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Ryan T. Muir, Shelly Wang and Benjamin C. Warf

OBJECTIVE

Pediatric hydrocephalus is one of the most common neurosurgical conditions and is a major contributor to the global burden of surgically treatable diseases. Significant health disparities exist for the treatment of hydrocephalus in developing nations due to a combination of medical, environmental, and socioeconomic factors. This review aims to provide the international neurosurgery community with an overview of the current challenges and future directions of neurosurgical care for children with hydrocephalus in low-income countries.

METHODS

The authors conducted a literature review around the topic of pediatric hydrocephalus in the context of global surgery, the unique challenges to creating access to care in low-income countries, and current international efforts to address the problem.

RESULTS

Developing countries face the greatest burden of pediatric hydrocephalus due to high birth rates and greater risk of neonatal infections. This burden is related to more general global health challenges, including malnutrition, infectious diseases, maternal and perinatal risk factors, and education gaps. Unique challenges pertaining to the treatment of hydrocephalus in the developing world include a preponderance of postinfectious hydrocephalus, limited resources, and restricted access to neurosurgical care. In the 21st century, several organizations have established programs that provide hydrocephalus treatment and neurosurgical training in Africa, Central and South America, Haiti, and Southeast Asia. These international efforts have employed various models to achieve the goals of providing safe, sustainable, and cost-effective treatment.

CONCLUSIONS

Broader commitment from the pediatric neurosurgery community, increased funding, public education, surgeon training, and ongoing surgical innovation will be needed to meaningfully address the global burden of untreated hydrocephalus.

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Shelly Wang, James Drake and Abhaya V. Kulkarni

OBJECTIVE

Spontaneous subaponeurotic fluid collection (SSFC) is an uncommon and newly described entity of unknown etiology, observed in infants less than 1 year of age. The authors report on series of infants who presented to the Hospital for Sick Children (HSC) with SSFC, focusing on the natural history of this condition.

METHODS

Data from the Hospital for Sick Children were retrospectively reviewed for the period between January 2004 and June 2015. Patient age and sex, birth history, medical history, laboratory findings, and symptoms were reviewed. SSFC location, imaging characteristics, management, and outcome were also analyzed. A MEDLINE and Embase literature search was performed on the condition, yielding previously reported cases of SSFC in the English language.

RESULTS

Nine cases involving patients who presented with SSFC during the study period were identified. The patients were 4 male and 5 female infants (age range 5 weeks to 11 months). All cases of SSFC developed spontaneously over a period of days, and the infants had no history of injuries, trauma, or hair manipulation in the immediate period preceding the development of the subgaleal collections. Six patients underwent remote forceps- or vacuum-assisted instrumented births, although none of the patients developed scalp collections or skin discoloration immediately after birth. All of the cases were managed conservatively on an outpatient (6 cases) or inpatient (3 cases) basis. In 1 case, the size of the fluid collection fluctuated over 4 months, but in all of the cases, the collections resolved spontaneously without structural or infectious complications.

CONCLUSIONS

This is the largest series describing SSFC to date and summarizes 9 cases managed at a large academic neurosurgical center. Although the specific pathophysiology of SSFC remains unknown, in some cases the condition may be associated with a remote history of instrumented delivery. SSFC occurs spontaneously without immediate preceding trauma, and an extensive hematology or child abuse workup is not necessary. A conservative approach with outpatient follow-up is advocated.

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Mostafa Fatehi, Christopher S. Ahuja, Shelly Wang and Howard J. Ginsberg

Tumoral calcinosis is an uncommon condition characterized by the calcification of periarticular soft tissue. In uremic patients the disease is secondary to metabolic disturbances in predisposed patients. The authors report the case of a 73-year-old woman who presented with a new painful cervical mass while undergoing continuous ambulatory peritoneal dialysis for long-standing end-stage renal disease (ESRD). A CT scan of the neck showed a lobulated, calcified mass in the left paraspinal soft tissue at C2–3. This mass affected the facet joint and also extended into the neural foramen but did not cause any neurological compromise. Due to the patient's significant medical comorbidities, resection was deferred and the patient was followed in the clinic. Subsequent repeat imaging has shown a significant decrease in the size of the mass. In the context of ESRD, a diagnosis of uremic tumoral calcinosis (UTC) was made. The authors conducted a search of the PubMed and EMBASE databases and identified 7 previously reported cases of UTC of the cervical spine. They present a summary of these cases and discuss the etiology, diagnosis, and management of the condition.

