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Costs of pediatric stroke care in the United States: a systematic and contemporary review Expert Rev. Pharmacoecon. Outcomes Res. 14(5), 643–650 (2014)
Charles Ellis*1, Katlyn McGrattan1, Patrick Mauldin2 and Bruce Ovbiagele3 1 Department of Health Sciences and Research, Medical University of South Carolina, Charleston, SC, USA 2 Department of Medicine, Section of Health Systems Research and Policy, Division of General Internal Medicine and Geriatrics, Medical University of South Carolina, Charleston, SC, USA 3 Department of Neurosciences, Medical University of South Carolina, Charleston, SC, USA *Author for correspondence: Tel.: +1 843 792 7492 Fax: +1 843 792 1358 [email protected]
A substantial literature exists regarding cost-of-care outcomes in adult stroke, however less is known about pediatric stroke. The objective of this review of the literature was to examine studies of costs associated with pediatric stroke care. Six studies reporting data from individuals who experienced a pediatric stroke were included in the review. Cost data (charges and payments) were generally limited to one year and ranged from approximately US$15,000–140,000 depending upon stroke type. Pediatric stroke is linked to substantial costs but studies primarily emphasize the direct cost of care during the first year post-stroke onset. However, since many pediatric stroke survivors experience normal lifespans, they can also accumulate a significantly greater long term cost of care than strokes that occur in adulthood. Future studies are needed to examine long term direct costs, short and long term indirect costs and other economic outcomes in this population. KEYWORDS: costs • economic • outcome • pediatric • stroke
Background Overview of stroke in the USA
The WHO estimates that annually approximately 15 million individuals experience strokes worldwide [1]. In the USA alone, 795,000 individuals experience strokes each year [2]. Recent projections in the USA indicate that the annual costs of stroke will increase substantially over the next two decades, underscoring a need for a greater emphasis on implementing effective preventive, acute care and rehabilitative services [3]. Interestingly, the USA stroke literature overwhelmingly focuses on adult-onset stroke, with relatively much less information about the burden of pediatric strokes. Although a condition believed to be of low incidence, the long-term impact of pediatric stroke can be substantial and the economic burden is unclear [4,5]. The overall direct and indirect cost of stroke was estimated to be US $36 billion in 2010 and those estimates are expected to triple by 2030 [2]. Although the cost of stroke has been studied extensively, it is unclear what percentage of those costs is attributed to pediatric stroke. A minimum informahealthcare.com
10.1586/14737167.2014.933672
estimate suggests that the overall annual cost of pediatric stroke in the USA is US$42 milllion [6]. However, these estimates are based on one study limited by analysis of only hospital discharge data. Therefore, a comprehensive review of studies could offer greater insights into the contributors of costs in pediatric stroke. Incidence & mortality
Recent data suggest the annual incidence of pediatric stroke in the USA is 4.6 per 100,000 among children aged 0–19 years [7]. Incidence data vary considerably based on the populations sampled and ages that are included in the analyses. George and colleagues recently reported an incidence of ischemic stroke of 4.2 per 10,000 for children aged 5–14 years in 2007–2008 compared to 3.2 per 10,000 in 1995–1996 [8]. Gandhi and colleagues reported a 2.24 per 100,000 ageadjusted incidence of stroke from 1994 to 2007 [9]. The Gandhi study also suggested that the incidence of pediatric stroke is increasing, although other reports indicate the incidence is stable [9,10]. Despite the technological advances
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in stroke care, not all infants and children experiencing pediatric strokes survive. Gandhi and colleagues reported a 30-day fatality rate of 12.3%, a 1-year fatality rate of 15.7% and a 5-year fatality rate of 17.5%. In addition, approximately 70% of deaths occurred within the first 30 days of hospitalization while 19% occurred within 1 year [9].
approaches and guiding health care policy [16]. For example, cost evaluations can provide critical perspective information about the ‘cost to whom’, that is, the patient (out-of-pocket expenses, co-pays, house modification and lost wages), the payer (payments to hospitals and health providers) or society (loss of taxable income to the federal government, loss of productivity to companies, etc.) [17].
Pathogenesis
Pediatric stroke can be organized into two categories based on pathogenic differences. Perinatal strokes occur at £28 days of life (which includes in utero strokes) and childhood strokes are defined as those occurring >28 days after birth [2]. The underlying causes of pediatric stroke differ in many ways from adultonset stroke with most resulting from a range of heterogeneous diseases [11]. Consequently, mortality rates and the impact on long-term costs are also substantially different. In the perinatal period, a complex interaction of maternal and fetal/neonatal factors can contribute to pediatric stroke. Maternal contributors can include thrombotic states, autoimmune disorders, drug abuse, preeclampsia, infection, infertility treatment and labor complications [12]. Fetal/neonatal factors can include inherited thrombolphilias, infections, perinatal asphyxia, congenital heart disease, preterm hypoglycemia, polycythermia and catheterrelated complications [12]. The general risk factors and causes of pediatric stroke can be classified as cardiac (disease, cardiac repair or catherization, embolic clots after valve repair), hemorrhagic (sickle cell disease, prothombotic disorders), infections, arteriovenous malformations, syndromic and metabolic disorders, vasculitis, oncologic disorders, trauma and drugs [13]. Pediatric stroke can be further classified as ischemic, resulting from arterial occlusion, or hemorrhagic, the result of bleeding from a cerebral artery. Ischemic strokes account for about 50% of strokes that occur in the pediatric population which is significantly less than that observed in the adult population [13]. In addition, those who experience a hemorrhagic stroke have a two-times greater risk of death than those experiencing an ischemic stroke [9].
