Experiences of people with multiple sclerosis at work: Towards the understanding of the needs for a job retention vocational rehabilitation intervention

Background: Supporting people with multiple sclerosis (MS) at work can be challenging due to the unpredictable nature and myriad of disease-related symptoms, and issues related to the work environment.Objective: To explore, amongst people with MS in employment, their experiences of and need for vocational rehabilitation (VR), and perceived barriers and facilitators to implementing VR.Methods: We conducted 20 semi-structured interviews with people with MS, employers, and healthcare professionals. Interviews were audio-recorded, transcribed, and analysed using the framework method.Results: We identified nine themes reflecting the main MS symptoms (e.g. cognition, fatigue), and environmental factors such as support provided at work (e.g. change of working hours) and workplace characteristics. Providing support tailored to the individual’s needs and early intervention were seen as important attributes for the intervention. The barriers identified referred to lack of resources and confidentiality issues; however, having flexibility in the content and delivery of the intervention were seen as facilitators.Conclusion: The impact of environmental factors (e.g. attitudes towards disability) on employment difficulties is equal to or greater than disease-related factors. Environmental changes in the attitudes of co-workers and workplace flexibility can enable people with MS to remain in work for longer

Development of cancer surveillance guidelines in ataxia telangiectasia: A Delphi-based consensus survey of international experts

Ataxia telangiectasia; Cancer predisposition; International surveyAtàxia telangiectàsia; Predisposició al càncer; Enquesta internacionalAtaxia telangiectasia; Predisposición al cáncer; Encuesta internacionalBackground/Objectives Ataxia telangiectasia (A-T) is a multiorgan disorder with increased vulnerability to cancer. Despite this increased cancer risk, there are no widely accepted guidelines for cancer surveillance in people affected by A-T. We aimed to understand the current international practice regarding cancer surveillance in A-T and agreed-upon approaches to develop cancer surveillance in A-T. Design/Methods We used a consensus development method, the e-Delphi technique, comprising three rounds. Round 1 consisted of a Delphi questionnaire and a survey that collected the details of respondents' professional background, experience, and current practice of cancer surveillance in A-T. Rounds 2 and 3 were designed based on previous rounds and modified according to the comments made by the panellists. The pre-specified consensus threshold was ≥75% agreement. Results Thirty-five expert panellists from 13 countries completed the study. The survey indicated that the current practice of cancer surveillance varies widely between experts and centres'. Consensus was reached that evidence-based guidelines are needed for cancer surveillance in people with A-T, with separate recommendations for adults and children. Statements relating to the tests that should be included, the age for starting and stopping cancer surveillance and the optimal surveillance interval were also agreed upon, although in some areas, the consensus was that further research is needed. Conclusion The international expert consensus statement confirms the need for evidence-based cancer surveillance guidelines in A-T, highlights key features that the guidelines should include, and identifies areas of uncertainty in the expert community. This elucidates current knowledge gaps and will inform the design of future clinical trials.This study is funded by a grant from Action for A-T (ref. 20NOT05). Renata Neves is supported by a Doctoral Fellowship awarded by the College of Radiographers (ref. DF021). This study is supported by the National Institute for Health Research (NIHR) Applied Research Collaboration East Midlands (ARC EM). The views expressed are those of the author(s) and not necessarily those of the NIHR or the Department of Health and Social Care

Efficient and effective assessment of deficits and their neural bases in stroke aphasia

ObjectiveMulti-assessment batteries are necessary for diagnosing and quantifying the multifaceted deficits observed post-stroke. Extensive batteries are thorough but impractically long for clinical settings or large-scale research studies. Clinically-targeted “shallow” batteries superficially cover a wide range of language skills relatively quickly but can struggle to identify mild deficits or quantify the impairment level. Our aim was to compare these batteries across a large group of chronic stroke aphasia and to test a novel data-driven reduced version of an extensive battery that maintained sensitivity to mild impairment, ability to grade deficits and the underlying component structure.MethodsWe tested 75 chronic left-sided stroke participants, spanning global to mild aphasia. The underlying structure of these three batteries was analysed using cross-validation and principal component analysis, in addition to univariate and multivariate lesion-symptom mapping.ResultsThis revealed a four-factor solution for the extensive and data-reduced batteries, identifying phonology, semantic skills, fluency and executive function in contrast to a two-factor solution using the shallow battery (language severity and cognitive severity). Lesion symptom mapping using participants’ factor scores identified convergent neural structures for phonology (superior temporal gyrus), semantics (inferior temporal gyrus), speech fluency (precentral gyrus) and executive function (lateral occipitotemporal cortex). The two shallow battery components converged with the phonology and executive function clusters. In addition, we show that multivariate models could predict the component scores using neural data, however not for every component.ConclusionsOverall, the data-driven battery appears to be an effective way to save time yet retain maintained sensitivity to mild impairment, ability to grade deficits and the underlying component structure observed in post-stroke aphasia

Comparing short and long batteries to assess deficits and their neural bases in stroke aphasia

