Patient-reported outcomes in chronic hepatitis delta: An exploratory analysis of the Phase III MYR301 trial of bulevirtide.

BACKGROUND & AIMS: Once-daily treatment of chronic hepatitis delta (CHD) with bulevirtide is well tolerated and associated with significant reductions in HDV RNA in the blood and in biochemical liver disease activity. This study explored the effects of 48-week bulevirtide treatment on health-related quality of life (HRQoL) in patients with CHD. METHODS: In an open-label, randomised, Phase 3 trial, 150 patients with CHD and compensated liver disease were stratified by liver cirrhosis status and randomised 1:1:1 to no treatment (control), bulevirtide 2 mg/day, or bulevirtide 10 mg/day for 48 weeks. HRQoL was evaluated by the following patient-reported outcome (PRO) instruments at baseline, 24 weeks, and 48 weeks: EQ-5D-3L, Hepatitis Quality of Life Questionnaire (HQLQ), and Fatigue Severity Scale (FSS). RESULTS: Patient characteristics and HRQoL scores were balanced at baseline between the treatment (2 mg, n = 49; 10 mg, n = 50) and control (n = 51) groups. Patients receiving 2-mg bulevirtide reported significant improvements compared with controls on the HQLQ domains of role physical, hepatitis-specific limitations, and hepatitis-specific health distress. Numerically higher scores for general health, hepatitis-specific limitations, and hepatitis-specific health distress domains were reported by patients with cirrhosis who received bulevirtide vs control. FSS scores remained stable across treatment groups throughout. At week 48, patients in the 2-mg group showed greater mean improvement from baseline in health status compared with controls on the EQ-5D-3L visual analogue scale. CONCLUSION: PROs indicate that 48-week treatment with bulevirtide monotherapy may improve aspects of HRQoL in patients with CHD. IMPACT AND IMPLICATIONS: Bulevirtide 2 mg is the only approved treatment for patients with chronic hepatitis delta (CHD) in the EU. Patients with CHD have worse quality of life scores than those with chronic hepatitis B. Bulevirtide treatment for 48 weeks reduced HDV RNA and alanine aminotransferase levels and was well tolerated among patients with CHD. For the first time, this study shows that patients who received bulevirtide therapy for 48 weeks reported improvements in physical and hepatitis-related quality of life domains compared to those who did not receive therapy (control group). CLINICAL TRIAL REGISTRATION: ClinicalTrials.gov Identifier, NCT03852719.

Systematic review of fatigue severity in ME/CFS patients: insights from randomized controlled trials.

BACKGROUND: Myalgic Encephalomyelitis/Chronic Fatigue Syndrome (ME/CFS) is a debilitating illness medically unexplained, affecting approximately 1% of the global population. Due to the subjective complaint, assessing the exact severity of fatigue is a clinical challenge, thus, this study aimed to produce comprehensive features of fatigue severity in ME/CFS patients. METHODS: We systematically extracted the data for fatigue levels of participants in randomized controlled trials (RCTs) targeting ME/CFS from PubMed, Cochrane Library, Web of Science, and CINAHL throughout January 31, 2024. We normalized each different measurement to a maximum 100-point scale and performed a meta-analysis to assess fatigue severity by subgroups of age, fatigue domain, intervention, case definition, and assessment tool, respectively. RESULTS: Among the total of 497 relevant studies, 60 RCTs finally met our eligibility criteria, which included a total of 7088 ME/CFS patients (males 1815, females 4532, and no information 741). The fatigue severity of the whole 7,088 patients was 77.9 (95% CI 74.7-81.0), showing 77.7 (95% CI 74.3-81.0) from 54 RCTs in 6,706 adults and 79.6 (95% CI 69.8-89.3) from 6 RCTs in 382 adolescents. Regarding the domain of fatigue, 'cognitive' (74.2, 95% CI 65.4-83.0) and 'physical' fatigue (74.3, 95% CI 68.3-80.3) were a little higher than 'mental' fatigue (70.1, 95% CI 64.4-75.8). The ME/CFS participants for non-pharmacological intervention (79.1, 95% CI 75.2-83.0) showed a higher fatigue level than those for pharmacological intervention (75.5, 95% CI 70.0-81.0). The fatigue levels of ME/CFS patients varied according to diagnostic criteria and assessment tools adapted in RCTs, likely from 54.2 by ICC (International Consensus Criteria) to 83.6 by Canadian criteria and 54.2 by MFS (Mental Fatigue Scale) to 88.6 by CIS (Checklist Individual Strength), respectively. CONCLUSIONS: This systematic review firstly produced comprehensive features of fatigue severity in patients with ME/CFS. Our data will provide insights for clinicians in diagnosis, therapeutic assessment, and patient management, as well as for researchers in fatigue-related investigations.