Review objective
The purpose of this review article is to examine the current literature related to costs associated with pediatric stroke care in the USA and discuss the important gaps for future research related to pediatric stroke. In addition to cost data, this review includes data related to length of stay (LOS), which is one of the most important indexes of health care utilization and a major determinant of hospital costs [18]. Methods
To conduct this review, the authors searched PubMed, Psych Info, CINAHL and Rehab Data using the following Medical Subject Headings terms: stroke, pediatric, child, childhood and cross-searched those with: cost(s), LOS(s), economic impact and economic outcomes. A separate in-depth search of the following journals was completed using the same Medical Subject Headings terms: Pediatrics, Pediatric Neurology, Journal of Child Neurology, Developmental Medicine and Child Neurology, Seminars in Pediatric Neurology and Journal of Pediatric Neurology. The search was limited to papers published over a 10-year period (2004–2013) and only considered papers including a USA patient population and written in English. Randomized controlled trials, quasi-randomized controlled trials and retrospective data analyses and published scientific conference presentations that reported economic outcomes associated with pediatric stroke were considered. Due to the heterogeneity of studies, an a priori decision was made to not perform a metaanalysis, but instead to perform a qualitative analysis of the findings from various studies.
Costs perspective
Regardless of the type, cause or outcome, little is known about the cost of pediatric stroke. Cost of care for stroke and other medical conditions consists of ‘direct costs’ or the cost of utilizing medical care in different settings (acute hospital, outpatient clinics, pharmacy, laboratory) and ‘indirect costs’ or those costs incurred by the individual, employer and society due to cessation or reduction of work (work loss, worker replacement, reduced productivity) [14]. Understanding cost data in pediatric stroke is critical for several reasons. First, pediatric stroke cost data can offer evidence of inappropriate or overutilization of services that may be targets for cost containment [15]. Second, pediatric stroke cost data can offer evidence of the true cost of pediatric stroke to the individual experiencing the stroke, their family and society. Third, an analysis of pediatric stroke cost data can offer insights into the different perspectives of cost measurement. Fourth, pediatric stroke cost data is critical to the identification of the most cost-effective treatment 644
Search outcome
About 329 reports were identified through the initial search of PubMed, Psych Info, CINAHL and Rehab Data. About 312 were excluded after a review of the abstracts. Seventeen full-text articles were examined to identify publications eligible for inclusion in the review. Ten reports were excluded because they did not report economic outcomes data despite having a title that suggested inclusion of such data. One study was excluded because the age range that included pediatric stroke extended to age 34. In total, six studies reporting data from individuals who experienced a pediatric stroke met inclusion criteria and were included in the review (See TABLE 1). Results Costs
Six studies were identified that reported cost-of-care data for pediatric stroke. Reported cost-of-care data for pediatric stroke Expert Rev. Pharmacoecon. Outcomes Res. 14(5), (2014)
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National Inpatient sample
Children’s Hospital Columbus, OH
HCUP-Kids Inpatient Database
HCUP-Kids Inpatient Database
Kaiser Pediatric Stroke Study data
Janjua et al. (2007)
Lo et al. (2008)
Perkins et al. (2009)
Engle and Ellis (2012)
Gardner et al. (2010)
Cost results All • US$39,400 Receiving thrombolysis • US$81,800 Not receiving thrombolysis • US$38,700 All • US$36,132 Ischemic • US$31,678 Hemorrhagic • US$67,860 All • US$20, 927 Ischemic stroke • US$15,003 Subarachnoid hemorrhage • US$31,653 Intracranial hemorrhage • US$24,117 Ischemic stroke • 2000 US$26,056 • 2003 US$40,815 • 2006 US$56,349 Subarachnoid hemorrhage • 2000 US$65,027 • 2003 US$97,985 • 2006 US$139,970 Intracranial hemorrhage • 2000 US$47,622 • 2003 US$64,000 • 2006 US$86,680 5-year post-stoke adjusted costs All • Mean US$110,921 • Median US$49,958 Neonatal (n = 53) • Mean US$51,719 • Median US$25,825 • Maximum US$792,648
Age 1–17 years
3 months –19 years
3–20
0–17
0–19
2904 children with ischemic stroke of which (2000–2003)
39 children with nontraumatic ischemic or hemorrhagic stroke
1070 actual pediatric stroke cases which equates to 2224 cases with statistical weighting
6857 neonates and children with ischemic, subarachnoid hemorrhage or intracranial hemorrhage (2000, 2003, 2006)
266 neonates and children with ischemic or hemorrhagic stroke (1993–2003)
Sample
HCUP: Health Care Utilization Project; IQR: Interquartile range; LOS: Length of stay.
Data source
Study (year)
Table 1. Summary of studies reporting hospital cost data in pediatric stroke.
[19]
[16]
[6]
[20]
[21]
Median total first-year costs across health care system only; did not include costs in other systems or out of pocket or other costs
Mean costs calculated by multiplying cost-to-charge ratios by charges for hospital services. Total costs reported in 2003 dollars
Mean costs
Mean 5-year costs. 5-year cost analysis using microcosting approach, directly tabulating actual cost of each individual component adjusted for inflation to 2003 dollars. Adjusted costs calculated by subtracting mean birth admission costs from each cases
Ref.
Mean yearly cost adjusted for 2005 inflation rate
Comment
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[15]
Median total costs. Costs were adjusted to account for regional differences. Hospital-specific cost-tocharge ratio for each year was applied to transform charges to total hospital costs. Costs adjusted to 2009 dollars
HCUP: Health Care Utilization Project; IQR: Interquartile range; LOS: Length of stay.
First discharge • Median US$19,548 Multiple discharges • Median US$39,451 All cases • Median US$19,548 Stroke + associated diagnoses • Median US$22,049 Stroke-associated diagnoses • Median US$16,150 IQR 5–15 1667 children with a primary diagnosis from 24 hospitals (2003–2009) Pediatric Health Information System
trio of pair-matched controls to isolate costs attributable to stroke Childhood • Mean US$135,161 • Median US$59,648 • Maximum US$984,210
Turney et al. (2011)
Comment Cost results Age Sample Data source Study (year)
Table 1. Summary of studies reporting hospital cost data in pediatric stroke (cont.).
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Ref.
