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Abbas Rattani, Coleman P. Riordan, John G. Meara and Mark R. Proctor

OBJECTIVE

Unilateral lambdoid synostosis is the premature fusion of a lambdoid suture or sutures and represents the least common form of craniosynostosis, occurring in 1 in 40,000 births. Cranial vault remodeling (CVR) and endoscopic suturectomy with helmet therapy (ES) are surgical approaches that are used to allow for normal brain growth and improved craniofacial symmetry. The authors conducted a comparative outcomes analysis of patients with lambdoid synostosis undergoing either CVR or ES.

METHODS

The authors conducted a retrospective consecutive cohort study of patients with nonsyndromic lambdoid synostosis who underwent surgical correction identified from a single-institution database of patients with craniosynostosis seen between 2000 and 2018. Cranial growth was measured in head circumference percentile and z score.

RESULTS

Nineteen patients (8 female and 11 male) with isolated unilateral lambdoid synostosis were identified (8 right and 11 left). Six underwent CVR and 13 underwent ES. No statistically significant differences were noted between surgical groups with respect to suture laterality, the patient’s sex, and length of follow-up. Patients treated with ES presented and underwent surgery at a younger age than those treated with CVR (p = 0.0002 and p = 0.0001, respectively). Operating and anesthesia time, estimated blood loss, and ICU and total hospital days were significantly lower in ES (all p < 0.05). No significant differences were observed in pre- and postoperative head circumference percentiles or z scores between groups up to 36 months postoperatively. No patients required reoperation as of last follow-up.

CONCLUSIONS

Endoscopic management of lambdoid synostosis is safe, efficient, and efficacious in terms of intraoperative and long-term cranial growth outcomes when compared to CVR. The authors recommend this minimally invasive approach as an option for correction of lambdoid synostosis in patients presenting early in their course.

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Abbas Rattani, Michael C. Dewan, Vickie Hannig, Robert P. Naftel, John C. Wellons III and Lori C. Jordan

The authors present a case of monozygotic twins with hereditary hemorrhagic telangiectasia (HHT) who experienced cerebral arteriovenous malformation (AVM) hemorrhage at a very young age. The clinical variables influencing HHT-related AVM rupture are discussed, and questions surrounding the timing of screening and intervention are explored. This is only the second known case of monozygotic HHT twins published in the medical literature, and the youngest pair of first-degree relatives to experience AVM-related cerebral hemorrhage. Evidence guiding the screening and management of familial HHT is lacking, and cases such as this underscore the need for objective and validated protocols.

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Michael C. Dewan, Ronnie E. Baticulon, Abbas Rattani, James M. Johnston Jr., Benjamin C. Warf and William Harkness

OBJECTIVE

The presence and capability of existing pediatric neurosurgical care worldwide is unknown. The objective of this study was to solicit the expertise of specialists to quantify the geographic representation of pediatric neurosurgeons, access to specialist care, and equipment and training needs globally.

METHODS

A mixed-question survey was sent to surgeon members of several international neurosurgical and general pediatric surgical societies via a web-based platform. Respondents answered questions on 5 categories: surgeon demographics and training, hospital and practice details, surgical workforce and access to neurosurgical care, training and equipment needs, and desire for international collaboration. Responses were anonymized and analyzed using Stata software.

RESULTS

A total of 459 surgeons from 76 countries responded. Pediatric neurosurgeons in high-income and upper-middle-income countries underwent formal pediatric training at a greater rate than surgeons in low- and lower-middle-income countries (89.5% vs 54.4%). There are an estimated 2297 pediatric neurosurgeons in practice globally, with 85.6% operating in high-income and upper-middle-income countries. In low- and lower-middle-income countries, roughly 330 pediatric neurosurgeons care for a total child population of 1.2 billion. In low-income countries in Africa, the density of pediatric neurosurgeons is roughly 1 per 30 million children. A higher proportion of patients in low- and lower-middle-income countries must travel > 2 hours to seek emergency neurosurgical care, relative to high-income countries (75.6% vs 33.6%, p < 0.001). Vast basic and essential training and equipment needs exist, particularly low- and lower-middle-income countries within Africa, South America, the Eastern Mediterranean, and South-East Asia. Eighty-nine percent of respondents demonstrated an interest in international collaboration for the purposes of pediatric neurosurgical capacity building.