Rasch analysis of the modified Fatigue Severity Scale in neuromuscular disorders and comparison between sex, age and diagnoses.

INTRODUCTION/AIMS: Fatigue is a common and debilitating symptom encountered in the neuromuscular clinic. The 7-item Fatigue Severity Scale (FSS-7) is a Rasch-modified assessment validated in inflammatory neuropathies but not across a typical neuromuscular patient population. The aim of this study was to validate this measure in neuromuscular disorders and to compare between patient sex, age and diagnoses. METHODS: The modified FSS-7 was mailed to patients recruited from a specialist neuromuscular clinic at the Walton Centre. Responses were subjected to Rasch analysis and descriptive statistics were performed on the Rasch converted data. RESULTS: The mFSS-7 met the Rasch model expectations with an overall Chi-square probability of 0.4918, a strict unidimensional scale free from differential item functioning (DIF) that satisfied the model with substantial test-retest reliability using Lin's concordance correlation coefficient 0.71 (95% CI 0.63-0.77). A 15.7% ceiling effect was observed in this patient cohort. Post hoc analysis did not show any significant difference in fatigue between sex, age or neuromuscular diagnoses. DISCUSSION: The self-completed Rasch mFSS-7 showed acceptable test-retest reliability across patients with varied disorders under follow-up in a specialist neuromuscular clinic. The ceiling effect constrains its use for those with the most severe fatigue. Future considerations could include assessment of the benefits of clinical interventions, particularly multidisciplinary team input or dedicated fatigue clinics.

Different subtypes of chronic fatigue in childhood cancer survivors: A DCCSS LATER study.

INTRODUCTION: The aim of the current study was to investigate whether subtypes of chronic fatigue (CF) can be identified in childhood cancer survivors (CCS), and if so, to determine the characteristics of participants with a specific subtype. METHODS: Participants were included from the nationwide DCCSS LATER cohort. The Checklist Individual Strength (CIS) was completed to assess fatigue. Participants with CF (scored ≥35 on the fatigue severity subscale and indicated to suffer from fatigue for ≥6 months) were divided into subgroups using two-step cluster analysis based on the CIS concentration, motivation, and physical activity subscales. Differences between groups on demographics, psychosocial, lifestyle, and treatment-related variables were determined using ANOVA and chi-square analyses (univariable) and multinomial regression analysis (multivariable). RESULTS: A total of 1910 participants participated in the current study (n = 450 with CF; n = 1460 without CF). Three CF subgroups were identified: Subgroup 1 (n = 133, 29% of participants) had CF with problems in physical activity; Subgroup 2 (n = 111, 25% of participants) had CF with difficulty concentrating; and Subgroup 3 (n = 206, 46% of participants) had multi-dimensional CF. Compared to Subgroup 1, Subgroup 2 more often reported sleep problems, limitations in social functioning, and less often have more than two comorbidities. Subgroup 3 more often reported depression, sleep problems, a lower self-esteem, and limitations in social functioning and a lower educational level compared to Subgroup 1. CONCLUSION: Different subgroups of CCS with CF can be identified based on fatigue dimensions physical activity, motivation and concentration. Results suggest that different intervention strategies, tailored for each subgroup, might be beneficial.

Minimal clinically important differenceof fatigue severity scale in patients with chronic stroke.

