Department of Rare Skeletal Disorders, IRCCS Istituto Ortopedico Rizzoli, Bologna, Italy.
Associazione Conto Alla Rovescia-ACAR Aps, Roma, Italy.
Sci Rep. 2024 Nov 1;14(1):26339. doi: 10.1038/s41598-024-77934-1.
Individuals with rare skeletal disorders like Multiple Osteochondromas and Ollier Disease often experience physical and psychological burdens. Adventure therapy, with activities like sailing in outdoor settings, promotes personal growth and psychological well-being, potentially improving health-related quality of life (HRQoL). This study aimed to evaluate the impact of a sailing program on health-related quality of life and participant satisfaction in individuals with Multiple Osteochondromas and Ollier Disease. A quasi-experimental one-group pre-post design was employed, with HRQoL assessed using the EQ-5D® instrument and participant satisfaction measured via a feedback survey. Data were collected before and after the five-day sailing program conducted in the Mediterranean Sea in 2022 and 2023, involving participants diagnosed with Multiple Osteochondromas and Ollier Disease. Statistical analyses were performed using the Wilcoxon signed-rank test and McNemar's test for paired data. A significance level of p < 0.05 and p < 0.10 was considered. A total of 25 participants, predominantly male (52%), with a median age of 16 years (ranking from 11 to 31), were included in the study. The sailing program had mixed impact on HRQoL. Specifically, individuals who were female (p = 0.03), aged 16 and older (p = 0.04), with higher educational attainment (p = 0.10) or stronger self-management (p = 0.09), resilience (p = 0.01) and self-engagement (p = 0.09) skills experienced enhanced HRQoL. Conversely, other participants exhibited an increase in self-care difficulties (p = 0.02) and a trend towards worsening pain/discomfort (p = 0.38). Overall satisfaction with the program was high, with 90% of participants expressing satisfaction.This is the first study which examined HRQoL in Multiple Osteochondromas and Ollier Disease patients within an outdoor adventure therapy setting. Findings suggest that adventure therapy, integrated into healthcare strategies, may offer a valuable complement to conventional treatments for rare skeletal disorders. Future research, including randomized controlled trials, are necessary to confirm these results and develop robust interventions for improving the well-being in this population.
患有多发性骨软骨瘤和奥利尔病等罕见骨骼疾病的个体常常承受身体和心理的双重负担。户外航海等冒险疗法可以促进个人成长和心理健康,从而提高健康相关生活质量(HRQoL)。本研究旨在评估航海项目对多发性骨软骨瘤和奥利尔病患者的 HRQoL 和参与者满意度的影响。采用准实验性单组前后设计,使用 EQ-5D®工具评估 HRQoL,通过反馈调查评估参与者满意度。数据收集于 2022 年和 2023 年在地中海进行的为期五天的航海项目前后,参与者均被诊断为多发性骨软骨瘤和奥利尔病。使用 Wilcoxon 符号秩检验和 McNemar 检验对配对数据进行统计分析。p 值<0.05 和 p 值<0.10 被认为具有统计学意义。共有 25 名参与者(52%为男性),年龄中位数为 16 岁(范围 11-31 岁),纳入本研究。航海项目对 HRQoL 的影响混合。具体而言,女性(p=0.03)、年龄 16 岁及以上(p=0.04)、教育程度较高(p=0.10)或自我管理能力(p=0.09)、适应力(p=0.01)和自我参与度(p=0.09)较强的个体,HRQoL 得到改善。相反,其他参与者自我护理困难增加(p=0.02),疼痛/不适恶化趋势(p=0.38)。对项目的总体满意度较高,90%的参与者表示满意。这是第一项在户外冒险疗法环境中研究多发性骨软骨瘤和奥利尔病患者 HRQoL 的研究。研究结果表明,冒险疗法作为医疗策略的一部分,可能为罕见骨骼疾病的常规治疗提供有价值的补充。未来需要进行随机对照试验等研究,以确认这些结果并为改善该人群的幸福感制定稳健的干预措施。