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Leonardo Rangel-Castilla, Steven W. Hwang, William E. Whitehead, Daniel J. Curry, Thomas G. Luerssen and Andrew Jea

Tuberculosis (TB) is a common disease worldwide that is caused by Mycobacterium tuberculosis. Tuberculosis of the spine, also called Pott disease, is the most common site of bony dissemination. Although children are disproportionately affected, spinal TB is nonetheless rare in very young children. Cases involving infants requiring surgical intervention have been previously reported, and they are often associated with greater management challenges given the technical difficulty with instrumentation in very young children.

This case involved a 3-year-old girl with TB centered at T-6, who presented with myelopathy from spinal cord compression and a severe kyphotic deformity (> 60°). She underwent a single-stage costotransversectomy for vertebral column resection, followed by reconstruction with an anterior expandable titanium cage and posterior pedicle screw instrumentation. At last follow-up, the patient was clinically and radiographically stable.

The authors report on the youngest patient with spinal TB treated surgically with this strategy and review the literature regarding prior cases involving young children. Although limited by the paucity of cases in the literature, surgical debridement and spinal fusion appear to provide a safe alternative to prolonged bed rest or casting and may offer additional benefits of a faster recovery and ambulation.

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Leonardo Rangel-Castilla, Jaime Torres-Corzo, Roberto Rodriguez Della Vecchia, Aaron Mohanty and Haring J. W. Nauta

Object

Arachnoid cysts are congenital lesions that arise during development by splitting of the arachnoid membrane. Large cysts can be adjacent to CSF pathways causing a marked midline shift and hydrocephalus. The association between a large arachnoid cyst and hydrocephalus has been commonly described as being due to a mass effect, but these previous reports have not focused closely on any associated intraventricular abnormalities.

Methods

Seven patients who were previously treated with a cystoperitoneal shunt presented with shunt failure, hydrocephalus, and/or cyst expansion. All of these patients had giant arachnoid cysts extending to the periventricular region from the original site, which was the sylvian fissure in 4 patients, and the suprasellar cistern, quadrigeminal cistern, and interhemispheric fissure in 1 patient each. Endoscopic exploration of the ventricular system and cyst fenestration was then performed in all patients.

Results

The endoscopic findings were obstruction of the cerebral aqueduct by a membrane not related to the cyst in 5 patients, occlusion of the foramen of Monro in 6, septum pellucidum hypoplasia in 2, and occlusion of the cerebral aqueduct by a quadrigeminal arachnoid cyst in 1. Endoscopic procedures performed were septum pellucidum fenestration and/or foraminoplasty in 5 patients, aqueductoplasty in 2, endoscopic third ventriculostomy in 5, fenestration of the lamina terminalis in 1, and direct cystocisternostomy in 1. After the endoscopic procedure, signs and symptoms of increased intracranial pressure and hydrocephalus improved in all patients, with a reduction in size of the cyst and the ventricle.

Conclusions

Ventricular abnormalities contributing to hydrocephalus may be associated with arachnoid cysts. These abnormalities may more likely reflect a common origin than a casual relation. Foramen of Monro stenosis and cerebral aqueduct occlusion associated with an arachnoid cyst can be more frequent than has been previously believed. In cases of periventricular giant arachnoid cysts, endoscopic exploration is a good alternative for examining the ventricular system and identifying and treating CSF obstructions caused by and/or related to arachnoid cysts.

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Jaime Gasco, Brodus Franklin, Leonardo Rangel-Castilla, Gerald A. Campbell, Mahmoud Eltorky and Paul Salinas

Angioleiomyomas are benign neoplasms most often located in the subcutaneous tissue of middle-aged individuals and usually confined to the subcuticular and deep dermal layers of the lower extremities. An intracranial site for this tumor is exceedingly rare, with very few reports documenting locations in the neuraxis. To the authors' knowledge the present case represents the first reported instance of an infratentorial angioleiomyoma. The authors conducted a review of selected English-language papers published since 1960 describing well-documented cases of intracranial vascular leiomyomas, with detailed information on the clinical presentation, radiology, pathology, and particulars of surgical management in each case.

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Steven W. Hwang, Loyola V. Gressot, Leonardo Rangel-Castilla, William E. Whitehead, Daniel J. Curry, Robert J. Bollo, Thomas G. Luerssen and Andrew Jea

Object

The most common cause of cervical spine arthrodesis in the pediatric population is instability related to congenital or traumatic pathology. Instrumenting the cervical spine can be challenging given smaller anatomical structures, less ossified bone, and future growth potential and development. Studies in adult patients have suggested that using screw constructs results in improved outcomes with lower rates of instrumentation failure. However, the pediatric literature is limited to small retrospective series. Based on a review of the literature and their own patient series, the authors report that instrumenting the pediatric cervical spine with screw constructs may be safer and more effective than using wiring techniques.