BACKGROUND: One of the most prevalent symptoms of stroke is fatigue. Fatigue severity scale is the most often used tool for evaluating fatigue in stroke patients, its minimal clinically important difference threshold has not been determined. This study aimed to identify the minimal clinically important difference of fatigue severity scale in stroke patients. METHODS: All study participants were examined using fatigue severity scale and multidimensional fatigue symptom inventory-short form before and after the intervention. The 6-week intervention combined graded activity training and pacing therapy employed to reduce fatigue severity. Participants reported changes in their fatigue severity after the intervention with the global rating of change and visual analog scale. The minimal clinically important difference of the fatigue severity scale calculated using both anchor- and distribution-based methods. RESULTS: A total of 117 stroke patients were included in the study. Using multidimensional fatigue symptom inventory-short form, global rating of change, and visual analog scale as an anchor, the minimal clinically important difference of fatigue severity scale was obtained at 3.5, 4.5, and 4.5, respectively. The minimal clinically important difference for fatigue severity scale varied from 4.28 to 12.90 using the distribution-based method, with SEM = 4.28 displaying the best sensitivity and specificity for use as minimal clinically important difference. CONCLUSIONS: The minimal clinically important difference value for the fatigue severity scale was estimated at 3.5_12.90 using anchor-based and distribution-based methods. The study's results can be utilized to understand the effectiveness of fatigue interventions in stroke patients in clinical and research settings.

The association of personality traits with poststroke fatigue in daily life: An exploratory experience sampling method and cross-sectional study.

Fatigue is a frequently occurring and persistent symptom after stroke. Many biological, psychosocial, and behavioural factors are associated with poststroke fatigue, but research into associations with personality traits is relatively sparse. In this study, we explored whether personality traits were related to poststroke fatigue measured with conventional fatigue questionnaires as well as experience sampling methodology (ESM). Twenty-four individuals with stroke completed 10 daily questionnaires about momentary (here-and-now) fatigue for six consecutive days using the mHealth ESM application PsyMateTM. Further, they completed questionnaires assessing personality (NEO-FFI and LOR-T) and fatigue (FSS). Results showed that higher extraversion (ß = -.44, SE = .12, p = .001; 95% CI = -.67-.19) and optimism (ß = -.18, SE = .06, p = .007; 95% CI = -.30-.05) were associated with lower momentary fatigue. No association was found between neuroticism and momentary fatigue, but higher neuroticism (r = 0.531, p = .008, 95% CI = .160-.759; r = .574, p = .003, 95% CI = .245-.767) was associated with higher scores on the retrospective FSS scales. We conclude that personality traits differentially influence poststroke fatigue, but this also depends on the way fatigue is measured (with retrospective or with momentary measures). When functional gains are not in line with expected progress during the rehabilitation treatment of fatigue, it may be appropriate to take into account how person characteristics are related to momentary fatigue.

Automatic Assessment of the 2-Minute Walk Distance for Remote Monitoring of People with Multiple Sclerosis.

The aim of this study was to investigate the feasibility of automatically assessing the 2-Minute Walk Distance (2MWD) for monitoring people with multiple sclerosis (pwMS). For 154 pwMS, MS-related clinical outcomes as well as the 2MWDs as evaluated by clinicians and derived from accelerometer data were collected from a total of 323 periodic clinical visits. Accelerometer data from a wearable device during 100 home-based 2MWD assessments were also acquired. The error in estimating the 2MWD was validated for walk tests performed at hospital, and then the correlation (r) between clinical outcomes and home-based 2MWD assessments was evaluated. Robust performance in estimating the 2MWD from the wearable device was obtained, yielding an error of less than 10% in about two-thirds of clinical visits. Correlation analysis showed that there is a strong association between the actual and the estimated 2MWD obtained either at hospital (r = 0.71) or at home (r = 0.58). Furthermore, the estimated 2MWD exhibits moderate-to-strong correlation with various MS-related clinical outcomes, including disability and fatigue severity scores. Automatic assessment of the 2MWD in pwMS is feasible with the usage of a consumer-friendly wearable device in clinical and non-clinical settings. Wearable devices can also enhance the assessment of MS-related clinical outcomes.

Comparison of assessment scores for fatigue between multidimensional fatigue inventory (MFI-K) and modified chalder fatigue scale (mKCFQ).

