Abstract
BackgroundTo investigate the association between the structural quality of care and patient satisfaction with care in individuals with disorders/ differences of sex development (DSD).MethodsA multicenter cross-sectional comparative study was conducted in 14 clinics in six European countries. We assessed the level of structural quality of care in each center using a self-constructed measure (Center Score) and the level of participant satisfaction with care using the customer satisfaction questionnaire (CSQ-4) and an adopted version of the Youth Health Care – Satisfaction, Utilization & Needs (YHC-SUN-SF). Data were obtained from individuals with Turner Syndrome (261), Klinefelter Syndrome (173), 46, XX congenital adrenal hyperplasia (190) and XY-DSD (257).ResultsWe found large variations between the scores for structural quality of care both within a diagnostic group and within a country; the overall association between participant satisfaction with the center score was significant.ConclusionsComparative effectiveness research across Europe can lead to more insight on beneficial structures and processes and the overall strategy to care for people with rare diseases in general and specific conditions such as disorders/ differences of sex development. Appreciation of higher levels of structural quality of the centers in this study supports the concept of comprehensive care.Trial registrationGerman Clinical Trials Register: Registration identification number: DRKS00006072, date of registration April 17th, 2014.DRKS00006072 (German Clinical Trials Register).
Highlights
To investigate the association between the structural quality of care and patient satisfaction with care in individuals with disorders/ differences of sex development (DSD)
Instruments and measurements Scoring instrument for structural quality of centers (Center score): We developed the assessment of the extent of multidisciplinary care provided in the 14 centers by probing 17 items per the four diagnostic groups (TS, Klinefelter syndrome (KS), Congenital adrenal hyperplasia (CAH) and XY-DSD)
The recruitment strategies and sizes of the corresponding clinics among the participating centers showed large variations, and the numbers of diagnostic groups were unequally distributed among the centers
Summary
To investigate the association between the structural quality of care and patient satisfaction with care in individuals with disorders/ differences of sex development (DSD). 80% of rare diseases have a genetic background, and 50% manifest in early childhood. In the latter case, the diseases are often associated with high mortality. Because of the low prevalence and geographic distribution of patients and researchers, both the research and organization of care on rare diseases suffer from infrastructural deficits [3]. In Europe, EURORDIS, an umbrella organization of patient associations in rare diseases, has been supporting the work of the European commission since the early 2000s to improve care for people with rare diseases. Joint efforts on the EU level to build Reference Networks for various diagnostic groups (ERN-European Reference Networks for Rare Conditions) have been the latest cornerstone in improving access to high-quality care and establishing standards in diagnostic care, including the development of quality and safety benchmarks [4,5,6]
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