Economic benefit of neurosurgical intervention for infant hydrocephalus in Haiti

Zachary S. Hubbard Department of Neurosurgery, Medical University of South Carolina, Charleston, South Carolina;

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Ashish H. Shah Department of Neurological Surgery, University of Miami Miller School of Medicine, Miami, Florida; and

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Michael Ragheb Department of Neurological Surgery, University of Miami Miller School of Medicine, Miami, Florida; and

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Shelly Wang Department of Neurosurgery, Medical University of South Carolina, Charleston, South Carolina;

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Sarah Jernigan Department of Neurosurgery, Carolina Neurosurgery and Spine Associates, Charlotte, North Carolina

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John Ragheb Department of Neurological Surgery, University of Miami Miller School of Medicine, Miami, Florida; and

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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.

ABBREVIATIONS

CPC = choroid plexus cauterization; DALY = disability-adjusted life year; ETV = endoscopic third ventriculostomy; HBM = Hospital Bernard Mevs; IE = income elasticity; LMIC = low- and middle-income country; PIH = postinfectious hydrocephalus; PMHSS = Project Medishare Hydrocephalus Specialty Surgery; VPS = ventriculoperitoneal shunt; VSL = value of a statistical life.

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.

In Brief

This is the first study to quantify and characterize the enormous long-term socioeconomic impact of treating hydrocephalus in Haiti. Importantly, it defines both the need and potential impact of neurosurgery in Haiti. Based on the authors’ calculations, neurosurgical management has the capability to reduce the disease burden of hydrocephalus by nearly 50%. This work demonstrates the substantial utility Haitian-based neurosurgical programs would provide to improve the health state.

Surgical intervention has the potential to significantly reduce the global burden of disease; however, in developing areas of the world such as Haiti, access to surgical treatment is limited. Surgically treatable diseases remain a major cause of worldwide mortality with approximately 17 million deaths per year. Additionally, lack of access to affordable and appropriate surgical care affects over 80% of the world’s population, with the most severe disparity evident in low- and middle-income countries (LMICs).15,20 Left untreated, the natural course of surgically treatable diseases results in a great amount of disability, morbidity, and mortality, which has deleterious effects on the healthcare state of a country.23 In Haiti, limited instruments, implants, and supplies, as well as the absence of formal neurosurgical training, have left the vast burden of neurological disease untreated.1

Among neurosurgically treated conditions, pediatric hydrocephalus remains one of the most common causes of lifelong disability. It has been estimated that over 100,000 cases per year occur in Sub-Saharan Africa alone.18,23 At the CURE Children’s Hospital of Uganda, the economic benefit of hydrocephalus treatment was estimated to be $3 million to $5 million based on disability-adjusted life years (DALYs) avoided.24 In Haiti, where access to neurosurgical care remains even more limited, the economic benefit of treating hydrocephalus may be more immense. Despite effective treatments for hydrocephalus throughout the developed world, the lack of access to treatment in Haiti has created a significant economic burden. The present study seeks to estimate the economic benefit provided by the treatment of pediatric hydrocephalus at Hospital Bernard Mevs (HBM) in Port-au-Prince, Haiti, by the Project Medishare Hydrocephalus Specialty Surgery (PMHSS) program using the previously published methodology.

Methods

Data Collection

The data for this study were collected prospectively by the surgeons of the PMHSS program in the course of caring for children and were maintained in a surgical database. This database was analyzed after IRB approval, and patient data including date of birth, location of birth, sex, surgical timing, neurological examination findings, head circumference, etiology, and surgical intervention were recorded. Patients were evaluated with an ultrasound or CT scanner at the time of consultation. Data were analyzed for 401 patients seen and operated on by the PMHSS program beginning July 31, 2008, through March 2, 2015.

DALYs

The DALY, established in the early 1990s as a health measure that extended beyond deaths avoided, encompasses healthy years of life lost as a result of disability or premature death.4,16 The DALY itself is calculated by assigning a particular health state with a corresponding disability weight ranging from 0 to 1 (0 = perfect health, 1 = death). DALY values for this analysis were obtained from the global burden of disease studies, involving the input from over 30,000 individuals on hypothetical health states due to disease, as well as a Dutch study that incorporated the valuation of a panel of medical experts, all of whom were physicians. The value of a DALY is impacted based on adjustments made to discounting and age weighting. Discounting arises from the belief that individuals value a healthy year of life at the present, decreasing the value of a life 1 year in the future. Age weighting places more value on a year of middle life compared to years at the beginning or the end of life. Due to the debate over the accuracy of these principles, calculations were made with and without age weighting and discounting.16

Scenarios

The DALY calculations allow one to interpret and quantify multiple health states. Importantly, this allows for the calculations of DALYs averted due to an intervention, by establishing two scenarios that reflect the course of the disease with and without intervention (Fig. 1). In the first scenario, the assumption is made that no neurosurgical intervention is carried out, and DALYs are calculated (Fig. 2). In the second scenario, DALYs are calculated when neurosurgical intervention is carried out successfully (Fig. 3). The difference obtained by subtracting total DALYs in scenario 2 from scenario 1 is representative of the DALYs averted due to neurosurgical intervention, and is representative of the improved healthcare state as a consequence.

