Surgical Disconnection of a Residual Pediatric Pial Arterio-Venous Fistula Following Partial Embolization: A Case Study.


Journal

World neurosurgery
ISSN: 1878-8769
Titre abrégé: World Neurosurg
Pays: United States
ID NLM: 101528275

Informations de publication

Date de publication:
06 2020
Historique:
received: 08 02 2020
accepted: 22 02 2020
pubmed: 17 3 2020
medline: 18 7 2020
entrez: 17 3 2020
Statut: ppublish

Résumé

Pial arteriovenous fistulas (AVFs) are rare intracranial vascular lesions consisting of 1 or more feeder arteries connecting directly to a venous system without a nidus, in the subpial space. Because of the high-flow system, they are commonly associated with a large varix. They are thought to represent between 1.6% and 7.3% of all pediatric arteriovenous malformations (AVMs). Morbidity and mortality is high in this condition and surgical or endovascular treatment options are usually considered. There have been limited reports on the clinical features, treatment options, and outcomes of pial AVMs due to its rarity. We present a case study of a pediatric patient in our institution and her clinical course, focusing on her presenting clinical features and management. A 1-year-old girl presents with progressively prominent and dilated facial veins and no other features suggestive of pial AVF. She was diagnosed with pial AVF with two feeder arteries and a large varix on imaging. Embolization was undertaken 3 times before successful surgical disconnection was done. Genetic testing for associated syndromes were all negative. Prominence of facial veins could be 1 of the more uncommon presenting features of pial AVFs. Genetic testing should always be considered in the pediatric population diagnosed with AVFs because of their association to various syndromes. Despite endovascular embolization being considered the less invasive choice, decision on mode of treatment should be a multifactorial decision.

Sections du résumé

BACKGROUND
Pial arteriovenous fistulas (AVFs) are rare intracranial vascular lesions consisting of 1 or more feeder arteries connecting directly to a venous system without a nidus, in the subpial space. Because of the high-flow system, they are commonly associated with a large varix. They are thought to represent between 1.6% and 7.3% of all pediatric arteriovenous malformations (AVMs). Morbidity and mortality is high in this condition and surgical or endovascular treatment options are usually considered. There have been limited reports on the clinical features, treatment options, and outcomes of pial AVMs due to its rarity. We present a case study of a pediatric patient in our institution and her clinical course, focusing on her presenting clinical features and management.
CASE DESCRIPTION
A 1-year-old girl presents with progressively prominent and dilated facial veins and no other features suggestive of pial AVF. She was diagnosed with pial AVF with two feeder arteries and a large varix on imaging. Embolization was undertaken 3 times before successful surgical disconnection was done. Genetic testing for associated syndromes were all negative.
CONCLUSIONS
Prominence of facial veins could be 1 of the more uncommon presenting features of pial AVFs. Genetic testing should always be considered in the pediatric population diagnosed with AVFs because of their association to various syndromes. Despite endovascular embolization being considered the less invasive choice, decision on mode of treatment should be a multifactorial decision.

Identifiants

pubmed: 32173546
pii: S1878-8750(20)30421-6
doi: 10.1016/j.wneu.2020.02.146
pii:
doi:

Types de publication

Case Reports

Langues

eng

Sous-ensembles de citation

IM

Pagination

227-230

Informations de copyright

Crown Copyright © 2020. Published by Elsevier Inc. All rights reserved.

Auteurs

Neeraj Kalra (N)

Leeds Children's Hospital, Leeds Teaching Hospitals, NHS Trust, Leeds, UK. Electronic address: neeraj.kalra@nhs.net.

Liting Tong (L)

Leeds Children's Hospital, Leeds Teaching Hospitals, NHS Trust, Leeds, UK.

Helen McCullagh (H)

Leeds Children's Hospital, Leeds Teaching Hospitals, NHS Trust, Leeds, UK.

Tony Goddard (T)

Leeds General Infirmary, Leeds Teaching Hospitals, NHS Trust, Leeds, UK.

Atul Tyagi (A)

Leeds Children's Hospital, Leeds Teaching Hospitals, NHS Trust, Leeds, UK.

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