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Spontaneous thrombosis of high flow pediatric arteriovenous fistulae: Case series of two patients and a comprehensive literature review

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Abstract

Pediatric pial arteriovenous shunts in the brain and spine are challenging to understand because of low incidence, variable presentation, and associations with genetic syndromes. What is known about their natural history comes from reviews of small series. To better understand the natural history and role for intervention, two cases are presented followed by a review of the literature. In the first case, an infant with a prior history of intracranial hemorrhage from a ruptured pial fistula returns for elective embolization for a second pial fistula which was found to be spontaneously thrombosed 2 weeks later. In the second case, a 5-year-old with a vertebro-vertebral fistula, identified on work up for a heart murmur and documented with diagnostic angiography, is brought for elective embolization 6 weeks later where spontaneous thrombosis is identified. In reviewing the literature on pediatric single-hole fistulae of the brain and spine, the authors offer some morphologic considerations for identifying which high-flow fistulae may undergo spontaneous thrombosis to decrease the potentially unnecessary risk associated with interventions in small children.

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Authors and Affiliations

  1. Edward B. Singleton Department of Radiology and Department of Neurosurgery, Texas Children’s Hospital, Baylor College of Medicine, 6701 Fannin St, Suite 470, Houston, TX, 77030, USA

    Karen S. Chen

  2. Department of Neurology, Texas Children’s Hospital, Baylor College of Medicine, 6701 Fannin St, Suite 1250, Houston, TX, 77030, USA

    Daniel Davila Williams

  3. Department of Pediatrics, Baylor College of Medicine, Vascular Anomalies Center at Texas Children’s Cancer and Hematology Centers, Texas Children’s Hospital, 6701 Fannin St, Suite 1510, Houston, TX, 77030, USA

    Ionela Iacobas

  4. Department of Neurosurgery, Texas Children’s Hospital, Baylor College of Medicine, 6701 Fannin St, Suite 1230, Houston, TX, 77030, USA

    Samuel G. McClugage & Nisha Gadgil

  5. Department of Neurosurgery, University of Texas Medical Branch, 1005 Harborside Dr, 5th floor, Galveston, TX, 77555, USA

    Peter Kan

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KC and PK conceptualized the manuscript and KC prepared the original draft. II, DDW, SM, and NG procured data for the manuscript. KC prepared the table and all figures. All authors reviewed and edited the manuscript.

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Chen, K.S., Williams, D.D., Iacobas, I. et al. Spontaneous thrombosis of high flow pediatric arteriovenous fistulae: Case series of two patients and a comprehensive literature review. Childs Nerv Syst 40, 1405–1414 (2024). https://doi.org/10.1007/s00381-023-06241-3

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