Experienced fatigue in people with rare disorders: a scoping review on characteristics of existing research

Bathen, Trine; Johansen, Heidi; Strømme, Hilde; Velvin, Gry

doi:10.1186/s13023-021-02169-6

Cited by 7 publications

(10 citation statements)

References 79 publications

(97 reference statements)

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“…Similar criteria have been employed previously to categorize CF cases in the Norwegian population (Loge et al, 1998). Nonetheless, the self‐reported CF and the broad variety of defining and measuring CF (Bathen et al, 2022; Finsterer & Mahjoub, 2014) reduces generalization of our results across different medical disorders. Also, insomnia was determined by the ISI rather than clinical diagnostic assessment.…”

Section: Discussionmentioning

confidence: 74%

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Digital cognitive behaviour therapy for insomnia in individuals with self‐reported insomnia and chronic fatigue: A secondary analysis of a large scale randomized controlled trial

Ramfjord

Faaland

Scott

et al. 2023

Journal of Sleep Research

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SummaryInsomnia is associated with fatigue, but it is unclear whether response to cognitive behaviour therapy for insomnia is altered in individuals with co‐occurring symptoms of insomnia and chronic fatigue. This is a secondary analysis using data from 1717 participants with self‐reported insomnia in a community‐based randomized controlled trial of digital cognitive behaviour therapy for insomnia compared with patient education. We employed baseline ratings of the Chalder Fatigue Questionnaire to identify participants with more or fewer symptoms of self‐reported chronic fatigue (chronic fatigue, n = 592; no chronic fatigue, n = 1125). We used linear mixed models with Insomnia Severity Index, Short Form‐12 mental health, Short Form‐12 physical health, and the Hospital Anxiety and Depression Scale separately as outcome variables. The main covariates were main effects and interactions for time (baseline versus 9‐week follow‐up), intervention, and chronic fatigue. Participants with chronic fatigue reported significantly greater improvements following digital cognitive behaviour therapy for insomnia compared with patient education on the Insomnia Severity Index (Cohen's d = 1.36, p < 0.001), Short Form‐12 mental health (Cohen's d = 0.19, p = 0.029), and Hospital Anxiety and Depression Scale (Cohen's d = 0.18, p = 0.010). There were no significant differences in the effectiveness of digital cognitive behaviour therapy for insomnia between chronic fatigue and no chronic fatigue participants on any outcome. We conclude that in a large community‐based sample of adults with insomnia, co‐occurring chronic fatigue did not moderate the effectiveness of digital cognitive behaviour therapy for insomnia on any of the tested outcomes. This may further establish digital cognitive behaviour therapy for insomnia as an adjunctive intervention in individuals with physical and mental disorders.

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Section: Discussionmentioning

confidence: 74%

“…Nonetheless, the self-reported CF and the broad variety of defining and measuring CF (Bathen et al, 2022;Finsterer & Mahjoub, 2014) reduces generalization of our results across different medical disorders.…”

Section: Discussionmentioning

confidence: 87%

Digital cognitive behaviour therapy for insomnia in individuals with self‐reported insomnia and chronic fatigue: A secondary analysis of a large scale randomized controlled trial

Ramfjord

Faaland

Scott

et al. 2023

Journal of Sleep Research

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“…In recent years, fatigue has increasingly been studied in genetic conditions, and recognized as an important clinical factor affecting several aspects of patients’ lives [ 21 ]. Through our work in a resource center for rare diseases, we frequently encounter patients who report fatigue as a serious problem.…”

Section: Introductionmentioning

confidence: 99%

“…There is a risk that health professionals do not pay attention to fatigue because it is overshadowed by other more potential life-threatening aspects of the disease. Often, medical causes of fatigue may be ignored [ 21 ].…”

Section: Introductionmentioning

confidence: 99%

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Fatigue in patients with syndromic heritable thoracic aortic disease: a systematic review of the literature and a qualitative study of patients’ experiences and perceptions

