A cross-sectional study was carried out between June and December 2018. This study was approved by Ethics Committee of Clinical research at the San Juan of Alicante University General Hospital (18/303 on 5 February 2018).
A total of 480 subjects were randomly invited to participate considering an error sampling of 5% and lost expected responses of 20%.
The Spanish Rare Diseases Federation [Federación Española de Enfermedades Raras] (FEDER) collaborated in this study sending a set of his affiliates invitations to reply. They received a mail explaining the aim and the procedure for replying. This message included a link directing to the online questionnaire to be self-administered by subjects. Voluntary response and anonymized treatment were emphasized. A remainder was done to promote the response.
A modified version of the IEXPAC instrument[i] was applied. This instrument includes 11 items, plus an additional item for patients recently hospitalized. IEXPAC explores characteristics and content of interactions between patients and professionals designed to improve outcomes, the ability of individuals to cope with their diseases, manage their own care and improve their well-being, based on interventions mediated by healthcare professionals, and experience of use the new forms of patient interaction with the healthcare system. For each item, patients responded on a 5-point Likert scale (range from never to always).
Four questions of this instrument were modified to adjust to the rare diseases context (reinforcing the measure of patient-centered-care, support for engagement, and integrate care) and four specific items were added to explore the experience with care in case of emergency care, homecare, being receiving support from social services and information received about diagnosis. Adaptation was done by a selection of specialist working on rare diseases and the research team applying consensus technique. Responses were transformed in scores following criteria applied by Orozco et al[ii] using the IEXPAC considering always (score 10), mostly (7.5), sometimes (5), seldom (2.5) or never (0). The overall score was calculated as the sum of individual scores for the 11 common items divided by 11 between 0 (worst experience) and 10 (best experience). This procedure allowed to compare data with the national study conducted by Orozco et al including 1618 chronic patients suffering diabetes mellitus, human immunodeficiency virus infection, inflammatory bowel disease, or rheumatic diseases. Additionally, percentages of subjects who answered with the always option was calculated. These percentages allows identification of range in which improvement is needed and compare data with the Spanish studies conducted asking chronic patients9 and caregivers of chronic patients (relatives suffering Alzheimer, mental illness, and Chronic Obstructive Pulmonary Disease)[iii] using the same scale.
This version of the IEXPAC was assessed by a set of patients representatives and their wording changes suggested were introduced to assure content validity and legibility. Additionally, metric properties of this instrument was assessed to assure the validity (applying exploratory factorial analysis) and reliability (using Cronbach’s Alpha and Rho coefficient) of the measurement conducted in this study. The reliability measures using Cronbach’s Alpha of this version of the IEXPAC instrument for rare diseases was 0.87 and coefficient Rho was 0.86 (for the 11 elements applied in all cases). Items converged in two factors explaining 59.4 of total variance (Appendix I).
Only subjects replaying all items were included. Descriptive information (mean, standard deviation, SD) were displayed for each item, and the overall score. The distribution of responses to individual items was also displayed, as is the percent of “always” responses to each item. Frequencies or percent for qualitative variables were also used. Student’s t test or analysis of variance (ANOVA) were used to compare continuous variables. Time to diagnosis was grouped into categories (0 to 3 years, 4 to 6, 7 to 15, and 16 or more). Chi-Square was used to compare qualitative and continuous variables. Given the overall descriptive nature of the results, no multiplicity adjustments were made. Lineal regression was calculated to establish the relation of age, number of drugs they are taking, number of times they have been admitted to the hospital in the last year, length of time in years that they have been diagnosed with the main illness (overall score on the PREM).
[i] Mira, J J, Nuño-Solinís, R, Guilabert-Mora, M, Solas-Gaspar, O, Fernández-Cano, P, González-Mestre, M A, Contel, J C and del Río-Cámara, M. Development and Validation of an Instrument for Assessing Patient Experience of Chronic Illness Care. Int J Integr Care. 2016; 16:13.
[ii] Orozco-Beltrán, D., de Toro, J., Galindo, M.J. et al. Patient. Healthcare Experience and their Relationship with Demographic, Disease and Healthcare‑Related Variables: A Cross‑Sectional Survey of Patients with Chronic Diseases Using the IEXPAC Scale. Patient. 2018. https://doi.org/10.1007/s40271-018-0345-1.
[iii] Guilabert M, Amil P, González-Mestre A, Gil-Sánchez E, Vila A, Carles Contel J, Ansotegui JC, Solas O, Bacigalupe MT, Fernández-Cano P, Arteagoita M, Mira JJ. The Measure of the Family Caregivers’ Experience. Int J Environ Res Public Health. 2018; 15: 2040.