BACKGROUND: Because of the absence of biological parameters for fatigue, appropriate instruments for assessing the degree of fatigue are important in the diagnosis and management of people complaining of fatigue-like symptoms. This study statistically analyzed the fatigue scores from two typical questionnaire-based instruments: the Korean version of the Multidimensional Fatigue Inventory (MFI-K) and the modified Chalder Fatigue Scale (mKCFQ). METHODS: Seventy participants (males n = 40, females n = 30, median age 48 years old, range of 25-67) were grouped into three groups ('mild' = 20, 'moderate' = 42, and 'severe' = 8) according to self-reported fatigue levels using a 7-point Likert scale. The similarities and differences between two instrument-derived scores were analyzed using correlations (r) and multidimensional scaling (MDS). RESULTS: The total scores of the two assessments were significantly correlated (r = 75%, p < 0.001), as were the subscores ('Total Physical fatigue': r = 76%, p < 0.001, 'Total Mental fatigue': r = 56%, p < 0.001). Relative overestimation of the MFI-K (45.8 ± 11.3) compared to the mKCFQ (36.1 ± 16.2) was observed, which was especially prominent in the 'mild' group. The scores of the three groups were more easily distinguished by the mKCFQ than by the MFI-K. In terms of the five dimension scores, we found a higher correlation of the two assessments for 'general fatigue' (r = 79%, p < 0.001) and 'physical fatigue' (r = 66%, p < 0.001) than for the reductions in 'motivation' (r = 41%, p < 0.01) and 'activity' (r = 26%, p > 0.05). CONCLUSIONS: Our results may indicate the usefulness of the two instruments, especially for the physical symptoms of fatigue ('general' and 'physical' fatigue). Furthermore, the MFI-K may be useful for conditions of moderate-to-severe fatigue, such as chronic fatigue syndrome, but the mKCFQ may be useful for all spectra of fatigue, including in subhealthy people.

Rasch analysis of fatigue severity scale in patients with epilepsy.

OBJECTIVE: To utilize the Rasch model to validate and assess the psychometric properties of the Fatigue Severity Scale (FSS) in patients with epilepsy. METHODS: A total of 307 patients (age > 18 years) with a confirmed diagnosis of epilepsy were consented to participate. Exclusion criteria included patients with psychogenic nonepileptic events, cognitive disabilities, and patients who did not speak/understand English. The nine-step FSS was programmed into software administered to patients on electronic tablets, and patient responses were auto-scored. The Rasch rating scale model (RSM) was used to evaluate the unidimensionality, reliability, and targeting of the FSS. To assess unidimensionality, we examined infit and outfit mean squares. We also assessed unidimensionality of the FSS using a principal component analysis of Rasch residuals, where residuals are understood as the difference between observed and expected data values. We evaluated the internal consistency of person and item performance by examining separation reliability estimates and separation ratio. Differential Item Functioning (DIF) was calculated for gender. RESULTS: There was mixed evidence regarding the extent to which the FSS fit the Rasch model. Outfit values ranged from 0.52 to 2.72 and infit values were 0.60 to 2.18, strongly suggesting the presence of misfitting items: Item 1 ("My motivation is lower when I am fatigued") and Item 2 ("Exercise brings on my fatigue"). SIGNIFICANCE: The nine-item FSS showed fair psychometric properties in this sample of patients with epilepsy. Our study provides unique, supportive information for the use of a modified version of the FSS, omitting the first two items, in patients with epilepsy. Given the prevalence of fatigue and other neuropsychiatric comorbidities of epilepsy, having a validated fatigue scale can aid healthcare providers to identify moderate-to-severe fatigue levels in patients with epilepsy and address the plausible risk factors.

Evaluating fatigue in patients recovering from COVID-19: validation of the fatigue severity scale and single item screening questions.

BACKGROUND: Fatigue is a common symptom in hospitalized and non-hospitalized patients recovering from COVID-19, but no fatigue measurement scales or questions have been validated in these populations. The objective of this study was to perform validity assessments of the fatigue severity scale (FSS) and two single-item screening questions (SISQs) for fatigue in patients recovering from COVID-19. METHODS: We examined patients ≥ 28 days after their first SARS-CoV-2 infection who were hospitalized for their acute illness, as well as non-hospitalized patients referred for persistent symptoms. Patients completed questionnaires through 1 of 4 Post COVID-19 Recovery Clinics in British Columbia, Canada. Construct validity was assessed by comparing FSS scores to quality of life and depression measures. Two SISQs were evaluated based on the ability to classify fatigue (FSS score ≥ 4). RESULTS: Questionnaires were returned in 548 hospitalized and 546 non-hospitalized patients, with scores computable in 96.4% and 98.2% of patients respectively. Cronbach's alpha was 0.96 in both groups. The mean ± SD FSS score was 4.4 ± 1.8 in the hospitalized and 5.2 ± 1.6 in the non-hospitalized group, with 62.5% hospitalized and 78.9% non-hospitalized patients classified as fatigued. Ceiling effects were 7.6% in the hospitalized and 16.1% in non-hospitalized patients. FSS scores negatively correlated with EQ-5D scores in both groups (Spearman's rho - 0.6 in both hospitalized and non-hospitalized; p < 0.001) and were higher among patients with a positive PHQ-2 depression screen (5.4 vs. 4.0 in hospitalized and 5.9 vs. 4.9 in non-hospitalized; p < 0.001). An SISQ asking whether there was "fatigue present" had a sensitivity of 70.6% in hospitalized and 83.2% in non-hospitalized patients; the "always feeling tired" SISQ, had a sensitivity of 70.5% and 89.6% respectively. CONCLUSIONS: Fatigue was common and severe in patients referred for post COVID-19 assessment. Overall, the FSS is suitable for measuring fatigue in these patients, as there was excellent data quality, strong internal consistency, and construct validity. However, ceiling effects may be a limitation in the non-hospitalized group. SISQs had good sensitivity for identifying clinically relevant fatigue in non-hospitalized patients but only moderate sensitivity in the hospitalized group, indicating that there were more false negatives.

