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Hasan A. Zaidi, M. Yashar S. Kalani, Robert F. Spetzler, Cameron G. McDougall and Felipe C. Albuquerque

OBJECT

Pediatric cerebral arteriovenous fistulas (AVFs) are rare but potentially lethal vascular lesions. Management strategies for these lesions have undergone considerable evolution in the last decade with the advent of new endovascular, surgical, and radiosurgical technologies. This study sought to review current treatment strategies and long-term clinical outcomes at a high-volume cerebrovascular institute.

METHODS

A retrospective chart review was performed on patients with a diagnosis of cerebral AVF from 1999 to 2012. Patients with carotid-cavernous fistulas, vein of Galen malformations, and age > 18 years were excluded from final analysis. Medical history, surgical and nonsurgical treatment, and clinical outcomes were documented. Pre- and postoperative angiograms were analyzed to assess for obliteration of the fistula.

RESULTS

Seventeen patients with pial AVFs (29.4%), dural AVFs (64.7%), or mixed pial/dural AVFs (5.9%) were identified. The majority of lesions were paramedian (70.6%) and supratentorial (76.5%). The study population had a mean age of 6.4 years, with a slight male predominance (52.9%), and the most common presenting symptoms were seizures (23.5%), headaches (17.6%), congestive heart failure (11.7%), and enlarging head circumference (11.7%). Among patients who underwent intervention (n = 16), 56.3% were treated with endovascular therapy alone, 6.3% were treated with open surgery alone, and 37.5% required a multimodal approach. Overall, 93.8% of the treated patients received endovascular treatment, 43.8% received open surgery, and 12.5% received radiosurgery. Endovascular embolysates included Onyx (n = 5), N-butyl cyanoacrylate (NBCA; n = 4), or coil embolization (n = 7) with or without balloon assistance (n = 2). Complete angiographic obliteration was achieved in 87.5% at the last follow-up evaluation (mean follow-up 3.1 years). One infant with incomplete AVF obliteration died of congestive heart failure, and 1 patient with complete obliteration died of acute sinus thrombosis, with an overall complication rate of 18.8%.

CONCLUSIONS

Pediatric cerebral AVFs are challenging neurosurgical lesions. Although advancements in endovascular therapy in the last decade have greatly changed the natural course of this disease, a multidisciplinary approach remains necessary for a large subset of patients. Surgeon experience with a thorough analysis of preoperative imaging is paramount to achieving acceptable clinical outcomes.

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M. Yashar S. Kalani, Ali M. Elhadi, Wyatt Ramey, Peter Nakaji, Felipe C. Albuquerque, Cameron G. McDougall, Joseph M. Zabramski and Robert F. Spetzler

Object

Aneurysms are relatively rare in the pediatric population and tend to include a greater proportion of large and giant lesions. A subset of these large and giant aneurysms are not amenable to direct surgical clipping and require complex treatment strategies and revascularization techniques. There are limited data available on the management of these lesions in the pediatric population. This study was undertaken to evaluate the outcome of treatment of large and giant aneurysms that required microsurgical revascularization and vessel sacrifice in this population.

Methods

The authors retrospectively identified all cases in which pediatric patients (age < 18 years) with aneurysms were treated using cerebral revascularization in combination with other treatment modalities at their institution between 1989 and 2013.