Although the metabolic disturbances seen in patients undergoing dialysis can lead to tumoral calcinosis, most reported cases involve large joints such as the shoulder or the hip; however, the spine can also be affected and should be considered in the differential diagnosis of patients with uremia as it can mimic aggressive bone-forming neoplasms.

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Zachary S. Hubbard, Ashish H. Shah, Michael Ragheb, Shelly Wang, Sarah Jernigan and John Ragheb

OBJECTIVE

Previous models have been utilized in other low- and middle-income countries (LMICs) to explore and assess the cost, sustainability, and effectiveness of infant hydrocephalus treatment. However, similar models have not been implemented in Haiti due to a paucity of data, epidemiology, and outcomes for hydrocephalus. Therefore, the authors utilized previously described economic modeling to estimate the annual cost and benefit of treating hydrocephalus in infants at a neurosurgery referral center, Hospital Bernard Mevs (HBM), in Port-au-Prince, Haiti.

METHODS

The authors conducted a retrospective review of data obtained in all children treated for hydrocephalus at the HBM from 2008 to 2015. The raw data were pooled with previously described surgical outcomes for hydrocephalus in other LMICs. Modeling was performed to determine outcomes, neurosurgical costs, disability-adjusted life years (DALYs), and economic benefits of Haitian hydrocephalus treatment during this time frame. Standard account methodology was employed to calculate cost per procedure. Using these formulas, the net economic benefit and cost/DALY were determined for hydrocephalus treatment at HBM from 2008 to 2015.

RESULTS

Of the 401 patients treated during the study period, 158 (39.4%) met criteria for postinfectious hydrocephalus, 54 (13.5%) had congenital hydrocephalus, 38 (9.5%) had myelomeningocele, 19 (4.7%) had aqueductal stenosis, and 132 (33%) were not placed into a category. Overall, 317 individuals underwent surgical treatment of their hydrocephalus, averting 3077 DALYs. The total cost of the procedures was $754,000, and the cost per DALY ranged between $86 and $245. The resulting net economic benefit for neurosurgical intervention ranged from $2.5 to $5.5 million.

CONCLUSIONS

This work demonstrates the substantial economic benefit of neurosurgical intervention for the treatment of pediatric hydrocephalus at a single hospital in Haiti. Based on DALYs averted, the need for additional centers offering basic neurosurgical services is apparent. A single center offering these services for several days each month was able to generate between $2.5 to $5.5 million in economic benefits, suggesting the need to develop neurosurgical capacity building in Haiti. Ultimately, prevention, screening, and early surgical treatment of these infants represent a public health and socioeconomic requisite for Haiti.

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Ghassan Awad Elkarim, Naif M. Alotaibi, Nardin Samuel, Shelly Wang, George M. Ibrahim, Aria Fallah, Alexander G. Weil and Abhaya V. Kulkarni

OBJECTIVE

A recent survey has shown that caregivers of children with shunt-treated hydrocephalus frequently use social media networks for support and information gathering. The objective of this study is to describe and assess social media utilization among users interested in hydrocephalus.

METHODS

Publicly accessible accounts and videos dedicated to the topic of hydrocephalus were comprehensively searched across 3 social media platforms (Facebook, Twitter, and YouTube) throughout March 2016. Summary statistics were calculated on standard metrics of social media popularity. A categorization framework to describe the purpose of pages, groups, accounts, channels, and videos was developed following the screening of 100 titles. Categorized data were analyzed using nonparametric tests for statistical significance.

RESULTS

The authors’ search identified 30 Facebook pages, 213 Facebook groups, 17 Twitter accounts, and 253 YouTube videos. These platforms were run by patients, caregivers, nonprofit foundations, and patient support groups. Most accounts were from the United States (n = 196), followed by the United Kingdom (n = 31), Canada (n = 17), India (n = 15), and Germany (n = 12). The earliest accounts were created in 2007, and a peak of 65 new accounts were created in 2011. The total number of users in Facebook pages exceeded those in Facebook groups (p < 0.001). The majority of users in Facebook groups were in private groups, in contrast to public groups (p < 0.001). The YouTube videos with the highest median number of views were for surgical products and treatment procedures.