Methods

The authors reviewed the existing pediatric cervical spine arthrodesis literature and contributed 31 of their own cases from September 1, 2007, to January 1, 2011. They reviewed 204 abstracts from January 1, 1966, to December 31, 2010, and 80 manuscripts with 883 total patients were included in the review. They recorded demographic, radiographic, and outcomes data—as well as surgical details—with a focus on fusion rates and complications.

Patients were then grouped into categories based upon the procedure performed: 1) patients who underwent fusions bridging the occipitocervical junction and 2) patients who underwent fusion of the cervical spine that did not include the occiput, thus including atlantoaxial and subaxial fusions. Patients were further subdivided according to the type of instrumentation used—some had posterior cervical fusion with wiring (with or without rod implantation); others had posterior cervical fusion with screws.

Results

The entire series comprised 914 patients with a mean age of 8.30 years. Congenital abnormalities were encountered most often (in 55% of cases), and patients had a mean follow-up of 32.5 months. From the entire cohort, 242 patients (26%) experienced postsurgical complications, and 50 patients (5%) had multiple complications. The overall fusion rate was 94.4%.

For occipitocervical fusions (N = 285), both screw and wiring groups had very high fusion rates (99% and 95%, respectively, p = 0.08). However, wiring was associated with a higher complication rate. From a sample of 252 patients, 14% of those treated with screw instrumentation had complications, compared with 50% of patients treated with wiring (p < 0.05).

In cervical fusions not involving the occipitocervical junction (N = 181), screw constructs had a 99% fusion rate, whereas wire instrumentation only had an 83% fusion rate (p < 0.05). Similarly, patients who underwent screw fixation had a lower complication profile (15%) when compared with those treated with wiring constructs (54%, p < 0.05).

Conclusions

The results of this study are limited by variations in construct design, use of orthoses, follow-up duration, and newer adjuvant products promoting fusions. However, a literature review and the authors' own series of pediatric cases suggest that instrumentation of the cervical spine in children may be safer and more efficacious using screw constructs rather than wiring techniques.

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Leonardo Rangel-Castilla, Jonathan J. Russin, Hasan A. Zaidi, Eduardo Martinez-del-Campo, Min S. Park, Felipe C. Albuquerque, Cameron G. McDougall, Peter Nakaji and Robert F. Spetzler

Object

Spinal arteriovenous fistulas (AVFs) and arteriovenous malformations (AVMs) are rare, complex spinal vascular lesions that are challenging to manage. Recently, understanding of these lesions has increased thanks to neuroimaging technology. Published reports of surgical results and clinical outcome are limited to small series. The authors present a large contemporary series of patients with spinal AVFs and AVMs who were treated at Barrow Neurological Institute in Phoenix, Arizona.

Methods

Retrospective detailed review of a prospective vascular database was performed for all patients with spinal AVFs and AVMs treated between 2000 and 2013. Patient demographic data, AVF and AVM characteristics, surgical results, clinical outcomes, complications, and long-term follow-up were reviewed.

Results

Between 2000 and 2013, 110 patients (57 male and 53 female) underwent obliteration of spinal AVFs and AVMs. The mean age at presentation was 42.3 years (range 18 months–81 years). There were 44 patients with AVFs and 66 with AVMs. The AVM group included 27 intramedullary, 21 conus medullaris, 12 metameric, and 6 extradural. The most common location was thoracic spine (61%), followed by cervical (22.7%), lumbar (14.5%), and sacral (1.8%). The most common presenting signs and symptoms included paresis/paralysis (75.5%), paresthesias (60%), pain (51.8%), bowel/bladder dysfunction (41.8%), and myelopathy (36.4%). Evidence of rupture was seen in 26.4% of patients. Perioperative embolization was performed in 42% of patients. Resection was performed in 95 patients (86.4%). Embolization alone was the only treatment in 14 patients (12.7%). One patient was treated with radiosurgery alone. Angiographically verified AVF and AVM obliteration was achieved in 92 patients (83.6%). At a mean follow-up duration of 30.5 months (range 1–205 months), 43 patients (97.7%) with AVFs and 57 (86.4%) with AVMs remained functionally independent (McCormick Scale scores ≤ 2). Perioperative complications were seen in 8 patients (7%). No deaths occurred. Temporary neurological deficits were observed in 27 patients (24.5%). These temporary deficits recovered 6–8 weeks after treatment. Recurrence was identified in 6 patients (13.6%) with AVFs and 10 (15.2%) with AVMs.