CONCLUSIONS

Wide disparity in the access to pediatric neurosurgical care exists globally. In low- and lower-middle-income countries, wherein there exists the greatest burden of pediatric neurosurgical disease, there is a grossly insufficient presence of capable providers and equipped facilities. Neurosurgeons across income groups and geographic regions share a desire for collaboration and partnership.

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Swagoto Mukhopadhyay, Maria Punchak, Abbas Rattani, Ya-Ching Hung, James Dahm, Serena Faruque, Michael C. Dewan, Sophie Peeters, Sonal Sachdev and Kee B. Park

OBJECTIVE

In 2000, the global density of neurosurgeons was estimated at 1 per 230,000 population, which remains the most recent estimate of the global neurosurgeon workforce density. In 2004, the World Health Organization (WHO) estimated that there were 33,193 neurosurgeons worldwide, including trainees. There have been no updates to this estimate in the past decade. Moreover, only WHO region–level granularity regarding neurosurgeon distribution exists; country-level estimates are limited. The neurosurgery workforce is a crucial component to meeting the growing burden of neurosurgical diseases, which not only represent high absolute incidences and prevalences, but also represent correspondingly high disability-adjusted life years affecting hundreds of millions of people worldwide. Combining the lack of knowledge about the availability of the neurosurgical workforce and the increasing demand for neurosurgical services underscores the need for a system of neurosurgical workforce density surveillance.

METHODS

This study involved 3 key steps: 1) global survey/literature review to obtain the number of working neurosurgeons per WHO-recognized country, 2) regression to interpolate any missing data, and 3) calculation of workforce densities and comparison to available historical data by WHO region.

RESULTS

Data for 198 countries were collected (158) or interpolated (40). The global total number of neurosurgeons was estimated at 49,940. Overall, neurosurgeon density ranged from 0 to 58.95 (standardized to per 1,000,000 population) with a median of 3.56 (IQR 0.29–8.26). Thirty-three countries were found to have no neurosurgeons (zero). The highest density, 58.95, was in Japan, where 7495 neurosurgeons are taking care of a population of 127,131,800.

CONCLUSIONS

In 2015, the Lancet Commission on Global Surgery estimated that 143 million additional surgical procedures are needed in low- and middle-income countries each year, and a subsequent study revealed that approximately 15% of those surgical procedures are neurosurgical. Based on our results, we can conclude that there are approximately 49,940 neurosurgeons currently, worldwide. The availability of neurosurgeons appears to have increased in all geographic regions over the past decade, with Southeast Asia experiencing the greatest growth. Such remarkable expansion should be assessed to determine factors that could play a role in other regions where the acceleration of growth would be beneficial.

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Kerry A. Vaughan, Christian Lopez Ramos, Vivek P. Buch, Rania A. Mekary, Julia R. Amundson, Meghal Shah, Abbas Rattani, Michael C. Dewan and Kee B. Park

OBJECTIVE

Epilepsy is one of the most common neurological disorders, yet its global surgical burden has yet to be characterized. The authors sought to compile the most current epidemiological data to quantify global prevalence and incidence, and estimate global surgically treatable epilepsy. Understanding regional and global epilepsy trends and potential surgical volume is crucial for future policy efforts and resource allocation.

METHODS

The authors performed a systematic literature review and meta-analysis to determine the global incidence, lifetime prevalence, and active prevalence of epilepsy; to estimate surgically treatable epilepsy volume; and to evaluate regional trends by WHO regions and World Bank income levels. Data were extracted from all population-based studies with prespecified methodological quality across all countries and demographics, performed between 1990 and 2016 and indexed on PubMed, EMBASE, and Cochrane. The current and annual new case volumes for surgically treatable epilepsy were derived from global epilepsy prevalence and incidence.

RESULTS

This systematic review yielded 167 articles, across all WHO regions and income levels. Meta-analysis showed a raw global prevalence of lifetime epilepsy of 1099 per 100,000 people, whereas active epilepsy prevalence is slightly lower at 690 per 100,000 people. Global incidence was found to be 62 cases per 100,000 person-years. The meta-analysis predicted 4.6 million new cases of epilepsy annually worldwide, a prevalence of 51.7 million active epilepsy cases, and 82.3 million people with any lifetime epilepsy diagnosis. Differences across WHO regions and country incomes were significant. The authors estimate that currently 10.1 million patients with epilepsy may be surgical treatment candidates, and 1.4 million new surgically treatable epilepsy cases arise annually. The highest prevalences are found in Africa and Latin America, although the highest incidences are reported in the Middle East and Latin America. These regions are primarily low- and middle-income countries; as expected, the highest disease burden falls disproportionately on regions with the fewest healthcare resources.