CONCLUSIONS

This study presents novel observations into the characteristics of social media use in the topic of hydrocephalus. Users interested in hydrocephalus seek privacy for support communications and are attracted to treatment procedure and surgical products videos. These findings provide insight into potential avenues of hydrocephalus outreach, support, or advocacy in social media.

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Shelly Wang, Scellig Stone, Alexander G. Weil, Aria Fallah, Benjamin C. Warf, John Ragheb, Sanjiv Bhatia and Abhaya V. Kulkarni

OBJECTIVE

Endoscopic third ventriculostomy (ETV)/choroid plexus cauterization (CPC) has become an increasingly common technique for the treatment of infant hydrocephalus. Both flexible and rigid neuroendoscopy can be used, with little empirical evidence directly comparing the two. Therefore, the authors used a propensity score–matched cohort and survival analysis to assess the comparative efficacy of flexible and rigid neuroendoscopy.

METHODS

Individual data were collected through retrospective review of infants younger than 2 years of age, treated at 1 of 2 hospitals: 1) Boston Children's Hospital, exclusively utilizing flexible neuroendoscopy, and 2) Nicklaus Children's Hospital-Jackson Memorial Hospital, exclusively utilizing rigid neuroendoscopy. Patient characteristics and postoperative outcomes were assessed. A propensity score model was developed to balance patient characteristics in the case mix.

RESULTS

A propensity score model for neuroendoscope type was developed with 5 independent variables: chronological age, sex, hydrocephalus etiology, prior CSF diversion, and prepontine scarring. Propensity score decile-adjusted and 1-to-1 nearest-neighbor matching analysis revealed that compared with flexible neuroendoscopy, rigid neuroendoscopy had an ETV/CPC failure odds ratio (OR) of 1.43 (p = 0.31) and 1.31 (p = 0.47), respectively, compared with an unadjusted OR of 2.40 (p = 0.034). Furthermore, in a Cox regression analysis controlled by propensity score, rigid neuroendoscopy had a hazard ratio (HR) of 1.10 (p = 0.70), compared with an unadjusted HR of 1.61 (p = 0.031).

CONCLUSIONS

Although unadjusted analysis suggested worse ETV/CPC outcomes for infants treated by rigid neuroendoscopy, much of the difference could be attributed to the case mix and other predictors of outcome. A larger sample observational study or randomized controlled trials are required to provide evidence-based guidelines on ETV/CPC technique.

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Eric L. Chang, Almon S. Shiu, Ehud Mendel, Leni A. Mathews, Anita Mahajan, Pamela K. Allen, Jeffrey S. Weinberg, Barry W. Brown, Xin Shelly Wang, Shiao Y. Woo, Charles Cleeland, Moshe H. Maor and Laurence D. Rhines

Object.

The authors report data concerning the safety, effectiveness, and patterns of failure obtained in a Phase I/II study of stereotactic body radiotherapy (SBRT) for spinal metastatic tumors.

Methods.

Sixty-three cancer patients underwent near-simultaneous computed tomography–guided SBRT. Spinal magnetic resonance imaging was conducted at baseline and at each follow-up visit. The National Cancer Institute Common Toxicity Criteria 2.0 assessments were used to evaluate toxicity.

Results.

The median tumor volume of 74 spinal metastatic lesions was 37.4 cm3 (range 1.6–358 cm3). No neuropathy or myelopathy was observed during a median follow-up period of 21.3 months (range 0.9–49.6 months). The actuarial 1-year tumor progression–free incidence was 84% for all tumors. Pattern-of-failure analysis showed two primary mechanisms of failure: 1) recurrence in the bone adjacent to the site of previous treatment, and 2) recurrence in the epidural space adjacent to the spinal cord. Grade 3 or 4 toxicities were limited to acute Grade 3 nausea, vomiting, and diarrhea (one case); Grade 3 dysphagia and trismus (one case); and Grade 3 noncardiac chest pain (one case). There was no subacute or late Grade 3 or 4 toxicity.

Conclusions.

Analysis of the data obtained in the present study supports the safety and effectiveness of SBRT in cases of spinal metastatic cancer. The authors consider it prudent to routinely treat the pedicles and posterior elements using a wide bone margin posterior to the diseased vertebrae because of the possible direct extension into these structures. For patients without a history of radiotherapy, more liberal spinal cord dose constraints than those used in this study could be applied to help reduce failures in the epidural space.