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consisted of ‘charges’ for services rendered by hospital systems and ‘payments’ made on behalf of patients by third-party payers (health insurance companies) to hospital systems. Janjua and colleagues calculated mean yearly cost (hospital charges) of stroke among children aged 1–17 years and found an average cost of US$39,400 after adjusting for the 2005 inflation rate [19]. Children who received thrombolytic therapy had higher charges (US$81,800) than those who did not receive therapy (US$38,700). Similar median total first-year costs (direct and indirect costs across the health system excluding costs in other health care systems) were found by Lo et al. in an examination of 39 children aged 3 months to 19 years [16]. The median cost for all children was US$36,132; however, lower costs were found in those children experiencing an ischemic stroke (US$31,678) compared to those with hemorrhagic strokes (US$67,860). Two studies reported cost data and distinguished the costs between ischemic and hemorrhagic stroke. Both Perkins and colleagues and Engle and Ellis found lower cost of care among children experiencing ischemic strokes when compared to other stroke types [6,20]. Perkins found mean overall costs (hospital charges) of US$20,927, with lower costs among those with ischemic stroke (US$15,003) when compared to subarachnoid hemorrhage (US$31,653) and intracranial hemorrhage (US $24,117). Engle and Ellis also noted lower ischemic stroke costs (hospital charges) in 2000, 2003 and 2006 (US$26,056, 40,815, 56,349) compared to subarachnoid hemorrhage (US$65,027, 97,985, 139,970) and intracranial hemorrhage (US$47,622, 64,000, 86,680) for each year reported. More long-term cost of stroke care for children and neonates was reported by Gardner and colleagues [21]. They examined 5-year costs using a microcosting approach and found that the mean cost (payments made by the Kaiser Permanente Medical Care Program) for the 5-year period was US$110,921, with higher 5-year costs for children US$135,161 than neonates US $51,719. Maximum reported costs for stroke care in children were US$984,210 and 792,648 for neonates. Finally, Turney and colleagues examined costs (charges) in children with stroke in 24 hospital across the USA and found that the median charges for a first discharge were US$19,548 [15]. Children with multiple discharges for stroke care incurred US$39,451 in median charges, with US$22,049 reported for children with stroke and associated diagnoses and US$16,150 for those with stroke without associated diagnoses. Length of stay
Four of the six identified studies reported LOS outcomes data (See TABLE 2). In a study of more than 2000 children who experienced an ischemic stroke between 2000 and 2003, Janjua and colleagues found a median LOS of 7.6 days for all children with stroke, but significant higher for those who received thrombolysis (15.3 days) compared to those who did not (7.4 days) [19]. Lo and colleagues found a median LOS of 5.0 days among children who experienced an ischemic stroke and 8.5 days for those who experienced a hemorrhagic Expert Rev. Pharmacoecon. Outcomes Res. 14(5), (2014)
Costs of pediatric stroke care in the USA
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Table 2. Summary of studies reporting hospital length of stay data in pediatric stroke. Study (year)
Data source
Sample
Age
LOS results
Comment
Ref.
Janjua et al. (2007)
National Inpatient sample
2904 children with ischemic stroke of which (2000–2003)
1–17 years
All • 7.6 days Receiving thrombolysis • 15.3 days Not receiving thrombolysis • 7.4 days
Median LOS reported
[19]
Lo et al. (2008)
Children’s Hospital Columbus, OH
39 children with nontraumatic ischemic or hemorrhagic stroke
3 months–19 years
Ischemic • 5.0 days Hemorrhagic • 8.5 days
Median LOS reported
[16]
Perkins et al. (2009)
HCUP-Kids Inpatient Database
1070 actual pediatric stroke cases which equates to 2224 cases with statistical weighting
3–20
All • 8.3 days Ischemic stroke • 6.6 days Subarachnoid hemorrhage • 11.2 days Intracranial hemorrhage • 9.6 days
Mean LOS reported Small constant added to avoid zero value when calculating LOS
[6]
Engle and Ellis (2012)
HCUP-Kids Inpatient Database
2161 neonates and children with ischemic, subarachnoid hemorrhage or intracranial hemorrhage (2000, 2003, 2006)
0–17
Ischemic stroke 2000 6.9 days 2003 7.2 days 2006 6.9 days Subarachnoid hemorrhage 2000 11.5 days 2003 13.2 days 2006 14.2 days Intracranial hemorrhage 2000 10.4 days 2003 11.0 days 2006 10.9 days
Descriptive total cost nonweighted data from HCUP
[20]
HCUP: Health Care Utilization Project; LOS: Length of stay.
stroke [16]. Shorter LOSs among children with ischemic stroke were also reported by Perkins and colleagues in a study of data from the Kids Inpatient Database [6]. The mean LOS regardless of stroke type was 8.3 days compared to 6.6 days for ischemic stroke, 11.2 days for subarachnoid hemorrhage and 9.6 days for intracranial hemorrhage. Engle and Ellis also reported shorter LOSs among children with ischemic stroke (6.9 days), compared to subarachnoid hemorrhage (14.2 days) and intracranial hemorrhage (10.9 days) in 2006 [20]. Expert commentary
Pediatric stroke is generally considered a rare event, and pediatric stroke-related cost data have been slow to emerge. Early management of pediatric stroke is similar to adult management, and therefore it is generally assumed the cost of pediatric stroke is equivalent to adult stroke [6]. However, specific clinical differences in stroke etiology suggest that cost differences may exist between children and adults [6]. In congruence with the adult literature, the cost of pediatric stroke is substantial and influenced by a number of factors associated with the stroke (cause, type, severity, stroke-related neurophysiological changes, age of onset, etc.), as well as the number and types of services required. At informahealthcare.com
least two studies in the review reported higher costs for hemorrhagic pediatric strokes compared to those experiencing ischemic strokes. Similarly, four studies reported longer LOSs among those with hemorrhagic strokes (including subdural hematoma and intracranial hemorrhage) [6,16,19,20]. This is not surprising as stroke type affects initial deficit profile which subsequently dictates the type, intensity and range of services required for optimal management. In addition, the receipt of specialized services contributes to higher overall costs of care [22]. According to Turney and colleagues, hemorrhagic strokes and ischemic strokes differ in the specific categories of care needed (nursing, pharmacy, supplies, etc.) and overall LOSs, thereby resulting in longer LOSs among patients with hemorrhagic stroke [15]. Similarly, in a study of 256 cases of pediatric stroke, Fox and colleagues found that 62% were admitted to intensive care units (ICUs), 32% were intubated and 11% were treated with decompressive neurosurgery [23]. Admission to ICU units is associated with a higher level of complex care, and independent predictors of ICU admission include younger age and presentation with altered mental status. The LOS data reported here offers additional insight into the cost of pediatric stroke care. The mean LOS for pediatric 647
