Health care resource use in preschool children with cerebral palsy.

AIM: To estimate the burden of disease and evaluate which factors affect health care resource use (HCRU) in young children with cerebral palsy (CP). METHOD: Data were collected as part of a prospective, longitudinal cohort study of children with CP born in Queensland, Australia between 2006 and 2009. HCRU questionnaires were administered at six time points. Data on resource use, socio-demographics, and disease severity were collected. Costs were sourced from Medicare, the Australian National Hospital Cost Data Collection, and market prices. A generalized linear model was used to identify factors influencing CP-related costs. RESULTS: A total of 794 questionnaires were completed by 222 participants (mean = 3.6 per participant). Physiotherapy (94%, n = 208) was the most widely accessed allied health care therapy; almost half of the participants (45%; 354 of 794) reported one or more hospital admissions. From the health care funder perspective, a child with CP costs on average A$24 950 per annum (A$12 475 per 6 months). Higher costs were associated with increased motor impairment (Gross Motor Function Classification System, p < 0.001) and increased comorbidities (p = 0.012). INTERPRETATION: HCRU in preschool children with CP can be analysed according to disease severity. Both increased motor impairments and increased comorbidities were associated with higher health care costs. WHAT THIS PAPER ADDS: Children with a higher number of comorbidities had higher health care costs, and more specifically, higher hospitalization costs. No significant change in costs was found over time as children aged from 18 months to 5 years.

Supporting Ultra Poor People with Rehabilitation and Therapy among families of children with Cerebral Palsy in rural Bangladesh (SUPPORT CP): Protocol of a randomised controlled trial.

INTRODUCTION: Poverty is a key contributor to delayed diagnosis and limited access to early intervention and rehabilitation for children with cerebral palsy (CP) in rural Bangladesh. 97% of families of children with CP live below the poverty line in Bangladesh. Therefore, in low-and middle-income countries (LMICs), efforts to improve outcomes for children with CP (including health-related quality of life, motor function, communication, and nutritional attainments) should also include measures to improve family economic and social capital. We propose a randomised controlled trial (RCT) to evaluate the effectiveness of an integrated microfinance/livelihood and community-based rehabilitation (IMCBR) program for ultra-poor families of children with CP in rural Bangladesh. MATERIAL AND METHODS: This will be a cluster RCT comparing three arms: (a) integrated microfinance/livelihood and community-based rehabilitation (IMCBR); (b) community-based rehabilitation (CBR) alone; and (c) care-as-usual (i.e. no intervention). Seven clusters will be recruited within each arm. Each cluster will consist of 10 child-caregiver dyads totalling 21 clusters with 210 dyads. Parents recruited in the IMCBR arm will take part in a microfinance/livelihood program and Parent Training Module (PTM), their children with CP will take part in a Goal Directed Training (GDT) program. The programs will be facilitated by specially trained Community Rehabilitation Officers. The CBR arm includes the same PTM and GDT interventions excluding the microfinance/livelihood program. The care-as-usual arm will be provided with information about early intervention and rehabilitation. The assessors will be blinded to group allocation. The duration of the intervention will be 12 months; outcomes will be measured at baseline, 6 months, 12 months, and 18 months. CONCLUSION: This will be the first RCT of an integrated microfinance/livelihood and CBR program for children with CP in LMIC settings. Evidence from the study could transform approaches to improving wellbeing of children with CP and their ultra-poor families.

Polypharmacy Among Privately Insured Adults with Cerebral Palsy: A Retrospective Cohort Study.

