Preference-based utility weights for the Individualized Neuromuscular Quality of Life Questionnaire (INQoL), with a focus on non-dystrophic myotonia (NDM).
Eur J Health Econ
; 2024 Feb 28.
Article
em En
| MEDLINE
| ID: mdl-38416296
ABSTRACT
INTRODUCTION:
The Individualized Neuromuscular Quality of Life Questionnaire (INQoL) is used to measure quality of life in neuromuscular disorders such as non-dystrophic myotonia (NDM). Here we report methods to estimate utilities, with a focus on NDM, from this questionnaire based on two preference elicitation exercises.METHODS:
Eight items from the INQoL were selected with input from three neuromuscular disorder clinical experts with expertise in treating NDM. A discrete choice experiment (DCE) survey of UK general public respondents (n = 508) described outcomes defined by the INQoL items. The same 8 items were also valued using time trade-off (TTO) face-to-face interviews (n = 200). A hybrid regression modelling approach combined both datasets to inform the utility weights.RESULTS:
Hybrid modelling of DCE and TTO data in conjunction improved out-of-sample predictive accuracy. The selected INQoL utility model indicates substantial disutility associated with all eight dimensions of health, with the greatest losses associated with subjective items such as pain and depression.DISCUSSION:
The hybrid modelling approach allows us to combine data from the two methodologies and maximize the information from each to inform the utility weights for the INQoL. The TTO is the more conventional valuation method, but combined with the larger DCE study produced better descriptive coverage. This is a relatively novel method for estimating weights which we think is particularly well suited to economic evaluations of orphan drugs.
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Bases de dados:
MEDLINE
Idioma:
En
Revista:
Eur J Health Econ
Assunto da revista:
SAUDE PUBLICA
/
SERVICOS DE SAUDE
Ano de publicação:
2024
Tipo de documento:
Article
País de afiliação:
Reino Unido