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.
Similar content being viewed by others
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
References
Baharvahdat H, Ooi YC, Kim WJ, Mowla A, Coon AL, Colby GP (2020) Updates in the management of cranial dural arteriovenous fistula. Stroke Vasc Neurol 5(1):50–58
Ide S, Kiyosue H (2021) Dural Arteriovenous Fistula. No Shinkei Geka 49(2):362–367
The ISPN Guide to Pediatric Neurosurgery (USA): history of management of dural arteriovenous malformations in Children. The International Society for Pediatric Neurosurgery 2023; https://ispn.guide/vascular-disorders-of-the-nervous-system-in-children/dural-arteriovenous-fistulas-in-children-homepage/history-of-management-of-dural-arteriovenous-malformations-in-children/
Johnson JN, Hartman TK, Barbaresi W, Raffel C, Colby CE (2009) Developmental outcomes for neonatal dural arteriovenous fistulas. J Neurosurg Pediatr 3(2):105–109
Kincaid PK, Duckwiler GR, Gobin YP, Viñuela F (2001) Dural arteriovenous fistula in children: endovascular treatment and outcomes in seven cases. AJNR Am J Neuroradiol Jun-Jul 22(6):1217–1225
Chu CL, Lu YJ, Lee TH, Jung SM, Chu YC, Wong HF (2022) Concomitant spinal dural arteriovenous fistula and nodular fasciitis in an adolescent: case report. BMC Pediatr 22(1):30
Song GF, Wang H, Li X, He C, Mao ML (2019) Efficacy of microsurgery for dural arteriovenous fistula: a systematic review protocol. Medicine (Baltimore) 98(45):e17288
Jermakowicz WJ, Weil AG, Vlasenko A, Bhatia S, Niazi TN (2017) Cognard Type V intracranial dural arteriovenous fistula presenting in a pediatric patient with rapid, progressive myelopathy. J Neurosurg Pediatr 20(2):158–163
Zyck S, De Jesus O, Gould GC (2023) Dural arteriovenous fistula. StatPearls. Treasure Island (FL): StatPearls Publishing Copyright © 2023, StatPearls Publishing LLC
Gomez J, Amin AG, Gregg L, Gailloud P (2012) Classification schemes of cranial dural arteriovenous fistulas. Neurosurg Clin N Am 23(1):55–62
Pan HC, Sheehan J, Huang CF, Yang DY (2007) Two consecutive dural arteriovenous fistulae in a child: a case report of successful treatment with gamma knife radiosurgery. Childs Nerv Syst 23(10):1185–1190
Hong CS, Beckta JM, Kundishora AJ, Elsamadicy AA, Chiang VL (2020) Laser interstitial thermal therapy for treatment of cerebral radiation necrosis. Int J Hyperth 37(2):68–76
See AP, Kochis MA, Orbach DB (2017) Segmental upper mid-basilar artery sacrifice in a child using a Micro Vascular Plug device for treatment of a basilar arteriovenous fistula compressing the brainstem. BMJ Case Rep
Page MJ, McKenzie JE, Bossuyt PM et al (2021) The PRISMA 2020 statement: an updated guideline for reporting systematic reviews. Int J Surg 88:105906
Brinjikji W, Flemming KD, Lanzino G (2017) De novo formation of a large cavernoma associated with a congenital torcular dural arteriovenous fistula: case report. J Neurosurg Pediatr 19(5):567–570
Kuo MF, Tu YK, Liu HM (1995) Spinal intradural arteriovenous fistula: a case with an unusual presentation. Pediatr Neurosurg 22(3):158–161
Alonso Fernández L, Nzau M, Ventureyra E (2008) Spinal intradural arteriovenous fistula with unusual presentation: case report and literature review. Childs Nerv Syst 24(11):1349–1353
Crowley RW, Evans AJ, Jensen ME, Kassell NF, Dumont AS (2009) Combined surgical/endovascular treatment of a complex dural arteriovenous fistula in 21-month-old. Technical note. J Neurosurg Pediatr3(6):501–506
Vassilyadi M, Mehrotra N, Shamji MF, Michaud J (2009) Pediatric traumatic dural arteriovenous fistula. Can J Neurol Sci 36(6):751–756