Results

The authors identified 27 consecutive patients (19 male and 8 female) with 29 aneurysms. The mean age of the patients at the time of treatment was 11.5 years (median 13 years, range 1–17 years). Five patients presented with subarachnoid hemorrhage, 11 with symptoms related to mass effect, 2 with stroke, and 3 with seizures; in 6 cases, the aneurysms were incidental findings. Aneurysms were located along the internal carotid artery (n = 7), posterior cerebral artery (PCA) (n = 2), anterior cerebral artery (n = 2), middle cerebral artery (MCA) (n = 14), basilar artery (n = 2), vertebral artery (n = 1), and at the vertebrobasilar junction (n = 1). Thirteen were giant aneurysms (45%). The majority of the aneurysms were fusiform (n = 19, 66%), followed by saccular (n = 10, 34%). Three cases were previously treated using microsurgery (n = 2) or an endovascular procedure (n = 1). A total of 28 revascularization procedures were performed, including superficial temporal artery (STA) to MCA (n = 6), STA to PCA (n = 1), occipital artery to PCA (n = 1), extracranial-intracranial (EC-IC) bypass using radial artery graft (n = 3), EC-IC using a saphenous vein graft (n = 7), STA onlay (n = 3), end-to-end anastomosis (n = 1), and in situ bypasses (n = 6). Perioperative stroke occurred in 4 patients, but only one remained dependent (Glasgow Outcome Scale [GOS] score 3). At a mean clinical follow-up of 46 months (median 14 months, range 1–232 months), 26 patients had a good outcome (GOS score 4 or 5). There were no deaths. Five patients had documented occlusion of the bypass graft. The majority of aneurysms (n = 24) were obliterated at last follow-up. There was a single case of a residual aneurysm and one case of recurrence. Angiographic follow-up was unavailable in 3 cases.

Conclusions

Cerebral revascularization remains an essential tool in the treatment of complex cerebral aneurysms in children.

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Al-Wala Awad, Karam Moon, Nam Yoon, Marcus D. Mazur, M. Yashar S. Kalani, Philipp Taussky, Cameron G. McDougall, Felipe C. Albuquerque and Min S. Park

OBJECTIVE

Flow diversion has proven to be an efficacious means of treating cerebral aneurysms that are refractory to other therapeutic means. Patients with tandem aneurysms treated with flow diversion have been included in larger, previously reported series; however, there are no dedicated reports on using this technique during a single session to treat this unique subset of patients. Therefore, the authors analyzed the outcomes of patients who had undergone single-session flow diversion for the treatment of tandem aneurysms.

METHODS

The authors conducted a retrospective review of flow diversion with the Pipeline embolization device (PED) for the treatment of tandem aneurysms in a single session at 2 participating medical centers: University of Utah, Salt Lake City, Utah, and Barrow Neurological Institute, Phoenix, Arizona. Patient demographic data, aneurysm characteristics, treatment strategy and results, complications, and follow-up data were collected from the medical record and analyzed.

RESULTS

Between January 2011 and December 2015, 17 patients (12 female, 5 male) with a total of 38 aneurysms (mean size 4.7 ± 2.7 mm, mean ± SD) were treated. Sixteen patients had aneurysms in the anterior circulation, and 1 patient had tandem aneurysms in the posterior circulation. Twelve patients underwent only placement of a PED, whereas 5 underwent adjunctive coil embolization of at least 1 aneurysm. One PED was used in each of 9 patients, and 2 PEDs were required in each of 8 patients. There were 2 intraprocedural complications; however, in both instances, the patients were asymptomatic at the last follow-up. The follow-up imaging studies were available for 15 patients at a mean of 7 months after treatment (216 days, range 0–540 days). The mean initial Raymond score after treatment was 2.7 ± 0.7, and the mean final score was 1.3 ± 0.7.

CONCLUSIONS

In this series, the use of flow diversion for the treatment of tandem cerebral aneurysms had an acceptable safety profile, indicating that it should be considered as an effective therapy for this complicated subset of patients. Further prospective studies must be performed before more definitive conclusions can be made.

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Michael R. Levitt, Randall J. Hlubek, Karam Moon, M. Yashar S. Kalani, Peter Nakaji, Kris A. Smith, Andrew S. Little, Kerry Knievel, Jane W. Chan, Cameron G. McDougall and Felipe C. Albuquerque

OBJECTIVE

Cerebral venous pressure gradient (CVPG) from dural venous sinus stenosis is implicated in headache syndromes such as idiopathic intracranial hypertension (IIH). The incidence of CVPG in headache patients has not been reported.

