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Characteristics and outcomes of pediatric dural arteriovenous fistulas: a systematic review

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Abstract

Background

Dural arteriovenous fistulas (dAVF) are arteriovenous shunts in communication with the dural vasculature in the brain or spine. Apart from single-center series, risk factors and treatment outcomes for pediatric dAVFs are largely undescribed.

Methods

We performed a systematic literature review of pediatric (< 18 years at diagnosis) intracranial and spinal dAVF according to PRISMA guidelines. We queried PubMed, CINAHL, SCOPUS, and Embase databases without time/date restriction. Search strings included a variety of MeSH keywords relating to dural AV fistulas in combination with MeSH keywords related to pediatric cases (see Appendix). Manuscripts describing patients diagnosed with dural sinus malformations or pial AVF were excluded.

Results

We identified 61 studies describing 69 individual patients. Overall, dAVF were more common in males (55.1%) with a mean age of diagnosis (5.17 ± 4.42 years). Approximately 20.2% of patients presented with cardiovascular disease (CVD), and 31.9% were discovered incidentally on neuroimaging studies. Transverse-sigmoid junction was the most common location (17.3%). Ninety-three percent (64 patients) were treated, most commonly using endovascular embolization (68.1%) followed by surgery (8.7%) and radiosurgery (2.9%). Almost half (43.8%) of dAVFs were completely obliterated. Of the 64 procedures, there were 19 neurological complications (29.7%) of varying severity where 12.5% were considered transient (i.e., pseudomeningocele) and 17.2% permanent (i.e., mortality secondary to acute sinus thrombosis, etc.).

Conclusion

There is a paucity of information on pediatric dAVFs. This systematic review summarizes the published cases of dAVFs in the pediatric population. While the rate of missing data is high, there is publication bias, and precise details regarding complications are difficult to ascertain, this review serves as a descriptive summary of pediatric dAVFs.

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Availability of data and material

Datasets and search terms for this systematic review can be found in the Appendix below.

Abbreviations

dAVF:

Dural arteriovenous fistulas

AVM:

Arteriovenous malformation

CT:

Computed tomography

MRI:

Magnetic resonance imaging

CVD:

Cardiovascular disease

AVM:

Arteriovenous malformation

PRISMA:

Preferred Reporting Items for Systematic Reviews and Meta-Analyses

NBCA:

N-butyl cyanoacrylate

EVOH:

Ethylene vinyl alcohol copolymer

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Author notes

  1. Pedram D. Maleknia and Andrew T. Hale contributed equally to this work.

Authors and Affiliations

  1. Heersink School of Medicine, University of Alabama at Birmingham, Birmingham, AL, USA

    Pedram D. Maleknia & Cody Savage

  2. Department of Neurosurgery, University of Alabama at Birmingham, FOT Suite 1060, 1720 2nd Ave S, Birmingham, AL, 35294, USA

    Andrew T. Hale & Jesse G. A. Jones

  3. Division of Pediatric Neurosurgery, Department of Neurosurgery, University of Alabama at Birmingham, Birmingham, AL, USA

    Jeffrey P. Blount, Brandon G. Rocque, Curtis J. Rozzelle, James M. Johnston & Jesse G. A. Jones

  4. Department of Diagnostic Radiology, University of Alabama at Birmingham, Birmingham, AL, USA

    Jesse G. A. Jones

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  1. Pedram D. Maleknia
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Contributions

All authors contributed to the study conception and design. Material preparation, data collection and analysis were performed by Pedram D. Maleknia and Andrew T. Hale. The first draft of the manuscript was written by Pedram D. Maleknia, and all authors commented on previous versions of the manuscript. All authors read and approved the final manuscript.

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Appendix

Appendix

PubMed

  1. 1.

    (("paediatrics"[All Fields] OR "pediatrics"[MeSH Terms] OR "pediatrics"[All Fields] OR "paediatric"[All Fields] OR "pediatric"[All Fields]) AND ("arteriovenous fistula"[MeSH Terms] OR ("arteriovenous"[All Fields] AND "fistula"[All Fields]) OR "arteriovenous fistula"[All Fields] OR ("av"[All Fields] AND "fistula"[All Fields]) OR "av fistula"[All Fields])) AND (casereports[Filter] OR clinicaltrial[Filter])

    1. (a)

      435 Results

CINAHL

  1. 1.

    pediatric arteriovenous fistula: Expanders—Apply equivalent subjects; Narrow by SubjectAge:—all child; Search modes—Find all my search terms; Interface—EBSCOhost Research Databases; Search Screen—Basic Search; Database—CINAHL Plus with Full Text

    1. (a)

      84 Results

SCOPUS

  1. 1.

    pediatric AND arteriovenous AND fistula AND dural

    1. (a)

      87 Results

Embase

  1. 1.

    'pediatric arteriovenous fistula' OR (('pediatric'/exp OR pediatric) AND arteriovenous AND ('fistula'/exp OR fistula)) AND 'case report'/de AND ([adolescent]/lim OR [child]/lim OR [fetus]/lim OR [45]/lim OR [newborn]/lim OR [preschool]/lim OR [46]/lim) AND ('clinical article'/de OR 'cohort analysis'/de OR 'retrospective study'/de)

    1. (a)

      241 Results

Google Scholar

  1. 1.

    Pediatric dural arteriovenous fistula -renal

    1. (a)

      6,680 Results

  2. 2.

    Pediatric dural arteriovenous fistula neurological pediatric "pediatric dural arteriovenous fistula" -renal

    1. (a)

      4 Results

  3. 3.

    Pediatric dural arteriovenous fistula neurological pediatric "dural arteriovenous fistula" -renal

    1. (a)

      2,620 Results

  4. 4.

    Pediatric dural arteriovenous fistula neurological pediatric "dural arteriovenous fistula" -renal “Review articles”

    1. (a)

      601 Results

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Maleknia, P.D., Hale, A.T., Savage, C. et al. Characteristics and outcomes of pediatric dural arteriovenous fistulas: a systematic review. Childs Nerv Syst 40, 197–204 (2024). https://doi.org/10.1007/s00381-023-06156-z

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