Reig AS, Simon SD, Neblett WW 3rd, Mericle RA (2010) Eight-year follow-up after palliative embolization of a neonatal intracranial dural arteriovenous fistula with high-output heart failure: management strategies for symptomatic fistula growth and bilateral femoral occlusions in pediatric patients. J Neurosurg Pediatr 6(6):553–558
Solarte CE, Levin AV, Armstrong D (2010) Acute proptosis in a newborn infant: a presentation of infantile dural fistula. J aapos 14(1):88–89
Teranishi K, Takaya J, Yamahara T, Numa Y, Yamanouchi Y, Kaneko K (2010) Case of dural arteriovenous fistula with proptosis. Pediatr Int 52(4):674–676
Ventura E, Ormitti F, Summa A, Menozzi R, Cerasti D, Crisi G (2010) Occasional finding of a dural arteriovenous fistula in a newborn with orbital lymphangioma. A case report. Neuroradiol J 23(2):191–194
Abla AA, Albuquerque FC, Theodore N, Spetzler RF (2011) Delayed presentation of traumatic cerebral and dural arteriovenous fistulae after a BB gun accident in a pediatric patient: case report. Neurosurgery 68(6):E1750–1754; discussion E1754–1755
Prasanna Karanam LS, Baddam SR, Joseph S (2011) Role of endovascular embolisation in treatment of pediatric dural arteriovenous fistula: a case report with a review of the literature. Neurol India Nov-Dec 59(6):917–918
Cohen JE, Constantini S, Gomori JM, Benifla M, Itshayek E (2013) Pediatric perimedullary arteriovenous fistula of the conus medullaris supplied by the artery of Desproges-Gotteron. J Neurosurg Pediatr 11(4):426–430
Leu S, Valavanis A, Baltsavias G (2015) Langer-Giedion syndrome associated with congenital dural arterio-venous fistula. Childs Nerv Syst 31(5):801–804
Zaidi HA, Kalani MY, Spetzler RF, McDougall CG, Albuquerque FC (2015) Multimodal treatment strategies for complex pediatric cerebral arteriovenous fistulas: contemporary case series at Barrow Neurological Institute. J Neurosurg Pediatr 15(6):615–624
Hayward DM, Johans SJ, Rosenblum JD, Loftus CM, Ashley WW (2016) Balloon-occlusion catheter onyx embolization of a spinal dural arteriovenous fistula presenting with subarachnoid hemorrhage in a pediatric patient. J Stroke Cerebrovasc Dis 25(4):e46-49
Oshiro T, Nakayama O, Ohba C et al (2016) Transumbilical arterial embolization of a large dural arteriovenous fistula in a low-birth-weight neonate with congestive heart failure. Childs Nerv Syst 32(4):723–726
Hetts SW, Moftakhar P, Maluste N et al (2016) Pediatric intracranial dural arteriovenous fistulas: age-related differences in clinical features, angioarchitecture, and treatment outcomes. J Neurosurg Pediatr 18(5):602–610
Soni V, Vaidya PC, Sahu JK, Yadav M, Singhi P (2017) Spinal dural arteriovenous fistula and cecal arteriovenous malformation in a boy. APSP J Case Rep 8(1):3
Tomycz L, Haider AS, Miley J (2017) A combined open and endovascular approach for the treatment of a torcular dural arteriovenous fistula. Cureus 9(11):e1874
Koutsouras GW, Rahmani R, Schmidt T, Silberstein H, Bhalla T (2018) Coil and Onyx embolization of a torcular herophili dural arteriovenous fistula in a full-term neonate with advanced heart failure using a transumbilical approach. J Neurosurg Pediatr 23(1):80–85
Agnoletto GJ, Imbarrato GJ, Granja MF, Monteiro A, Aldana PR, Hanel RA (2019) A de novo sphenoparietal dural arteriovenous fistula: unveiling the deceitful culprit. World Neurosurg 127:375–380
Gatto LAM, De Souza TFS, Naves de Lima Alves G, Padilha Miranda T, Koppe GL, Demartini Z Jr (2019) Endovascular treatment of a dural arteriovenous fistula after cerebral sinovenous thrombosis in a child. Pediatr Neurosurg 54(1):66–70
Guo L, Liu T, Lv X (2019) Endovascular embolization of dural arteriovenous fistula in a child presented with slight conjunctival hyperemia. Childs Nerv Syst 35(12):2435–2437