FIG. 1.
FIG. 1.

Flow sheet demonstrating an overview of the DALYs-averted model. USD = US dollars; VSLY = VSL year.

FIG. 2.
FIG. 2.

Scenario 1 in which no neurosurgical intervention is performed. Estimations are based on assumptions from the referenced papers. DW = disability weighted. Figure is available in color online only.

FIG. 3.
FIG. 3.

Scenario 2 in which neurosurgical intervention is performed. Estimations are based on assumptions from the referenced papers. Figure is available in color online only.

Calculating DALYs

DALYs were calculated and converted to US dollars by means of two different methodologies: the human capital approach and the value of a statistical life (VSL) approach. The human capital approach assumes that individuals are valued at their contribution to the economy. The notation DALY (r,K,B) represents the adjustments to the DALY based on discount-weighting (r), age-weighting (K), and the age-weighting (B) factor, respectively. For the human capital approach, DALYs (3,1,0.04) were calculated under the conditions of 3% discounting, with age weighting (1 = use of age weighting), at an age-weighting constant of 0.04. Additionally, DALYs (0,0,0) were calculated under the conditions without discounting (0 = no discounting) or age weighting (0 = no age weighting). These two scenarios provide an upper and lower limit of DALY values, as the use of age weighting and discounting greatly impacts the total number of DALYs calculated. Using these parameters, DALYs were calculated using the following equation used in previous literature.4,24 Equation 1—Human capital approach DALYs:
Where L is the mean country-specific life expectancy of Haiti across the years involved in this study (61.93). The factor “a” is the age at onset of disease, which is unknown in our population. A conservative assumption of 12 months was made. K is the age-weighting modulation constant (0 = no age weighting, 1 = age weighting), C is the age-weighting disability constant (0.1658), x is the age integrated over the duration of disease and/or years of life lost, r is the discount rate (0 = age weighting, 0.03 = age weighting), and b is the age-weighting constant from the global burden of disease study (0.04).8 To calculate DALYs using the VSL approach, we calculated DALY (3,1,B). Equation 2—Value of a statistical life DALYs:

The calculations using a VSL have two significant assumptions that differ from the human capital approach. First, DALYs are always age weighted as indicated by the absence of K. Second, the variables Q and B indicate specific age weighting that is modified throughout an individual’s life. These values were obtained as described in Warf et al. by B = 1/(2/3*L), as the value of a statistical life year reaches its peak at 2/3 the life expectancy.24 A cubic polynomial was fit using Table 5.2 in Global Burden of Disease and Risk Factors and a value of Q (0.0782) was calculated for the given value of B (0.0242).14

An indirect sensitivity analysis was carried out to determine the magnitude of change in DALY output assuming 25% change in each variable. The resultant % change in DALYs was graphed into a tornado plot to identify which assumptions held the greatest impact in our model.

Valuing DALYs

A brief summary of DALY valuation is provided in this section; for more detailed methods see Warf et al.24 The VSL approach was calculated using the mean established value of a statistical life in the US, accounting for inflation across the years involved in the study ($8,073,000).17 Three different values for the income elasticity (IE) were used in the calculation of the VSL (Equation 3). These included IEVSL = 1.5, IEVSL = 1, and IE = 0.5. Equation 3— Converting US VSL to Haitian VSL:

The human capital approach valued averted DALYs at a lower bound of DALYs (3,1,0.04)*GNI/capita and an upper bound of DALYs (0,0,0)*GNI/capita. The GNI/capita was calculated by averaging the Haitian GNI/capita reported by the World Bank (https://data.worldbank.org) from the years involved in this study. IE represents the income elasticity between the two countries; representing decreased or increased proportions individuals are willing to pay of their annual income in order to decrease their risk of death. Three different values for IE were used: 0.55,1,1.5. IE of 0.55 represents the upper limit of VSL estimates, while an IE of 1.5 estimates the lower limit of VSL estimates.8,21

