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Benjamin C. Kennedy, Joshua Katz, Jacob Lepard, and Jeffrey P. Blount

OBJECTIVE

Stereoelectroencephalography (SEEG) has become widespread in the United States during the past decade. Many pediatric neurosurgeons practicing SEEG may not have had experience with this technique during their formal training, and the literature is mostly limited to single-center series. As a result, implementation of this relatively new technique may vary at different institutions. The authors hypothesized that aspects of SEEG experience, techniques, and outcomes would vary widely among programs across the country.

METHODS

An electronic survey with 35 questions addressing the categories of training and experience, technique, electrode locations, and outcomes was sent to 128 pediatric epilepsy surgeons who were potential SEEG users.

RESULTS

Sixty-one pediatric fellowship-trained epilepsy surgeons in the United States responded to the survey. Eighty-nine percent were actively using SEEG in their practice. Seventy-two percent of SEEG programs were in existence for less than 5 years, and 68% were using SEEG for > 70% of their invasive monitoring. Surgeons at higher-volume centers operated on younger patients (p < 0.001). Most surgeons (70%) spent 1–3 hours per case planning electrode trajectories. Two-thirds of respondents reported a median implant duration of 5–7 days, but 16% reported never having an implant duration > 5 days, and 16% reported having had implants stay in place for > 4 weeks. The median response for the median number of electrodes initially implanted was 12 electrodes, although 19% of respondents reported median implants of 5–8 electrodes and 17% reported median implants of 15–18 electrodes. Having a higher volume of SEEG cases per year was associated with a higher median number of electrodes implanted (p < 0.001). Most surgeons found SEEG helpful in defining an epileptic network and reported that most of their SEEG patients undergo focal surgical treatment.

CONCLUSIONS

SEEG has been embraced by the pediatric epilepsy surgery community. Higher case volume is correlated with a tendency to place more electrodes and operate on younger patients. For most parameters addressed in the survey, responses from surgeons clustered around a norm, though additional findings of substantial variations highlight differences in implementation and philosophy among pediatric epilepsy programs.

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Benjamin C. Kennedy, Michael B. Cloney, Richard C. E. Anderson, and Neil A. Feldstein

OBJECT

Choroid plexus papillomas (CPPs) are rare neoplasms, often found in the atrium of the lateral ventricle of infants, and cause overproduction hydrocephalus. The extensive vascularity and medially located blood supply of these tumors, coupled with the young age of the patients, can make prevention of blood loss challenging. Preoperative embolization has been advocated to reduce blood loss and prevent the need for transfusion, but this mandates radiation exposure and the additional risks of vessel injury and stroke. For these reasons, the authors present their experience using the superior parietal lobule approach to CPPs of the atrium without adjunct therapy.

METHODS

A retrospective review was conducted of all children who presented to Columbia University/Morgan Stanley Children's Hospital of New York with a CPP in the atrium of the lateral ventricle and who underwent surgery using a superior parietal lobule approach without preoperative embolization.

RESULTS

Nine children were included, with a median age of 7 months. There were no perioperative complications or new neurological deficits. All patients had intraoperative blood loss of less than 100 ml, with a mean minimum hematocrit of 26.9% (range 19.6%–36.2%). No patients required a blood transfusion. The median follow-up was 39 months, during which time no patient demonstrated residual or recurrent tumor on MRI, nor did any have an increase in ventricular size or require CSF diversion.

CONCLUSIONS

The superior parietal lobule approach is safe and effective for very young children with CPPs in the atrium of the lateral ventricle. The results suggest that preoperative embolization is not essential to avoid transfusion or achieve overall good outcomes in these patients. This management strategy avoids radiation exposure and the additional risks associated with embolization.

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Cameron Brimley, Vivek P. Buch, Jared M. Pisapia, and Benjamin C. Kennedy

Hemispheric disconnection in the form of hemispherectomy or hemispherotomy is the most effective way of treating intractable hemispheric epilepsy. Anatomical hemispherectomy approaches have largely been abandoned in most cases due to a higher risk of superficial hemosiderosis, intraoperative blood loss, hydrocephalus, prolonged hospital stay, and mortality compared to the variety of tissue-sparing hemispherotomy techniques. Disconnective hemispherotomy approaches utilize the lateral ventricle as a key component of the surgical corridor. Without a lateral ventricle, disconnective surgery becomes significantly challenging, typically leading to a hemispherectomy. The authors present the case of a patient with severe hemispheric dysplasia without a lateral ventricle on the pathologic side and detail a novel surgical technique for a prone, occipital interhemispheric, tissue-sparing, purely disconnective aventricular hemispherotomy with an excellent surgical outcome.