BACKGROUND: Adults with cerebral palsy (CP) have increased risk for developing various secondary chronic diseases, especially when they have other neurodevelopmental disabilities (NDDs). Multiple medications are likely prescribed to manage the greater morbidity-related burden for adults with CP; however, because health care delivery and care coordination is suboptimal for this population, adults with CP may have an increased risk for polypharmacy. To date, very little is known about the prescribing practices and extent of polypharmacy for adults with CP. OBJECTIVE: To determine the prevalence and adjusted odds of polypharmacy among adults with CP only and those with CP+NDDs, compared with adults without CP. METHODS: Data from 2017 Optum Clinformatics Data Mart, a U.S. private administrative database, was used for this retrospective cohort study. Diagnosis codes were used to identify adults (aged ≥ 18 years) with CP, NDDs (e.g., intellectual disabilities, epilepsy, and autism spectrum disorders), and 24 relevant morbidities. Polypharmacy was examined as 0-4 versus ≥ 5, 0-9 versus ≥ 10, and 0-14 versus ≥ 15 medications. Logistic regression estimated the OR and 95% CI of polypharmacy before and after adjusting for age, sex, region of residence, and multimorbidity (as 0, 1, 2, 3, 4-5, and ≥ 6 morbidities). Exploratory analyses were conducted to compare polypharmacy among young (18-40 years) and middle-aged (41-64 years) adults with CP only and CP + NDDs with elderly (≥ 65 years) adults without CP. RESULTS: Adults with CP only (n = 5,603) and CP + NDDs (n = 2,474) had higher unadjusted prevalence and adjusted OR for each polypharmacy definition compared with adults without CP (n = 9.0 million; e.g., ≥ 5 medications: adjusted OR for CP only = 1.38, 95% CI = 1.30-1.47; CP + NDDs: OR = 2.42, 95% CI = 2.20-2.67). Adults with CP+NDDs had higher unadjusted prevalence and adjusted OR of each polypharmacy definition compared with CP only. Compared with elderly without CP, the unadjusted prevalence of polypharmacy was lower for young adults with CP only (e.g., ≥ 5 medications: 60.2%, 43.8%), similar for young adults with CP+NDDs (e.g., ≥ 15 medications: 10.9%, 12.5%), and elevated for middle-aged CP only and CP + NDDs (e.g., ≥ 10 medications: 28.7%, 34.3%, 41.7%). CONCLUSIONS: Privately insured adults with CP only and CP + NDDs have an elevated prevalence of polypharmacy compared with adults without CP, even after accounting for multimorbidity. Importantly, adults aged 18-40 years with CP have a similar (CP + NDDs) prevalence of polypharmacy compared with the general geriatric population, with the prevalence increasing further for CP by middle age. DISCLOSURES: Whitney was supported by the University of Michigan Office of Health Equity and Inclusion Diversity Fund and the American Academy of Cerebral Palsy and Developmental Medicine. These funding sources had no role in the design or conduct of the study; collection, management, analysis, or interpretation of the data; preparation, review, or approval of the manuscript; or the decision to submit the manuscript for publication. The other authors have no conflicts of interest to disclose.

Cost-effectiveness of botulinum neurotoxin A versus surgery for drooling: a randomized clinical trial.

AIM: This study compared the cost-effectiveness of botulinum neurotoxin A (BoNT-A) injections with two-duct ligation of the submandibular glands as treatment for severe drooling after one treatment cycle. METHOD: The study was part of a larger, partly single-blinded, randomized clinical trial (trialregister.nl identifier NTR3537). Data were collected between 2012 and 2017. Evaluation was at 32 weeks after one treatment cycle. Fifty-seven patients with cerebral palsy or other neurological, non-progressive disorders and severe drooling classified as having a drooling frequency ≥3 or a drooling severity ≥2, in whom conservative treatment was deemed ineffective, were randomized to treatment by BoNT-A or two-duct ligation. An incremental cost-effectiveness ratio (ICER) was calculated using the success rates as the measure of benefit. Treatment success was defined as a decrease ≥50% from baseline to 32 weeks in the subjective visual analogue scale for the severity of drooling or the objective drooling quotient. RESULTS: Fifty-three patients were analysed (22 females, 31 males; mean age 11y, range 8-22y). Average costs for one treatment cycle, which included one BoNT-A injection, were €1929 (standard error 62) for BoNT-A and €3155 (standard error 99) for two-duct ligation. Treatment success was in favour of two-duct ligation (63% vs 27%; number needed to treat 3). The ICER was €34 per 1% gain in treatment success in favour of two-duct ligation versus BoNT-A. INTERPRETATION: The additional cost of two-duct ligation is to some extent offset by a larger treatment success rate compared with BoNT-A. WHAT THIS PAPER ADDS: Botulinum neurotoxin A (BoNT-A) is less expensive per percentage of success than two-duct ligation. The additional cost of two-duct ligation over BoNT-A is offset by greater treatment success.

Continuity of Care Is Associated with Medical Costs and Inpatient Days in Children with Cerebral Palsy.

Background: Children with cerebral palsy (CP) place a considerable burden on medical costs and add to an increased number of inpatient days in Taiwan. Continuity of care (COC) has not been investigated in this population thus far. Materials and Methods: We designed a retrospective population-based cohort study using Taiwan's National Health Insurance Research Database. Patients aged 0 to 18 years with CP catastrophic illness certificates were enrolled. We investigated the association of COC index (COCI) with medical costs and inpatient days. We also investigated the possible clinical characteristics affecting the outcome. Results: Over five years, children with CP with low COCI levels had higher medical costs and more inpatient days than did those with high COCI levels. Younger age at CP diagnosis, more inpatient visits one year before obtaining a catastrophic illness certificate, pneumonia, and nasogastric tube use increased medical expenses and length of hospital stay. Conclusions: Improving COC reduces medical costs and the number of inpatient days in children with CP. Certain characteristics also influence these outcomes.

