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      Health-related quality of life and associated risk factors in patients with Multiple Osteochondromas: a cross-sectional study

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          Abstract

          Purpose

          To evaluate the health-related quality of life and associated risk factors for Multiple Osteochondromas patients.

          Methods

          A cross-sectional, observational study was conducted from May to December 2022 during the routine visit to the referral center for rare skeletal disorders. All patients with Multiple Osteochondromas aged ≥ 3 years were included. EuroQol 5-dimension questionnaires, and demographic, clinical, and surgical history data were collected. Descriptive statistics, Fisher’s exact test, One-sample t-test, Spearman’s correlation, and multiple linear and logistic regression were performed to analyze the data. Results are reported following STROBE guidelines.

          Results

          A total of 128 patients were included in the study, with a mean age of 14 [SD, 10] years. The mean EQ-5D Index Value was 0.863 [SD, 0.200] and the EQ-VAS was 84 [SD, 19] with a positive correlation between two scores [ r = 0.541, p < 0.001]. Patients frequently referred problems in pain/discomfort [78.8%], anxiety/depression [50%], and usual activities [38.8%] dimensions. Increasing age was the common risk factor for health-related quality of life [ p < 0.000], as well as Index Value and VAS scores were significantly lower in surgical patients [ p = 0.001 and p < 0.001, respectively].

          Conclusion

          Increasing age and surgical procedures were found highly associated with reduced health-related quality of life in Multiple Osteochondromas patients. Our findings provide relevant information to support the establishment of patient-centered healthcare pathways and pave the way for further research into medical and non-medical therapeutic strategies for these patients.

          Supplementary Information

          The online version contains supplementary material available at 10.1007/s11136-024-03604-4.

          Related collections

          Most cited references41

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          The Strengthening the Reporting of Observational Studies in Epidemiology (STROBE) Statement: guidelines for reporting observational studies.

          Much biomedical research is observational. The reporting of such research is often inadequate, which hampers the assessment of its strengths and weaknesses and of a study's generalisability. The Strengthening the Reporting of Observational Studies in Epidemiology (STROBE) Initiative developed recommendations on what should be included in an accurate and complete report of an observational study. We defined the scope of the recommendations to cover three main study designs: cohort, case-control, and cross-sectional studies. We convened a 2-day workshop in September 2004, with methodologists, researchers, and journal editors to draft a checklist of items. This list was subsequently revised during several meetings of the coordinating group and in e-mail discussions with the larger group of STROBE contributors, taking into account empirical evidence and methodological considerations. The workshop and the subsequent iterative process of consultation and revision resulted in a checklist of 22 items (the STROBE Statement) that relate to the title, abstract, introduction, methods, results, and discussion sections of articles. 18 items are common to all three study designs and four are specific for cohort, case-control, or cross-sectional studies. A detailed Explanation and Elaboration document is published separately and is freely available on the Web sites of PLoS Medicine, Annals of Internal Medicine, and Epidemiology. We hope that the STROBE Statement will contribute to improving the quality of reporting of observational studies.
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            EuroQol - a new facility for the measurement of health-related quality of life

            (1990)
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              EQ-5D and the EuroQol Group: Past, Present and Future

              Over the period 1987–1991 an inter-disciplinary five-country group developed the EuroQol instrument, a five-dimensional three-level generic measure subsequently termed the ‘EQ-5D’. It was designed to measure and value health status. The salient features of its development and its consolidation and expansion are discussed. Initial expansion came, in particular, in the form of new language versions. Their development raised translation and semantic issues, experience with which helped feed into the design of two further instruments, the EQ-5D-5L and the youth version EQ-5D-Y. The expanded usage across clinical programmes, disease and condition areas, population surveys, patient-reported outcomes, and value sets is outlined. Valuation has been of continued relevance for the Group as this has allowed its instruments to be utilised as part of the economic appraisal of health programmes and their incorporation into health technology assessments. The future of the Group is considered in the context of: (1) its scientific strategy, (2) changes in the external environment affecting the demand for EQ-5D, and (3) a variety of issues it is facing in the context of the design of the instrument, its use in health technology assessment, and potential new uses for EQ-5D outside of clinical trials and technology appraisal. Electronic supplementary material The online version of this article (doi:10.1007/s40258-017-0310-5) contains supplementary material, which is available to authorized users.
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                Author and article information

                Contributors
                manuela.locatelli@ior.it
                Journal
                Qual Life Res
                Qual Life Res
                Quality of Life Research
                Springer International Publishing (Cham )
                0962-9343
                1573-2649
                8 March 2024
                8 March 2024
                2024
                : 33
                : 5
                : 1323-1334
                Affiliations
                [1 ]Department of Rare Skeletal Disorders, IRCCS Istituto Ortopedico Rizzoli, ( https://ror.org/02ycyys66) Via di Barbiano 1/10, 40136 Bologna, Italy
                [2 ]Unit of Pediatric Orthopedics and Traumatology, IRCCS Istituto Ortopedico Rizzoli, ( https://ror.org/02ycyys66) Bologna, Italy
                [3 ]3rd Orthopaedic and Traumatologic Clinic Prevalently Oncologic, IRCCS Istituto Ortopedico Rizzoli, ( https://ror.org/02ycyys66) Bologna, Italy
                Author information
                http://orcid.org/0000-0002-7594-8774
                Article
                3604
                10.1007/s11136-024-03604-4
                11045590
                38457053
                dd6e926b-c9f5-4f67-bbbd-6e42f8f2dfd6
                © The Author(s) 2024

                Open Access This article is licensed under a Creative Commons Attribution 4.0 International License, which permits use, sharing, adaptation, distribution and reproduction in any medium or format, as long as you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons licence, and indicate if changes were made. The images or other third party material in this article are included in the article's Creative Commons licence, unless indicated otherwise in a credit line to the material. If material is not included in the article's Creative Commons licence and your intended use is not permitted by statutory regulation or exceeds the permitted use, you will need to obtain permission directly from the copyright holder. To view a copy of this licence, visit http://creativecommons.org/licenses/by/4.0/.

                History
                : 8 January 2024
                Categories
                Article
                Custom metadata
                © Springer Nature Switzerland AG 2024

                Public health
                hrqol,skeletal dysplasia,rare disease,multiple osteochondromas,pros,healthcare pathway
                Public health
                hrqol, skeletal dysplasia, rare disease, multiple osteochondromas, pros, healthcare pathway

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