Cardiovascular autonomic dysfunction in multiple sclerosis-findings and relationships with clinical outcomes and fatigue severity.

OBJECTIVES: This study evaluates cardiovascular autonomic dysfunction (CAD) in multiple sclerosis (MS) and explores if CAD is related to clinical outcomes and fatigue severity. METHODS: A total of 53 MS patients (30 relapsing-remitting, RRMS; 23 progressive, PMS) and 30 healthy controls were evaluated. TaskForce® Monitor was used to assess impedance cardiography parameters, heart rate (HRV), and blood pressure (BPV) variability during head-up tilt test (HUTT). Expiration/inspiration (E/I) ratio was assessed in response to a deep breathing test. Fatigue severity was evaluated using Chalder Fatigue Scale (CFQ). RESULTS: Compared to controls, PMS patients were characterized by increased sympathetic-parasympathetic ratio at rest (p < 0.01), decreased resting values of parasympathetic parameters (high-frequency HRV, p < 0.05; E/I ratio, p < 0.001), and index of contractility (p < 0.05), whereas RRMS patients showed reduced E/I ratio (p < 0.01). Compared to RRMS group, PMS patients had higher sympathovagal ratio and lower cardiac inotropy parameters (p < 0.05). No intergroup differences were observed for cardiovascular and autonomic function test parameters after HUTT. PMS and low CFQ physical score were identified as independent predictors of sympathetic hyper-reactivity as measured with HRV. Greater disability and male sex were predictors of diastolic BP increase and reduced cardiac inotropy parameters, and older age was predictor of decreased vagal tone (E/I ratio, high-frequency HRV). CONCLUSION: Cardiovascular autonomic modulation is altered in MS and highly dependent on disease variant, disability level, fatigue severity, and patients' demographics.

Real-time measurement of post-stroke fatigue in daily life and its relationship with the retrospective Fatigue Severity Scale.

Improving our understanding of post-stroke fatigue is crucial to develop more effective interventions. This effort may be hampered by the methods used to assess fatigue, which usually rely on retrospective memory reports. However, such reports are prone to memory bias and may not capture variability in fatigue in daily life; thereby failing to adequately represent symptom experience. This study aimed to assess the strength of the relationship between real-time experience of post-stroke fatigue and the commonly used retrospective Fatigue Severity Scale (FSS). Thirty individuals with stroke completed 10 daily questionnaires about momentary (here-and-now) fatigue for six consecutive days using the mHealth application PsyMateTM (Experience Sampling Method). From these real-time fatigue ratings (N = 1012), we calculated three indices: total average, peak fatigue, and fatigue on the final day. Afterwards, participants rated their fatigue retrospectively with the FSS. Results showed weak to moderate and strong correlations (range: .334, .667), with retrospective reports capturing up to 44% of the variance in the indices of momentary fatigue. Exploratory analyses also revealed that even individuals with similar total FSS scores demonstrated highly different day-to-day fatigue patterns. We conclude that retrospective measures may provide an incomplete view of post-stroke fatigue and diurnal variation therein.

Validation of a three-item Fatigue Severity Scale for patients with substance use disorder: a cohort study from Norway for the period 2016-2020.

