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Nisha Gadgil, Sandi Lam, Monika Pyarali, Michael Paldino, I-Wen Pan and Robert C. Dauser

OBJECTIVE

Numerous surgical procedures facilitate revascularization of the ischemic brain in patients with moyamoya disease. Dural inversion is a technique in which flaps of dura mater centered around the middle meningeal artery are inverted, encouraging the formation of a rich collateral blood supply. This procedure has been used in combination with encephaloduroarteriosynangiosis for more than 20 years at the authors’ institution for the treatment of pediatric moyamoya disease. The objective of this study was to describe the clinical and radiographic outcomes for a cohort of consecutive pediatric moyamoya patients undergoing dural inversion.

METHODS

Clinical and radiographic data on patients who had undergone dural inversion in the period from 1997 to 2016 were reviewed. Univariate and multivariate logistic regression and Kaplan-Meier analyses were performed to assess the risk of postoperative stroke, functional outcome, and the angiographic degree of revascularization.

RESULTS

Dural inversion was performed on 169 hemispheres in 102 patients. Median follow-up was 4.3 years. Six patients (3.6% of hemispheres) suffered postoperative ischemic or hemorrhagic stroke. Overall mortality was 1.0%. Good postoperative neurological status (modified Rankin Scale [mRS] score ≤ 2) was observed in 90 patients (88%); preoperative and postoperative mRS scores showed significant improvement (p < 0.001). Eighty-six percent of hemispheres had Matsushima grade A or B revascularization. Younger age was associated with postoperative stroke and poor functional outcome. Patients with secondary moyamoya syndrome had a significantly worse radiographic outcome. The cumulative 5-year Kaplan-Meier risk for stroke was 6.4%.

CONCLUSIONS

Dural inversion is a useful technique of cerebral revascularization in pediatric moyamoya disease. A 20-year experience demonstrates the safety and efficacy of this technique with a relatively low rate of postoperative stroke, good functional outcomes, and favorable angiographic results.

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Melissa A. LoPresti, Vijay M. Ravindra, Monika Pyarali, Eric Goethe, Nisha Gadgil, Kathyrn Wagner, Peter Kan and Sandi Lam

OBJECTIVE

Intracranial arteriovenous malformations (AVMs) are a common cause of intracranial hemorrhage in children. Exploring outcomes of treatment and factors that predict recurrence may help guide the management of pediatric AVMs. In this study, the authors review a single-center experience with pediatric AVMs, aiming to identify factors contributing to recurrence and areas for improvement in treatment.

METHODS

A single-center retrospective review of patients ages 0–18 years with AVMs in the period from 2005 to 2018 was conducted. Demographic, clinical, and radiographic data were collected and examined to compare the clinical and functional outcomes of patients managed with and those managed without open microsurgical resection.

RESULTS

One hundred five patients were diagnosed and treated during the study period. Fifty (47.6%) had been treated surgically and 35 (33.3%) nonsurgically, and 20 (19%) had been treated primarily at an outside institution and thus were excluded from further analysis. Of the 50 treated surgically, 92% had complete obliteration on postoperative imaging, with the remaining patients having residual AVM and undergoing re-resection or SRS, ultimately resulting in a 96% radiographic cure rate. The surgically treated patients were followed up for an average of 3.34 years. Eighty-two percent had a modified Rankin Scale (mRS) score of 0–2. There was a recurrence rate of 12%, with the time to recurrence averaging 327.86 days (range 85–596 days). Of the 35 patients managed without open surgery, 77% underwent serial imaging surveillance, 20% underwent endovascular embolization, and 3% underwent stereotactic radiosurgery. Radiographic cure was seen in 6% of those treated nonsurgically, and all had undergone endovascular treatment alone. The nonsurgically treated patients were followed up for an average of 2.14 years. Eighty percent had an mRS score of 0–2.

CONCLUSIONS

The data revealed that patients with lower-grade AVMs treated surgically and those with higher-grade AVMs managed without surgery have good functional and clinical outcomes. A high rate of recurrence was noted, and the possible contributing factors were explored. The authors encourage further study into mechanisms to reduce AVM recurrence in pediatric patients and look forward to advances in the medical management of lesions that are considered unresectable.