Cost-effectiveness analysis of a surveillance program to prevent hip dislocation in children with cerebral palsy

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Abstract Objective In this study we conducted an economic evaluation of a surveillance programme to prevent hip dislocation in children with cerebral palsy. Method We developed a model that compared costs and health outcomes of children with cerebral palsy with and without a surveillance programme. Information from a number of sources was combined into a decision analytical model, primarily based on data from a comparative study with a 20-year follow-up. Effectiveness was measured using Quality-Adjusted Life Years (QALYs). The analysis took the perspective of the Spanish National Health Service. We undertook extensive sensitivity analyses including a probabilistic sensitivity analysis. Results The surveillance programme led to higher QALYs and higher health care costs, with an estimated incremental cost per QALY gained of 12,282€. The results were robust to model assumptions. The probability that the programme was cost-effective was estimated to be over 80% at the threshold of 25.000€/QALY recommended in Spain. Conclusion This study indicates that surveillance programmes to prevent hip dislocation in children with cerebral palsy are likely to be cost-effective.

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Challenges in health state valuation in paediatric economic evaluation: are QALYs contraindicated?

Wendy J Ungar (2011)

With the growth in the use of health economic evaluation to inform healthcare resource allocation decisions, the challenges in applying standard methods to child health have become apparent. A unique limitation is the paucity of child-specific preference-based measures. A single, valid, preference-based measure of utility that can be used in children of all ages does not exist. Thus, the ability to derive a QALY for use in cost-utility analysis (CUA) is compromised. This paper presents and discusses existing and novel options for deriving utilities for paediatric health states for use in CUAs. While a direct elicitation may be preferred, a child's ability to complete a standard gamble or time trade-off task is hampered by cognitive and age limitations. The abstract notions contained in indirect instruments such as the EQ-5D and Health Utilities Index may also pose challenges for young children. Novel approaches to overcome these challenges include the development of age-appropriate instruments such as the EQ-5D-Y, the development of new child-specific utility instruments such as the Child Health Utility-9D and the re-calibration of existing adult instruments to derive preference weights for health states from children themselves. For children aged <6 years, researchers have little choice but to use a proxy reporter such as parents. While parents may be reliable reporters for physical activity limitations and externally manifest symptoms, their ability to accurately report on subjective outcomes such as emotion is questionable. Catalogues of utility weights for a range of conditions are increasingly becoming available but retain many of the same limitations as valuing health states from children or from proxies. Given the dynamic relationship in quality of life (QOL) between family members when a child is ill, it seems appropriate to consider a 'family perspective' rather than an individual perspective in child health state valuation. In a collective approach, health state utilities derived from multiple family members may be combined mathematically. Alternatively, in a unitary approach, a single utility estimate may be determined to represent the family's perspective. This may include deriving utilities through parent-child dyad estimation or by using a household model that combines the utility weights of the patient and family members, incorporating reciprocal QOL effects. While these various approaches to child health state valuation represent novel research developments, the measurement challenges and threats to validity persist. Given the importance of non-health benefits to child health, especially in the domains of education and public policy, it may be worthwhile to consider an approach that allows incorporation of externalities to produce a cost-benefit analysis. The use of discrete-choice methods to assess willingness to pay for novel child health interventions holds promise as a means to produce meaningful economic evidence. Regardless of the approach taken, the highest degree of methodological rigour is essential. The increasing attention being paid by health economic researchers to the measurement challenges of paediatric health state valuation can only increase the value of child health economic evidence for decision making.

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Prevention of dislocation of the hip in children with cerebral palsy: 20-year results of a population-based prevention programme.

G Hägglund, A Alriksson-Schmidt, H Lauge-Pedersen … (2014)

In 1994 a cerebral palsy (CP) register and healthcare programme was established in southern Sweden with the primary aim of preventing dislocation of the hip in these children. The results from the first ten years were published in 2005 and showed a decrease in the incidence of dislocation of the hip, from 8% in a historical control group of 103 children born between 1990 and 1991 to 0.5% in a group of 258 children born between 1992 and 1997. These two cohorts have now been re-evaluated and an additional group of 431 children born between 1998 and 2007 has been added. By 1 January 2014, nine children in the control group, two in the first study group and none in the second study group had developed a dislocated hip (p < 0.001). The two children in the first study group who developed a dislocated hip were too unwell to undergo preventive surgery. Every child with a dislocated hip reported severe pain, at least periodically, and four underwent salvage surgery. Of the 689 children in the study groups, 91 (13%) underwent preventive surgery. A population-based hip surveillance programme enables the early identification and preventive treatment, which can result in a significantly lower incidence of dislocation of the hip in children with CP. ©2014 The British Editorial Society of Bone & Joint Surgery.

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Improving decision analyses: parent preferences (utility values) for pediatric health outcomes.

Aaron Carroll, Stephen Downs (2009)

To gather and calculate utilities for a wide range of health states in the pediatric population. The study subjects, parents or guardians at least 18 years of age with at least 1 child under age 18 years, were recruited through our Pediatric Research Network (PResNet). Recruitment locations included pediatric clinics, the Indiana State Fair, and public and private conventions. Each subject's utilities were assessed on 3 random health states out of 29 chosen for the study. Both the time trade-off and standard gamble methods were used to measure utilities. Utilities were assessed in a total of 4016 participants (a recruitment rate of 88%). Utility values ranged from a high for acute otitis media (0.96 by standard gamble; 0.97 by time trade-off) to a low for severe mental retardation (0.59 by standard gamble; 0.51 by time trade-off). Our extensive data set of utility assessments for a wide range of disease states can aid future economic evaluations of pediatric health care.

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Title: Gaceta Sanitaria

Abbreviated Title: Gaceta Sanitaria

Publisher: Elsevier BV

ISSN (Print): 02139111

Publication date Created: September 2019

Publication date (Print): September 2019

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DOI: 10.1016/j.gaceta.2019.05.005

SO-VID: 3f1b4ad0-706f-41b2-b180-d8b9e674b94c

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https://www.elsevier.com/tdm/userlicense/1.0/

http://creativecommons.org/licenses/by-nc-nd/4.0/

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Cost-effectiveness analysis of a surveillance program to prevent hip dislocation in children with cerebral palsy

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Most cited references 32

Challenges in health state valuation in paediatric economic evaluation: are QALYs contraindicated?

Prevention of dislocation of the hip in children with cerebral palsy: 20-year results of a population-based prevention programme.

Improving decision analyses: parent preferences (utility values) for pediatric health outcomes.

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