CONCLUSIONS

Understanding of the global epilepsy burden has evolved as more regions have been studied. This up-to-date worldwide analysis provides the first estimate of surgical epilepsy volume and an updated comprehensive overview of current epidemiological trends. The disproportionate burden of epilepsy on low- and middle-income countries will require targeted diagnostic and treatment efforts to reduce the global disparities in care and cost. Quantifying global epilepsy provides the first step toward restructuring the allocation of healthcare resources as part of global healthcare system strengthening.

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Michael C. Dewan, Abbas Rattani, Graham Fieggen, Miguel A. Arraez, Franco Servadei, Frederick A. Boop, Walter D. Johnson, Benjamin C. Warf and Kee B. Park

OBJECTIVE

Worldwide disparities in the provision of surgical care result in otherwise preventable disability and death. There is a growing need to quantify the global burden of neurosurgical disease specifically, and the workforce necessary to meet this demand.

METHODS

Results from a multinational collaborative effort to describe the global neurosurgical burden were aggregated and summarized. First, country registries, third-party modeled data, and meta-analyzed published data were combined to generate incidence and volume figures for 10 common neurosurgical conditions. Next, a global mapping survey was performed to identify the number and location of neurosurgeons in each country. Finally, a practitioner survey was conducted to quantify the proportion of disease requiring surgery, as well as the median number of neurosurgical cases per annum. The neurosurgical case deficit was calculated as the difference between the volume of essential neurosurgical cases and the existing neurosurgical workforce capacity.

RESULTS

Every year, an estimated 22.6 million patients suffer from neurological disorders or injuries that warrant the expertise of a neurosurgeon, of whom 13.8 million require surgery. Traumatic brain injury, stroke-related conditions, tumors, hydrocephalus, and epilepsy constitute the majority of essential neurosurgical care worldwide. Approximately 23,300 additional neurosurgeons are needed to address more than 5 million essential neurosurgical cases—all in low- and middle-income countries—that go unmet each year. There exists a gross disparity in the allocation of the surgical workforce, leaving large geographic treatment gaps, particularly in Africa and Southeast Asia.

CONCLUSIONS

Each year, more than 5 million individuals suffering from treatable neurosurgical conditions will never undergo therapeutic surgical intervention. Populations in Africa and Southeast Asia, where the proportion of neurosurgeons to neurosurgical disease is critically low, are especially at risk. Increasing access to essential neurosurgical care in low- and middle-income countries via neurosurgical workforce expansion as part of surgical system strengthening is necessary to prevent severe disability and death for millions with neurological disease.

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Michael C. Dewan, Abbas Rattani, Rania Mekary, Laurence J. Glancz, Ismaeel Yunusa, Ronnie E. Baticulon, Graham Fieggen, John C. Wellons III, Kee B. Park and Benjamin C. Warf

OBJECTIVE

Hydrocephalus is one of the most common brain disorders, yet a reliable assessment of the global burden of disease is lacking. The authors sought a reliable estimate of the prevalence and annual incidence of hydrocephalus worldwide.

METHODS

The authors performed a systematic literature review and meta-analysis to estimate the incidence of congenital hydrocephalus by WHO region and World Bank income level using the MEDLINE/PubMed and Cochrane Database of Systematic Reviews databases. A global estimate of pediatric hydrocephalus was obtained by adding acquired forms of childhood hydrocephalus to the baseline congenital figures using neural tube defect (NTD) registry data and known proportions of posthemorrhagic and postinfectious cases. Adult forms of hydrocephalus were also examined qualitatively.

RESULTS

Seventy-eight articles were included from the systematic review, representative of all WHO regions and each income level. The pooled incidence of congenital hydrocephalus was highest in Africa and Latin America (145 and 316 per 100,000 births, respectively) and lowest in the United States/Canada (68 per 100,000 births) (p for interaction < 0.1). The incidence was higher in low- and middle-income countries (123 per 100,000 births; 95% CI 98–152 births) than in high-income countries (79 per 100,000 births; 95% CI 68–90 births) (p for interaction < 0.01). While likely representing an underestimate, this model predicts that each year, nearly 400,000 new cases of pediatric hydrocephalus will develop worldwide. The greatest burden of disease falls on the African, Latin American, and Southeast Asian regions, accounting for three-quarters of the total volume of new cases. The high crude birth rate, greater proportion of patients with postinfectious etiology, and higher incidence of NTDs all contribute to a case volume in low- and middle-income countries that outweighs that in high-income countries by more than 20-fold. Global estimates of adult and other forms of acquired hydrocephalus are lacking.