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stroke ranged from 6.6 to 14.2 days depending upon stroke type. Data from the Centers for Disease Control indicate that the average LOS for adults 65 was 5.3 days in 2009 [24]. Although only four studies are reported here, evidence suggests the cost of pediatric stroke is substantial and at a minimum equivalent to the cost of adult-onset stroke. The studies reported in this review are primarily limited to direct costs of care during the first year after stroke. Gardner and colleagues extended their study beyond the typical 1-year measurement point and reported 5-year direct medical cost data as a measure of the societal burden of pediatric stroke [21]. Pediatric survivors can go on to lead full lives of average lifespans and to our knowledge, few if any studies have carefully considered the long-term impact of pediatric stroke and more specially the indirect costs of pediatric stroke. Limiting cost data to 1 year offers little in terms of clarifying the true overall costs of the stroke event. The economic cost of pediatric stroke is unique in that the span of time that costs will be expended differs dramatically from adult-onset stroke [6]. Although many pediatric stroke survivors will live full lives with average life expectancies, a portion will have persisting motor, sensory, cognitive, language and other behavioral deficits [25–28]. These deficits can occur prior to or at the same time that they are undergoing normal development of the same skills [29]. Consequently, other direct costs required to address persisting deficits and to reduce risk of recurrent stroke can be substantial. These can include the cost of physician visits, rehabilitative therapies, medications and other treatments that will be expended over an exponentially long period of time. Evidence from the general disability literature also suggests that experiencing a pediatric stroke results in unique costs that are not common among adult-onset stroke survivors. These include the costs of services required to facilitate the child’s participation in school and play as well as specialized child-care services outside of the educational system [30]. Some costs are long term and reside outside of the health care system. State and local educational systems may require additional resources to educate children with disabilities depending on the level of residual deficits they experience. Currently, data do not exist specific to stroke; however, deficits that can result from stroke such as developmental delay, intellectual disability, specific learning disabilities and speech and language impairments are known to increase the cost of childhood education [31]. The economic cost of pediatric stroke likely extends beyond the direct medical costs related to the child. Indirect and difficult-to-measure family unit costs can be incurred due to reductions in the family unit’s health-related quality of life. However, in the absence of focused research related to the indirect impact of pediatric stroke on the family unit, a number of questions remain. First, what are the negative financial and psychosocial implications of childhood illness on the family unit [30]? Second, are the mothers of children with pediatric stroke-related disabilities limited in their ability to engage in competitive employment [32,33]? Third, is there additional 648
burden among mothers caring for children with mental health conditions after pediatric stroke [34]. Fourth, are there longer term issues such as unemployment and underemployment that are the result of mental health, cognitive and other deficits that persist into adulthood [5]? Further study of these issues is needed to assess the relationship between changes in costs and resource utilization over time with the disease condition, as well as with changes in clinical and socio-demographic characteristics. As an example, acute-care utilization has been shown to be associated with socioeconomic and neighborhood factors, and these often change through time [35]. Other non-monetary family unit costs can be linked to caregiver illness and family psychosocial distress related to caregiving for children with disabilities. Childhood disability has been associated with significant reductions in caregiver physical health and sleep quality, as well as elevations in levels of depression and stress [36]. More specifically, parents of children with stroke can have significant and wide-ranging health difficulties including mental health issues and issues associated with personal and social development [37]. Ultimately, these issues can translate into poor health among parents of children with disabilities, which result in greater health care costs for the parents [30]. It is tenable then that these issues are not limited to parents/caregivers but the entire family unit. For example, families that have a child with health issues have greater odds of experiencing separation or divorce [38]. Likewise, siblings of children with disabilities experience limited parental availability and more negative psychological functioning including internalizing problems (e.g., depression and anxiety) and externalizing problems (e.g., behavioral problems, aggression and social problems) [39]. Five-year view
The cost of pediatric stroke is substantial. Previous studies have primarily focused on the 1-year direct medical care cost of caring for the child with stroke. Evidence suggests that some stroke-related deficits persist into adulthood [5]. Consequently, ‘direct costs’ of care may also continue for some including long-term health care costs for the child, the costs of educating children who experience disabilities resulting from their stroke and the long-term family-related health care costs due to their experiencing poorer health. It is important to note that not all pediatric stroke survivors experience persisting disabilities and these associated long-term costs. However, a research agenda is currently needed to determine the long-term economic impact of pediatric stroke including the direct and indirect cost to the stroke survivor, to their parents and to society. A longitudinal epidemiological approach could offer important information about the short- and long-term clinical, functional and economic outcomes associated with pediatric stroke. Given the magnitude of costs associated with pediatric stroke, longitudinal studies will offer critical information about current approaches to treating, educating and managing patient and family-related issues and to determine their cost–effectiveness which will in turn reduce costs and improve outcomes. Expert Rev. Pharmacoecon. Outcomes Res. 14(5), (2014)
Costs of pediatric stroke care in the USA
Financial & competing interests disclosure
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The authors have no relevant affiliations or financial involvement with any organization or entity with a financial interest in or financial conflict with the subject matter or materials discussed in the manuscript. This
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includes employment, consultancies, honoraria, stock ownership or options, expert testimony, grants or patents received or pending, or royalties. No writing assistance was utilized in the production of this manuscript.