Cost of Neurosurgical Intervention

The cost per procedure for endoscopic third ventriculostomy (ETV) and ventriculoperitoneal shunt (VPS) placement was determined for Bernard Mevs by means of standard accounting methods. Costs of supplies, medicine, and equipment accounted for 46.3% of total costs including operating room/anesthesia charges and overhead. Monthly costs of general surgeon, nursing staff, and technicians totaled 31.2% total costs, including gross salaries and annual leave. Volunteer costs amounted for 22.4%, including meals, accommodation, electricity/fuel, maintenance, and transportation. The estimated costs were $1125 per ETV/choroid plexus cauterization (CPC). VPS costs equate to 2.36*ETV/CPC costs (accounting for two revision surgeries), amounting to $2655 per VPS. Patients in whom ETV/CPC failed were assumed to revert to VPS placement. Total costs for a failed ETV with subsequent VPS placement and two revisions amounted to $3781. Importantly, 19 procedures were aborted, resulting in $21,375 in costs. In total, this amounted to $754,000 in total costs for the 317 procedures.

Results

Clinical Data

In total, 401 children received neurosurgical intervention from 2008 to 2015 at HBM (Table 1). The mean age was 15.1 months, with a mean head circumference of 55.1 ± 8.6 cm. Head severity was predominantly very severe, defined as greater than or equal to 6 cm above the 95th percentile. Of the 401 patients, 158 (39.4%) met criteria for postinfectious hydrocephalus (PIH), 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. Of the patients who met criteria for PIH, 84 (53.2%) were treated with ETV with CPC, 25 (16.1%) received ventriculoperitoneal shunt (VPS) placement, 5 (3.2%) had VPS revision, 14 (9%) were treated with ETV alone, 14 (9%) underwent aborted ETV/CPC, 9 (5.8%) were treated with CPC alone, and 7 were not reported/cancelled. A total of 245 patients underwent endoscopic procedures (ETV, ETV+CPC, CPC), while 72 underwent VPS placement. There were 19 aborted endoscopic procedures included in the analysis. Patients receiving treatment for myelomeningocele and primary shunt repair and procedures that were unspecified were excluded from the analysis. This resulted in 336 individuals undergoing index surgery for treatment of their hydrocephalus from 2008 to 2015.

TABLE 1.

Patient characteristics

CharacteristicValue
Sex, n = 401
 Male197 (49.1%)
 Female162 (40.4%)
 Not reported42 (10.5%)
Age (mos)
 Mean (SD)15.1 (24.7)
 Median7.5
Head circumference (cm)
 Mean (SD)55.1 (8.6)
 Median55.5
Head severity (percentile), n = 194
 Normal (50%–95%)18 (9.3%)
 Mild (0–1.9 cm above 95%)12 (6.2%)
 Moderate (2–3.9 cm above 95%)10 (5.2%)
 Severe (4–5.9 cm above 95%)18 (9.3%)
 Very severe (>6 cm above 95%)136 (70.1%)
Etiology, n = 401
 Aqueductal stenosis19 (4.7%)
 Congenital54 (13.5)
 Myelomeningocele38 (9.5%)
 Other132 (33%)
 Postinfectious hydrocephalus158 (39.4%)
Surgical intervention, n = 401*
 Aborted ETV/CPC19 (4.7%)
 CPC27 (6.9%)
 ETV43 (10.7%)
 ETV/CPC175 (43.6%)
 Myelomeningocele16 (4.0%)
 Other8 (2.0%)
 VPS insertion72 (18.0%)
 VPS removal18 (4.5%)
 Not reported23 (5.7%)

Values are expressed as the number (%) of patients unless otherwise stated.

Percentages corrected from primary data set.

DALYs Averted by Neurosurgical Intervention

In scenario 1, we assumed no neurosurgical intervention was performed using all three approaches. The DALYs for VSL (3,1,B) are shown in Table 2 and Table 3. These patients were assumed to have age-specific mortality rates secondary to hydrocephalus as established by Laurence and Coates.12 Based on these estimations, hydrocephalus-related mortality and disability (motor, sensory, epilepsy, developmental delay, etc.) would be 83.2% (n = 279) and 16.7% (n = 57), respectively. This calculation results in 6551 DALYs from lack of neurosurgical intervention.9,13

TABLE 2.

Overall, 279 patients with hydrocephalus-related deaths stratified by age at death

Age at DeathMortality RateNo. of PtsDWDALYs/DWTotal DALYs (3,1,B)
2 mos21%71121.691530
5 mos24%81121.691749
8 mos10%34121.69729
12 mos6%20121.69437
24 mos6%20121.69437
5 yrs7%23121.69510
10 yrs4%13121.69292
15 yrs3%10121.69219
18 yrs2%7121.69146
Total6049

DW = disability weight; Pts = patients.

