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Health and Quality of Life Outcomes
Published in: Health and Quality of Life Outcomes 1/2012

Open Access 01-12-2012 | Study protocol

Development of a disease-specific quality of life questionnaire for adult patients with hereditary angioedema due to C1 inhibitor deficiency (HAE-QoL): Spanish multi-centre research project

Authors: Nieves Prior, Eduardo Remor, Carmen Gómez-Traseira, Concepción López-Serrano, Rosario Cabañas, Javier Contreras, Ángel Campos, Victoria Cardona, Stefan Cimbollek, Teresa González-Quevedo, Mar Guilarte, Dolores Hernández Fernández de Rojas, Carmen Marcos, María Rubio, Miguel Ángel Tejedor-Alonso, Teresa Caballero

Published in: Health and Quality of Life Outcomes | Issue 1/2012

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Abstract

Background

There is a need for a disease-specific instrument for assessing health-related quality of life in adults with hereditary angioedema due to C1 inhibitor deficiency, a rare, disabling and life-threatening disease. In this paper we report the protocol for the development and validation of a specific questionnaire, with details on the results of the process of item generation, domain selection, and the expert and patient rating phase.

Methods/Design

Semi-structured interviews were completed by 45 patients with hereditary angioedema and 8 experts from 8 regions in Spain. A qualitative content analysis of the responses was carried out. Issues raised by respondents were grouped into categories. Content analysis identified 240 different responses, which were grouped into 10 conceptual domains. Sixty- four items were generated. A total of 8 experts and 16 patients assessed the items for clarity, relevance to the disease, and correct dimension assignment. The preliminary version of the specific health-related quality of life questionnaire for hereditary angioedema (HAE-QoL v 1.1) contained 44 items grouped into 9 domains.

Discussion

To the best of our knowledge, this is the first multi-centre research project that aims to develop a specific health-related quality of life questionnaire for adult patients with hereditary angioedema due to C1 inhibitor deficiency. A preliminary version of the specific HAE-QoL questionnaire was obtained. The qualitative analysis of interviews together with the expert and patient rating phase helped to ensure content validity. A pilot study will be performed to assess the psychometric properties of the questionnaire and to decide on the final version.
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Metadata
Title
Development of a disease-specific quality of life questionnaire for adult patients with hereditary angioedema due to C1 inhibitor deficiency (HAE-QoL): Spanish multi-centre research project
Authors
Nieves Prior
Eduardo Remor
Carmen Gómez-Traseira
Concepción López-Serrano
Rosario Cabañas
Javier Contreras
Ángel Campos
Victoria Cardona
Stefan Cimbollek
Teresa González-Quevedo
Mar Guilarte
Dolores Hernández Fernández de Rojas
Carmen Marcos
María Rubio
Miguel Ángel Tejedor-Alonso
Teresa Caballero
Publication date
01-12-2012
Publisher
BioMed Central
Published in
Health and Quality of Life Outcomes / Issue 1/2012
Electronic ISSN: 1477-7525
DOI
https://doi.org/10.1186/1477-7525-10-82

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