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Heather J McCrea, Jacques Lara-Reyna, Imali Perera, Rafael Uribe, Silky Chotai, Nicole Savage, Eliza H Hersh, Therese Haussner, and Mark M Souweidane

OBJECTIVE

The rarity of colloid cysts in children makes it difficult to characterize this entity and offer meaningful advice on treatment. Infrequent case reports exist, but to date there has been no age-specific assessment. The purpose of this study was to define any differences between children and adults who are evaluated and treated for colloid cysts of the third ventricle.

METHODS

Patients with colloid cysts were reviewed and stratified by age. Individuals ≤ 18 years of age were defined as pediatric patients and those > 18 years of age as adults. Clinical and radiographic data, treatment, and postoperative outcomes were compared between both groups. Bivariate analysis was conducted using the Fisher exact test for categorical variables and Mann-Whitney U-test for continuous variables.

RESULTS

Of 132 endoscopic resections (121 primary, 10 secondary, and 1 tertiary) of a colloid cyst, 9 (6.8%) were performed in pediatric patients (mean age 14.1 years, range 9–18 years) and 123 (93.2%) were performed in adult patients (mean age 43.8 years, range 19–73 years). Cases were found incidentally more commonly in pediatric than adult patients (66.7% vs 37.4%, p > 0.05), and pediatric patients had lower rates of hydrocephalus than adult patients (11.1% vs 63.4%, p < 0.05). Acute decompensation at presentation was found in 8 adults (6.5%) but no children. Complete cyst removal (88.9% vs 90.2%, p > 0.05) and length of stay (1.6 days vs 2.9 days, p > 0.05) were not significantly different between the groups. Postoperative complications (6.5% in adults, 0% in children) and recurrence (2.4% in adults, 0% in children) were rare in both groups, and there were no treatment-related deaths. The mean postoperative radiological follow-up was longer in pediatric patients (45 months, range 4–89 months) than adults (44.1 months, range 1–171 months).

CONCLUSIONS

While differences exist between children and adults regarding colloid cyst presentation, these are in keeping with the predicted evolution of a slow-growing lesion. Consistent with this observation, children had lower rates of hydrocephalus and a smaller mean maximal cyst diameter. Contrary to the published literature, however, sudden deterioration was not observed in pediatric patients but occurred in adult patients. In this limited pediatric sample size, the authors have not recorded any postoperative complications or recurrences to date. These encouraging results with endoscopic removal may positively impact future decisions related to children given their protracted life expectancy and projected rates of progression.

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Giorge Voutsas, Aaron St-Laurent, Crystal Hutchinson, Reshma Amin, James Drake, and Indra Narang

OBJECTIVE

Chiari malformation type I (CM-I) involves the herniation of the cerebellar tonsils through the foramen magnum. CM-I is associated with both obstructive sleep apnea (OSA) and central sleep apnea (CSA) in children. The primary management of symptomatic CM-I remains surgical decompression. There is, however, a paucity of data evaluating the efficacy of decompression surgery on outcomes related to sleep-disordered breathing (SDB). The objective of this study was to evaluate SDB outcomes, specifically the need for respiratory support following decompression in pediatric patients with CM-I.

METHODS

This was a retrospective chart review of all children diagnosed with CM-I when younger than 18 years of age who had polysomnography (PSG) studies pre- and postsurgery, between January 2008 and October 2018 at the Hospital for Sick Children in Toronto. Patient demographics, symptoms, PSG data, ongoing respiratory support, and surgical notes were recorded. Differences in PSG studies obtained pre- and postsurgery were compared using the Wilcoxon test for paired samples.