Scenario 1 DALYs calculated assuming no neurosurgical intervention (scenario 1). Percentages based on 336 patients.

TABLE 3.

Overall, 57 total patients surviving with hydrocephalus-related morbidity by condition

Condition% AffectedNo. of PtsDWDALYs/DWTotal DALYs (3,1,B)
Motor defects60%340.173.687126
Visual/auditory defects25%140.337.158102
Epilepsy30%170.1132.45142
IQ 70–8430%170.091.95233
IQ 50–6921%120.296.29075
IQ <5016%90.6213.448123
Total502

Scenario 1 DALYs calculated assuming no neurosurgical intervention (scenario 1).

In scenario 2, 336 cases of hydrocephalus were then assumed to have clinical outcomes similar to a previously published cohort in LMICs using a VSL (3,1,B) approach are shown in Table 4.22 Assuming a 7% surgery-related mortality rate (n = 22) from previous series without age-related mortality and 19 aborted procedures with age-related mortality, 3447 DALYs were obtained.22

TABLE 4.

DALYs calculated assuming neurosurgical intervention (scenario 2)

Condition% AffectedNo. of PtsDWDALYs/DWTotal DALYs (3,1,B)
Aborted procedures19373
Surgery deaths7%221.0021.6902481
Motor defects60%1770.173.6873652
Visual/auditory defects25%740.337.1578528
Epilepsy30%880.1132.4510217
IQ 70–8430%880.091.9521173
IQ 50–6921%620.296.2902389
IQ <5016%470.6213.4479634
Total3447

This table reflects the calculations prior to performing a re-analysis for aborted procedures. There were a total of 336 patients. Nineteen of these patients had aborted procedures, and 22 patients had surgery-related mortality. The remaining patients (n = 295) experienced hydrocephalus-related morbidity with a variety of health states that were not mutually exclusive.

As previously stated, Tables 2 and 3 display DALYs calculated using the VSL approach (VSL [3,1,B]). Additionally, DALYs were calculated using a human capital approach without age weighting or discounting (human capital [0,0,0]) and using a human capital approach with both age weighting and discounting (human capital [3,1,0.04]). The resultant subtraction of scenario 2 from scenario 1 resulted in 8730, 4592, and 3077 DALYs averted for human capital (0,0,0), human capital (3,1,0.04), and VSL (3,1,B) approaches, respectively (Table 5).

TABLE 5.

Total DALYs averted by neurosurgical intervention using 3 approaches

VSL (3,1,B)Human Capital (3,1,0.04)Human Capital (0,0,0)
3077 DALYs4592 DALYs8730 DALYs

Indirect sensitivity analysis for scenario 1 identified premature death due to hydrocephalus at 5 months, 2 months, and 8 months as the most influential variables in our DALY equation. Assuming 25% variation of each variable, total DALYs were altered by 6.7%, 5.8%, and 2.7% respectively (Fig. 4). For scenario 2, motor defects, IQ < 50, and visual/auditory defects were the most influential variables, with 25% variation leading to 5.5%, 5.1%, and 4.3% change in total DALY output (Fig. 5). Tornado plots for each scenario were generated.

FIG. 4.
FIG. 4.

Indirect sensitivity analysis for scenario 1. Figure is available in color online only.

FIG. 5.
FIG. 5.

Indirect sensitivity analysis for scenario 2. Figure is available in color online only.

Valuing DALYs

This resulted in a range of gross economic benefit of neurosurgical intervention between $3.29 million and $6.26 million utilizing the human capital approach (Table 6). The resulting gross economic benefit of neurosurgical intervention utilizing the VSL year totaled $399,000, $3.37 million, and $23.0 million, respectively (Table 7).

TABLE 6.

Gross economic benefit calculated using human capital approach

Human Capital (3,1,0.04)Human Capital (0,0,0)
4592 DALYs8730 DALYs
$3,290,000$6,260,000
TABLE 7.

Gross economic benefit calculated using a VSL year approach

IE-VSL 1.5 (3,1,B)IE-VSL 1 (3,1,B)IE-VSL 0.5 (3,1,B)
$399,000$3,370,000$23,000,000

Net Economic Benefit of Neurosurgical Intervention

Taking into account our DALYs calculated for each scenario, both the human capital approaches and the IE-VSL of 1 provided a relatively tight clustering of economic benefit. We examined these three scenarios further to calculate the net economic benefit. ETV costs were calculated to be $297,000, while VPS costs were calculated at $191,000. Given the reported success rate of 62%, failed endoscopic procedures were assumed to have subsequent shunt placement with two revisions, which totaled $266,000. Altogether, total costs of the procedures were estimated to be $754,000. Subtracting the total costs from the gross economic benefit calculated for each of the three conditions provided the net economic benefit of neurosurgical intervention. The net economic benefit was calculated to be $2.54, $2.62, and $5.51 million for human capital (3,1,0.04), IE-VSL (3,1,B), and human capital (0,0,0) approaches, respectively (Table 8). The cost per DALY averted was calculated to be $164, $245, $86 for human capital (3,1,0.04), IE-VSL (3,1,B), and human capital (0,0,0) approaches, respectively.

