Rupture risk and outcomes of giant aneurysms in pediatric patients: a multi-institutional case series and systematic review

Saksham Gupta Department of Neurosurgery, Brigham and Women’s Hospital, Harvard Medical School, Boston, Massachusetts;
Departments of Neurosurgery and

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Blake M. Hauser Department of Neurosurgery, Brigham and Women’s Hospital, Harvard Medical School, Boston, Massachusetts;

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Joshua S. Catapano Department of Neurosurgery, Barrow Neurologic Institute, St. Joseph’s Medical Center, Phoenix; and

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Dara Farhadi Department of Neurosurgery, Barrow Neurologic Institute, St. Joseph’s Medical Center, Phoenix; and
Department of Neurosurgery, Banner University Medical Center/The University of Arizona, Tucson, Arizona

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Patrick R. Ng Department of Neurosurgery, Brigham and Women’s Hospital, Harvard Medical School, Boston, Massachusetts;

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Joshua D. Bernstock Department of Neurosurgery, Brigham and Women’s Hospital, Harvard Medical School, Boston, Massachusetts;
Departments of Neurosurgery and

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Alfred Pokmeng See Departments of Neurosurgery and
Interventional Neuroradiology, Boston Children’s Hospital, Harvard Medical School, Boston, Massachusetts;

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Michael T. Lawton Department of Neurosurgery, Barrow Neurologic Institute, St. Joseph’s Medical Center, Phoenix; and

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Edward R. Smith Departments of Neurosurgery and

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Rose Du Department of Neurosurgery, Brigham and Women’s Hospital, Harvard Medical School, Boston, Massachusetts;

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OBJECTIVE

Giant aneurysms in pediatric patients are vascular lesions that can cause significant neurological morbidity and mortality. Their rarity has precluded large cohort studies to inform their management. The objective of this study was to understand the clinical course and outcomes of giant aneurysms in pediatric patients.

METHODS

The authors performed a multi-institutional cohort study of cases from Boston Children’s Hospital and Barrow Neurological Institute, as well as a systematic review and pooled cohort analysis of previously reported cases using descriptive statistics and multivariate regression modeling.

RESULTS

Fifteen patients were included in the multi-institutional cohort, and an additional 88 patients were included from 14 series, yielding 103 patients within the pooled cohort. Among the pooled cohort, the most common aneurysm locations were in the middle cerebral artery (36%), internal carotid artery (27%), vertebral artery (11%), and vertebrobasilar junction (8%). Within 69 cases containing radiographic data in the analysis, 38% of aneurysms were saccular. Twenty-eight cases presented with aneurysm rupture (28%), including 0% of cavernous carotid aneurysms, 26% of other anterior circulation aneurysms, and 44% of posterior circulation aneurysms (p = 0.003). In multivariate analysis, posterior circulation location (OR 2.66, 95% CI 1.03–6.86) and younger age (OR 0.90 per year, 95% CI 0.81–1.00) were associated with aneurysm rupture presentation. Most cases were treated (97%) rather than observed (3%). The mortality rate was 3% for unruptured aneurysms and 18% for ruptured aneurysms. A favorable neurological outcome occurred in 80% of unruptured aneurysm cases and 54% of ruptured cases. In multivariate analysis, unruptured aneurysm presentation (OR 3.74, 95% CI 1.24–11.29) and endovascular treatment modality (OR 5.05, 95% CI 1.56–16.29) were associated with a favorable outcome.

CONCLUSIONS

Giant aneurysms are rare entities in pediatric patients that are unlikely to be discovered incidentally and usually merit treatment. Most patients survive with good neurological outcome, even in ruptured aneurysm cases. These data reveal that posterior circulation location and younger age are risk factors that correlate with an increased risk of aneurysm rupture.

ABBREVIATIONS

BA = basilar artery; BCH = Boston Children’s Hospital; BNI = Barrow Neurological Institute; CN = cranial nerve; ePVO = endovascular parent vessel occlusion; GOS = Glasgow Outcome Scale; GRADE = Grading of Recommendations Assessment, Development and Evaluation; HH = Hunt and Hess; ICA = internal carotid artery; MCA = middle cerebral artery; mRS = modified Rankin Scale; PCA = posterior cerebral artery; ROBINS-I = Risk of Bias in Non-randomized Studies – of Interventions; VA = vertebral artery; VBJ = vertebrobasilar junction.

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Illustration from Caklili et al. (pp 223–235). © Savas Ceylan, published with permission.

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