Elsevier

Social Science & Medicine

Volume 69, Issue 6, September 2009, Pages 920-925
Social Science & Medicine

Patient involvement in health research: A contribution to a systematic review on the effectiveness of treatments for degenerative ataxias

https://doi.org/10.1016/j.socscimed.2009.07.005Get rights and content

Abstract

This study aims to incorporate patients’ perspective in the design of a systematic review of scientific literature on the effectiveness of degenerative ataxias (DA) treatments. 53 patients with DA from different regions of Spain were consulted using the Delphi method, with three rounds via e-mail. In the first round, obtained information was on treatments used and relevant self-perceived health problems related to DA. The following two rounds were used to prioritize and achieve a consensus on the answers.

The participation rate was 100% for all rounds. The most relevant self-perceived health problems were limitations in activities of daily living (ADL), visual and auditory problems and diminished self-esteem. The bibliographic search for the systematic review was enriched by these patient contributions. No study offered information on treatment effectiveness for the following problems prioritized by patients: ADL, social relationships, disease acceptance and quality of life.

Thus some of the self-perceived DA-related health problems identified by the patients have never been investigated and should be considered to improve future research projects which should be adapted to meet patients’ needs. Effective participation of patients can extend the value of systematic reviews to ensure they respond to both clinicians’ information needs and patients’ expectations.

Section snippets

Presentation of the case

Degenerative ataxias (DA) are an heterogeneous group of genetically-based rare diseases with diverse patterns of inheritance and with many genes and genetic loci implicated (Cuadrado-Gamarra, Sevillano-García, & de Pedro-Cuesta, 1999). Overall, their prevalence in Spain is 20.2 cases/100.000 population (López-Vega, Calleja, Combarros, Polo, & Berciano, 1988). DA are clinically characterized by progressive ataxia resulting from degeneration of the cerebellum, brain stem and spino-cerebellar

Methodology

A consultation was conducted between January and May 2007 with a sample of DA patients recruited from different regions of Spain. This study was approved by the Ethics Committee of the Canary Islands since its coordination was carried out by the Planning and Evaluation Unit of the Canary Islands Health Service. As part of the recruitment process, an attempt was made to enroll as many patients as possible, and was managed by the regional patient associations in Spain (Hispataxia and the Spanish

Results

A total of 53 patients were invited to participate. The participation rate was 100% (53/53) for the three rounds of the Delphi method. Patients’ general characteristics are shown in Table 1: their mean age was 39.2 ± 2.0 years, 60.4% (32/53) were women and most of them (67.9%) suffered Friedreich's ataxia. Mean time of disease progression was 15.0 ± 1.6 years and 41.5% (22/53) required some help to perform activities of daily living (ADL). Participants’ place of residence was distributed across 12

Analysis and discussion

The case example provides a learning experience concerning the feasibility and efficiency of patient involvement, by means of the Delphi method, in designing a systematic review of the scientific literature. The participatory process was found to be particularly effective in enhancing the design and conduct of our research. According to the James Lind Alliance proposals, lived experiences of individual patients with their illnesses extended the value of the systematic review ensuring that it

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    This work was supported by the Ministry of Health and Consumption of Spain in cooperation with the “Fundación Canaria de Investigación y Salud” (FUNCIS), in the framework of the Quality Plan for the National Health Service.

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