Cerebral palsy in Canada, 2011-2031: results of a microsimulation modelling study of epidemiological and cost impacts. / La paralysie cérébrale au Canada, 2011-2031 : résultats d'une étude de modélisation par microsimulation des répercussions épidémiologiques et économiques.

INTRODUCTION: The objective of our study was to present model-based estimates and projections on current and future health and economic impacts of cerebral palsy in Canada over a 20-year time horizon (2011-2031). METHODS: We used Statistics Canada's Population Health Model (POHEM)-Neurological to simulate individuals' disease states, risk factors and health determinants and to describe and project health outcomes, including disease incidence, prevalence, life expectancy, health-adjusted life expectancy, health-related quality of life and health care costs over the life cycle of Canadians. Cerebral palsy cases were identified from British Columbia's health administrative data sources. A population-based cohort was then used to generate the incidence and mortality rates, enabling the projection of future incidence and mortality rates. A utility-based measure (Health Utilities Index Mark 3) was also included in the model to reflect various states of functional health to allow projections of health-related quality of life. Finally, we estimated caregiving parameters and health care costs from Canadian national surveys and health administrative data and included them as model parameters to assess the health and economic impact of cerebral palsy. RESULTS: Although the overall crude incidence rate of cerebral palsy is projected to remain stable, newly diagnosed cases of cerebral palsy will rise from approximately 1800 in 2011 to nearly 2200 in 2031. In addition, the number of people with the condition is expected to increase from more than 75 000 in 2011 to more than 94 000 in 2031. Direct health care costs in constant 2010 Canadian dollars were about $11 700 for children with cerebral palsy aged 1-4 years versus about $600 for those without the condition. In addition, people with cerebral palsy tend to have longer periods in poorer health-related quality of life. CONCLUSION: Individuals with cerebral palsy will continue to face challenges related to an ongoing need for specialized medical care and a rising need for supportive services. Our study offers important insights into future costs and impacts associated with cerebral palsy and provides valuable information that could be used to develop targeted health programs and strategies for Canadians living with this condition.

CP-North: living life in the Nordic countries? A retrospective register research protocol on individuals with cerebral palsy and their parents living in Sweden, Norway, Denmark, Finland and Iceland.

INTRODUCTION: Cerebral palsy (CP) is one of the most common neurodevelopmental disabilities. Yet, most individuals with CP are adults. How individuals with CP fare in terms of health, quality of life (QoL), education, employment and income is largely unknown. Further, little is known about the effects of having a child with CP on the parents. The Nordic countries are known for their strong welfare systems, yet it is unknown to what extent the added burden related to disability is actually compensated for. We will explore how living with CP affects health, QoL, healthcare utilisation, education, labour market outcomes, socioeconomic status and mortality throughout the lifespan of individuals with CP and their parents. We will also investigate if these effects differ between subgroups, within and across the Nordic countries. METHODS AND ANALYSES: CP-North is a multidisciplinary 4-year (1 August 2017 to 31 July 2021) register research project. The research consortium comprises researchers and users from Sweden, Norway, Denmark, Iceland and Finland. Data from CP registries and follow-up programmes, or cohorts of individuals with CP, will be merged with general national registries. All individual studies are structured under three themes: medical outcomes, social and public health outcomes, and health economics. Both case-control and cohort designs will be included depending on the particular research question. Data will be analysed in the individual countries and later merged across nations. ETHICS AND DISSEMINATION: The ethics approval processes in each individual country are followed. Findings will be published (open access) in international peer-reviewed journals in related fields. Updates on CP-North will be published online at http://rdi.arcada.fi/cpnorth/en/.

Prevalence, Patterns, and Cost of Care for Children with Cerebral Palsy Enrolled in Medicaid Managed Care.

