title: International multicenter cohort study of pediatric brain arteriovenous malformations. Part 1: Predictors of hemorrhagic presentation.
abstract: OBJECTIVE Brain arteriovenous malformations (AVMs) are the most common cause of spontaneous intracranial hemorrhage in pediatric patients (age < 18 years). Since the cumulative lifetime risk of AVM hemorrhage is considerable in children, an improved understanding of the risk factors influencing hemorrhagic presentation may aid in the management of pediatric AVMs. The aims of this first of a 2-part multicenter, retrospective cohort study are to evaluate the incidence and determine the predictors of hemorrhagic presentation in pediatric AVM patients. METHODS The authors analyzed pooled AVM radiosurgery data from 7 institutions participating in the International Gamma Knife Research Foundation (IGKRF). Patients younger than 18 years at the time of radiosurgery and who had at least 12 months of follow-up were included in the study cohort. Patient and AVM characteristics were compared between unruptured and ruptured pediatric AVMs. RESULTS A total of 357 pediatric patients were eligible for analysis, including 112 patients in the unruptured and 245 patients in the ruptured AVM cohorts (69% incidence of hemorrhagic presentation). The annual hemorrhage rate prior to radiosurgery was 6.3%. Hemorrhagic presentation was significantly more common in deep locations (basal ganglia, thalamus, and brainstem) than in cortical locations (frontal, temporal, parietal, and occipital lobes) (76% vs 62%, p = 0.006). Among the factors found to be significantly associated with hemorrhagic presentation in the multivariate logistic regression analysis, deep venous drainage (OR 3.2, p < 0.001) was the strongest independent predictor, followed by female sex (OR 1.7, p = 0.042) and smaller AVM volume (OR 1.1, p < 0.001). CONCLUSIONS Unruptured and ruptured pediatric AVMs have significantly different patient and nidal features. Pediatric AVM patients who possess 1 or more of these high-risk features may be candidates for relatively more aggressive management strategies.
authors.affiliation: University of Virginia, Department of Neurosurgery, Charlottesville, Virginia.
authors.forename: Dale
authors.initials: D
authors.lastname: Ding
authors.affiliation: University of Miami, Department of Neurological Surgery, Miami, Florida.
authors.forename: Robert M
authors.initials: RM
authors.lastname: Starke
authors.affiliation: University of Pittsburgh, Department of Neurological Surgery, Pittsburgh, Pennsylvania.
authors.forename: Hideyuki
authors.initials: H
authors.lastname: Kano
authors.affiliation: University of Sherbrooke, Division of Neurosurgery, Sherbrooke, Quebec, Canada; and.
authors.forename: David
authors.initials: D
authors.lastname: Mathieu
authors.affiliation: New York University Langone Medical Center, Department of Neurosurgery, New York, New York.
authors.forename: Paul P
authors.initials: PP
authors.lastname: Huang
authors.affiliation: University of Puerto Rico, Section of Neurological Surgery, San Juan, Puerto Rico.
authors.forename: Caleb
authors.initials: C
authors.lastname: Feliciano
authors.affiliation: University of Puerto Rico, Section of Neurological Surgery, San Juan, Puerto Rico.
authors.forename: Rafael
authors.initials: R
authors.lastname: Rodriguez-Mercado
authors.affiliation: University of Puerto Rico, Section of Neurological Surgery, San Juan, Puerto Rico.
authors.forename: Luis
authors.initials: L
authors.lastname: Almodovar
authors.affiliation: Beaumont Health System, Department of Radiation Oncology, Royal Oak, Michigan.
authors.forename: Inga S
authors.initials: IS
authors.lastname: Grills
authors.affiliation: Cleveland Clinic Foundation, Department of Neurosurgery, Cleveland, Ohio.
authors.forename: Danilo
authors.initials: D
authors.lastname: Silva
authors.affiliation: Cleveland Clinic Foundation, Department of Neurosurgery, Cleveland, Ohio.
authors.forename: Mahmoud
authors.initials: M
authors.lastname: Abbassy
authors.affiliation: Cleveland Clinic Foundation, Department of Neurosurgery, Cleveland, Ohio.
authors.forename: Symeon
authors.initials: S
authors.lastname: Missios
authors.affiliation: New York University Langone Medical Center, Department of Neurosurgery, New York, New York.
authors.forename: Douglas
authors.initials: D
authors.lastname: Kondziolka
authors.affiliation: Cleveland Clinic Foundation, Department of Neurosurgery, Cleveland, Ohio.
authors.forename: Gene H
authors.initials: GH
authors.lastname: Barnett
authors.affiliation: University of Pittsburgh, Department of Neurological Surgery, Pittsburgh, Pennsylvania.
authors.forename: L
authors.initials: L
authors.lastname: Dade Lunsford
authors.affiliation: University of Virginia, Department of Neurosurgery, Charlottesville, Virginia.
authors.forename: Jason P
authors.initials: JP
authors.lastname: Sheehan
mesh_terms: D000293:Adolescent; D002648:Child; D005260:Female; D005500:Follow-Up Studies; D006801:Humans; D038622:Internationality; D002538:Intracranial Arteriovenous Malformations / Q000453:epidemiology* / Q000601:surgery*; D016015:Logistic Models; D008297:Male; D015999:Multivariate Analysis; D019106:Postoperative Hemorrhage / Q000453:epidemiology*; D011379:Prognosis; D016634:Radiosurgery / Q000009:adverse effects*; D012189:Retrospective Studies; D012307:Risk Factors; D012737:Sex Factors; D016896:Treatment Outcome
keywords: AVM = arteriovenous malformation; Gamma Knife; ICH = intracranial hemorrhage; IGKRF = International Gamma Knife Research Foundation; RBAS = radiosurgery-based AVM score; VRAS = Virginia Radiosurgery AVM Scale; intracranial arteriovenous malformation; intracranial hemorrhages; pediatric; stereotactic radiosurgery; stroke; vascular disorders; vascular malformations