Conclusions

Spinal AVFs and AVMs are complex lesions that should be considered for surgical obliteration. Over the last several decades the authors have changed surgical strategies and management to achieve better clinical outcomes. Transient neurological deficit postoperatively is a risk associated with intervention; however, clinical outcomes appear to exceed the natural history based on patients’ ability to recover during the follow-up period. Due to the recurrence rate associated with these lesions, long-term follow-up is required.

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Leonardo Rangel-Castilla, Stephan A. Munich, Naser Jaleel, Marshall C. Cress, Chandan Krishna, Ashish Sonig, Kenneth V. Snyder, Adnan H. Siddiqui and Elad I. Levy

OBJECTIVE

The Pipeline Embolization Device (PED) has become increasingly used for the treatment of intracranial aneurysms. Given its high metal surface area coverage, there is concern for the patency of branch vessels that become covered by the device. Limited data exist regarding the patency of branch vessels adjacent to aneurysms that are covered by PEDs. The authors assessed the rate of intracranial internal carotid artery, anterior circulation branch vessel patency following PED placement at their institution.

METHODS

The authors retrospectively reviewed the records of 82 patients who underwent PED treatment between 2009 and 2014 and in whom the PED was identified to cover branch vessels. Patency of the anterior cerebral, posterior communicating, anterior choroidal, and ophthalmic arteries was evaluated using digital subtraction angiography preoperatively and postoperatively after PED deployment and at longer-term follow-up.

RESULTS

Of the 127 arterial branches covered by PEDs, there were no immediate postoperative occlusions. At angiographic follow-up (mean 10 months, range 3–34.7 months), arterial side branches were occluded in 13 (15.8%) of 82 aneurysm cases and included 2 anterior cerebral arteries, 8 ophthalmic arteries, and 3 posterior communicating arteries. No cases of anterior choroidal artery occlusion were observed. Patients with branch occlusion did not experience any neurological symptoms.

CONCLUSIONS

In this large series, the longer-term rate of radiographic side branch arterial occlusion after coverage by a flow diverter was 15.8%. Terminal branch vessels, such as the anterior choroidal artery, remained patent in this series. The authors' series suggests that branch vessel occlusions are clinically silent and should not deter aneurysm treatment with flow diversion.

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Leonardo Rangel-Castilla, Gary B. Rajah, Hakeem J. Shakir, Hussain Shallwani, Sirin Gandhi, Jason M. Davies, Kenneth V. Snyder, Elad I. Levy and Adnan H. Siddiqui

OBJECTIVE

Acute tandem occlusions of the cervical internal carotid artery and an intracranial large vessel present treatment challenges. Controversy exists regarding which lesion should be addressed first. The authors sought to evaluate the endovascular approach for revascularization of these lesions at Gates Vascular Institute.

METHODS

The authors performed a retrospective review of a prospectively maintained, single-institution database. They analyzed demographic, procedural, radiological, and clinical outcome data for patients who underwent endovascular treatment for tandem occlusions. A modified Rankin Scale (mRS) score ≤ 2 was defined as a favorable clinical outcome.

RESULTS

Forty-five patients were identified for inclusion in the study. The average age of these patients was 64 years; the mean National Institutes of Health Stroke Scale score at presentation was 14.4. Fifteen patients received intravenous thrombolysis before undergoing endovascular treatment. Thirty-seven (82%) of the 45 proximal cervical internal carotid artery occlusions were atherothrombotic in nature. Thirty-eight patients underwent a proximal-to-distal approach with carotid artery stenting first, followed by intracranial thrombectomy, whereas 7 patients underwent a distal-to-proximal approach (that is, intracranial thrombectomy was performed first). Thirty-seven (82%) procedures were completed with local anesthesia. For intracranial thrombectomy procedures, aspiration alone was used in 15 cases, stent retrieval alone was used in 5, and a combination of aspiration and stent-retriever thrombectomy was used in the remaining 25. The average time to revascularization was 81 minutes. Successful recanalization (thrombolysis in cerebral infarction Grade 2b/3) was achieved in 39 (87%) patients. Mean National Institutes of Health Stroke Scale scores were 9.3 immediately postprocedure (p < 0.05) (n = 31), 5.1 at discharge (p < 0.05) (n = 31), and 3.6 at 3 months (p < 0.05) (n = 30). There were 5 in-hospital deaths (11%); and 2 patients (4.4%) had symptomatic intracranial hemorrhage within 24 hours postprocedure. Favorable outcomes (mRS score ≤ 2) were achieved at 3 months in 22 (73.3%) of 30 patients available for follow-up, with an mRS score of 3 for 7 of 30 (23%) patients.