BACKGROUND: In the United States, many children with cerebral palsy (CP) obtain health care coverage through managed Medicaid, but little is known about the current demographics or management of this high-need, complex population. OBJECTIVE: To develop U.S. population-level information about the prevalence of CP, management patterns, and costs. METHODS: Data (2013-2015) were analyzed from a managed Medicaid database with coverage of children and adolescents in 15 states. Analyses included demographic information and use of 10 prespecified CP management options often used to manage spasticity. Code-based algorithms were applied to indicate presence of spasticity and determine the likely ambulatory status. RESULTS: In this claims analysis, the prevalence estimate of CP was 1.78 per 1,000 patients. Most (69.8%) children with CP had spasticity, of which 20.8% had hemiplegia, 15.6% diplegia, 32.9% quadriplegia, and 30.5% CP unspecified. Overall, 42.4% of children with CP were not treated with any of the 10 CP management options via Medicaid. Among treated children, the most common management options were physical therapy (37.1%), orthotics (29.9%), oral baclofen (13.5%) and botulinum toxins (9.4%). Overall annualized Medicaid costs were higher for children with CP versus children in the overall database population ($22,383 vs. $1,358). Within the CP population, costs were higher for those children who were likely nonambulatory than for those who were likely ambulatory ($43,687 vs. $10,368, respectively). CONCLUSIONS: Most children with CP have spasticity, and the costs of care are high. This study highlights wide variation in the way CP is managed, with many young patients not receiving CP management options via Medicaid. DISCLOSURES: This analysis was funded by Ipsen Biopharmaceuticals and conducted by Milliman. Pulgar and Bains were employees of Ipsen Biopharmaceuticals during the conduct of this study. Chambers is a consultant for OrthoPediatrics and an employee of the University of California. Pyenson and Ferro are employees of Milliman, as was Sawhney during the analysis. Gooch, Noritz, and Wright report no conflicts of interest. Part of this work was presented as a poster at TOXINS 2017: Basic Science and Clinical Aspects of Botulinum and Other Neurotoxins, held January 18-21, 2017, in Madrid, Spain.

[Epidemiology, cost and economic impact of cerebral palsy in Hungary]. / A cerebralis paresis epidemiológiája, költségei és közgazdasági hatásai Magyarországon.

BACKGROUND AND PURPOSE: The purpose of our communication was to determine the total cost of cerebral paretic patients in Hungary between 0 and 18 years and to assess their impact on the national budget. METHODS: Based on the data of Borsod county we calculated the CP characteristics. The cost of CP was determined by routine care of individuals. Lost Parental Income and Tax were calculated on the basis of average earnings. The ratio of GDP, Health and Social Budget and Health Budget to CP is based on CP annual average cost and frequency. We have developed a repeatable computational model. RESULTS: Of the risk groups, premature birth (30.97%), low birth weight (29.64%), perinatal asphyxia (19.47%) were the most common. Source is unknown of 37.61% of the cases. CP prevalence was 2.1‰. The two-sided (59.7%) and the one-sided (19.0%) spastic pareses dominated. The most serious form is the two-sided spastic paresis (42.5% GMFCS 3-5 degrees). Epilepsy was 22.0%, incontinence was 27%, mental involvement was 46%. Care for one child up to 18 years of age costs an average of 73 million HUF (€ 251,724). The lost family income was 27.36 million HUF (€ 94,345), and lost tax and health care contributions were 14.46 million HUF (€ 49,862). Additionally, 0.525% of the GDP, 0.88% of the full health and social budget and 1.83% of direct medical costs were spent for CP families. CONCLUSION: The cost of CP disease is significant. Costs can be reduced by improving primary prevention. From the perspective of the family and government, it is better to care for families so they can take care of their disabled children.

Profile of the judicialization of the Therasuit Method and its direct cost in the scope of the state of Rio de Janeiro. / Perfil da judicialização do Método Therasuit e seu custo direto no âmbito do Estado do Rio de Janeiro.

INTRODUCTION: The lack of availability of the Therasuit Method by the Unified Health System associated with its high cost has led to the prosecution of this treatment. The study aimed to outline the profile of this judicialization, as well as to estimate the direct costs resulting from compliance with the deferred judicial decisions. METHOD: Weanalyzed the cases submitted to the Court of Justice of Rio de Janeiro between January 2013 and January 2017, in which the Therasuit Method was applied. Demographic, clinical, advocacy and legal data were extracted, as was the timing of the court's decision and the required technology budgets. RESULTS: Atotal of 11 processes was analyzed. The authors had a mean age of 6.8 years and a median of 6, the majority being male, and resident in thestate capital. Quadriparesis was the most reported condition. The gratuity of justice was requested by all, and the Public Defender's Officewas used by 9 of the 11 processes. The judicial decisions at first instance were considered. In all of processes there was application of the legal tool called guardianship. The time of the judicial decision was on average of 266.5 days with a median of 35.5. The deferral index was 90%, totaling an annual direct cost of R$501,894.09. DISCUSSION: The judicialization of this treatment can cause an unforeseen displacement of public funds, transgressing the principles of equity and the integrality of Unified Health System. CONCLUSION: It was observed a high rate of deferred processes, resulting in a high cost spent by the Public Power to attend a small portion of patients.