Key issues • The long-term direct and indirect financial burden of pediatric stroke may be greater than adult stroke. • Cost-related data have yet to emerge beyond 5 years, although pediatric stroke survivors can live into adulthood. • Cost-of-care data are critical to understanding service utilization patterns and which treatments and services are most cost–effective in achieving optimal outcomes. • Current cost-of-care data for pediatric stroke include both reports of ‘charges’ and ‘payments’, thus limiting an interpretation of the overall cost perspective. • Studies are needed to examine the long-term indirect cost of pediatric stroke or the cost of care outside of the primary hospital where care was received. • Studies are needed to examine the ‘indirect costs’ of pediatric stroke to the stroke survivor and their families.
case fatality of stroke among children from 1994 to 2007. Neurology 2012;78(24): 1923-9
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Costs of pediatric stroke care in the United States: a systematic and contemporary review Expert Rev. Pharmacoecon. Outcomes Res. 14(5), 643–650 (2014)
Charles Ellis*1, Katlyn McGrattan1, Patrick Mauldin2 and Bruce Ovbiagele3 1 Department of Health Sciences and Research, Medical University of South Carolina, Charleston, SC, USA 2 Department of Medicine, Section of Health Systems Research and Policy, Division of General Internal Medicine and Geriatrics, Medical University of South Carolina, Charleston, SC, USA 3 Department of Neurosciences, Medical University of South Carolina, Charleston, SC, USA *Author for correspondence: Tel.: +1 843 792 7492 Fax: +1 843 792 1358 [email protected]
A substantial literature exists regarding cost-of-care outcomes in adult stroke, however less is known about pediatric stroke. The objective of this review of the literature was to examine studies of costs associated with pediatric stroke care. Six studies reporting data from individuals who experienced a pediatric stroke were included in the review. Cost data (charges and payments) were generally limited to one year and ranged from approximately US$15,000–140,000 depending upon stroke type. Pediatric stroke is linked to substantial costs but studies primarily emphasize the direct cost of care during the first year post-stroke onset. However, since many pediatric stroke survivors experience normal lifespans, they can also accumulate a significantly greater long term cost of care than strokes that occur in adulthood. Future studies are needed to examine long term direct costs, short and long term indirect costs and other economic outcomes in this population. KEYWORDS: costs • economic • outcome • pediatric • stroke
Background Overview of stroke in the USA
The WHO estimates that annually approximately 15 million individuals experience strokes worldwide [1]. In the USA alone, 795,000 individuals experience strokes each year [2]. Recent projections in the USA indicate that the annual costs of stroke will increase substantially over the next two decades, underscoring a need for a greater emphasis on implementing effective preventive, acute care and rehabilitative services [3]. Interestingly, the USA stroke literature overwhelmingly focuses on adult-onset stroke, with relatively much less information about the burden of pediatric strokes. Although a condition believed to be of low incidence, the long-term impact of pediatric stroke can be substantial and the economic burden is unclear [4,5]. The overall direct and indirect cost of stroke was estimated to be US $36 billion in 2010 and those estimates are expected to triple by 2030 [2]. Although the cost of stroke has been studied extensively, it is unclear what percentage of those costs is attributed to pediatric stroke. A minimum informahealthcare.com
10.1586/14737167.2014.933672
estimate suggests that the overall annual cost of pediatric stroke in the USA is US$42 milllion [6]. However, these estimates are based on one study limited by analysis of only hospital discharge data. Therefore, a comprehensive review of studies could offer greater insights into the contributors of costs in pediatric stroke. Incidence & mortality
Recent data suggest the annual incidence of pediatric stroke in the USA is 4.6 per 100,000 among children aged 0–19 years [7]. Incidence data vary considerably based on the populations sampled and ages that are included in the analyses. George and colleagues recently reported an incidence of ischemic stroke of 4.2 per 10,000 for children aged 5–14 years in 2007–2008 compared to 3.2 per 10,000 in 1995–1996 [8]. Gandhi and colleagues reported a 2.24 per 100,000 ageadjusted incidence of stroke from 1994 to 2007 [9]. The Gandhi study also suggested that the incidence of pediatric stroke is increasing, although other reports indicate the incidence is stable [9,10]. Despite the technological advances
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in stroke care, not all infants and children experiencing pediatric strokes survive. Gandhi and colleagues reported a 30-day fatality rate of 12.3%, a 1-year fatality rate of 15.7% and a 5-year fatality rate of 17.5%. In addition, approximately 70% of deaths occurred within the first 30 days of hospitalization while 19% occurred within 1 year [9].
approaches and guiding health care policy [16]. For example, cost evaluations can provide critical perspective information about the ‘cost to whom’, that is, the patient (out-of-pocket expenses, co-pays, house modification and lost wages), the payer (payments to hospitals and health providers) or society (loss of taxable income to the federal government, loss of productivity to companies, etc.) [17].
Pathogenesis
Pediatric stroke can be organized into two categories based on pathogenic differences. Perinatal strokes occur at £28 days of life (which includes in utero strokes) and childhood strokes are defined as those occurring >28 days after birth [2]. The underlying causes of pediatric stroke differ in many ways from adultonset stroke with most resulting from a range of heterogeneous diseases [11]. Consequently, mortality rates and the impact on long-term costs are also substantially different. In the perinatal period, a complex interaction of maternal and fetal/neonatal factors can contribute to pediatric stroke. Maternal contributors can include thrombotic states, autoimmune disorders, drug abuse, preeclampsia, infection, infertility treatment and labor complications [12]. Fetal/neonatal factors can include inherited thrombolphilias, infections, perinatal asphyxia, congenital heart disease, preterm hypoglycemia, polycythermia and catheterrelated complications [12]. The general risk factors and causes of pediatric stroke can be classified as cardiac (disease, cardiac repair or catherization, embolic clots after valve repair), hemorrhagic (sickle cell disease, prothombotic disorders), infections, arteriovenous malformations, syndromic and metabolic disorders, vasculitis, oncologic disorders, trauma and drugs [13]. Pediatric stroke can be further classified as ischemic, resulting from arterial occlusion, or hemorrhagic, the result of bleeding from a cerebral artery. Ischemic strokes account for about 50% of strokes that occur in the pediatric population which is significantly less than that observed in the adult population [13]. In addition, those who experience a hemorrhagic stroke have a two-times greater risk of death than those experiencing an ischemic stroke [9].