Koduri S, Wilkinson DA, Griauzde JM, Gemmete JJ, Maher CO (2019) Development of bilateral dural arteriovenous fistulae following pial synangiosis for moyamoya syndrome: case report. J Neurosurg Pediatr 24(1):9–13
Rajadurai J, Kohan S, Wenderoth J (2020) Management of spinal dural arteriovenous fistula in a child with myelopathy. Surg Neurol Int 11:91
Jordan L, Rodgers N, Roberts KD (2021) Transverse sinus dural arteriovenous fistula: a reversible cause of severe pulmonary hypertension in an extremely premature infant. BMJ Case Rep 14(2)
Niimi Y, Sato S, Ryu B, Inoue T, Shima S (2021) Unusual pediatric epidural arteriovenous fistula with venous ectasia presented with spontaneous spinal epidural hematoma: case report. Interv Neuroradiol 27(6):763–769
Saenz A, Badaloni E, Rugilo C, Requejo F, Argañaraz R, Mantese B (2021) Giant dural arteriovenous fistula in a pediatric patient: positive outcome following surgical treatment. Childs Nerv Syst 37(6):2063–2068
Yadav V, Bhatt S, Athaullah, Dangwal S (2021) Rare occurrence of dural arteriovenous fistula in a child: multi-modality imaging and literature review. Radiol Case Rep 16(4):879–883
Roth SG, Feldman MJ, Borst AJ, Froehler MT (2022) Formation of a de novo intracranial arteriovenous fistula in a child with PTEN hamartoma tumor syndrome. Childs Nerv Syst 38(5):1029–1033
Linfante I, Delgado-Mederos R, Andreone V, Gounis M, Hendricks L, Wakhloo AK (2008) Angiographic and hemodynamic effect of high concentration of intra-arterial nicardipine in cerebral vasospasm. Neurosurgery 63(6):1080–1086; discussion 1086–1087
McGuire AM, Weiner B, Park ST et al (2012) Comparative analysis of Mycobacterium and related Actinomycetes yields insight into the evolution of Mycobacterium tuberculosis pathogenesis. BMC Genom 13:120
Author information
Authors and Affiliations
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.
Corresponding author
Ethics declarations
Ethics approval and consent to participate
Approved by all participating parties
Conflict of interest
The authors declare no competing interests.
Additional information
Publisher's Note
Springer Nature remains neutral with regard to jurisdictional claims in published maps and institutional affiliations.
Appendix
Appendix
PubMed
-
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])
-
(a)
435 Results
-
(a)
CINAHL
-
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
-
(a)
84 Results
-
(a)
SCOPUS
-
1.
pediatric AND arteriovenous AND fistula AND dural
-
(a)
87 Results
-
(a)
Embase
-
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)
-
(a)
241 Results
-
(a)
Google Scholar
-
1.
Pediatric dural arteriovenous fistula -renal
-
(a)
6,680 Results
-
(a)
-
2.
Pediatric dural arteriovenous fistula neurological pediatric "pediatric dural arteriovenous fistula" -renal
-
(a)
4 Results
-
(a)
-
3.
Pediatric dural arteriovenous fistula neurological pediatric "dural arteriovenous fistula" -renal
-
(a)
2,620 Results
-
(a)
-
4.
Pediatric dural arteriovenous fistula neurological pediatric "dural arteriovenous fistula" -renal “Review articles”
-
(a)
601 Results
-
(a)
Rights and permissions
Springer Nature or its licensor (e.g. a society or other partner) holds exclusive rights to this article under a publishing agreement with the author(s) or other rightsholder(s); author self-archiving of the accepted manuscript version of this article is solely governed by the terms of such publishing agreement and applicable law.
About this article
Cite this article
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
Received:
Accepted:
Published:
Issue Date:
DOI: https://doi.org/10.1007/s00381-023-06156-z