INTRODUÇÃO: O Método Therasuit possui alto custo e não é disponibilizado pelo Sistema Único de Saúde, o que tem ocasionado a judicialização desse tratamento. O estudo visou traçar o perfil dessa judicialização, bem como estimar os custos diretos decorrentes do cumprimento das decisões judiciais deferidas. MÉTODO: Foram analisados processos submetidos ao Tribunal de Justiça do Rio de Janeiro entre janeiro de 2013 e janeiro de 2017, no qual foi solicitado o Método Therasuit. Os dados demográficos, clínicos, advocatícios e jurídicos foram extraídos, assim como o tempo da decisão judicial e os orçamentos da tecnologia requerida. RESULTADOS: O total de 11 processos foi analisado. Os autores tinham uma média de idade de 6,8 anos e mediana de 6, sendo a maioria do sexo masculino e residentes na capital do estado. A quadriparesia foi a condição mais relatada. A gratuidade de justiça foi solicitada por todos, e a Defensoria Pública foi utilizada em 9 dos 11 processos analisados. Foram consideradas as decisões judiciais em primeira instância. O tempo da decisão judicial foi em média de 266,5 dias com uma mediana de 35,5. Em todos houve requerimento da ferramenta jurídica tutela antecipada. O índice de deferimento foi de 90%, totalizando um custo direto anual de R$501.894,09. DISCUSSÃO: A judicialização desse tratamento pode ocasionar um deslocamento não previsto de verba pública, podendo impactar nos princípios da equidade e na integralidade do Sistema Único de Saúde. CONCLUSÃO: Constatou-se elevada taxa de processos deferidos, acarretando um alto custo despendido pelo Poder Público para atender a uma pequena parcela de pacientes.

Economic cost of congenital CMV in the UK.

OBJECTIVE: Congenital cytomegalovirus (cCMV) is the most common infectious cause of congenital disability. It can disrupt neurodevelopment, causing lifelong impairments including sensorineural hearing loss and developmental delay. This study aimed, for the first time, to estimate the annual economic burden of managing cCMV and its sequelae in the UK. DESIGN: The study collated available secondary data to develop a static cost model. SETTING: The model aimed to estimate costs of cCMV in the UK for the year 2016. PATIENTS: Individuals of all ages with cCMV. MAIN OUTCOME MEASURES: Direct (incurred by the public sector) and indirect (incurred personally or by society) costs associated with management of cCMV and its sequelae. RESULTS: The model estimated that the total cost of cCMV to the UK in 2016 was £732 million (lower and upper estimates were between £495 and £942 million). Approximately 40% of the costs were directly incurred by the public sector, with the remaining 60% being indirect costs, including lost productivity. Long-term impairments caused by the virus had a higher financial burden than the acute management of cCMV. CONCLUSIONS: The cost of cCMV is substantial, predominantly stemming from long-term impairments. Costs should be compared against investment in educational strategies and vaccine development programmes that aim to prevent virus transmission, as well as the value of introducing universal screening for cCMV to both increase detection of children who would benefit from treatment, and to build a more robust evidence base for future research.

Needs of families of children with cerebral palsy in Bangladesh: A qualitative study.

PURPOSE: Families of children with disabilities often have needs related to the care of their child with a disability. Although there has been extensive exploration of family needs in high-income contexts, there is little known about this issue in low and middle-income countries like Bangladesh. In this study, we explored the needs of families of children with cerebral palsy in Bangladesh. Such understanding is important as it will help to improve services for children with disabilities and their families. METHODS: We used a qualitative approach and interviewed 20 family members of children with cerebral palsy who visited the Centre for the Rehabilitation of the Paralysed, Bangladesh. We thematically analyzed data from semistructured interviews. RESULTS: Five different themes were found on needs of families with children with disabilities: (a) financial needs, (b) needs for disability-related services, (c) needs for family and community cohesion, (d) informational needs, and (e) emotional needs. Participants overwhelmingly reported that financial needs were their highest priority. CONCLUSION: Needs of families of children with disabilities must be considered in rehabilitation services to improve children's outcomes. Further studies are required to explore needs of families of children with disabilities who do not have access to rehabilitation services.