CONCLUSIONS

For the first time in a global model, the annual incidence of pediatric hydrocephalus is estimated. Low- and middle-income countries incur the greatest burden of disease, particularly those within the African and Latin American regions. Reliable incidence and burden figures for adult forms of hydrocephalus are absent in the literature and warrant specific investigation. A global effort to address hydrocephalus in regions with the greatest demand is imperative to reduce disease incidence, morbidity, mortality, and disparities of access to treatment.

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Michael C. Dewan, Abbas Rattani, Saksham Gupta, Ronnie E. Baticulon, Ya-Ching Hung, Maria Punchak, Amit Agrawal, Amos O. Adeleye, Mark G. Shrime, Andrés M. Rubiano, Jeffrey V. Rosenfeld and Kee B. Park

OBJECTIVE

Traumatic brain injury (TBI)—the “silent epidemic”—contributes to worldwide death and disability more than any other traumatic insult. Yet, TBI incidence and distribution across regions and socioeconomic divides remain unknown. In an effort to promote advocacy, understanding, and targeted intervention, the authors sought to quantify the case burden of TBI across World Health Organization (WHO) regions and World Bank (WB) income groups.

METHODS

Open-source epidemiological data on road traffic injuries (RTIs) were used to model the incidence of TBI using literature-derived ratios. First, a systematic review on the proportion of RTIs resulting in TBI was conducted, and a meta-analysis of study-derived proportions was performed. Next, a separate systematic review identified primary source studies describing mechanisms of injury contributing to TBI, and an additional meta-analysis yielded a proportion of TBI that is secondary to the mechanism of RTI. Then, the incidence of RTI as published by the Global Burden of Disease Study 2015 was applied to these two ratios to generate the incidence and estimated case volume of TBI for each WHO region and WB income group.

RESULTS

Relevant articles and registries were identified via systematic review; study quality was higher in the high-income countries (HICs) than in the low- and middle-income countries (LMICs). Sixty-nine million (95% CI 64–74 million) individuals worldwide are estimated to sustain a TBI each year. The proportion of TBIs resulting from road traffic collisions was greatest in Africa and Southeast Asia (both 56%) and lowest in North America (25%). The incidence of RTI was similar in Southeast Asia (1.5% of the population per year) and Europe (1.2%). The overall incidence of TBI per 100,000 people was greatest in North America (1299 cases, 95% CI 650–1947) and Europe (1012 cases, 95% CI 911–1113) and least in Africa (801 cases, 95% CI 732–871) and the Eastern Mediterranean (897 cases, 95% CI 771–1023). The LMICs experience nearly 3 times more cases of TBI proportionally than HICs.

CONCLUSIONS

Sixty-nine million (95% CI 64–74 million) individuals are estimated to suffer TBI from all causes each year, with the Southeast Asian and Western Pacific regions experiencing the greatest overall burden of disease. Head injury following road traffic collision is more common in LMICs, and the proportion of TBIs secondary to road traffic collision is likewise greatest in these countries. Meanwhile, the estimated incidence of TBI is highest in regions with higher-quality data, specifically in North America and Europe.

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Michael C. Dewan, Abbas Rattani, Ronnie E. Baticulon, Serena Faruque, Walter D. Johnson, Robert J. Dempsey, Michael M. Haglund, Blake C. Alkire, Kee B. Park, Benjamin C. Warf and Mark G. Shrime

OBJECTIVE

The global magnitude of neurosurgical disease is unknown. The authors sought to estimate the surgical and consultative proportion of diseases commonly encountered by neurosurgeons, as well as surgeon case volume and perceived workload.

METHODS

An electronic survey was sent to 193 neurosurgeons previously identified via a global surgeon mapping initiative. The survey consisted of three sections aimed at quantifying surgical incidence of neurological disease, consultation incidence, and surgeon demographic data. Surgeons were asked to estimate the proportion of 11 neurological disorders that, in an ideal world, would indicate either neurosurgical operation or neurosurgical consultation. Respondent surgeons indicated their confidence level in each estimate. Demographic and surgical practice characteristics—including case volume and perceived workload—were also captured.

