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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

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...

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Autores principales: Prior, Nieves, Remor, Eduardo, Gómez-Traseira, Carmen, López-Serrano, Concepción, Cabañas, Rosario, Contreras, Javier, Campos, Ángel, Cardona, Victoria, Cimbollek, Stefan, González-Quevedo, Teresa, Guilarte, Mar, de Rojas, Dolores Hernández Fernández, Marcos, Carmen, Rubio, María, Tejedor-Alonso, Miguel Ángel, Caballero, Teresa
Formato: Online Artículo Texto
Lenguaje:English
Publicado: BioMed Central 2012
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC3489868/
https://www.ncbi.nlm.nih.gov/pubmed/22817696
http://dx.doi.org/10.1186/1477-7525-10-82
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author Prior, Nieves
Remor, Eduardo
Gómez-Traseira, Carmen
López-Serrano, Concepción
Cabañas, Rosario
Contreras, Javier
Campos, Ángel
Cardona, Victoria
Cimbollek, Stefan
González-Quevedo, Teresa
Guilarte, Mar
de Rojas, Dolores Hernández Fernández
Marcos, Carmen
Rubio, María
Tejedor-Alonso, Miguel Ángel
Caballero, Teresa
author_facet Prior, Nieves
Remor, Eduardo
Gómez-Traseira, Carmen
López-Serrano, Concepción
Cabañas, Rosario
Contreras, Javier
Campos, Ángel
Cardona, Victoria
Cimbollek, Stefan
González-Quevedo, Teresa
Guilarte, Mar
de Rojas, Dolores Hernández Fernández
Marcos, Carmen
Rubio, María
Tejedor-Alonso, Miguel Ángel
Caballero, Teresa
author_sort Prior, Nieves
collection PubMed
description 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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spelling pubmed-34898682012-11-06 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 Prior, Nieves Remor, Eduardo Gómez-Traseira, Carmen López-Serrano, Concepción Cabañas, Rosario Contreras, Javier Campos, Ángel Cardona, Victoria Cimbollek, Stefan González-Quevedo, Teresa Guilarte, Mar de Rojas, Dolores Hernández Fernández Marcos, Carmen Rubio, María Tejedor-Alonso, Miguel Ángel Caballero, Teresa Health Qual Life Outcomes Study Protocol 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. BioMed Central 2012-07-20 /pmc/articles/PMC3489868/ /pubmed/22817696 http://dx.doi.org/10.1186/1477-7525-10-82 Text en Copyright ©2012 Prior et al.; licensee BioMed Central Ltd. http://creativecommons.org/licenses/by/2.0 This is an Open Access article distributed under the terms of the Creative Commons Attribution License (http://creativecommons.org/licenses/by/2.0), which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.
spellingShingle Study Protocol
Prior, Nieves
Remor, Eduardo
Gómez-Traseira, Carmen
López-Serrano, Concepción
Cabañas, Rosario
Contreras, Javier
Campos, Ángel
Cardona, Victoria
Cimbollek, Stefan
González-Quevedo, Teresa
Guilarte, Mar
de Rojas, Dolores Hernández Fernández
Marcos, Carmen
Rubio, María
Tejedor-Alonso, Miguel Ángel
Caballero, Teresa
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
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
title_full 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
title_fullStr 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
title_full_unstemmed 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
title_short 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
title_sort 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
topic Study Protocol
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC3489868/
https://www.ncbi.nlm.nih.gov/pubmed/22817696
http://dx.doi.org/10.1186/1477-7525-10-82
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