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Characteristics and outcomes of pediatric dural arteriovenous fistulas: a systematic review
Pedram D Maleknia1, Andrew T Hale2, Cody Savage1
1Heersink School of Medicine, University of Alabama at Birmingham, Birmingham, AL, USA.
Insights
This systematic review highlights the limited data on pediatric dural arteriovenous fistulas (dAVF). It summarizes 69 cases, noting male predominance and common locations, with varied treatment outcomes and complication rates.
Area of Science:
- Neurology
- Vascular Surgery
- Pediatric Neurosurgery
Background:
- Dural arteriovenous fistulas (dAVF) are rare in children, with limited data on risk factors and treatment outcomes.
- Existing literature primarily consists of single-center case series, necessitating a broader review.
Approach:
- A systematic literature review was conducted following PRISMA guidelines.
- Searched major databases (PubMed, CINAHL, SCOPUS, Embase) for pediatric dAVF cases (<18 years).
- Excluded studies on dural sinus malformations or pial AVF.
Key Points:
- Identified 69 pediatric dAVF cases from 61 studies; males predominated (55.1%), with a mean age of 5.17 years.
- Transverse-sigmoid junction was the most common location (17.3%); 20.2% had cardiovascular disease.
- Endovascular embolization was the primary treatment (68.1%), achieving obliteration in 43.8%; neurological complications occurred in 29.7%.
Conclusions:
- There is a significant paucity of information regarding pediatric dAVFs.
- This review provides a descriptive summary of dAVF in the pediatric population, despite data limitations and publication bias.
- Further research is needed to better understand risk factors and optimize treatment strategies for pediatric dAVF.
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.