RESULTS

Eighty-five neurosurgeons from 57 countries, representing all WHO regions and World Bank income levels, completed the survey. Neurological conditions estimated to warrant neurosurgical consultation with the highest frequency were brain tumors (96%), spinal tumors (95%), hydrocephalus (94%), and neural tube defects (92%), whereas stroke (54%), central nervous system infection (58%), and epilepsy (40%) carried the lowest frequency. Similarly, surgery was deemed necessary for an average of 88% cases of hydrocephalus, 82% of spinal tumors and neural tube defects, and 78% of brain tumors. Degenerative spine disease (42%), stroke (31%), and epilepsy (24%) were found to warrant surgical intervention less frequently. Confidence levels were consistently high among respondents (lower quartile > 70/100 for 90% of questions), and estimates did not vary significantly across WHO regions or among income levels. Surgeons reported performing a mean of 245 cases annually (median 190). On a 100-point scale indicating a surgeon’s perceived workload (0—not busy, 100—overworked), respondents selected a mean workload of 75 (median 79).

CONCLUSIONS

With a high level of confidence and strong concordance, neurosurgeons estimated that the vast majority of patients with central nervous system tumors, hydrocephalus, or neural tube defects mandate neurosurgical involvement. A significant proportion of other common neurological diseases, such as traumatic brain and spinal injury, vascular anomalies, and degenerative spine disease, demand the attention of a neurosurgeon—whether via operative intervention or expert counsel. These estimates facilitate measurement of the expected annual volume of neurosurgical disease globally.

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Faith C. Robertson, Jacob R. Lepard, Rania A. Mekary, Matthew C. Davis, Ismaeel Yunusa, William B. Gormley, Ronnie E. Baticulon, Muhammad Raji Mahmud, Basant K. Misra, Abbas Rattani, Michael C. Dewan and Kee B. Park

OBJECTIVE

Central nervous system (CNS) infections cause significant morbidity and mortality and often require neurosurgical intervention for proper diagnosis and treatment. However, neither the international burden of CNS infection, nor the current capacity of the neurosurgical workforce to treat these diseases is well characterized. The objective of this study was to elucidate the global incidence of surgically relevant CNS infection, highlighting geographic areas for targeted improvement in neurosurgical capacity.

METHODS

A systematic literature review and meta-analysis were performed to capture studies published between 1990 and 2016. PubMed, EMBASE, and Cochrane databases were searched using variations of terms relating to CNS infection and epidemiology (incidence, prevalence, burden, case fatality, etc.). To deliver a geographic breakdown of disease, results were pooled using the random-effects model and stratified by WHO region and national income status for the different CNS infection types.

RESULTS

The search yielded 10,906 studies, 154 of which were used in the final qualitative analysis. A meta-analysis was performed to compute disease incidence by using data extracted from 71 of the 154 studies. The remaining 83 studies were excluded from the quantitative analysis because they did not report incidence. A total of 508,078 cases of CNS infections across all studies were included, with a total sample size of 130,681,681 individuals. Mean patient age was 35.8 years (range: newborn to 95 years), and the male/female ratio was 1:1.74. Among the 71 studies with incidence data, 39 were based in high-income countries, 25 in middle-income countries, and 7 in low-income countries. The pooled incidence of studied CNS infections was consistently highest in low-income countries, followed by middle- and then high-income countries. Regarding WHO regions, Africa had the highest pooled incidence of bacterial meningitis (65 cases/100,000 people), neurocysticercosis (650/100,000), and tuberculous spondylodiscitis (55/100,000), whereas Southeast Asia had the highest pooled incidence of intracranial abscess (49/100,000), and Europe had the highest pooled incidence of nontuberculous vertebral spondylodiscitis (5/100,000). Overall, few articles reported data on deaths associated with infection. The limited case fatality data revealed the highest case fatality for tuberculous meningitis/spondylodiscitis (21.1%) and the lowest for neurocysticercosis (5.5%). In all five disease categories, funnel plots assessing for publication bias were asymmetrical and suggested that the results may underestimate the incidence of disease.

CONCLUSIONS

This systematic review and meta-analysis approximates the global incidence of neurosurgically relevant infectious diseases. These results underscore the disproportionate burden of CNS infections in the developing world, where there is a tremendous demand to provide training and resources for high-quality neurosurgical care.