Amajjar Ihsane, Vergauwen Kuni, Willigenburg Nienke W, Ham S John, Smeets Rob J E M
Department of Orthopaedic Surgery, OLVG, Amsterdam, The Netherlands.
Department of Rehabilitation Medicine, CAPHRI, Maastricht University, Maastricht, The Netherlands.
PLoS One. 2024 Jul 17;19(7):e0305640. doi: 10.1371/journal.pone.0305640. eCollection 2024.
Multiple Osteochondromas (MO) is a rare genetic disorder characterised by the presence of numerous benign bone tumours, known as osteochondromas. Within the spectrum of debilitating symptoms associated with MO, pain is recognized as a major problem. Interestingly, our clinical observations suggest that fatigue is also a significant concern but has merely been touched upon in MO literature. This study aims to (1) assess the level of pain and fatigue in adult patients with MO; (2) compare fatigue in MO to healthy subjects and patients with Rheumatoid Arthritis (RA); (3) identify associated variables for pain and fatigue in patients with MO.
In this cross-sectional study, 353 adult MO patients completed a survey with validated questionnaires on pain, fatigue and psychosocial factors. Pain and fatigue were assessed with the Numeric Rating Scale (NRS), and fatigue was also measured with the Checklist Individual Strength (CIS). Fatigue (CIS) was compared with reference scores of healthy subjects and patients with RA, using a one-sample t-test. Multiple linear regression models for pain and fatigue were developed using a-priori selected independent variables based on a theoretical framework (ICF-model).
Pain was reported by 87.8% (NRS = 3.19±2.6) and fatigue by 90.4% (NRS = 4.1±2.6) of patients with MO. Fatigue scores for MO (CIS = 84.1±15.3) were significantly higher (p<0.001) compared to reference scores of healthy subjects and patients with RA. The multivariable analysis for pain provided a final regression model with six variables (R2 = 0.445, p<0.001) of which fear avoidance beliefs and fatigue had the strongest association. For the fatigue models NRS (R2 = 0.455, p<0.001) and CIS (R2 = 0.233, p<0.001), the strongest associations were found with anxiety and depression respectively.
Pain and fatigue are highly prevalent in patients with MO. Fatigue is significantly higher compared to healthy subjects and patients with RA. Several variables associated with pain and fatigue have been identified that could help improve multidisciplinary treatment plans.
多发性骨软骨瘤（MO）是一种罕见的遗传性疾病，其特征是存在大量被称为骨软骨瘤的良性骨肿瘤。在与MO相关的一系列使人衰弱的症状中，疼痛被认为是一个主要问题。有趣的是，我们的临床观察表明，疲劳也是一个重要问题，但在MO的文献中只是略有提及。本研究旨在：（1）评估成年MO患者的疼痛和疲劳程度；（2）将MO患者的疲劳情况与健康受试者及类风湿关节炎（RA）患者进行比较；（3）确定MO患者疼痛和疲劳的相关变量。
在这项横断面研究中，353名成年MO患者完成了一项包含经过验证的关于疼痛、疲劳和心理社会因素的问卷的调查。使用数字评定量表（NRS）评估疼痛和疲劳，同时也用个体力量检查表（CIS）测量疲劳。使用单样本t检验将疲劳（CIS）与健康受试者和RA患者的参考分数进行比较。基于理论框架（国际功能、残疾和健康分类模型），使用预先选定的自变量建立疼痛和疲劳的多元线性回归模型。
87.8%的MO患者报告有疼痛（NRS = 3.19±2.6），90.4%的患者报告有疲劳（NRS = 4.1±2.6）。与健康受试者和RA患者的参考分数相比，MO患者的疲劳得分（CIS = 84.1±15.3）显著更高（p<0.001）。疼痛的多变量分析得出了一个包含六个变量的最终回归模型（R2 = 0.445，p<0.001），其中恐惧回避信念和疲劳的关联最强。对于疲劳模型NRS（R2 = 0.455，p<0.001）和CIS（R2 = 0.233，p<0.001），分别发现与焦虑和抑郁的关联最强。
疼痛和疲劳在MO患者中非常普遍。与健康受试者和RA患者相比，疲劳程度显著更高。已经确定了一些与疼痛和疲劳相关的变量，这有助于改进多学科治疗方案。