Review objective
The purpose of this review article is to examine the current literature related to costs associated with pediatric stroke care in the USA and discuss the important gaps for future research related to pediatric stroke. In addition to cost data, this review includes data related to length of stay (LOS), which is one of the most important indexes of health care utilization and a major determinant of hospital costs [18]. Methods
To conduct this review, the authors searched PubMed, Psych Info, CINAHL and Rehab Data using the following Medical Subject Headings terms: stroke, pediatric, child, childhood and cross-searched those with: cost(s), LOS(s), economic impact and economic outcomes. A separate in-depth search of the following journals was completed using the same Medical Subject Headings terms: Pediatrics, Pediatric Neurology, Journal of Child Neurology, Developmental Medicine and Child Neurology, Seminars in Pediatric Neurology and Journal of Pediatric Neurology. The search was limited to papers published over a 10-year period (2004–2013) and only considered papers including a USA patient population and written in English. Randomized controlled trials, quasi-randomized controlled trials and retrospective data analyses and published scientific conference presentations that reported economic outcomes associated with pediatric stroke were considered. Due to the heterogeneity of studies, an a priori decision was made to not perform a metaanalysis, but instead to perform a qualitative analysis of the findings from various studies.
Costs perspective
Regardless of the type, cause or outcome, little is known about the cost of pediatric stroke. Cost of care for stroke and other medical conditions consists of ‘direct costs’ or the cost of utilizing medical care in different settings (acute hospital, outpatient clinics, pharmacy, laboratory) and ‘indirect costs’ or those costs incurred by the individual, employer and society due to cessation or reduction of work (work loss, worker replacement, reduced productivity) [14]. Understanding cost data in pediatric stroke is critical for several reasons. First, pediatric stroke cost data can offer evidence of inappropriate or overutilization of services that may be targets for cost containment [15]. Second, pediatric stroke cost data can offer evidence of the true cost of pediatric stroke to the individual experiencing the stroke, their family and society. Third, an analysis of pediatric stroke cost data can offer insights into the different perspectives of cost measurement. Fourth, pediatric stroke cost data is critical to the identification of the most cost-effective treatment 644
Search outcome
About 329 reports were identified through the initial search of PubMed, Psych Info, CINAHL and Rehab Data. About 312 were excluded after a review of the abstracts. Seventeen full-text articles were examined to identify publications eligible for inclusion in the review. Ten reports were excluded because they did not report economic outcomes data despite having a title that suggested inclusion of such data. One study was excluded because the age range that included pediatric stroke extended to age 34. In total, six studies reporting data from individuals who experienced a pediatric stroke met inclusion criteria and were included in the review (See TABLE 1). Results Costs
Six studies were identified that reported cost-of-care data for pediatric stroke. Reported cost-of-care data for pediatric stroke Expert Rev. Pharmacoecon. Outcomes Res. 14(5), (2014)
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National Inpatient sample
Children’s Hospital Columbus, OH
HCUP-Kids Inpatient Database
HCUP-Kids Inpatient Database
Kaiser Pediatric Stroke Study data
Janjua et al. (2007)
Lo et al. (2008)
Perkins et al. (2009)
Engle and Ellis (2012)
Gardner et al. (2010)
Cost results All • US$39,400 Receiving thrombolysis • US$81,800 Not receiving thrombolysis • US$38,700 All • US$36,132 Ischemic • US$31,678 Hemorrhagic • US$67,860 All • US$20, 927 Ischemic stroke • US$15,003 Subarachnoid hemorrhage • US$31,653 Intracranial hemorrhage • US$24,117 Ischemic stroke • 2000 US$26,056 • 2003 US$40,815 • 2006 US$56,349 Subarachnoid hemorrhage • 2000 US$65,027 • 2003 US$97,985 • 2006 US$139,970 Intracranial hemorrhage • 2000 US$47,622 • 2003 US$64,000 • 2006 US$86,680 5-year post-stoke adjusted costs All • Mean US$110,921 • Median US$49,958 Neonatal (n = 53) • Mean US$51,719 • Median US$25,825 • Maximum US$792,648
Age 1–17 years
3 months –19 years
3–20
0–17
0–19
2904 children with ischemic stroke of which (2000–2003)
39 children with nontraumatic ischemic or hemorrhagic stroke
1070 actual pediatric stroke cases which equates to 2224 cases with statistical weighting
6857 neonates and children with ischemic, subarachnoid hemorrhage or intracranial hemorrhage (2000, 2003, 2006)
266 neonates and children with ischemic or hemorrhagic stroke (1993–2003)
Sample
HCUP: Health Care Utilization Project; IQR: Interquartile range; LOS: Length of stay.
Data source
Study (year)
Table 1. Summary of studies reporting hospital cost data in pediatric stroke.
[19]
[16]
[6]
[20]
[21]
Median total first-year costs across health care system only; did not include costs in other systems or out of pocket or other costs
Mean costs calculated by multiplying cost-to-charge ratios by charges for hospital services. Total costs reported in 2003 dollars
Mean costs
Mean 5-year costs. 5-year cost analysis using microcosting approach, directly tabulating actual cost of each individual component adjusted for inflation to 2003 dollars. Adjusted costs calculated by subtracting mean birth admission costs from each cases
Ref.
Mean yearly cost adjusted for 2005 inflation rate
Comment
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[15]
Median total costs. Costs were adjusted to account for regional differences. Hospital-specific cost-tocharge ratio for each year was applied to transform charges to total hospital costs. Costs adjusted to 2009 dollars
HCUP: Health Care Utilization Project; IQR: Interquartile range; LOS: Length of stay.
First discharge • Median US$19,548 Multiple discharges • Median US$39,451 All cases • Median US$19,548 Stroke + associated diagnoses • Median US$22,049 Stroke-associated diagnoses • Median US$16,150 IQR 5–15 1667 children with a primary diagnosis from 24 hospitals (2003–2009) Pediatric Health Information System
trio of pair-matched controls to isolate costs attributable to stroke Childhood • Mean US$135,161 • Median US$59,648 • Maximum US$984,210
Turney et al. (2011)
Comment Cost results Age Sample Data source Study (year)
Table 1. Summary of studies reporting hospital cost data in pediatric stroke (cont.).