RESULTS

A total of 15 children with 15 interventions met inclusion criteria with pre- and postsurgery PSG studies and were considered for statistical analysis. Of the 15 subjects included for analysis, preoperative OSA was present in 2 (13.3%), CSA in 5 (33.3%), mixed SDB (both OSA and CSA) in 4 (26.7%), and no significant SDB in 4 (26.7%). Postoperatively, OSA was present in 3 (20.0%), CSA in 4 (26.7%), mixed SDB in 0 (0%), and no significant SDB in 8 (53.3%). The presence of severe OSA decreased from 4/15 (26.7%) to 2/15 (13.3%) postoperatively, and severe CSA decreased from 5/15 (33.3%) to 2/15 (13.3%) postoperatively. Following decompression surgery, 7/15 subjects (46.7%) required positive airway pressure for management of their SDB. Overall, significant improvements were observed in a number of respiratory parameters following decompression including the following: the total apnea-hypopnea index (AHI) (17.5 ± 48.2 vs 6.1 ± 32.7 events/hour; p = 0.001), obstructive AHI (2.1 ± 16.1 vs 1.0 ± 6.6 events/hour; p = 0.005), central AHI (6.3 ± 48.9 vs 2.7 ± 33.0 events/hour; p = 0.005), and the desaturation index (16.7 ± 49.6 vs 3.8 ± 25.3; p = 0.001).

CONCLUSIONS

Although decompression surgery led to a significant reduction in obstructive and central events, many children continued to have persistent SDB and required additional positive airway pressure therapy. This information is important and relevant for anticipatory guidance around decompression surgery and the necessity for respiratory support for the management of SDB in pediatric patients with CM-I.

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Elizabeth N Alford, Travis J Atchley, Tofey J Leon, Nicholas M. B Laskay, Anastasia A Arynchyna, Burkely P Smith, Inmaculada Aban, James M Johnston, Jeffrey P Blount, Curtis J Rozzelle, W. Jerry Oakes, and Brandon G Rocque

OBJECTIVE

In Chiari malformation type I (CM-I), a variety of imaging findings have been purported to be important; however, results have been inconclusive, inconsistent, or not replicated in independent studies. The purpose of this study was to report imaging characteristics for a large cohort of patients with CM-I and identify the imaging findings associated with surgical decompression.

METHODS

Patients were identified using ICD-9 codes for CM-I for the period from 1996 to 2017. After review of the medical records, patients were excluded if they 1) did not have a diagnosis of CM-I, 2) were not evaluated by a neurosurgeon, or 3) did not have available preoperative MRI. Retrospective chart review was performed to collect demographic and clinical data. Imaging parameters were measured according to the Chiari I Malformation Common Data Elements.

RESULTS

A total of 731 patients were included for analysis, having a mean follow-up duration of 25.5 months. The mean age at presentation was 8.5 years. The mean tonsil position was 11.4 mm below the foramen magnum, and 62.8% of patients had a pegged tonsil shape. Two hundred patients (27.4%) underwent surgery for life-dominating tussive headache, lower cranial nerve dysfunction, syrinx, and/or brainstem dysfunction. Surgical treatment was associated with a syrinx (OR 20.4, 95% CI 12.3–33.3, p < 0.0001), CM-1.5 (OR 1.797, 95% CI 1.08–2.98, p = 0.023), lower tonsil position (OR 1.130, 95% CI 1.08–1.18, p < 0.0001), and congenital fusion of cervical vertebrae (OR 5.473, 95% CI 1.08–27.8, p = 0.040). Among patients with benign CM-I, tonsil position was statistically significantly associated with future surgery.

CONCLUSIONS

Comprehensive imaging characteristics for a large cohort of patients with CM-I are reported. Analysis showed that a lower tonsillar position, a syrinx, and CM-1.5 were associated with undergoing posterior fossa decompression. This study demonstrates the importance of considering imaging findings in the context of patient symptomatology.

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Marianne I. J. Tissot, Andre E. Boyke, Alvin Onyewuenyi, Gregory Glauser, Evalyn S. Mackenzie, Bethany J. Thach, and Donald K. E. Detchou

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Philippe De Vloo, Luc Joyeux, Gregory G. Heuer, N. Scott Adzick, Jan Deprest, and Frank Van Calenbergh

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Kevin Hines, Zachary T. Wilt, Daniel Franco, Aria Mahtabfar, Nicholas Elmer, Glenn A. Gonzalez, Thiago S. Montenegro, Lohit Velagapudi, Parthik D. Patel, Maxwell Detweiler, Umma Fatema, Gregory D. Schroeder, and James Harrop

OBJECTIVE

Posterior cervical decompression and fusion (PCDF) is a commonly performed procedure to address cervical myelopathy. A significant number of these patients require revision surgery for adjacent-segment disease (ASD) or pseudarthrosis. Currently, there is no consensus among spine surgeons on the inclusion of proximal thoracic spine instrumentation. This study investigates the benefits of thoracic extension in long-segment cervical fusions and the potential drawbacks. The authors compare outcomes in long-segment subaxial cervical fusion for degenerative cervical myelopathy with caudal vertebral levels of C6, C7, and T1.

