Langues

eng

Sous-ensembles de citation

IM

Informations de copyright

© Author(s) (or their employer(s)) 2023. No commercial re-use. See rights and permissions. Published by BMJ.

Déclaration de conflit d'intérêts

Competing interests: None declared.

Auteurs

Conner D Reynolds (CD)

Department of Medical Imaging, The University of Arizona College of Medicine Tucson, Tucson, Arizona, USA.
ORCID: 0000-0001-6320-1131

M Travis Caton (MT)

Department of Neurosurgery, Mount Sinai Health System, New York, New York, USA.
Department of Radiology and Biomedical Imaging, University of California San Francisco, San Francisco, California, USA.

Amanda Baker (A)

Department of Radiology and Biomedical Imaging, University of California San Francisco, San Francisco, California, USA.
ORCID: 0000-0001-9973-5792

Eric R Smith (ER)

Department of Neurointerventional Radiology, University of California San Francisco, San Francisco, California, USA.
ORCID: 0000-0002-6855-5104

Matthew R Amans (MR)

Department of Radiology and Biomedical Imaging, University of California San Francisco, San Francisco, California, USA.
Department of Neurointerventional Radiology, University of California San Francisco, San Francisco, California, USA.
Department of Neurological Surgery, University of California San Francisco, San Francisco, California, USA.
ORCID: 0000-0002-8209-0534

Daniel L Cooke (DL)

Department of Radiology and Biomedical Imaging, University of California San Francisco, San Francisco, California, USA.
Department of Neurointerventional Radiology, University of California San Francisco, San Francisco, California, USA.
Department of Neurological Surgery, University of California San Francisco, San Francisco, California, USA.

Christopher F Dowd (CF)

Department of Radiology and Biomedical Imaging, University of California San Francisco, San Francisco, California, USA.
Department of Neurointerventional Radiology, University of California San Francisco, San Francisco, California, USA.
Department of Neurological Surgery, University of California San Francisco, San Francisco, California, USA.

Randall T Higashida (RT)

Department of Radiology and Biomedical Imaging, University of California San Francisco, San Francisco, California, USA.
Department of Neurointerventional Radiology, University of California San Francisco, San Francisco, California, USA.
Department of Neurological Surgery, University of California San Francisco, San Francisco, California, USA.

Nalin Gupta (N)

Department of Neurological Surgery, University of California San Francisco, San Francisco, California, USA.
Department of Pediatrics, University of California San Francisco, San Francisco, California, USA.

Adib A Abla (AA)

Department of Radiology and Biomedical Imaging, University of California San Francisco, San Francisco, California, USA.
Department of Neurointerventional Radiology, University of California San Francisco, San Francisco, California, USA.
Department of Neurological Surgery, University of California San Francisco, San Francisco, California, USA.

Kurtis Auguste (K)

Department of Neurological Surgery, University of California San Francisco, San Francisco, California, USA.
Department of Pediatrics, University of California San Francisco, San Francisco, California, USA.

Christine H Fox (CH)

Department of Pediatrics, University of California San Francisco, San Francisco, California, USA.
Department of Neurology, University of California San Francisco, San Francisco, California, USA.

Heather Fullerton (H)

Department of Pediatrics, University of California San Francisco, San Francisco, California, USA.
Department of Neurology, University of California San Francisco, San Francisco, California, USA.

Steven W Hetts (SW)

Department of Radiology and Biomedical Imaging, University of California San Francisco, San Francisco, California, USA steven.hetts@ucsf.edu.
Department of Neurointerventional Radiology, University of California San Francisco, San Francisco, California, USA.
Department of Neurological Surgery, University of California San Francisco, San Francisco, California, USA.
ORCID: 0000-0001-5885-7259

Classifications MeSH