TABLE 8.

Net economic benefit for a neurosurgical intervention

VariableIE-VSL 1 (3,1,B)Human Capital (3,1,0.04)Human Capital (0,0,0)
Total DALYs307745928730
Gross economic benefit$3,370,000$3,290,000$6,260,000
Total cost$754,000$754,000$754,000
Net economic benefit$2,620,000$2,540,000$5,510,000
Cost/DALY averted$245$164$86

Discussion

This study is the first to assess the economic benefit of treating hydrocephalus in infants in Haiti. As the most common neurosurgical disease in the pediatric population, hydrocephalus is one of the leading causes of preventable disability and mortality. Although the epidemiology of hydrocephalus has yet to be defined in Haiti, it is estimated that the incidence is greater than 150–300 per 100,000 live births in developing nations.5,24 Similar to previously published series in other LMICs, the most common cause of hydrocephalus identified in Haiti in this surgical series was postinfectious hydrocephalus (39.4% of all cases), which is typically due to neonatal bacterial sepsis. Although its incidence has yet to be defined in Haiti, neonatal sepsis accounts for over 50% of admissions in pediatric hospitals in Haiti, with a 23% mortality rate.2 The combination of increased neonatal infections and the increased incidence of postinfectious hydrocephalus in Haiti establishes the need for durable surgical intervention for hydrocephalus. In the current study, we attempted to illustrate the economic benefit of neurosurgical intervention for pediatric hydrocephalus at a single neurosurgical hospital (HBM) in Port-au-Prince, Haiti, over 7 years.

The surgical burden of disease in LMICs such as Haiti remains immense, with only one surgical subspecialty tertiary care hospital for nearly 10 million people.3 However, as of yet, the concept of DALYs has not yet been applied to analyze the benefit of surgical intervention in Haiti. Here, we strived to implement previously described techniques to quantitatively demonstrate the socioeconomic benefit of treating a subset of neurosurgical diseases. The results of our analyses demonstrate a net economic benefit of over $3.49 million with 3077 VSL DALYs averted for hydrocephalus treatment alone at one institution in Haiti (using the human capital approach, the potential economic benefit would amount to nearly $2.54–$5.51 million US). These estimates demonstrate the long-term economic impact of durable pediatric hydrocephalus surgery in an LMIC such as Haiti.

The cost of hydrocephalus treatment in Haiti—$86 to $245 (mean $165/DALY)—remained comparable to other published series ($59–$126/DALY).24 Additionally, the cost-benefit analysis for hydrocephalus treatment seems to be favorable compared to other surgical specialties in Haiti such as trauma ($223/DALY) or orthopedic treatment ($362/DALY), indicating further allocation of resources to treat hydrocephalus may be favorable (Table 9).6,7,10,11,19,24 We estimated that the cost of neurosurgical intervention was estimated at nearly $1125 per endoscopic procedure and $2656 per shunt placement, which is largely due to foreign dependency and lack of infrastructure and supplies. We anticipate that the costs of each procedure will decrease as our program matures. Nevertheless, with these favorable ratios, there remains a strong need to cultivate neurosurgical capacity building that reduces the need for foreign support.

TABLE 9.

Cost/DALY of other public health interventions

InterventionCost/DALY
Rotavirus vaccination$500
Pneumococcal vaccination$670
Trauma surgery$223
Elective orthopedic surgery$362
Hernia surgery$12
Hydrocephalus$165

It is important to note that our manuscript focuses on secondary/tertiary surgical treatment of infant hydrocephalus, which is limited and costly. Primary prevention of hydrocephalus, especially PIH, could be possible by implementing large-scale public health strategies that focus on maternal education, fetal screening, sanitation, and early treatment of neonatal infections. Work is being conducted to help define the epidemiology of pediatric hydrocephalus in Haiti and identify socioeconomic, environmental, and geographic risk factors. Ultimately, prevention, screening, and early treatment of these infants remain a priority within Haiti, and two sustainable capacity-building programs (pediatric and neurosurgery residency programs) are actively addressing these concerns at HBM.