METHODS

The authors reviewed all cerebral venograms with manometry performed for headache between January 2008 and May 2015. Patient demographics, headache etiology, intracranial pressure (ICP) measurements, and radiographic and manometric results were recorded. CVPG was defined as a difference ≥ 8 mm Hg by venographic manometry.

RESULTS

One hundred sixty-four venograms were performed in 155 patients. There were no procedural complications. Ninety-six procedures (58.5%) were for patients with IIH. The overall incidence of CVPG was 25.6% (42 of 164 procedures): 35.4% (34 of 96 procedures) in IIH patients and 11.8% (8 of 68 procedures) in non-IIH patients. Sixty procedures (36.6%) were performed in patients with preexisting shunts. Seventy-seven patients (49.7%) had procedures preceded by an ICP measurement within 4 weeks of venography, and in 66 (85.7%) of these patients, the ICP had been found to be elevated. CVPG was seen in 8.3% (n = 5) of the procedures in the 60 patients with a preexisting shunt and in 0% (n = 0) of the 11 procedures in the 77 patients with normal ICP (p < 0.001 for both). Noninvasive imaging (MR venography, CT venography) was assessed prior to venography in 112 (68.3%) of 164 cases, and dural venous sinus abnormalities were demonstrated in 73 (65.2%) of these cases; there was a trend toward CVPG (p = 0.07). Multivariate analysis demonstrated an increased likelihood of CVPG in patients with IIH (OR 4.97, 95% CI 1.71–14.47) and a decreased likelihood in patients with a preexisting shunt (OR 0.09, 95% CI 0.02–0.44).

CONCLUSIONS

CVPG is uncommon in IIH patients, rare in those with preexisting shunts, and absent in those with normal ICP.

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Christopher D. Wilson, Sam Safavi-Abbasi, Hai Sun, M. Yashar S. Kalani, Yan D. Zhao, Michael R. Levitt, Ricardo A. Hanel, Eric Sauvageau, Timothy B. Mapstone, Felipe C. Albuquerque, Cameron G. McDougall, Peter Nakaji and Robert F. Spetzler

OBJECTIVE

Aneurysmal subarachnoid hemorrhage (aSAH) may be complicated by hydrocephalus in 6.5%–67% of cases. Some patients with aSAH develop shunt dependency, which is often managed by ventriculoperitoneal shunt placement. The objectives of this study were to review published risk factors for shunt dependency in patients with aSAH, determine the level of evidence for each factor, and calculate the magnitude of each risk factor to better guide patient management.

METHODS

The authors searched PubMed and MEDLINE databases for Level A and Level B articles published through December 31, 2014, that describe factors affecting shunt dependency after aSAH and performed a systematic review and meta-analysis, stratifying the existing data according to level of evidence.

RESULTS

On the basis of the results of the meta-analysis, risk factors for shunt dependency included high Fisher grade (OR 7.74, 95% CI 4.47–13.41), acute hydrocephalus (OR 5.67, 95% CI 3.96–8.12), in-hospital complications (OR 4.91, 95% CI 2.79–8.64), presence of intraventricular blood (OR 3.93, 95% CI 2.80–5.52), high Hunt and Hess Scale score (OR 3.25, 95% CI 2.51–4.21), rehemorrhage (OR 2.21, 95% CI 1.24–3.95), posterior circulation location of the aneurysm (OR 1.85, 95% CI 1.35–2.53), and age ≥ 60 years (OR 1.81, 95% CI 1.50–2.19). The only risk factor included in the meta-analysis that did not reach statistical significance was female sex (OR 1.13, 95% CI 0.77–1.65).

CONCLUSIONS

The authors identified several risk factors for shunt dependency in aSAH patients that help predict which patients are likely to require a permanent shunt. Although some of these risk factors are not independent of each other, this information assists clinicians in identifying at-risk patients and managing their treatment.

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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.