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Cameron Brimley, Vivek P. Buch, Jared M. Pisapia, and Benjamin C. Kennedy

Hemispheric disconnection in the form of hemispherectomy or hemispherotomy is the most effective way of treating intractable hemispheric epilepsy. Anatomical hemispherectomy approaches have largely been abandoned in most cases due to a higher risk of superficial hemosiderosis, intraoperative blood loss, hydrocephalus, prolonged hospital stay, and mortality compared to the variety of tissue-sparing hemispherotomy techniques. Disconnective hemispherotomy approaches utilize the lateral ventricle as a key component of the surgical corridor. Without a lateral ventricle, disconnective surgery becomes significantly challenging, typically leading to a hemispherectomy. The authors present the case of a patient with severe hemispheric dysplasia without a lateral ventricle on the pathologic side and detail a novel surgical technique for a prone, occipital interhemispheric, tissue-sparing, purely disconnective aventricular hemispherotomy with an excellent surgical outcome.

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Benjamin C. Kennedy, Kathleen M. Kelly, Michelle Q. Phan, Samuel S. Bruce, Michael M. McDowell, Richard C. E. Anderson, and Neil A. Feldstein

OBJECT

Symptomatic pediatric Chiari malformation Type I (CM-I) is most often treated with posterior fossa decompression (PFD), but controversy exists over whether the dura needs to be opened during PFD. While dural opening as a part of PFD has been suggested to result in a higher rate of resolution of CM symptoms, it has also been shown to lead to more frequent complications. In this paper, the authors present the largest reported series of outcomes after PFD without dural opening surgery, as well as identify risk factors for recurrence.

METHODS

The authors performed a retrospective review of 156 consecutive pediatric patients in whom the senior authors performed PFD without dural opening from 2003 to 2013. Patient demographics, clinical symptoms and signs, radiographic findings, intraoperative ultrasound results, and neuromonitoring findings were reviewed. Univariate and multivariate regression analyses were performed to determine risk factors for recurrence of symptoms and the need for reoperation.

RESULTS

Over 90% of patients had a good clinical outcome, with improvement or resolution of their symptoms at last follow-up (mean 32 months). There were no major complications. The mean length of hospital stay was 2.0 days. In a multivariate regression model, partial C-2 laminectomy was an independent risk factor associated with reoperation (p = 0.037). Motor weakness on presentation was also associated with reoperation but only with trend-level significance (p = 0.075). No patient with < 8 mm of tonsillar herniation required reoperation.

CONCLUSIONS

The vast majority (> 90%) of children with symptomatic CM-I will have improvement or resolution of symptoms after a PFD without dural opening. A non–dural opening approach avoids major complications. While no patient with tonsillar herniation < 8 mm required reoperation, children with tonsillar herniation at or below C-2 have a higher risk for failure when this approach is used.

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Benjamin C. Kennedy, Michael M. McDowell, Peter H. Yang, Caroline M. Wilson, Sida Li, Todd C. Hankinson, Neil A. Feldstein, and Richard C. E. Anderson

Object

Pediatric patients with sickle cell anemia (SCA) carry a significant risk of developing moyamoya syndrome (MMS) and brain ischemia. The authors sought to review the safety and efficacy of pial synangiosis in the treatment of MMS in children with SCA by performing a comprehensive review of all previously reported cases in the literature.

Methods

The authors retrospectively reviewed the clinical and radiographic records in 17 pediatric patients with SCA treated at the Morgan Stanley Children's Hospital of New York (MSCHONY) who developed radiological evidence of MMS and underwent pial synangiosis between 1996 and 2012. The authors then added any additional reported cases of pial synangiosis for this population in the literature for a combined analysis of clinical and radiographic outcomes.

Results

The combined data consisted of 48 pial synangiosis procedures performed in 30 patients. Of these, 27 patients (90%) presented with seizure, stroke, or transient ischemic attack, whereas 3 (10%) were referred after transcranial Doppler screening. At the time of surgery, the median age was 12 years. Thirteen patients (43%) suffered an ischemic stroke while on chronic transfusion therapy. Long-term follow-up imaging (MR angiography or catheter angiography) at a mean of 25 months postoperatively was available in 39 (81%) treated hemispheres. In 34 (87%) of those hemispheres there were demonstrable collateral vessels on imaging. There were 4 neurological events in 1590 cumulative months of follow-up, or 1 event per 33 patient-years. In the patients in whom complete data were available (MSCHONY series, n = 17), the postoperative stroke rate was reduced more than 6-fold from the preoperative rate (p = 0.0003).