BACKGROUND: Little attention has been paid to customising fatigue questionnaires for patients with Substance Use Disorders (SUDs). The present study aims to validate and shorten the nine-item Fatigue Severity Scale (FSS-9) and Visual Analogue Fatigue Scale (VAFS) for use with this population. METHODS: We used data from a nested cohort with annual health assessments with responses on the FSS-9 and VAFS. During the period 2016-2020, 917 health assessments were collected from 655 patients with SUD in Bergen and Stavanger, Norway. A total of 225 patients answered the health assessment at least twice. We defined baseline as the first annual health assessment when the health assessments were sorted chronologically per patient. We checked for internal consistency, and we used longitudinal confirmatory factor analysis (CFA) and linear mixed model (LMM) analysis to validate and shorten the FSS-9 and VAFS. RESULTS: The internal consistency of the FSS-9 was excellent with a Cronbach's α of 0.94 at baseline and 0.93 at the second annual health assessment. When shortening the FSS-9 to a three-item FSS (FSS-3, items 5-7), the Cronbach's α was 0.87 at baseline and 0.84 at the second health assessment. The internal consistency was not affected when the VAFS was added to the FSS-3 and the FSS-9. The longitudinal CFA model showed a well-fitting model for the FSS-3 (χ2 = 13.33, degree of freedom = 8, P = 0.101). The LMM analysis showed equal linear changes at the individual level for the FSS-3 (slope: 0.00, P > 0.05) and FSS-9 (slope: 0.01, P > 0.05) between the health assessments. CONCLUSION: The FSS-9 could be shortened to the FSS-3 with high validity and reliability for patients with SUDs and the addition of VAFS did not provide much added variability.

Fatigue and its relationship with disease-related factors in patients with systemic sclerosis: A cross-sectional study

Background/aim: Fatigue is very common symptom in patients with systemic sclerosis (SSc) and adversely affects health-related quality of life and the ability to perform daily living activities. This study aimed to determine the severity of fatigue, and its related factors, in patients with SSc. Materials and methods: A total of 35 patients with SSc (6 males and 29 females, mean age of 50.71 ± 10.09 years) and 35 healthy control subjects (8 males and 27 females, mean age of 54.14 ± 9.51 years) were included in this study. The Fatigue Impact Scale for fatigue, Modified Medical Research Council Scale for dyspnea severity, 6-Minute Walking Test for functional capacity, Health Assessment Questionnaire Disability Index, Scleroderma Health Assessment Questionnaire and Short Form-36 Quality of Life Questionnaire for health-related quality of life were used in the evaluation of the subjects. Furthermore, pulmonary functions, diffusion capacity, and respiratory and peripheral muscle strength were evaluated. Results: Of the SSc patients, 80% experienced fatigue and presented with higher total and cognitive, physical, and psychosocial subscale fatigue scores than the healthy control subjects (P < 0.05). Moreover, the SSc patients exhibited significantly increased dyspnea severity, impaired pulmonary function-diffusion capacity, decreased respiratory-peripheral muscle strength, reduced functional capacity, and worsened health-related quality of life when compared to the control group (P < 0.05). Fatigue in the SSc group was significantly associated with age, dyspnea severity, diffusion capacity, respiratory and peripheral muscle strength, functional capacity, and health- related quality of life (P < 0.05). Conclusions: Along with the decrease in diffusion capacity, increase dyspnea, a decrease in both peripheral and respiratory muscle strength, and worsening functional capacity may have an effect on increased fatigue in SSc patients. Increased fatigue can also affect the life quality and daily life activities of a patient. Therefore, multidisciplinary approaches are recommended to evaluate and improve these parameters in the treatment of fatigue from the early period in SSc patients.

Fatigue in adults with Osteogenesis Imperfecta.

