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Shih-Shan Lang, Lauren A. Beslow, Robert L. Bailey, Arastoo Vossough, Joanna Ekstrom, Gregory G. Heuer, and Phillip B. Storm

Object

The true postoperative incidence of arteriovenous malformation (AVM) recurrence in the pediatric population remains largely unreported. Some literature suggests that delayed imaging studies should be obtained at 6 months to 1 year after negative findings on a postoperative angiogram. The aim of this study was to describe the timing of AVM recurrences after resection and the neuroimaging modalities on which the recurrences were detected.

Methods

This study was performed in a retrospective cohort of all pediatric patients treated surgically for AVM resection by a single neurosurgeon between 2005 and 2010. Patients were followed after resection with MR angiography (MRA) or conventional angiography, when possible, at various time points. A visual scale for compactness of the initial AVM nidus was used, and the score was correlated with probability of recurrence after surgery.

Results

A total of 28 patients (13 female, 15 male) underwent an AVM resection. In 18 patients (64.3%) an intraoperative angiogram was obtained. In 4 cases the intraoperative angiogram revealed residual AVM, and repeat resections were performed immediately. Recurrent AVMs were found in 4 children (14.3%) at 50, 51, 56, and 60 weeks after the initial resection. Recurrence risk was 0.08 per person-year. No patient with normal results on an angiogram obtained at 1 year developed a recurrence on either a 5-year angiogram or one obtained at 18 years of age. All patients with recurrence had a compactness score of 1 (diffuse AVM); a lower compactness score was associated with recurrence (p = 0.0003).

Conclusions

All recurrences in this cohort occurred less than 15 months from the initial resection. The authors recommend intraoperative angiography to help ensure complete resection at the time of the surgery. Follow-up vascular imaging is crucial for detecting recurrent AVMs, and conventional angiography is preferred because MRA can miss smaller AVMs. One-year follow-up imaging detected these recurrences, and no one who had negative results on an angiogram obtained at 1 year had a late recurrence. However, not all of the patients have been followed for 5 years or until 18 years of age, so longer follow-up is required for these patients. A lower compactness score predicted recurrent AVM in this cohort.

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Fabio A. Frisoli, Shih-Shan Lang, Arastoo Vossough, Anne Marie Cahill, Gregory G. Heuer, Hisham M. Dahmoush, Phillip B. Storm, and Lauren A. Beslow

Object

Cerebral arteriovenous malformations (AVMs) have a higher postresection recurrence rate in children than in adults. The authors' previous study demonstrated that a diffuse AVM (low compactness score) predicts postresection recurrence. The aims of this study were to evaluate the intra- and interrater reliability of the AVM compactness score.

Methods

Angiograms of 24 patients assigned a preoperative compactness score (scale of 1–3; 1 = most diffuse, 3 = most compact) in the authors' previous study were rerated by the same pediatric neuroradiologist 9 months later. A pediatric neurosurgeon, pediatric neuroradiology fellow, and interventional radiologist blinded to each other's ratings, the original ratings, and AVM recurrence also rated each AVM's compactness. Intrarater and interrater reliability were calculated using the κ statistic.

Results

Of the 24 AVMs, scores by the original neuroradiologist were 1 in 6 patients, 2 in 16 patients, and 3 in 2 patients. Intrarater reliability was 1.0. The κ statistic among the 4 raters was 0.69 (95% CI 0.44–0.89), which indicates substantial reliability. The interrater reliability between the neuroradiologist and neuroradiology fellow was moderate (κ = 0.59 [95%CI 0.20–0.89]) and was substantial between the neuroradiologist and neurosurgeon (κ = 0.74 [95% CI 0.41–1.0]). The neuroradiologist and interventional radiologist had perfect agreement (κ = 1.0).

Conclusions

Intrarater and interrater reliability of the AVM compactness score were excellent and substantial, respectively. These results demonstrate that the AVM compactness score is reproducible. However, the neuroradiologist and interventional radiologist had perfect agreement, which indicates that the compactness score is applied most accurately by those with extensive angiography experience.

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Shih-Shan Lang, Alexander M. Tucker, Craig Schreiber, Phillip B. Storm, Hongyan Liu, Yimei Li, Rebecca Ichord, Lauren A. Beslow, Neda I. Sedora-Roman, Mougnyan Cox, Hussein Nasser, Arastoo Vossough, Michael J. Fisher, Todd J. Kilbaugh, and Jimmy W. Huh

OBJECTIVE

Digital subtraction angiography (DSA) is commonly performed after pial synangiosis surgery for pediatric moyamoya disease to assess the degree of neovascularization. However, angiography is invasive, and the risk of ionizing radiation is a concern in children. In this study, the authors aimed to identify whether arterial spin labeling (ASL) can predict postoperative angiogram grading. In addition, they sought to determine whether patients who underwent ASL imaging without DSA had similar postoperative outcomes when compared with patients who received ASL imaging and postoperative DSA.

METHODS

The medical records of pediatric patients who underwent pial synangiosis for moyamoya disease at a quaternary children’s hospital were reviewed during a 10-year period. ASL-only and ASL+DSA cohorts were analyzed. The frequency of preoperative and postoperative symptoms was analyzed within each cohort. Three neuroradiologists assigned a visual ASL grade for each patient indicating the change from the preoperative to postoperative ASL perfusion sequences. A postoperative neovascularization grade was also assigned for patients who underwent DSA.

RESULTS

Overall, 21 hemispheres of 14 patients with ASL only and 14 hemispheres of 8 patients with ASL+DSA were analyzed. The groups had similar rates of MRI evidence of acute or chronic stroke preoperatively (61.9% in the ASL-only group and 64.3% in the ASL+DSA group). In the entire cohort, transient ischemic attack (TIA) (p = 0.027), TIA composite (TIA or unexplained neurological symptoms; p = 0.0006), chronic headaches (p = 0.035), aphasia (p = 0.019), and weakness (p = 0.001) all had decreased frequency after intervention. The authors found a positive association between revascularization observed on DSA and the visual ASL grading (p = 0.048). The visual ASL grades in patients with an angiogram indicating robust neovascularization demonstrated improved perfusion when compared with the ASL grades of patients with a poor neovascularization.

CONCLUSIONS

Noninvasive ASL perfusion imaging had an association with postoperative DSA neoangiogenesis following pial synangiosis surgery in children. There were no significant postoperative stroke differences between the ASL-only and ASL+DSA cohorts. Both cohorts demonstrated significant improvement in preoperative symptoms after surgery. Further study in larger cohorts is necessary to determine whether the results of this study are validated in order to circumvent the invasive catheter angiogram.