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Michael C. Dewan, Justin Onen, Hansen Bow, Peter Ssenyonga, Charles Howard and Benjamin C. Warf

There is inadequate pediatric neurosurgical training to meet the growing burden of disease in low- and middle-income countries (LMIC). Subspecialty expertise in the management of hydrocephalus and spina bifida—two of the most common pediatric neurosurgical conditions—offers a high-yield opportunity to mitigate morbidity and avoid unnecessary death. The CURE Hydrocephalus and Spina Bifida (CHSB) fellowship offers an intensive subspecialty training program designed to equip surgeons from LMIC with the state-of-the-art surgical skills and equipment to most effectively manage common neurosurgical conditions of childhood. Prospective fellows and their home institution undergo a comprehensive evaluation before being accepted for the 8-week training period held at CURE Children’s Hospital of Uganda (CCHU) in Mbale, Uganda. The fellowship combines anatomy review, treatment paradigms, a flexible endoscopic simulation lab, daily ward and ICU rounds, radiology rounds, and clinic exposure. The cornerstone of the fellowship is the unique operative experience that includes a high volume of endoscopic third ventriculostomy with choroid plexus cauterization, myelomeningocele closure, and ventriculoperitoneal shunting, among many other procedures performed at CCHU. Upon completion, fellows return to their home institution to establish or rejuvenate a robust pediatric practice as part of a worldwide network of CHSB trainees committed to the care of underserved children. To date, the fellowship has graduated 33 surgeons from 20 different LMIC who are independently performing thousands of hydrocephalus and spina bifida operations each year.

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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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Eric W. Sankey, C. Rory Goodwin, Ignacio Jusué-Torres, Benjamin D. Elder, Jamie Hoffberger, Jennifer Lu, Ari M. Blitz and Daniele Rigamonti

OBJECT

Endoscopic third ventriculostomy (ETV) is the treatment of choice for obstructive hydrocephalus; however, the success of ETV in patients who have previously undergone shunt placement remains unclear. The present study analyzed 103 adult patients with aqueductal stenosis who underwent ETV for obstructive hydrocephalus and evaluated the effect of previous shunt placement on post-ETV outcomes.

METHODS

This study was a retrospective review of 151 consecutive patients who were treated between 2007 and 2013 with ETV for hydrocephalus. One hundred three (68.2%) patients with aqueductal stenosis causing obstructive hydrocephalus were included in the analysis. Postoperative ETV patency and aqueductal and cisternal flow were assessed by high-resolution, gradient-echo MRI. Post-ETV Mini-Mental State Examination, Timed Up and Go, and Tinetti scores were compared with preoperative values. Univariate and multivariate analyses were performed comparing the post-ETV outcomes in patients who underwent a primary (no previous shunt) ETV (n = 64) versus secondary (previous shunt) ETV (n = 39).

RESULTS

The majority of patients showed significant improvement in symptoms after ETV; however, no significant differences were seen in any of the quantitative tests performed during follow-up. Symptom recurrence occurred in 29 (28.2%) patients after ETV, after a median of 3.0 (interquartile range 0.8–8.0) months post-ETV failure. Twenty-seven (26.2%) patients required surgical revision after their initial ETV. Patients who received a secondary ETV had higher rates of symptom recurrence (p = 0.003) and surgical revision (p = 0.003), particularly in regard to additional shunt placement/revision post-ETV (p = 0.005). These differences remained significant after multivariate analysis for both symptom recurrence (p = 0.030) and surgical revision (p = 0.043).

CONCLUSIONS

Patients with obstructive hydrocephalus due to aqueductal stenosis exhibit symptomatic improvement after ETV, with a relatively low failure rate. Patients with a primary history of shunt placement who undergo ETV as a secondary intervention are at increased risk of symptom recurrence and need for surgical revision post-ETV.

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Helen J. Sims-Williams, Hugh P. Sims-Williams, Edith Mbabazi Kabachelor, James Fotheringham and Benjamin C. Warf

OBJECTIVE

Myelomeningocele (MM) is a neural tube defect complicated by neurological deficits below the level of the spinal lesion and, in many cases, hydrocephalus. Long-term survival of infants treated for MM in a low- and middle-income country has never been reported. This retrospective cohort study reports 10-year outcomes and factors affecting survival for infants undergoing MM repair at CURE Children's Hospital of Uganda.