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consisted of ‘charges’ for services rendered by hospital systems and ‘payments’ made on behalf of patients by third-party payers (health insurance companies) to hospital systems. Janjua and colleagues calculated mean yearly cost (hospital charges) of stroke among children aged 1–17 years and found an average cost of US$39,400 after adjusting for the 2005 inflation rate [19]. Children who received thrombolytic therapy had higher charges (US$81,800) than those who did not receive therapy (US$38,700). Similar median total first-year costs (direct and indirect costs across the health system excluding costs in other health care systems) were found by Lo et al. in an examination of 39 children aged 3 months to 19 years [16]. The median cost for all children was US$36,132; however, lower costs were found in those children experiencing an ischemic stroke (US$31,678) compared to those with hemorrhagic strokes (US$67,860). Two studies reported cost data and distinguished the costs between ischemic and hemorrhagic stroke. Both Perkins and colleagues and Engle and Ellis found lower cost of care among children experiencing ischemic strokes when compared to other stroke types [6,20]. Perkins found mean overall costs (hospital charges) of US$20,927, with lower costs among those with ischemic stroke (US$15,003) when compared to subarachnoid hemorrhage (US$31,653) and intracranial hemorrhage (US $24,117). Engle and Ellis also noted lower ischemic stroke costs (hospital charges) in 2000, 2003 and 2006 (US$26,056, 40,815, 56,349) compared to subarachnoid hemorrhage (US$65,027, 97,985, 139,970) and intracranial hemorrhage (US$47,622, 64,000, 86,680) for each year reported. More long-term cost of stroke care for children and neonates was reported by Gardner and colleagues [21]. They examined 5-year costs using a microcosting approach and found that the mean cost (payments made by the Kaiser Permanente Medical Care Program) for the 5-year period was US$110,921, with higher 5-year costs for children US$135,161 than neonates US $51,719. Maximum reported costs for stroke care in children were US$984,210 and 792,648 for neonates. Finally, Turney and colleagues examined costs (charges) in children with stroke in 24 hospital across the USA and found that the median charges for a first discharge were US$19,548 [15]. Children with multiple discharges for stroke care incurred US$39,451 in median charges, with US$22,049 reported for children with stroke and associated diagnoses and US$16,150 for those with stroke without associated diagnoses. Length of stay
Four of the six identified studies reported LOS outcomes data (See TABLE 2). In a study of more than 2000 children who experienced an ischemic stroke between 2000 and 2003, Janjua and colleagues found a median LOS of 7.6 days for all children with stroke, but significant higher for those who received thrombolysis (15.3 days) compared to those who did not (7.4 days) [19]. Lo and colleagues found a median LOS of 5.0 days among children who experienced an ischemic stroke and 8.5 days for those who experienced a hemorrhagic Expert Rev. Pharmacoecon. Outcomes Res. 14(5), (2014)
Costs of pediatric stroke care in the USA
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Table 2. Summary of studies reporting hospital length of stay data in pediatric stroke. Study (year)
Data source
Sample
Age
LOS results
Comment
Ref.
Janjua et al. (2007)
National Inpatient sample
2904 children with ischemic stroke of which (2000–2003)
1–17 years
All • 7.6 days Receiving thrombolysis • 15.3 days Not receiving thrombolysis • 7.4 days
Median LOS reported
[19]
Lo et al. (2008)
Children’s Hospital Columbus, OH
39 children with nontraumatic ischemic or hemorrhagic stroke
3 months–19 years
Ischemic • 5.0 days Hemorrhagic • 8.5 days
Median LOS reported
[16]
Perkins et al. (2009)
HCUP-Kids Inpatient Database
1070 actual pediatric stroke cases which equates to 2224 cases with statistical weighting
3–20
All • 8.3 days Ischemic stroke • 6.6 days Subarachnoid hemorrhage • 11.2 days Intracranial hemorrhage • 9.6 days
Mean LOS reported Small constant added to avoid zero value when calculating LOS
[6]
Engle and Ellis (2012)
HCUP-Kids Inpatient Database
2161 neonates and children with ischemic, subarachnoid hemorrhage or intracranial hemorrhage (2000, 2003, 2006)
0–17
Ischemic stroke 2000 6.9 days 2003 7.2 days 2006 6.9 days Subarachnoid hemorrhage 2000 11.5 days 2003 13.2 days 2006 14.2 days Intracranial hemorrhage 2000 10.4 days 2003 11.0 days 2006 10.9 days
Descriptive total cost nonweighted data from HCUP
[20]
HCUP: Health Care Utilization Project; LOS: Length of stay.
stroke [16]. Shorter LOSs among children with ischemic stroke were also reported by Perkins and colleagues in a study of data from the Kids Inpatient Database [6]. The mean LOS regardless of stroke type was 8.3 days compared to 6.6 days for ischemic stroke, 11.2 days for subarachnoid hemorrhage and 9.6 days for intracranial hemorrhage. Engle and Ellis also reported shorter LOSs among children with ischemic stroke (6.9 days), compared to subarachnoid hemorrhage (14.2 days) and intracranial hemorrhage (10.9 days) in 2006 [20]. Expert commentary
Pediatric stroke is generally considered a rare event, and pediatric stroke-related cost data have been slow to emerge. Early management of pediatric stroke is similar to adult management, and therefore it is generally assumed the cost of pediatric stroke is equivalent to adult stroke [6]. However, specific clinical differences in stroke etiology suggest that cost differences may exist between children and adults [6]. In congruence with the adult literature, the cost of pediatric stroke is substantial and influenced by a number of factors associated with the stroke (cause, type, severity, stroke-related neurophysiological changes, age of onset, etc.), as well as the number and types of services required. At informahealthcare.com
least two studies in the review reported higher costs for hemorrhagic pediatric strokes compared to those experiencing ischemic strokes. Similarly, four studies reported longer LOSs among those with hemorrhagic strokes (including subdural hematoma and intracranial hemorrhage) [6,16,19,20]. This is not surprising as stroke type affects initial deficit profile which subsequently dictates the type, intensity and range of services required for optimal management. In addition, the receipt of specialized services contributes to higher overall costs of care [22]. According to Turney and colleagues, hemorrhagic strokes and ischemic strokes differ in the specific categories of care needed (nursing, pharmacy, supplies, etc.) and overall LOSs, thereby resulting in longer LOSs among patients with hemorrhagic stroke [15]. Similarly, in a study of 256 cases of pediatric stroke, Fox and colleagues found that 62% were admitted to intensive care units (ICUs), 32% were intubated and 11% were treated with decompressive neurosurgery [23]. Admission to ICU units is associated with a higher level of complex care, and independent predictors of ICU admission include younger age and presentation with altered mental status. The LOS data reported here offers additional insight into the cost of pediatric stroke care. The mean LOS for pediatric 647