Limitations

Our study is intrinsically limited to certain assumptions that were made about the data that were available. First, surgical outcomes in our cohort remain limited due to a lack of long-term follow-up; therefore, we were required to derive surgical outcomes from previously published results in LMICs.24 Second, morbidity and mortality of untreated hydrocephalus were extrapolated from a 1962 manuscript from the United Kingdom, which may not accurately depict the severity of untreated hydrocephalus in an underdeveloped nation such as Haiti. Furthermore, the concept of DALYs was not originally intended to be employed in the determination of economic benefit; however, since it was utilized in previous studies as such, we judiciously retained the concept in our study. Given assumptions in variables used in total DALY calculation, we conducted a sensitivity analysis; we demonstrated that even 25% variations in our most influential assumptions only led to 6% variation in the total number of DALYs, thereby demonstrating the robust significance of hydrocephalus treatment in Haiti.

Conclusions

Overall, our work demonstrates the substantial economic benefit of neurosurgical intervention for the treatment of pediatric hydrocephalus. Our data were limited to the cases performed at only one referral hospital in Haiti and therefore likely underestimate the potential economic impact of a self-dependent pediatric neurosurgery program. Moreover, our analysis was limited to assumed surgical outcomes derived from cohorts in other LMICs. A single center offering these services for several days each month generated between $2.5 and $5.5 million in net economic benefits, suggesting the need to develop Haitian neurosurgery programs. Given this need, we have secured and initiated the first Haitian neurosurgical training program that will be aimed to mitigate the overall burden of neurosurgical disease in Haiti, especially pediatric hydrocephalus. Future work characterizing the long-term socioeconomic and health impact of these training programs will be necessary.

Disclosures

The authors report no conflict of interest concerning the materials or methods used in this study or the findings specified in this paper.

Author Contributions

Conception and design: all authors. Acquisition of data: all authors. Analysis and interpretation of data: all authors. Drafting the article: all authors. Critically revising the article: all authors. Reviewed submitted version of manuscript: all authors. Approved the final version of the manuscript on behalf of all authors: Shah. Statistical analysis: all authors. Administrative/technical/material support: J Ragheb. Study supervision: J Ragheb.

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    • Export Citation
  • 4

    Devleesschauwer B, Havelaar AH, Maertens de Noordhout C, Haagsma JA, Praet N, Dorny P, et al.: Calculating disability-adjusted life years to quantify burden of disease. Int J Public Health 59:565569, 2014

    • Crossref
    • PubMed
    • Search Google Scholar
    • Export Citation
  • 5

    Dewan MC, Rattani A, Mekary R, Glancz LJ, Yunusa I, Baticulon RE, et al.: Global hydrocephalus epidemiology and incidence: systematic review and meta-analysis. J Neurosurg 130:10651079, 2019

    • Crossref
    • PubMed
    • Search Google Scholar
    • Export Citation
  • 6

    Gosselin RA, Gialamas G, Atkin DM: Comparing the cost-effectiveness of short orthopedic missions in elective and relief situations in developing countries. World J Surg 35:951955, 2011

    • Crossref
    • PubMed
    • Search Google Scholar
    • Export Citation
  • 7

    Gosselin RA, Maldonado A, Elder G: Comparative cost-effectiveness analysis of two MSF surgical trauma centers. World J Surg 34:415419, 2010

  • 8

    Hammitt JK, Robinson LA: The income elasticity of the value per statistical life: transferring estimates between high and low income populations. J Benefit Cost Anal 2:129, 2011

    • Crossref
    • PubMed
    • Search Google Scholar
    • Export Citation
  • 9

    Hoppe-Hirsch E, Laroussinie F, Brunet L, Sainte-Rose C, Renier D, Cinalli G, et al.: Late outcome of the surgical treatment of hydrocephalus. Childs Nerv Syst 14:9799, 1998

    • Crossref
    • PubMed
    • Search Google Scholar
    • Export Citation
  • 10

    Kim SY, Lee G, Goldie SJ: Economic evaluation of pneumococcal conjugate vaccination in The Gambia. BMC Infect Dis 10:260, 2010

  • 11

    Kim SY, Sweet S, Slichter D, Goldie SJ: Health and economic impact of rotavirus vaccination in GAVI-eligible countries. BMC Public Health 10:253, 2010

  • 12

    Laurence KM, Coates S: The natural history of hydrocephalus. Detailed analysis of 182 unoperated cases. Arch Dis Child 37:345362, 1962

  • 13

    Lindquist B, Carlsson G, Persson EK, Uvebrant P: Learning disabilities in a population-based group of children with hydrocephalus. Acta Paediatr 94:878883, 2005