Conclusions

Pial synangiosis in patients with SCA, MMS, and brain ischemia appears to be a safe and effective treatment option. Transcranial Doppler and/or MRI screening in asymptomatic patients with SCA is recommended for the diagnosis of MMS.

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Benjamin C. Kennedy, Taylor B. Nelp, Kathleen M. Kelly, Michelle Q. Phan, Samuel S. Bruce, Michael M. McDowell, Neil A. Feldstein, and Richard C. E. Anderson

OBJECT

Chiari malformation Type I (CM-I) is associated with a syrinx in 25%–85% of patients. Although posterior fossa decompression (PFD) without dural opening is an accepted treatment option for children with symptomatic CM-I, many surgeons prefer to open the dura if a syrinx exists. The purpose of this study was to investigate the frequency and timing of syrinx resolution in children undergoing PFD without dural opening for CM-I.

METHODS

A retrospective review of 68 consecutive pediatric patients with CM-I and syringomyelia who underwent PFD without dural opening was conducted. Patient demographics, presenting symptoms and signs, radiographic findings, and intraoperative ultrasound and neuromonitoring findings were studied as well as the patients’ clinical and radiographic follow-up.

RESULTS

During the mean radiographic follow-up period of 32 months, 70% of the syringes improved. Syrinx improvement occurred at a mean of 31 months postoperatively. All patients experienced symptom improvement within the 1st year, despite only 26% of patients showing radiographic improvement during that period. Patients presenting with sensory symptoms or motor weakness had a higher likelihood of having radiographic syrinx improvement postoperatively.

CONCLUSIONS

In children with CM-I and a syrinx undergoing PFD without dural opening, syrinx resolution occurs in approximately 70% of patients. Radiographic improvement of the syrinx is delayed, but this does not correlate temporally with symptom improvement. Sensory symptoms or motor weakness on presentation are associated with syrinx resolution after surgery.

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Richard C. E. Anderson, Benjamin Kennedy, Candix L. Yanes, James Garvin, Michael Needle, Peter Canoll, Neil A. Feldstein, and Jeffrey N. Bruce

Convection-enhanced delivery (CED) for the treatment of malignant gliomas is a technique that can deliver chemotherapeutic agents directly into the tumor and the surrounding interstitium through sustained, low-grade positive-pressure infusion. This allows for high local concentrations of drug within the tumor while minimizing systemic levels that often lead to dose-limiting toxicity. Diffuse intrinsic pontine gliomas (DIPGs) are universally fatal childhood tumors for which there is currently no effective treatment. In this report the authors describe CED of the topoisomerase inhibitor topotecan for the treatment of DIPG in 2 children.

As part of a pilot feasibility study, the authors treated 2 pediatric patients with DIPG. Stereotactic biopsy with frozen section confirmation of glial tumor was followed by placement of bilateral catheters for CED of topotecan during the same procedure. The first patient underwent CED 210 days after initial diagnosis, after radiation therapy and at the time of tumor recurrence, with a total dose of 0.403 mg in 6.04 ml over 100 hours. Her Karnofsky Performance Status (KPS) score was 60 before CED and 50 posttreatment. Serial MRI initially demonstrated a modest reduction in tumor size and edema, but the tumor progressed and the patient died 49 days after treatment. The second patient was treated 24 days after the initial diagnosis prior to radiation with a total dose of 0.284 mg in 5.30 ml over 100 hours. Her KPS score was 70 before CED and 50 posttreatment. Serial MRI similarly demonstrated an initial modest reduction in tumor size. The patient subsequently underwent fractionated radiation therapy, but the tumor progressed and she died 120 days after treatment.

Topotecan delivered by prolonged CED into the brainstem in children with DIPG is technically feasible. In both patients, high infusion rates (> 0.12 ml/hr) and high infusion volumes (> 2.8 ml) resulted in new neurological deficits and reduction in the KPS score, but lower infusion rates (< 0.04 ml/hr) were well tolerated. While serial MRI showed moderate treatment effect, CED did not prolong survival in these 2 patients. More studies are needed to improve patient selection and determine the optimal flow rates for CED of chemotherapeutic agents into DIPG to maximize safety and efficacy. Clinical trial registration no.: NCT00324844.