Perfil da judicialização do Método Therasuit e seu custo direto no âmbito do Estado do Rio de Janeiro / Profile of the judicialization of the Therasuit Method and its direct cost in the scope of the state of Rio de Janeiro

RESUMO: Introdução: O Método Therasuit possui alto custo e não é disponibilizado pelo Sistema Único de Saúde, o que tem ocasionado a judicialização desse tratamento. O estudo visou traçar o perfil dessa judicialização, bem como estimar os custos diretos decorrentes do cumprimento das decisões judiciais deferidas. Método: Foram analisados processos submetidos ao Tribunal de Justiça do Rio de Janeiro entre janeiro de 2013 e janeiro de 2017, no qual foi solicitado o Método Therasuit. Os dados demográficos, clínicos, advocatícios e jurídicos foram extraídos, assim como o tempo da decisão judicial e os orçamentos da tecnologia requerida. Resultados: O total de 11 processos foi analisado. Os autores tinham uma média de idade de 6,8 anos e mediana de 6, sendo a maioria do sexo masculino e residentes na capital do estado. A quadriparesia foi a condição mais relatada. A gratuidade de justiça foi solicitada por todos, e a Defensoria Pública foi utilizada em 9 dos 11 processos analisados. Foram consideradas as decisões judiciais em primeira instância. O tempo da decisão judicial foi em média de 266,5 dias com uma mediana de 35,5. Em todos houve requerimento da ferramenta jurídica tutela antecipada. O índice de deferimento foi de 90%, totalizando um custo direto anual de R$501.894,09. Discussão: A judicialização desse tratamento pode ocasionar um deslocamento não previsto de verba pública, podendo impactar nos princípios da equidade e na integralidade do Sistema Único de Saúde. Conclusão: Constatou-se elevada taxa de processos deferidos, acarretando um alto custo despendido pelo Poder Público para atender a uma pequena parcela de pacientes.

ABSTRACT: Introduction: The lack of availability of the Therasuit Method by the Unified Health System associated with its high cost has led to the prosecution of this treatment. The study aimed to outline the profile of this judicialization, as well as to estimate the direct costs resulting from compliance with the deferred judicial decisions. Method: Weanalyzed the cases submitted to the Court of Justice of Rio de Janeiro between January 2013 and January 2017, in which the Therasuit Method was applied. Demographic, clinical, advocacy and legal data were extracted, as was the timing of the court's decision and the required technology budgets. Results: Atotal of 11 processes was analyzed. The authors had a mean age of 6.8 years and a median of 6, the majority being male, and resident in thestate capital. Quadriparesis was the most reported condition. The gratuity of justice was requested by all, and the Public Defender's Officewas used by 9 of the 11 processes. The judicial decisions at first instance were considered. In all of processes there was application of the legal tool called guardianship. The time of the judicial decision was on average of 266.5 days with a median of 35.5. The deferral index was 90%, totaling an annual direct cost of R$501,894.09. Discussion: The judicialization of this treatment can cause an unforeseen displacement of public funds, transgressing the principles of equity and the integrality of Unified Health System. Conclusion: It was observed a high rate of deferred processes, resulting in a high cost spent by the Public Power to attend a small portion of patients.

Systematic review of the economic impact of cerebral palsy.

BACKGROUND: Cerebral palsy (CP) and its associated conditions can pose a significant economic burden on families, the health care system and the general economy. The boundary for inclusion of costs in research can vary substantially across studies. AIMS: To summarize the evidence for burden of disease for CP including the impacts on the health system, the community and carers. METHODS: Literature was identified from Ovid Medline, Embase, CINHAL, PsyInfo, Econlit, Health Economic Evaluation Database (HEED) and NHS Economic Evaluation Database (NHS EED) in the Cochrane Library. The search was restricted to articles published in English between 1970 and April 2016. All costs were converted to $USD 2016 price. RESULTS: Twenty-two articles were included. Studies varied from snapshot cost descriptions to more complex lifetime estimates, from prevalence-based to incidence-based studies, and from inclusion to exclusion of non-medical costs. There was a strong positive relationship between CP severity and expenditure. Significant costs were incurred by families and the welfare system to facilitate school and community engagement. CONCLUSION: Facilitating participation for people with CP involves substantial expense. The size, nature and distribution of the economic burden emphasises the importance of finding effective strategies to reduce the risk and severity of CP, together with how it is financed.

Associations of Coexisting Conditions with Healthcare Spending for Children with Cerebral Palsy.