METHODS

Patients were traced by telephone or home visit. Survival was estimated using the Kaplan-Meier method. Multivariate survival was analyzed using the Cox proportional hazards model, investigating the following variables: sex, age at surgery, weight-for-age at surgery, motor level, and presence and management of hydrocephalus.

RESULTS

A total of 145 children underwent MM repair between 2000 and 2004; complete data were available for 133 patients. The probability of 10-year survival was 55%, with 78% of deaths occurring in the first 5 years. Most of the deaths were not directly related to MM; infection and neglect were most commonly described. Lesions at motor level L-2 or above were associated with increased mortality (HR 3.176, 95% CI 1.557–6.476). Compared with repair within 48 hours of birth, surgery at 15–29 days was associated with increased mortality (HR 9.091, 95% CI 1.169–70.698).

CONCLUSIONS

Infants in low- and middle-income countries with MM can have long-term survival with basic surgical intervention. Motor level and age at surgery were significant factors influencing outcome. Education of local health care workers and families to ensure both urgent referral for initial treatment and subsequent access to basic medical care are essential to survival.

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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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Matthew D. Alvin, Jacob A. Miller, Daniel Lubelski, Benjamin P. Rosenbaum, Kalil G. Abdullah, Robert G. Whitmore, Edward C. Benzel and Thomas E. Mroz

Object

Cost-effectiveness research in spine surgery has been a prominent focus over the last decade. However, there has yet to be a standardized method developed for calculation of costs in such studies. This lack of a standardized costing methodology may lead to conflicting conclusions on the cost-effectiveness of an intervention for a specific diagnosis. The primary objective of this study was to systematically review all cost-effectiveness studies published on spine surgery and compare and contrast various costing methodologies used.

Methods

The authors performed a systematic review of the cost-effectiveness literature related to spine surgery. All cost-effectiveness analyses pertaining to spine surgery were identified using the cost-effectiveness analysis registry database of the Tufts Medical Center Institute for Clinical Research and Health Policy, and the MEDLINE database. Each article was reviewed to determine the study subject, methodology, and results. Data were collected from each study, including costs, interventions, cost calculation method, perspective of cost calculation, and definitions of direct and indirect costs if available.

Results

Thirty-seven cost-effectiveness studies on spine surgery were included in the present study. Twenty-seven (73%) of the studies involved the lumbar spine and the remaining 10 (27%) involved the cervical spine. Of the 37 studies, 13 (35%) used Medicare reimbursements, 12 (32%) used a case-costing database, 3 (8%) used cost-to-charge ratios (CCRs), 2 (5%) used a combination of Medicare reimbursements and CCRs, 3 (8%) used the United Kingdom National Health Service reimbursement system, 2 (5%) used a Dutch reimbursement system, 1 (3%) used the United Kingdom Department of Health data, and 1 (3%) used the Tricare Military Reimbursement system. Nineteen (51%) studies completed their cost analysis from the societal perspective, 11 (30%) from the hospital perspective, and 7 (19%) from the payer perspective. Of those studies with a societal perspective, 14 (38%) reported actual indirect costs.

Conclusions

Changes in cost have a direct impact on the value equation for concluding whether an intervention is cost-effective. It is essential to develop a standardized, accurate means of calculating costs. Comparability and transparency are essential, such that studies can be compared properly and policy makers can be appropriately informed when making decisions for our health care system based on the results of these studies.

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Benjamin D. Kuhns, Daniel Lubelski, Matthew D. Alvin, Jason S. Taub, Matthew J. McGirt, Edward C. Benzel and Thomas E. Mroz

OBJECT

Infections following spine surgery negatively affect patient quality of life (QOL) and impose a significant financial burden on the health care system. Postoperative wound infections occur at higher rates following dorsal cervical procedures than ventral procedures. Quantifying the health outcomes and costs associated with infections following dorsal cervical procedures may help to guide treatment strategies to minimize the deleterious consequences of these infections. Therefore, the goals of this study were to determine the cost and QOL outcomes affecting patients who developed deep wound infections following subaxial dorsal cervical spine fusions.