BACKGROUND: Osteogenesis Imperfecta (OI) is characterized by bone fragility, and features such as blue sclerae, dentinogenesis imperfecta, hearing loss, ligamentous laxity and short stature can be present. It has long been assumed that the functional ability and quality of life of patients with OI depends primarily on the severity of skeletal deformities. However, fatigue is often mentioned in clinic by patients with all types of OI as an important modifier of their quality of life and does not always seem to be related to their functional ability. The aim of this study is to investigate whether adults with Osteogenesis Imperfecta are significantly more fatigued than the normal population. METHODS: The Fatigue Severity Scale (FSS) was distributed by mobile phone application among 151 adult patients with different OI types. Results of the FSS in the OI group were compared with two control populations from America (n = 20) and the Netherlands (n = 113). RESULTS: Ninety-nine patients (OI type 1 (n = 72), OI type 3 (n = 13), OI type 4 (n = 14) completed the FSS questionnaire. The mean FSS score of this cohort was 4.4 and significantly higher than the control populations (2.3/2.9). 65% of our cohort reported at least moderate fatigue compared with 2 control populations from America and the Netherlands. CONCLUSION: Fatigue in patients with OI is a frequently encountered problem in our expert clinic but research into this topic is sparse. This pilot study is the largest study to date investigating fatigue in patients with OI and results have been compared with two control groups. The mean FSS score of 4.4 in the OI group indicates that people with OI are generally significantly more fatigued than the control population. Further evaluation of fatigue and its influencers in a larger group of OI patients is important for future management.

Effects of Maximal Strength Training on Perceived-Fatigue and Functional Mobility in Persons with Relapsing-Remitting Multiple Sclerosis.

Background and objectives: Fatigue is one of the most disabling symptoms that limit daily life activities in persons with multiple sclerosis (pwMS). This study aimed to evaluate the effects of maximal strength training (MST) on perceived-fatigue and functional mobility in pwMS. Materials and Methods: 26 participants with MS were balanced according to their pre-intervention fatigue scores and distributed into an MST group (n = 13) and a control group (CG; n = 13). The MST group completed eight weeks using high loads, evaluating detraining after ten weeks. Quadriceps and hamstring isokinetic (QPTIK; HPTIK) and isometric (QPTIM; HPTIM) peak torques were assessed using an isokinetic dynamometer. Effect size differences were estimated with the Hedges' g index (dg ). Fatigue was evaluated through the Fatigue Severity Scale (FSS), while functional mobility was assessed via the Timed Up and Go Test (TUG). Results: The MST significantly improved all the strength measurements after the intervention (Δ6.43-29.55%; p < 0.05) compared to the control group. FSS showed a significant reduction (59.97%, dg = 5.41, large). The MST group also reduced the TUG time (19.69%; dg = 0.93, large) compared to the control group. Improvements caused by the intervention did not remain after a 10-week follow-up, with decreases in strength performance from 4.40% to 13.86% (dg = 0.24-0.56, small to moderate), 112.08% in the FSS (dg = -3.88, large), and 16.93% in TUG (dg = -1.07, large). Conclusions: MST (up to 90% 1RM) seems to be a feasible and useful way to obtain clinically relevant improvements in the perceived-fatigue symptoms and functional mobility. Still, symptom improvements decrease after a 10-week detraining period.

Psychological and sleep-related functioning among women with unsettled infants in Victoria, Australia: a cross-sectional study.

Objective: To identify (a) clinical symptom profiles based on psychological and sleep-related functioning among women admitted to a residential early parenting service (REPS) and (b) factors associated with membership of profile groups. Background: Depression and anxiety are common among women with unsettled infants; less is known about other indicators of psychological distress and about maternal sleep. Methods: Women admitted to a REPS during a 5-month period completed validated measures of depression, anxiety, stress, irritability, alcohol use, fatigue, sleepiness and sleep quality. Latent class analysis was used to identify symptom profiles. Factors significantly associated with class membership were identified. Results: Surveys were completed by 167/380 women. Scores on all measures were statistically significantly poorer than community norms. Two classes were identified, characterised by high versus low psychological distress. Mean scores on measures of fatigue, sleepiness and sleep quality were high in both classes. High psychological distress was associated with having previous mental health problems and an unmet need for emotional support. Conclusion: Fatigue and poor sleep quality are universal among women admitted to REPS. Health services providing assistance with unsettled infant behaviour should include strategies to improve maternal sleep, and encourage social interaction among women to protect against social isolation.

Validation of the Russian version of the Fatigue Impact Scale and Fatigue Severity Scale in multiple sclerosis patients.