METHODS

A retrospective analysis identified 369 patients who underwent PCDF. Patients were grouped by caudal fusion level. Reoperation rates for ASD and pseudarthrosis, infection, and blood loss were examined. Data were analyzed with chi-square, 1-way ANOVA, and logistic regression.

RESULTS

The total reoperation rate for symptomatic pseudarthrosis or ASD was 4.8%. Reoperation rates, although not significant, were lower in the C3–6 group (2.6%, vs 8.3% for C3–7 and 3.8% for C3–T1; p = 0.129). Similarly, rates of infection were lower in the shorter-segment fusion without achieving statistical significance (2.6% for C3–6, vs 5.6% for C3–7 and 5.5% for C3–T1; p = 0.573). The mean blood loss was documented as 104, 125, and 224 mL for groups 1, 2, and 3, respectively (p < 0.001).

CONCLUSIONS

Given the lack of statistical difference in reoperation rates for long-segment cervical fusions ending at C6, C7, or T1, shorter fusions in high-risk surgical candidates or elderly patients may be performed without higher rates of reoperation.

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Nathan A Shlobin, Eytan Raz, Maksim Shapiro, Luke Moretti, Donald R Cantrell, Sandi K Lam, Michael C Hurley, Sameer A Ansari, Erez Nossek, Howard A Riina, Peter K Nelson, Babak S Jahromi, Ali Shaibani, and Matthew B Potts

OBJECTIVE

Cerebral aneurysms in the pediatric population are rare and optimal treatment strategies are not as well characterized as in adults. The Pipeline embolization device (PED) is an endoluminal flow diverter that is commonly used to treat aneurysms in adults, but experience with this device in children is limited. The authors sought to further characterize PED use and outcomes in this specific population by performing both a systematic review of patient-level data from studies reporting the use of the PED to treat pediatric aneurysms and a retrospective review of their experience.

METHODS

A systematic review of the PubMed, Embase, and Scopus databases was performed to identify studies reporting the use of the PED in pediatric patients (age ≤ 18 years). Disaggregated data regarding demographics, aneurysm characteristics, treatment, and outcomes were collected. Retrospective data from the authors’ two institutions were also included.

RESULTS

Thirty studies comprising patient-level data on 43 pediatric patients with 47 aneurysms were identified. An additional 9 patients with 9 aneurysms were included from the authors' institutions for a total of 52 patients with 56 aneurysms. The mean patient age was 11.1 years. Presentations included aneurysm rupture (17.3%) and symptomatic mass effect (23.1%). Aneurysms were located in the anterior circulation in 55.4% of cases, and 73.2% were described as nonsaccular. Imaging follow-up was available for 89.3% with a mean follow-up of 13.3 months. Aneurysm occlusion was reported in 75%, with 1 case each (1.8%) demonstrating significant in-stent stenosis and parent vessel occlusion. Clinical follow-up was reported in 90.4% with a mean follow-up of 14.7 months. Good functional outcomes (modified Rankin Scale score of 0–1 or Glasgow Outcome Scale score of 5) were reported in 65.4% of the total population. Two major complications were reported, including 1 death.

CONCLUSIONS

Despite substantial differences in aneurysm location and type between published pediatric and adult patient populations treated with the PED, the use of the PED in the pediatric population appears to be safe. While the short-term effectiveness is also similar to that of adults, additional studies are needed to further characterize the long-term outcomes and better define the use of this device in pediatric patients.