METHODS

The authors identified 22 (4.0%) of 551 patients undergoing dorsal cervical fusions who developed deep wound infections requiring surgical debridement. These patients were individually matched with control patients who did not develop infections. Health outcomes were assessed using the EQ-5D, Pain Disability Questionnaire (PDQ), Patient Health Questionnaire (PHQ-9), and visual analog scale (VAS). QOL outcome measures were collected preoperatively and after 6 and 12 months. Health resource utilization was recorded from patient electronic medical records over an average follow-up of 18 months. Direct costs were estimated using Medicare national payment amounts, and indirect costs were based on patients' missed workdays and income.

RESULTS

No significant differences in preoperative QOL scores were found between the 2 cohorts. At 6 months postsurgery, the noninfection cohort had significant pre- to postoperative improvement in EQ-5D (p = 0.02), whereas the infection cohort did not (p = 0.2). The noninfection cohort also had a significantly higher 6-month postoperative EQ-5D scores than the infection cohort (p = 0.04). At 1 year postsurgery, there was no significant difference in EQ-5D scores between the groups. Health care–associated costs for the infection cohort were significantly higher ($16,970 vs $7658; p < 0.0001). Indirect costs for the infection cohort and the noninfection cohort were $6495 and $2756, respectively (p = 0.03). Adjusted for inflation, the total costs for the infection cohort were $21,778 compared with $9159 for the noninfection cohort, reflecting an average cost of $12,619 associated with developing a postoperative deep wound infection (p < 0.0001).

CONCLUSIONS

Dorsal cervical infections temporarily decrease patient QOL postoperatively, but with no long-term impact; they do, however, dramatically increase the cost of care. Knowledge of the financial burden of wound infections following dorsal cervical fusion may stimulate the development and use of improved prophylactic and therapeutic techniques to manage this serious complication.

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Darin D. Dougherty, Tina Chou, Andrew K. Corse, Amanda R. Arulpragasam, Alik S. Widge, Cristina Cusin, Karleyton C. Evans, Benjamin D. Greenberg, Suzanne N. Haber and Thilo Deckersbach

OBJECTIVE

Deep brain stimulation (DBS) is a reversible, nonlesion-based treatment for patients with intractable obsessive-compulsive disorder (OCD). The first studies on DBS for OCD stimulating the ventral capsule/ventral striatum (VC/VS) yielded encouraging results for this neuroanatomical site's therapeutic efficacy. This investigation was conducted to better understand which regions of the cortico-striatal-thalamic-cortical network were acutely affected by VC/VS DBS for OCD. Furthermore, the objective was to identify which brain regions demonstrated changes in perfusion, as stimulation was applied across a dorsoventral lead axis that corresponded to different anatomical locations in the VC/VS.

METHODS

Six patients receiving VC/VS DBS for OCD underwent oxygen-15 positron emission tomography (15O-PET) scanning. Monopolar DBS was delivered at each of the 4 different electrodes on the stimulating lead in the VC/VS. The data were analyzed using SPM5. Paired t-tests were run in SPSS to identify significant changes in regional cerebral blood flow (rCBF) between stimulation conditions. Pearson's r correlations were run between these significant changes in rCBF and changes in OCD and depressive symptom severity.

RESULTS

Perfusion in the dorsal anterior cingulate cortex (dACC) significantly increased when monopolar DBS was turned on at the most ventral DBS contact, and this increase in dACC activity was correlated with reductions in depressive symptom severity (r(5) = −0.994, p = 0.001). Perfusion in the thalamus, striatum, and globus pallidus significantly increased when DBS was turned on at the most dorsal contact.

CONCLUSIONS

DBS of the VC/VS appears to modulate activity in the regions implicated in the pathophysiology of OCD. Different regions in the cortico-striatal-thalamic-cortical circuit showed increased perfusion based on whether the stimulation was more ventral or dorsal along the lead axis in the VC/VS. Evidence was found that DBS at the most ventral site was associated with clinical changes in depressive symptom severity, but not OCD symptom severity.