BACKGROUND: Fatigue is a common complaint in patients with multiple sclerosis (MS), and its detection and monitoring are based on self-reported questionnaires. The objective of this study was to validate the Russian translation of the Fatigue Impact Scale (FIS) and the Fatigue Severity Scale (FSS) in MS patients and controls. METHODS: We included 85 MS patients and 250 age-, sex-, and body mass index (BMI)-matched controls. We ascertained in all subjects levels of education, marital status, and comorbidities, such as sleepiness (using the Epworth Sleepiness Scale, ESS), anxiety and depression (using the Hospital Anxiety and Depression Scale, HADS). The expanded disability status scale (EDSS) reflected physical disability in MS. RESULTS: The Russian versions of the three FIS subscales (cognitive, physical, and psychosocial) and FSS had excellent internal consistencies (Cronbach's α coefficients 0.88-0.96), and good test-retest stability with intraclass coefficients between 0.78 and 0.89. Both convergent and discriminant validity of the Russian FIS and FSS appeared to be good, as expressed by strong inter-correlations between FIS subscales and FSS, and by absent associations between fatigue scales and BMI. Principal components analysis and scree plots indicated unidimensional structures of the physical and cognitive FIS subscales and FSS, but a multidimensional structure of the psychosocial subscale. We identified EDSS and anxiety scores as independent predictors of more severe fatigue in MS. SIGNIFICANCE: The Russian FIS and FSS represent reliable and valid tools for efficient quantification and monitoring of fatigue severity and its clinical impact in MS. EDSS and anxiety are important contributors to fatigue severity in MS.

Association between seizure frequency and fatigue levels in children with epilepsy.

AIM: The purpose of this study was to examine the association between seizure-related features and fatigue levels in children with epilepsy. METHODS: All children were classified into three subgroups based on the state of their seizure control: well-controlled epilepsy (WCE; seizure-free), intermediate-controlled epilepsy (ICE; seizure frequency < 1×/month) and uncontrolled epilepsy (UCE; seizure frequency > 1×/month). Participants were asked to rate on a 7-point scale, from 1 (strongly disagree) to 7 (strongly agree), how often they felt the ways described by nine items on the Fatigue Severity Scale (FSS). A higher score is suggestive of greater fatigue. RESULTS: The study participants comprised 58 children with epilepsy and 15 children without seizures, who served as the healthy (non-epilepsy) group. The mean FSS scores of the children with epilepsy were significantly higher than those of the healthy (non-epilepsy) group (4.40 vs. 1.55, respectively; P < 0.0001). Multiple linear regression analysis showed that seizure frequency was the only characteristic significantly associated with fatigue (P < 0.0001). In the three epilepsy subgroups, the mean FSS scores for the WCE, intermediate-controlled epilepsy and UCE groups were 2.30, 3.97 and 6.28, respectively. A higher seizure frequency was associated with more severe fatigue. In particular, children in the UCE group had significantly more severe fatigue than those in the WCE group (P < 0.0001). CONCLUSIONS: The results suggest that seizure frequency is also associated with fatigue in children with epilepsy. Improved control of seizures may help reduce fatigue levels and improve the quality of life of children with epilepsy.

Fatigue in early Parkinson's disease: the Norwegian ParkWest study.

BACKGROUND AND PURPOSE: Fatigue is a common and disabling non-motor symptom in Parkinson's disease (PD). The pathogenesis is unknown, and the treatment options are limited. The aim of the present study was to investigate the development of fatigue during the first year after diagnosis. METHODS: The study design was a prospective, controlled population-based longitudinal cohort study, comprising 181 de novo, drug-naïve patients with PD and 162 control participants. PD was diagnosed according to the Gelb criteria. Fatigue was assessed by the Fatigue Severity Scale (FSS). Both groups were assessed for fatigue at baseline and after 1 year. RESULTS: Patients reported more fatigue than the control subjects at baseline and at the 1-year follow-up evaluation. The FSS scores in the patient group improved from a mean score of 4.4 (SD 1.6) to 4.0 (SD 1.6). Patients with fatigue at baseline received higher doses of dopaminergic medication during follow-up. Patients who received dopamine agonists improved slightly more than patients who received levodopa. A regression analysis did not show a correlation between an improvement in fatigue and a change in disease severity, depressive symptoms, sleep problems, apathy or cognitive impairment. CONCLUSION: Fatigue is a common symptom in PD, also in early, untreated patients. During the first year of observation, an improvement in the fatigue scores was found. The improvement could not be attributed to a change in disease severity or depressive symptoms. The results indicate a better effect of dopamine agonists than of levodopa. This may have implications for treatment in patients with PD-associated fatigue.