CONCLUSIONS

Tandem occlusions present treatment challenges, but high recanalization rates were possible in the present series using acute carotid artery stenting and mechanical thrombectomy concurrently. Proximal-to-distal and aspiration approaches were most commonly used because they were safe, efficacious, and feasible. Further study in the setting of a randomized controlled trial is needed to determine the best sequence for the treatment approach and the best technology for tandem occlusion.

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Victor M. Lu, Christopher S. Graffeo, Avital Perry, Lucas P. Carlstrom, Leonardo Rangel-Castilla, Giuseppe Lanzino, Waleed Brinjikji, Eelco F. M. Wijdicks and Alejandro A. Rabinstein

OBJECTIVE

Delayed cerebral ischemia (DCI) and aneurysm rebleeding contribute to morbidity and mortality in aneurysmal subarachnoid hemorrhage (aSAH); however, the relationship between their impacts on overall functional outcome is incompletely understood.

METHODS

The authors conducted a cohort study of all aSAH during the study period from 2001 to 2016. Primary end points were overall functional outcome and ischemic aSAH sequelae, defined as delayed cerebral ischemia (DCI), DCI with infarction, symptomatic vasospasm (SV), and global cerebral edema (GCE). Outcomes were compared between the rebleed and nonrebleed cohorts overall and after propensity-score matching (PSM) for risk factors and treatment modality. Univariate and multivariate ordered logistic regression analyses for functional outcomes were performed in the PSM cohort to identify predictors of poor outcome.

RESULTS

Four hundred fifty-five aSAH cases admitted within 24 hours of aneurysm rupture were included, of which 411 (90%) experienced initial aneurysm ruptures only, while 44 (10%) had clinically confirmed rebleeding. In the overall cohort, rebleeding was associated with significantly worse functional outcome, longer intensive care unit length of stay (LOS), and GCE (all p < 0.01); treatment modality, overall LOS, DCI, DCI with infarction, and SV were nonsignificant. In the PSM analysis of 43 matched rebleed and 43 matched nonrebleed cases, only poor functional outcome and GCE remained significantly associated with rebleeding (p < 0.01 and p = 0.02, respectively). Multivariate regression identified that both rebleeding (HR 21.5, p < 0.01) and DCI (HR 10.1, p = 0.01) independently predicted poor functional outcome.

CONCLUSIONS

Rebleeding and DCI after aSAH are highly morbid and potentially deadly events after aSAH, which appear to have independent negative impacts on overall functional outcome. Early rebleeding did not significantly affect the risk of delayed ischemic complications.

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David S. Xu, Michael R. Levitt, M. Yashar S. Kalani, Leonardo Rangel-Castilla, Celene B. Mulholland, Isaac J. Abecassis, Ryan P. Morton, John D. Nerva, Adnan H. Siddiqui, Elad I. Levy, Robert F. Spetzler, Felipe C. Albuquerque and Cameron G. McDougall

OBJECTIVE

Fusiform dolichoectatic vertebrobasilar aneurysms are rare, challenging lesions. The natural history of these lesions and medium- and long-term patient outcomes are poorly understood. The authors sought to evaluate patient prognosis after diagnosis of fusiform dolichoectatic vertebrobasilar aneurysms and to identify clinical and radiographic predictors of neurological deterioration.

METHODS

The authors reviewed multiple, prospectively maintained, single-provider databases at 3 large-volume cerebrovascular centers to obtain data on patients with unruptured, fusiform, basilar artery dolichoectatic aneurysms diagnosed between January 1, 2000, and January 1, 2015.

RESULTS

A total of 50 patients (33 men, 17 women) were identified; mean clinical follow-up was 50.1 months and mean radiographic follow-up was 32.4 months. At last follow-up, 42% (n = 21) of aneurysms had progressed and 44% (n = 22) of patients had deterioration of their modified Rankin Scale scores. When patients were dichotomized into 2 groups— those who worsened and those who did not—univariate analysis showed 5 variables to be statistically significantly different: sex (p = 0.007), radiographic brainstem compression (p = 0.03), clinical posterior fossa compression (p < 0.001), aneurysmal growth on subsequent imaging (p = 0.001), and surgical therapy (p = 0.006). A binary logistic regression was then created to evaluate these variables. The only variable found to be a statistically significant predictor of clinical worsening was clinical symptoms of posterior fossa compression at presentation (p = 0.01).

CONCLUSIONS

Fusiform dolichoectatic vertebrobasilar aneurysms carry a poor prognosis, with approximately one-half of the patients deteriorating or experiencing progression of their aneurysm within 5 years. Despite being high risk, intervention—when carefully timed (before neurological decline)—may be beneficial in select patients.