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

被引:1
|
作者
Boarini, Manila [1 ]
Tremosini, Morena [1 ]
Di Cecco, Alessia [1 ]
Gnoli, Maria [1 ]
Brizola, Evelise [1 ]
Mordenti, Marina [1 ]
Pedrini, Elena [1 ]
Locatelli, Manuela [1 ]
Lanza, Marcella [1 ]
Antonioli, Diego [2 ]
Gallone, Giovanni [2 ]
Rocca, Gino [2 ]
Staals, Eric L. [3 ]
Trisolino, Giovanni [2 ]
Sangiorgi, Luca [1 ]
机构
[1] IRCCS Ist Ortoped Rizzoli, Dept Rare Skeletal Disorders, Via Barbiano 1-10, I-40136 Bologna, Italy
[2] IRCCS Ist Ortoped Rizzoli, Unit Pediat Orthoped & Traumatol, Bologna, Italy
[3] IRCCS Ist Ortoped Rizzoli, Orthopaed & Traumatol Clin Prevalently Oncol 3, Bologna, Italy
关键词
HRQoL; Skeletal dysplasia; Rare disease; Multiple Osteochondromas; PROs; Healthcare pathway; RARE; EXOSTOSES; CHILDREN; DISEASES; EQ-5D;
D O I
10.1007/s11136-024-03604-4
中图分类号
R19 [保健组织与事业(卫生事业管理)];
学科分类号
摘要
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.
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收藏
页码:1323 / 1334
页数:12
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