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Lorenzo Rinaldo, Deena M. Nasr, Kelly D. Flemming, Giuseppe Lanzino, and Waleed Brinjikji

OBJECTIVE

Symptomatic nonsaccular vertebrobasilar aneurysms (NSVBAs) are associated with high rates of aneurysm-related death. Anecdotal evidence suggests that brainstem infarction may be a harbinger of aneurysm rupture. The authors aimed to investigate the association between brainstem infarction and subsequent NSVBA rupture.

METHODS

The clinical records and radiographic imaging studies of patients presenting to the authors’ institution between 1996 and 2019 for evaluation and management of an NSVBA were retrospectively reviewed to determine the effect of perforating artery infarction on the natural history of NSVBAs. Kaplan-Meier curves for patients with and patients without perforator infarction were constructed, and predictors of aneurysm rupture were identified using a multivariate Cox proportional hazards model.

RESULTS

There were 98 patients with 591.3 person-years of follow-up who met the inclusion criteria for analysis. There were 20 patients who experienced perforator infarction during follow-up. Ten patients (10.2%) experienced aneurysm rupture during follow-up and 26 patients (26.5%) died due to aneurysm-related complications, with annual rates of rupture and aneurysm-related death of 1.7% and 4.4%, respectively. Five patients with a perforator infarction later experienced aneurysm rupture, with a median time between infarction and rupture of 3 months (range 0–35 months). On multivariate analysis, the presence of intraaneurysmal thrombus (risk ratio [RR] 4.01, 95% confidence interval [CI] 1.12–14.44, p = 0.033) and perforator infarction (RR 6.37, 95% CI 1.07–37.95, p = 0.042) were independently associated with risk of aneurysm rupture.

CONCLUSIONS

NSVBAs continue to be extremely challenging clinical entities with a poor prognosis. These results suggest that brainstem infarction due to perforating artery occlusion may be a harbinger of near-term aneurysm rupture.

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Mihir Gupta, Varun Sagi, Aditya Mittal, Anudeep Yekula, Devan Hawkins, Justin Shimizu, Pate J. Duddleston, Kathleen Thomas, Steven J. Goetsch, John F. Alksne, David W. Hodgens, Kenneth Ott, Kenneth T. Shimizu, Christopher Duma, and Sharona Ben-Haim

OBJECTIVE

Gamma Knife radiosurgery (GKRS) is an established surgical option for the treatment of trigeminal neuralgia (TN), particularly for high-risk surgical candidates and those with recurrent pain. However, outcomes after three or more GKRS treatments have rarely been reported. Herein, the authors reviewed outcomes among patients who had undergone three or more GKRS procedures for recurrent TN.

METHODS

The authors conducted a multicenter retrospective analysis of patients who had undergone at least three GKRS treatments for TN between July 1997 and April 2019 at two different institutions. Clinical characteristics, radiosurgical dosimetry and technique, pain outcomes, and complications were reviewed. Pain outcomes were scored on the Barrow Neurological Institute (BNI) scale, including time to pain relief (BNI score ≤ III) and recurrence (BNI score > III).

RESULTS

A total of 30 patients were identified, including 16 women and 14 men. Median pain duration prior to the first GKRS treatment was 10 years. Three patients (10%) had multiple sclerosis. Time to pain relief was longer after the third treatment (p = 0.0003), whereas time to pain recurrence was similar across each of the successive treatments (p = 0.842). Complete or partial pain relief was achieved in 93.1% of patients after the third treatment. The maximum pain relief achieved after the third treatment was significantly better among patients with no prior percutaneous procedures (p = 0.0111) and patients with shorter durations of pain before initiation of GKRS therapy (p = 0.0449). New or progressive facial sensory dysfunction occurred in 29% of patients after the third GKRS treatment and was reported as bothersome in 14%. One patient developed facial twitching, while another experienced persistent lacrimation. No statistically significant predictors of adverse effects following the third treatment were found. Over a median of 39 months of follow-up, 77% of patients maintained complete or partial pain relief. Three patients underwent a fourth GKRS treatment, including one who ultimately received five treatments; all of them reported sustained pain relief at the extended follow-up.

CONCLUSIONS

The authors describe the largest series to date of patients undergoing three or more GKRS treatments for refractory TN. A third treatment may produce outcomes similar to those of the first two treatments in terms of long-term pain relief, recurrence, and adverse effects.