Velvin,

Johansen,

Østertun-Geirdal

et al. 2023

Orphanet J Rare Dis

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Introduction The purpose of this study was to explore the literature on fatigue in patients with syndromic heritable thoracic aortic disease (sHTAD), including Marfan syndrome (MFS), Loeys-Dietz syndrome (LDS), vascular Ehlers Danlos syndrome (vEDS) and other sHTADs, critically appraise and synthesize the relevant literature. We also aimed to investigate how adults with sHTAD experience and perceive fatigue, and to discuss clinical implications and direction for further research. Methods First, a systematic review was performed by searching the published literature in all relevant databases and other sources until 20th October 2022. Second, a qualitative focus group interview study was conducted of 36 adults with sHTADs (LDS n = 11, MFS n = 14, vEDS n = 11). Results In the systematic review, 33 articles satisfied the eligibility criteria (3 reviews and 30 primary studies). Of the primary studies: 25 dealt with adults (MFS n = 17, MFS/EDS n = 1, EDS n = 2, LDS/vEDS n = 3, different sHTADs n = 2), 5 with children (MFS n = 4, different sHTADs n = 1). Twenty-two were cross-sectional quantitative studies, 4 prospective and 4 qualitative studies. The quality of the included studies was mostly good, but many had small sample sizes, low response rates and/or participants without verified diagnosis. Despite these limitations, studies indicated high prevalence of fatigue (ranging from 37 to 89%), and fatigue was associated with both health and psychosocial aspects. Few studies found that fatigue was associated with disease-related symptoms. In the qualitative focus groups most of the participants reported that they had experienced fatigue which influenced several aspects of life. Four themes related to fatigue were elucidated: (1) different diagnoses–different fatigue?, (2) the nature of fatigue, (3) searches for causes of fatigue, (4) dealing with fatigue in daily life. The four themes seemed mutually interrelated in terms of barriers, strategies and facilitators for dealing with fatigue. The participants experienced fatigue as a consistent dilemma between self-assertion and inadequacy. Fatigue seems to influence several aspects of daily life and may be one of the most debilitating symptoms of having a sHTAD. Conclusion Fatigue seems to negatively impact the lives of people with sHTADs and should be recognized as an important aspect in the lifelong follow-up of these patients. The life-threatening complications of sHTADs may result in emotional stress, including fatigue and the risk of developing a sedentary lifestyle. Research and clinical initiatives should consider rehabilitation interventions aiming at postponing the onset or reducing symptoms of fatigue.

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Development of a phone application for assessing fatigue levels in rare disorders: a feasibility and validity study

Chu,

Payne,

Hunter

et al. 2023

J Rare Dis

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Purpose Barth syndrome (BTHS) is a rare genetic disorder characterized by skeletal myopathy, cardiomyopathy, and exercise intolerance due to early fatigue. The purpose of this study was to test the feasibility and validity of a new phone application designed to capture multi-dimensional aspects of fatigue across the lifespan. The specific study aims were to (1) assess the feasibility of using the app to record perceived fatigue levels in real-time, (2) evaluate discriminant validity by assessing if the app can differentiate between those with and without BTHS, and (3) content validity by assessing the relationship between perceived energy levels and actual energy expenditure. Methods Eighteen participants with BTHS and 18 age-matched control participants completed the study. The participants wore an activity tracker for 14 days and were prompted to respond to an Android app to report their fatigue levels 6 × /day. Statistical analysis was completed to examine perceived fatigue and the relationship between reported fatigue and actual energy expenditure. Results Feasibility was supported by the majority of participants responding to at least 50% of the application prompts and scores indicative of good internal consistency between responses (92–95%) and reliability of the battery scale (p < .001). Discriminant validity of the app was only partially supported, with the number of “crashes” being significantly different between those with and without BTHS (p = 0.042). Other measures of perceived fatigue were not found to be significantly different between groups, even though individuals with BTHS showed significantly lower energy expenditure than control participants during the day as measured by actigraphy (p < 0.001). Content validity of the app was supported, with perceived energy levels significantly correlating with actual energy expenditure collected with the activity tracker (p < 0.001). Conclusions In summary, the phone app developed by our team allowed researchers to capture the lived experience of individuals with BTHS while also capturing objective data. We verified that the app was able to consistently and accurately capture participant-reported fatigue. The battery scale tested as part of our feasibility aim was successful in capturing perceived levels of energy and can be used as a valid measure of fatigue in future studies. It was interesting to note that “crashes” appear to be the main differentiating factor in fatigue between the BTHS and control participants, where other measures of perceived fatigue were not found to be significantly different. These results highlight the complex nature of measuring fatigue as a subjective construct. This study provides foundational information on methods for quantifying fatigue in adults, adolescents, and children with BTHS and can provide possible targets for future therapeutic trials.

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Experienced fatigue in people with rare disorders: a scoping review on characteristics of existing research

Cited by 7 publications

References 79 publications

Digital cognitive behaviour therapy for insomnia in individuals with self‐reported insomnia and chronic fatigue: A secondary analysis of a large scale randomized controlled trial

Digital cognitive behaviour therapy for insomnia in individuals with self‐reported insomnia and chronic fatigue: A secondary analysis of a large scale randomized controlled trial

Fatigue in patients with syndromic heritable thoracic aortic disease: a systematic review of the literature and a qualitative study of patients’ experiences and perceptions

Development of a phone application for assessing fatigue levels in rare disorders: a feasibility and validity study

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