    • Crossref
    • PubMed
    • Search Google Scholar
    • Export Citation
  • 14

    Lopez AD, Mathers CD, Ezzati M, Jamison DT, Murray CJL (eds): Global Burden of Disease and Risk Factors. New York: Oxford University Press, 2006

  • 15

    Meara JG, Greenberg SL: The Lancet Commission on Global Surgery Global surgery 2030: evidence and solutions for achieving health, welfare and economic development. Surgery 157:834835, 2015

    • Crossref
    • PubMed
    • Search Google Scholar
    • Export Citation
  • 16

    Murray CJ, Acharya AK: Understanding DALYs (disability-adjusted life years). J Health Econ 16:703730, 1997

  • 17

    Rudolfson N, Dewan MC, Park KB, Shrime MG, Meara JG, Alkire BC: The economic consequences of neurosurgical disease in low- and middle-income countries. J Neurosurg 130:11491156, 2019

    • Crossref
    • PubMed
    • Search Google Scholar
    • Export Citation
  • 18

    Schiff SJ, Ranjeva SL, Sauer TD, Warf BC: Rainfall drives hydrocephalus in East Africa. J Neurosurg Pediatr 10:161167, 2012

  • 19

    Shillcutt SD, Clarke MG, Kingsnorth AN: Cost-effectiveness of groin hernia surgery in the Western Region of Ghana. Arch Surg 145:954961, 2010

  • 20

    Shrime MG, Bickler SW, Alkire BC, Mock C: Global burden of surgical disease: an estimation from the provider perspective. Lancet Glob Health 3 (Suppl 2):S8S9, 2015

    • Crossref
    • PubMed
    • Search Google Scholar
    • Export Citation
  • 21

    Viscusi WK, Aldy JE: The value of a statistical life: a critical review of market estimates throughout the world. J Risk Uncertain 27:576, 2003

  • 22

    Warf BC: Comparison of endoscopic third ventriculostomy alone and combined with choroid plexus cauterization in infants younger than 1 year of age: a prospective study in 550 African children. J Neurosurg 103 (6 Suppl):475481, 2005

    • PubMed
    • Search Google Scholar
    • Export Citation
  • 23

    Warf BC: Pediatric hydrocephalus in East Africa: prevalence, causes, treatments, and strategies for the future. World Neurosurg 73:296300, 2010

  • 24

    Warf BC, Alkire BC, Bhai S, Hughes C, Schiff SJ, Vincent JR, et al.: Costs and benefits of neurosurgical intervention for infant hydrocephalus in sub-Saharan Africa. J Neurosurg Pediatr 8:509521, 2011

    • Crossref
    • PubMed
    • Search Google Scholar
    • Export Citation
  • Collapse
  • Expand
  • Flow sheet demonstrating an overview of the DALYs-averted model. USD = US dollars; VSLY = VSL year.

  • Scenario 1 in which no neurosurgical intervention is performed. Estimations are based on assumptions from the referenced papers. DW = disability weighted. Figure is available in color online only.

  • Scenario 2 in which neurosurgical intervention is performed. Estimations are based on assumptions from the referenced papers. Figure is available in color online only.

  • Indirect sensitivity analysis for scenario 1. Figure is available in color online only.

  • Indirect sensitivity analysis for scenario 2. Figure is available in color online only.

  • 1

    Barthélemy EJ, Benjamin E, Edouard Jean-Pierre MY, Poitevien G, Ernst S, Osborn I, et al.: A prospective emergency department-based study of pattern and outcome of neurologic and neurosurgical diseases in Haiti. World Neurosurg 82:948953, 2014

    • Crossref
    • PubMed
    • Search Google Scholar
    • Export Citation
  • 2

    Boulos A, Rand K, Johnson JA, Gautier J, Koster M: Neonatal sepsis in Haiti. J Trop Pediatr 63:7073, 2017

  • 3

    DeGennaro V Jr, DeGennaro VA, Bitar M, Bitar J, Thaller S: Building advanced surgical capacity at a hospital in Port-au-Prince, Haiti. J Craniofac Surg 26:10421047, 2015

    • Crossref
    • PubMed
    • Search Google Scholar
    • Export Citation
  • 4

    Devleesschauwer B, Havelaar AH, Maertens de Noordhout C, Haagsma JA, Praet N, Dorny P, et al.: Calculating disability-adjusted life years to quantify burden of disease. Int J Public Health 59:565569, 2014

    • Crossref
    • PubMed
    • Search Google Scholar
    • Export Citation
  • 5

    Dewan MC, Rattani A, Mekary R, Glancz LJ, Yunusa I, Baticulon RE, et al.: Global hydrocephalus epidemiology and incidence: systematic review and meta-analysis. J Neurosurg 130:10651079, 2019