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stroke ranged from 6.6 to 14.2 days depending upon stroke type. Data from the Centers for Disease Control indicate that the average LOS for adults 65 was 5.3 days in 2009 [24]. Although only four studies are reported here, evidence suggests the cost of pediatric stroke is substantial and at a minimum equivalent to the cost of adult-onset stroke. The studies reported in this review are primarily limited to direct costs of care during the first year after stroke. Gardner and colleagues extended their study beyond the typical 1-year measurement point and reported 5-year direct medical cost data as a measure of the societal burden of pediatric stroke [21]. Pediatric survivors can go on to lead full lives of average lifespans and to our knowledge, few if any studies have carefully considered the long-term impact of pediatric stroke and more specially the indirect costs of pediatric stroke. Limiting cost data to 1 year offers little in terms of clarifying the true overall costs of the stroke event. The economic cost of pediatric stroke is unique in that the span of time that costs will be expended differs dramatically from adult-onset stroke [6]. Although many pediatric stroke survivors will live full lives with average life expectancies, a portion will have persisting motor, sensory, cognitive, language and other behavioral deficits [25–28]. These deficits can occur prior to or at the same time that they are undergoing normal development of the same skills [29]. Consequently, other direct costs required to address persisting deficits and to reduce risk of recurrent stroke can be substantial. These can include the cost of physician visits, rehabilitative therapies, medications and other treatments that will be expended over an exponentially long period of time. Evidence from the general disability literature also suggests that experiencing a pediatric stroke results in unique costs that are not common among adult-onset stroke survivors. These include the costs of services required to facilitate the child’s participation in school and play as well as specialized child-care services outside of the educational system [30]. Some costs are long term and reside outside of the health care system. State and local educational systems may require additional resources to educate children with disabilities depending on the level of residual deficits they experience. Currently, data do not exist specific to stroke; however, deficits that can result from stroke such as developmental delay, intellectual disability, specific learning disabilities and speech and language impairments are known to increase the cost of childhood education [31]. The economic cost of pediatric stroke likely extends beyond the direct medical costs related to the child. Indirect and difficult-to-measure family unit costs can be incurred due to reductions in the family unit’s health-related quality of life. However, in the absence of focused research related to the indirect impact of pediatric stroke on the family unit, a number of questions remain. First, what are the negative financial and psychosocial implications of childhood illness on the family unit [30]? Second, are the mothers of children with pediatric stroke-related disabilities limited in their ability to engage in competitive employment [32,33]? Third, is there additional 648
burden among mothers caring for children with mental health conditions after pediatric stroke [34]. Fourth, are there longer term issues such as unemployment and underemployment that are the result of mental health, cognitive and other deficits that persist into adulthood [5]? Further study of these issues is needed to assess the relationship between changes in costs and resource utilization over time with the disease condition, as well as with changes in clinical and socio-demographic characteristics. As an example, acute-care utilization has been shown to be associated with socioeconomic and neighborhood factors, and these often change through time [35]. Other non-monetary family unit costs can be linked to caregiver illness and family psychosocial distress related to caregiving for children with disabilities. Childhood disability has been associated with significant reductions in caregiver physical health and sleep quality, as well as elevations in levels of depression and stress [36]. More specifically, parents of children with stroke can have significant and wide-ranging health difficulties including mental health issues and issues associated with personal and social development [37]. Ultimately, these issues can translate into poor health among parents of children with disabilities, which result in greater health care costs for the parents [30]. It is tenable then that these issues are not limited to parents/caregivers but the entire family unit. For example, families that have a child with health issues have greater odds of experiencing separation or divorce [38]. Likewise, siblings of children with disabilities experience limited parental availability and more negative psychological functioning including internalizing problems (e.g., depression and anxiety) and externalizing problems (e.g., behavioral problems, aggression and social problems) [39]. Five-year view
The cost of pediatric stroke is substantial. Previous studies have primarily focused on the 1-year direct medical care cost of caring for the child with stroke. Evidence suggests that some stroke-related deficits persist into adulthood [5]. Consequently, ‘direct costs’ of care may also continue for some including long-term health care costs for the child, the costs of educating children who experience disabilities resulting from their stroke and the long-term family-related health care costs due to their experiencing poorer health. It is important to note that not all pediatric stroke survivors experience persisting disabilities and these associated long-term costs. However, a research agenda is currently needed to determine the long-term economic impact of pediatric stroke including the direct and indirect cost to the stroke survivor, to their parents and to society. A longitudinal epidemiological approach could offer important information about the short- and long-term clinical, functional and economic outcomes associated with pediatric stroke. Given the magnitude of costs associated with pediatric stroke, longitudinal studies will offer critical information about current approaches to treating, educating and managing patient and family-related issues and to determine their cost–effectiveness which will in turn reduce costs and improve outcomes. Expert Rev. Pharmacoecon. Outcomes Res. 14(5), (2014)
Costs of pediatric stroke care in the USA
Financial & competing interests disclosure
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The authors have no relevant affiliations or financial involvement with any organization or entity with a financial interest in or financial conflict with the subject matter or materials discussed in the manuscript. This
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includes employment, consultancies, honoraria, stock ownership or options, expert testimony, grants or patents received or pending, or royalties. No writing assistance was utilized in the production of this manuscript.
Key issues • The long-term direct and indirect financial burden of pediatric stroke may be greater than adult stroke. • Cost-related data have yet to emerge beyond 5 years, although pediatric stroke survivors can live into adulthood. • Cost-of-care data are critical to understanding service utilization patterns and which treatments and services are most cost–effective in achieving optimal outcomes. • Current cost-of-care data for pediatric stroke include both reports of ‘charges’ and ‘payments’, thus limiting an interpretation of the overall cost perspective. • Studies are needed to examine the long-term indirect cost of pediatric stroke or the cost of care outside of the primary hospital where care was received. • Studies are needed to examine the ‘indirect costs’ of pediatric stroke to the stroke survivor and their families.
case fatality of stroke among children from 1994 to 2007. Neurology 2012;78(24): 1923-9
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