Pain and fatigue in adult patients with multiple osteochondromas: The Netherlands.

BACKGROUND: 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. METHODS: 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). RESULTS: 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. CONCLUSIONS: 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.

An exploratory study of discrepancies between objective and subjective measurement of the physical activity level in female patients with chronic fatigue syndrome.

OBJECTIVE: To explore the ability of a self-report activity diary to measure the physical activity level (PAL) in female patients with chronic fatigue syndrome (CFS) and whether illness-related complaints, health-related quality of life domains (HRQOL) or demographic factors are associated with discrepancies between self-reported and objectively measured PAL. METHODS: Sixty-six patients with CFS, recruited from the chronic fatigue clinic of a university hospital, and twenty matched healthy controls wore an accelerometer (Actical) for six consecutive days and registered their activities in an activity diary in the same period. Participants' demographic data was collected and all subjects completed the CFS Symptom List (illness-related complaints) daily and Short-Form-36 (HRQOL domains) during the first and second appointment. RESULTS: A significant, but weak association between the activity diary and Actical was present in patients with CFS (rs = 0.376 and rs = 0.352; p < 0.001) and a moderately strong association in healthy controls (rs = 0.605; and rs = 0.644; p < 0.001) between week and weekend days, respectively. A linear mixed model identified a negative association between age and the discrepancy between the self-reported and objective measure of PA in both patients with CFS and healthy controls. CONCLUSION: The activity diary showed limited ability to register the PAL in female patients with CFS. The discrepancy between measures was not explained by illness-related complaints, HRQOL domains or demographic factors. The activity diary cannot replace objective activity monitoring measured with an accelerometer, but may provide additional information about the perceived activity.

Assessment of activity limitations and participation restrictions with persons with chronic fatigue syndrome: a systematic review.

PURPOSE: To summarize measurement instruments used to evaluate activity limitations and participation restrictions in patients with chronic fatigue syndrome (CFS) and review the psychometric properties of these instruments. METHOD: General information of all included measurement instruments was extracted. The methodological quality was evaluated using the COSMIN checklist. Results of the measurement properties were rated based on the quality criteria of Terwee et al. Finally, overall quality was defined per psychometric property and measurement instrument by use of the quality criteria by Schellingerhout et al. RESULTS: A total of 68 articles were identified of which eight evaluated the psychometric properties of a measurement instrument assessing activity limitations and participation restrictions. One disease-specific and 37 generic measurement instruments were found. Limited evidence was found for the psychometric properties and clinical usability of these instruments. However, the CFS-activities and participation questionnaire (APQ) is a disease-specific instrument with moderate content and construct validity. CONCLUSION: The psychometric properties of the reviewed measurement instruments to evaluate activity limitations and participation restrictions are not sufficiently evaluated. Future research is needed to evaluate the psychometric properties of the measurement instruments, including the other properties of the CFS-APQ. If it is necessary to use a measurement instrument, the CFS-APQ is recommended. IMPLICATIONS FOR REHABILITATION: Chronic fatigue syndrome (CFS). Chronic fatigue syndrome causes activity limitations and participation restrictions in one or more areas of life. Standardized, reliable and valid measurement instruments are necessary to identify these limitations and restrictions. Currently, no measurement instrument is sufficiently evaluated with persons with CFS. If a measurement instrument is needed to identify activity limitations and participation restrictions with persons with CFS, it is recommended to use the CFS-APQ in clinical practice and scientific research.