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Developing a utility index for the Aberrant Behavior Checklist (ABC-C) for fragile X syndrome
PURPOSE: This study aimed to develop a utility index (the ABC-UI) from the Aberrant Behavior Checklist-Community (ABC-C), for use in quantifying the benefit of emerging treatments for fragile X syndrome (FXS). METHODS: The ABC-C is a proxy-completed assessment of behaviour and is a widely used measu...
Autores principales: | , , , , , , , |
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Formato: | Online Artículo Texto |
Lenguaje: | English |
Publicado: |
Springer International Publishing
2014
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Materias: | |
Acceso en línea: | https://www.ncbi.nlm.nih.gov/pmc/articles/PMC4317522/ https://www.ncbi.nlm.nih.gov/pubmed/25063082 http://dx.doi.org/10.1007/s11136-014-0759-8 |
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author | Kerr, Cicely Breheny, Katie Lloyd, Andrew Brazier, John Bailey, Donald B. Berry-Kravis, Elizabeth Cohen, Jonathan Petrillo, Jennifer |
author_facet | Kerr, Cicely Breheny, Katie Lloyd, Andrew Brazier, John Bailey, Donald B. Berry-Kravis, Elizabeth Cohen, Jonathan Petrillo, Jennifer |
author_sort | Kerr, Cicely |
collection | PubMed |
description | PURPOSE: This study aimed to develop a utility index (the ABC-UI) from the Aberrant Behavior Checklist-Community (ABC-C), for use in quantifying the benefit of emerging treatments for fragile X syndrome (FXS). METHODS: The ABC-C is a proxy-completed assessment of behaviour and is a widely used measure in FXS. A subset of ABC-C items across seven dimensions was identified to include in health state descriptions. This item reduction process was based on item performance, factor analysis and Rasch analysis performed on an observational study dataset, and consultation with five clinical experts and a methodological expert. Dimensions were combined into health states using an orthogonal design and valued using time trade-off (TTO), with lead-time TTO methods used where TTO indicated a state valued as worse than dead. Preference weights were estimated using mean, individual level, ordinary least squares and random-effects maximum likelihood estimation [RE (MLE)] regression models. RESULTS: A representative sample of the UK general public (n = 349; mean age 35.8 years, 58.2 % female) each valued 12 health states. Mean observed values ranged from 0.92 to 0.16 for best to worst health states. The RE (MLE) model performed best based on number of significant coefficients and mean absolute error of 0.018. Mean utilities predicted by the model covered a similar range to that observed. CONCLUSIONS: The ABC-UI estimates a wide range of utilities from patient-level FXS ABC-C data, allowing estimation of FXS health-related quality of life impact for economic evaluation from an established FXS clinical trial instrument. |
format | Online Article Text |
id | pubmed-4317522 |
institution | National Center for Biotechnology Information |
language | English |
publishDate | 2014 |
publisher | Springer International Publishing |
record_format | MEDLINE/PubMed |
spelling | pubmed-43175222015-02-06 Developing a utility index for the Aberrant Behavior Checklist (ABC-C) for fragile X syndrome Kerr, Cicely Breheny, Katie Lloyd, Andrew Brazier, John Bailey, Donald B. Berry-Kravis, Elizabeth Cohen, Jonathan Petrillo, Jennifer Qual Life Res Article PURPOSE: This study aimed to develop a utility index (the ABC-UI) from the Aberrant Behavior Checklist-Community (ABC-C), for use in quantifying the benefit of emerging treatments for fragile X syndrome (FXS). METHODS: The ABC-C is a proxy-completed assessment of behaviour and is a widely used measure in FXS. A subset of ABC-C items across seven dimensions was identified to include in health state descriptions. This item reduction process was based on item performance, factor analysis and Rasch analysis performed on an observational study dataset, and consultation with five clinical experts and a methodological expert. Dimensions were combined into health states using an orthogonal design and valued using time trade-off (TTO), with lead-time TTO methods used where TTO indicated a state valued as worse than dead. Preference weights were estimated using mean, individual level, ordinary least squares and random-effects maximum likelihood estimation [RE (MLE)] regression models. RESULTS: A representative sample of the UK general public (n = 349; mean age 35.8 years, 58.2 % female) each valued 12 health states. Mean observed values ranged from 0.92 to 0.16 for best to worst health states. The RE (MLE) model performed best based on number of significant coefficients and mean absolute error of 0.018. Mean utilities predicted by the model covered a similar range to that observed. CONCLUSIONS: The ABC-UI estimates a wide range of utilities from patient-level FXS ABC-C data, allowing estimation of FXS health-related quality of life impact for economic evaluation from an established FXS clinical trial instrument. Springer International Publishing 2014-07-26 2015 /pmc/articles/PMC4317522/ /pubmed/25063082 http://dx.doi.org/10.1007/s11136-014-0759-8 Text en © The Author(s) 2014 https://creativecommons.org/licenses/by/4.0/ Open AccessThis article is distributed under the terms of the Creative Commons Attribution License which permits any use, distribution, and reproduction in any medium, provided the original author(s) and the source are credited. |
spellingShingle | Article Kerr, Cicely Breheny, Katie Lloyd, Andrew Brazier, John Bailey, Donald B. Berry-Kravis, Elizabeth Cohen, Jonathan Petrillo, Jennifer Developing a utility index for the Aberrant Behavior Checklist (ABC-C) for fragile X syndrome |
title | Developing a utility index for the Aberrant Behavior Checklist (ABC-C) for fragile X syndrome |
title_full | Developing a utility index for the Aberrant Behavior Checklist (ABC-C) for fragile X syndrome |
title_fullStr | Developing a utility index for the Aberrant Behavior Checklist (ABC-C) for fragile X syndrome |
title_full_unstemmed | Developing a utility index for the Aberrant Behavior Checklist (ABC-C) for fragile X syndrome |
title_short | Developing a utility index for the Aberrant Behavior Checklist (ABC-C) for fragile X syndrome |
title_sort | developing a utility index for the aberrant behavior checklist (abc-c) for fragile x syndrome |
topic | Article |
url | https://www.ncbi.nlm.nih.gov/pmc/articles/PMC4317522/ https://www.ncbi.nlm.nih.gov/pubmed/25063082 http://dx.doi.org/10.1007/s11136-014-0759-8 |
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