    • Crossref
    • PubMed
    • Search Google Scholar
    • Export Citation
  • 6

    Gosselin RA, Gialamas G, Atkin DM: Comparing the cost-effectiveness of short orthopedic missions in elective and relief situations in developing countries. World J Surg 35:951955, 2011

    • Crossref
    • PubMed
    • Search Google Scholar
    • Export Citation
  • 7

    Gosselin RA, Maldonado A, Elder G: Comparative cost-effectiveness analysis of two MSF surgical trauma centers. World J Surg 34:415419, 2010

  • 8

    Hammitt JK, Robinson LA: The income elasticity of the value per statistical life: transferring estimates between high and low income populations. J Benefit Cost Anal 2:129, 2011

    • Crossref
    • PubMed
    • Search Google Scholar
    • Export Citation
  • 9

    Hoppe-Hirsch E, Laroussinie F, Brunet L, Sainte-Rose C, Renier D, Cinalli G, et al.: Late outcome of the surgical treatment of hydrocephalus. Childs Nerv Syst 14:9799, 1998

    • Crossref
    • PubMed
    • Search Google Scholar
    • Export Citation
  • 10

    Kim SY, Lee G, Goldie SJ: Economic evaluation of pneumococcal conjugate vaccination in The Gambia. BMC Infect Dis 10:260, 2010

  • 11

    Kim SY, Sweet S, Slichter D, Goldie SJ: Health and economic impact of rotavirus vaccination in GAVI-eligible countries. BMC Public Health 10:253, 2010

  • 12

    Laurence KM, Coates S: The natural history of hydrocephalus. Detailed analysis of 182 unoperated cases. Arch Dis Child 37:345362, 1962

  • 13

    Lindquist B, Carlsson G, Persson EK, Uvebrant P: Learning disabilities in a population-based group of children with hydrocephalus. Acta Paediatr 94:878883, 2005

    • Crossref
    • PubMed
    • Search Google Scholar
    • Export Citation
  • 14

    Lopez AD, Mathers CD, Ezzati M, Jamison DT, Murray CJL (eds): Global Burden of Disease and Risk Factors. New York: Oxford University Press, 2006

  • 15

    Meara JG, Greenberg SL: The Lancet Commission on Global Surgery Global surgery 2030: evidence and solutions for achieving health, welfare and economic development. Surgery 157:834835, 2015

    • Crossref
    • PubMed
    • Search Google Scholar
    • Export Citation
  • 16

    Murray CJ, Acharya AK: Understanding DALYs (disability-adjusted life years). J Health Econ 16:703730, 1997

  • 17

    Rudolfson N, Dewan MC, Park KB, Shrime MG, Meara JG, Alkire BC: The economic consequences of neurosurgical disease in low- and middle-income countries. J Neurosurg 130:11491156, 2019

    • Crossref
    • PubMed
    • Search Google Scholar
    • Export Citation
  • 18

    Schiff SJ, Ranjeva SL, Sauer TD, Warf BC: Rainfall drives hydrocephalus in East Africa. J Neurosurg Pediatr 10:161167, 2012

  • 19

    Shillcutt SD, Clarke MG, Kingsnorth AN: Cost-effectiveness of groin hernia surgery in the Western Region of Ghana. Arch Surg 145:954961, 2010

  • 20

    Shrime MG, Bickler SW, Alkire BC, Mock C: Global burden of surgical disease: an estimation from the provider perspective. Lancet Glob Health 3 (Suppl 2):S8S9, 2015

    • Crossref
    • PubMed
    • Search Google Scholar
    • Export Citation
  • 21

    Viscusi WK, Aldy JE: The value of a statistical life: a critical review of market estimates throughout the world. J Risk Uncertain 27:576, 2003

  • 22

    Warf BC: Comparison of endoscopic third ventriculostomy alone and combined with choroid plexus cauterization in infants younger than 1 year of age: a prospective study in 550 African children. J Neurosurg 103 (6 Suppl):475481, 2005

    • PubMed
    • Search Google Scholar
    • Export Citation
  • 23

    Warf BC: Pediatric hydrocephalus in East Africa: prevalence, causes, treatments, and strategies for the future. World Neurosurg 73:296300, 2010

  • 24

    Warf BC, Alkire BC, Bhai S, Hughes C, Schiff SJ, Vincent JR, et al.: Costs and benefits of neurosurgical intervention for infant hydrocephalus in sub-Saharan Africa. J Neurosurg Pediatr 8:509521, 2011

    • Crossref
    • PubMed
    • Search Google Scholar
    • Export Citation

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