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Paul Gigante, Michael M. McDowell, Samuel S. Bruce, Genevieve Chirelstein, Claudia A. Chiriboga, Joseph Dutkowsky, Elizabeth Fontana, Joshua Hyman, Heakyung Kim, Dean Morgan, Toni S. Pearson, Benjamin D. Roye, David P. Roye Jr., Patricia Ryan, Michael Vitale and Richard C. E. Anderson

Object

Randomized clinical trials have established that lumbar selective dorsal rhizotomy (SDR) reduces lower-extremity tone and improves functional outcome in children with spastic cerebral palsy. Significant data exist to support a secondary effect on upper-extremity function in patients with upper-extremity spasticity. The effects of SDR on upper-extremity tone, however, are not well characterized. In this report, the authors sought to assess changes in upper-extremity tone in individual muscle groups after SDR and tried to determine if these changes could be predicted preoperatively.

Methods

The authors retrospectively reviewed 42 children who underwent SDR at Columbia University Medical Center/Morgan Stanley Children's Hospital of NewYork-Presbyterian between 2005 and 2011. Twenty-five had upper-extremity spasticity. All underwent pre- and postoperative examination for measuring tone (Modified Ashworth Scale) and assessing functional outcome. Follow-up examinations with therapists were performed at least once at a minimum of 2 months postoperatively (mean 15 months).

Results

In the upper extremities, 23 (92%) of 25 patients had improvements of at least 1 Ashworth point in 2 or more independent motor groups on the Modified Ashworth Scale, and 12 (71%) of 17 families surveyed reported increases in motor control or spontaneous movement. The mean Modified Ashworth Scale scores for all upper-extremity muscle groups demonstrated an improvement from 1.34 to 1.22 (p < 0.001). Patients with a mean preoperative upper-extremity tone of 1.25–1.75 were most likely to benefit from reduction in tone (p = 0.0019). Proximal and pronator muscle groups were most likely to demonstrate reduced tone.

Conclusions

In addition to improvements in lower-extremity tone and function, SDR has demonstrable effects on upper extremities. Greater than 90% of our patients with elevated upper-extremity tone demonstrated reduction in tone in at least 2 muscle groups postoperatively. Patients with a mean Modified Ashworth Scale upper-extremity score of 1.25–1.75 may encounter the greatest reduction in upper-extremity tone.

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Joyce Koueik, Carolina Sandoval-Garcia, John R. W. Kestle, Brandon G. Rocque, David M. Frim, Gerald A. Grant, Robert F. Keating, Carrie R. Muh, W. Jerry Oakes, Ian F. Pollack, Nathan R. Selden, R. Shane Tubbs, Gerald F. Tuite, Benjamin Warf, Victoria Rajamanickam, Aimee Teo Broman, Victor Haughton, Susan Rebsamen, Timothy M. George and Bermans J. Iskandar

OBJECTIVE

Despite significant advances in diagnostic and surgical techniques, the surgical management of Chiari malformation type I (CM-I) with associated syringomyelia remains controversial, and the type of surgery performed is surgeon dependent. This study’s goal was to determine the feasibility of a prospective, multicenter, cohort study for CM-I/syringomyelia patients and to provide pilot data that compare posterior fossa decompression and duraplasty (PFDD) with and without tonsillar reduction.

METHODS

Participating centers prospectively enrolled children suffering from both CM-I and syringomyelia who were scheduled to undergo surgical decompression. Clinical data were entered into a database preoperatively and at 1–2 weeks, 3–6 months, and 1 year postoperatively. MR images were evaluated by 3 independent, blinded teams of neurosurgeons and neuroradiologists. The primary endpoint was improvement or resolution of the syrinx.

RESULTS

Eight clinical sites were chosen based on the results of a published questionnaire intended to remove geographic and surgeon bias. Data from 68 patients were analyzed after exclusions, and complete clinical and imaging records were obtained for 55 and 58 individuals, respectively. There was strong agreement among the 3 radiology teams, and there was no difference in patient demographics among sites, surgeons, or surgery types. Tonsillar reduction was not associated with > 50% syrinx improvement (RR = 1.22, p = 0.39) or any syrinx improvement (RR = 1.00, p = 0.99). There were no surgical complications.

CONCLUSIONS

This study demonstrated the feasibility of a prospective, multicenter surgical trial in CM-I/syringomyelia and provides pilot data indicating no discernible difference in 1-year outcomes between PFDD with and without tonsillar reduction, with power calculations for larger future studies. In addition, the study revealed important technical factors to consider when setting up future trials. The long-term sequelae of tonsillar reduction have not been addressed and would be an important consideration in future investigations.