OBJECTIVE: To determine which coexisting conditions have the strongest associations with healthcare use and spending among children with cerebral palsy (CP). STUDY DESIGN: Retrospective analysis of 16 695 children ages 0-18 years with CP - identified with International Classification of Diseases, Ninth Revision, Clinical Modification codes - using Medicaid from January 1, 2013 to December 31, 2013 from 10 states in the Truven MarketScan Medicaid Database. Using generalized linear models, we assessed which coexisting conditions (including medical technology) identified with Agency for Healthcare Research and Quality's Chronic Condition Indicators had the strongest associations with total healthcare spending across the healthcare continuum. RESULTS: Median per-patient annual Medicaid spending for children with CP was $12 299 (IQR $4826-$35 582). Most spending went to specialty (33.1%) and hospital (26.7%) care. The children had a median 6 (IQR 4-10) coexisting conditions; epilepsy was the most common (38.1%). Children with epilepsy accounted for 59.6% ($364 million) of all CP spending. In multivariable analysis, the coexisting conditions most strongly associated with increased spending were tracheostomy (median additional cost per patient = $56 567 [95%CI $51 386-61 748]) and enterostomy (median additional cost per patient = $25 707 [95%CI $23 753-27 660]). CONCLUSIONS: Highly prevalent in children with CP using Medicaid, coexisting conditions correlate strongly with healthcare spending. Tracheostomy and enterostomy, which indicate significant functional impairments in breathing and digestion, are associated with the highest spending. Families, providers, payers, and legislators may leverage these findings when designing policies positioned to enable the best health and care for children with cerebral palsy.

Comparing impact on the family and insurance coverage in children with cerebral palsy and children with another special healthcare need.

BACKGROUND: Families and caregivers of children with special healthcare needs (CSHCN) often experience financial difficulties, have unmet physical and mental health needs, and are at increased risk of marital problems due to the stress caused by carrying for their child. Within the larger population of CHSCN, young people with cerebral palsy (CP) have more unmet needs due to the complexity and potential severity of the disability. The purpose of this study was to identify factors associated with differences in insurance coverage and impact on the family of children with CP and other CHSCN. METHODS: The data were taken from the National Survey of Children with Special Health Care Needs, which was designed to examine state- and national-level estimates of CSHCN. Three variables examined differences in insurance coverage between those children diagnosed with CP versus all other CSHCN: insurance coverage for the previous year, current insurance coverage, and adequacy of insurance coverage. Four variables representing different indicators of family impact were used to assess differences between children with CP versus all other CSHCN: out-of-pocket expenses for healthcare, family financial burden, hours per week that family members spent caring for the child, and impact on family work life. RESULTS: The results of this study showed significant differences between households with a child with CP and a child with another health special need in terms of insurance coverage, indicating a tendency of children with CP to be insured the entire year. As for the impact on the family in households with children with CP versus other CSHCN, there were significant differences in all four variables that were analysed. CONCLUSIONS: There is limited evidence highlighting differences between the impact of caring for a child with CP and caring for other CSHCN. Caring for a child with CP has a significant impact on the family, despite insurance coverage.

Economic evaluation and cost of interventions for cerebral palsy: a systematic review.

AIM: Economic appraisal can help guide policy-making for purchasing decisions, and treatment and management algorithms for health interventions. We conducted a systematic review of economic studies in cerebral palsy (CP) to inform future research. METHOD: Economic studies published since 1970 were identified from seven databases. Two reviewers independently screened abstracts and extracted data following the Preferred Reporting Items for Systematic Reviews and Meta-Analyses (PRISMA) guidelines. Any discrepancies were resolved by discussion. RESULTS: Of 980 identified references, 115 were included for full-text assessment. Thirteen articles met standard criteria for a full economic evaluation, two as partial economic evaluations, and 18 as cost studies. Six were full economic evaluations alongside clinical studies or randomized controlled trials, whereas seven involved modelling simulations. The economic case for administration of magnesium sulfate for imminent preterm birth is compelling, achieving both health gain and cost savings. Current literature suggests intrathecal baclofen therapy and botulinum toxin injection are cost-effective, but stronger evidence for long-term effects is needed. Lifestyle and web-based interventions are inexpensive, but broader measurement of outcomes is required. INTERPRETATION: Prevention of CP would avoid significant economic burden. Some treatments and interventions have been shown to be cost-effective, although stronger evidence of clinical effectiveness is needed. What this paper adds Cost-effectiveness evidence shows prevention is the most significant strategy. Some treatments are cost-effective, but stronger evidence for long-term effectiveness is required. Comparison of treatment costs is challenging owing to variations in methodologies and varying clinical indications.

Value of botulinum toxin injections preceding a comprehensive rehabilitation period for children with spastic cerebral palsy: A cost-effectiveness study.

OBJECTIVE: Despite the widespread use of botulinum toxin in ambulatory children with spastic cerebral palsy, its value prior to intensive physiotherapy with adjunctive casting/orthoses remains unclear. DESIGN: A pragmatically designed, multi-centre trial, comparing the effectiveness of botulinum toxin + intensive physiotherapy with intensive physiotherapy alone, including economic evaluation. SUBJECTS/PATIENTS: Children with spastic cerebral palsy, age range 4-12 years, cerebral palsy-severity Gross Motor Function Classification System levels I-III, received either botulinum toxin type A + intensive physiotherapy or intensive physiotherapy alone and, if necessary, ankle-foot orthoses and/or casting. METHODS: Primary outcomes were gross motor func-tion, physical activity levels, and health-related quality-of-life, assessed at baseline, 12 (primary end-point) and 24 weeks (follow-up). Economic outcomes included healthcare and patient costs. Intention-to-treat analyses were performed with linear mixed models. RESULTS: There were 65 participants (37 males), with a mean age of 7.3 years (standard deviation 2.3 years), equally distributed across Gross Motor Function Classification System levels. Forty-one children received botulinum toxin type A plus intensive physio-therapy and 24 received intensive physiotherapy treatment only. At primary end-point, one statistically significant difference was found in favour of intensive physiotherapy alone: objectively measured percentage of sedentary behaviour (-3.42, 95% confidence interval 0.20-6.64, p=0.038). Treatment costs were significantly higher for botulinum toxin type A plus intensive physiotherapy (8,963 vs 6,182 euro, p=0.001). No statistically significant differences were found between groups at follow-up. CONCLUSION: The addition of botulinum toxin type A to intensive physiotherapy did not improve the effectiveness of rehabilitation for ambulatory children with spastic cerebral palsy and was also not cost-effective. Thus botulinum toxin is not recommended for use in improving gross motor function, activity levels or health-related quality-of-life in this cerebral palsy age- and severity-subgroup.

Impact of Non-medical Out-of-pocket Expenses on Families of Children With Cerebral Palsy Following Orthopaedic Surgery.

PURPOSE: Limited research has been conducted on the non-medical out-of-pocket expenses (NOOPEs) incurred by families of children with chronic health conditions. The study objectives were to: 1) calculate the estimated NOOPEs incurred by families during hospitalization of their child, 2) identify predictors of high NOOPEs, and 3) assess the impact of the child's chronic health condition on the family's finances. DESIGN AND METHODS: Prospective observational study. Parents were included if their child was 3-20years old, had severe, non-ambulatory cerebral palsy (CP), and scheduled for hip or spine surgery. Parents reported all NOOPEs incurred during their child's hospitalization using the Family Expense Diary. Families completed the subscales of the Impact on Family Scale and the Assessment of Caregivers Experience with Neuromuscular Disease. Descriptive and univariate and multiple hierarchical regression models were used in the analysis. RESULTS: Fifty two parents participated. The total NOOPEs ranged from $193.00 to $7192.71 (M=$2001.92) per hospitalization representing an average of 4% of the family's annual earned income. Caregiver age (F=8.393, p<0.001), income (F=7.535, p<0.001), and distance traveled to the hospital (F=4.497, p=0.039) were significant predictors of high NOOPEs. The subscale scores indicated that a child's chronic health condition had a significant impact on family finances. CONCLUSIONS AND PRACTICE IMPLICATIONS: Hospitalization is associated with numerous NOOPEs that create additional financial demands for families caring for a child with severe CP. NOOPEs should be addressed when preparing families for their children's planned hospital admissions, especially those families of CSHCN who experience significant financial impacts secondary to their children's care.

Therapy Use for Children With Developmental Conditions: Analysis of Colorado Medicaid Data.

STUDY PURPOSE: To examine therapy use and spending for Medicaid-enrolled infants and toddlers with developmental conditions. METHODS: Sample infants and toddlers had a diagnosis (eg, cerebral palsy) or developmental delay (DD). Colorado Children's Medicaid administrative outpatient therapy claims (2006-2008) were used to estimate differences, by condition type and number of comorbid chronic conditions (CCCs), of any physical therapy (PT)/occupational therapy (OT) and Medicaid PT/OT spending. RESULTS: The sample included 20 959 children. Children with at least 2 CCCs had higher odds of PT/OT than children with no CCC. Children with DD had 12-fold higher odds of having any PT/OT compared with children with diagnosis. Children with a DD and 2 CCCs had the highest PT/OT spending. CONCLUSIONS: Medicaid PT/OT use and spending are higher for children with more CCCs and those with DD because children with DD receive more specialized PT/OT.