Multiple language assessments are necessary for diagnosing, characterising and quantifying the multifaceted deficits observed in many patients’ post-stroke. Current language batteries, however, tend to be an imperfect trade-off between time and sensitivity of assessment. There have hitherto been two main types of battery. Extensive batteries provide thorough information but are impractically long for application in clinical settings or large-scale research studies. Clinically-targeted batteries tend to provide superficial information about a large number of language skills in a relatively short period of time by reducing the depth of each test but, consequently, can struggle to identify mild deficits, qualify the level of each impairment or reveal the underlying component structure. In the current study, we compared these batteries across a large group of individuals with chronic stroke aphasia to determine their utility. In addition, we developed a data-driven reduced version of an extensive battery that maintained sensitivity to mild impairment, ability to grade deficits and the component structure. The underlying structure of these three language batteries (extensive, shallow and data-reduced) was analysed using cross-validation analysis and principal component analysis. This revealed a four-factor solution for the extensive and data-reduced batteries, identifying phonology, semantic skills, fluency and executive function in contrast to a two-factor solution using the shallow battery (phonological/language severity and cognitive severity). Lesion symptom mapping using participants’ factor scores identified convergent neural structures based on existing language models for phonology (superior temporal gyrus), semantics (inferior temporal gyrus), speech fluency (precentral gyrus) and executive function (lateral occipitotemporal cortex) based on the extensive and data-reduced batteries. The two components in the shallow battery converged with the phonology and executive function clusters. In addition, we show that multivariate prediction models could be utilised to predict the component scores using neural data, however not for every component score within every test battery. Overall, the data-reduced battery appears to be an effective way to save assessment time yet retain the underlying structure of language and cognitive deficits observed in post stroke aphasia

Auditory beat perception is related to speech output fluency in post-stroke aphasia

Aphasia affects at least one third of stroke survivors, and there is increasing awareness that more fundamental deficits in auditory processing might contribute to impaired language performance in such individuals. We performed a comprehensive battery of psychoacoustic tasks assessing the perception of tone pairs and sequences across the domains of pitch, rhythm and timbre in 17 individuals with post-stroke aphasia and 17 controls. At the level of individual differences we demonstrated a correlation between metrical pattern (beat) perception and speech output fluency with strong effect (Spearman’s rho = 0.72). This dissociated from more basic auditory timing perception, which did not correlate with output fluency. This was also specific in terms of the language and cognitive measures, amongst which phonological, semantic and executive function did not correlate with beat detection. We interpret the data in terms of a requirement for the analysis of the metrical structure of sound to construct fluent output, with both being a function of higher-order “temporal scaffolding”. The beat perception task herein allows measurement of timing analysis without any need to account for motor output deficit, and could be a potential clinical tool to examine this. This work suggests strategies to improve fluency after stroke by training in metrical pattern perception

A qualitative study exploring how vocational rehabilitation for people with multiple sclerosis can be integrated within existing healthcare services in the United Kingdom

BackgroundTo explore how a vocational rehabilitation (VR) intervention can be integrated within existing healthcare services for people with multiple sclerosis (MS) in the United Kingdom (UK) National Health Service (NHS).MethodsData from 37 semi-structured interviews with 22 people with MS, eight employers, and seven healthcare professionals were analysed using a framework method informed by the Consolidated Framework for Implementation Research and an intervention logic model.ResultsFour themes were identified relating to the structure of current NHS services, how to improve access to and awareness of VR services, the collaboration between internal and external networks, and the benefits of integrating VR within the NHS services. Participants identified several implementation barriers such as poor links with external organisations, staffing issues, and lack of funding. To overcome these barriers, participants suggested enablers such as technology (such as apps or online assessments) and collaboration with third-sector organisations to reduce the pressure on the NHS.ConclusionSignificant organisational changes are required to ensure a successful implementation of a VR intervention within current NHS services. Despite this, the NHS was seen as a trustworthy organisation to offer support that can optimise the health and professional lives of people with MS

Preventing job loss for people with Multiple Sclerosis

Multiple Sclerosis (MS) is the most common chronic neurological condition affecting young adults. Many people are diagnosed with MS while they are of working age, and many leave the workplace prematurely. Vocational Rehabilitation (VR) aims to support those with illness or disability to find new employment, remain in or return to work (RTW). The effectiveness of VR for people with MS is inconclusive. This thesis aimed to develop, implement, and evaluate a job retention VR intervention for employed people with MS. The VR intervention was developed following the Medical Research Council Framework for developing and evaluating complex interventions and the person-based approach (PBA). The first study was a systematic review to identify VR interventions implemented to support people with MS to find new employment, remain in or RTW. Fourteen studies met the inclusion criteria describing thirteen interventions. There was considerable variability across the interventions and no clear conclusion about the most effective intervention characteristics or components was reached due to the poor reporting of the interventions. The second study was a qualitative study to explore the experiences of people with MS who work, their needs for VR, and perceived barriers and facilitators to implementing the intervention. I conducted 20 semi-structured interviews with people with MS, healthcare professionals, and employers. Analysis was informed by the framework method and interviews were underpinned by theoretical frameworks. I identified nine themes reflecting the main MS symptoms (e.g., cognition, fatigue), difficulties at work, and support received (e.g., change of working hours). Providing tailored support and early intervention were seen as important attributes for the intervention. The main barrier identified to delivering VR support referred to lack of resources. Having flexibility in the intervention delivery was seen as a facilitator to receiving VR. The findings were combined following the PBA to develop the intervention’s guiding principles, logic model, and a job retention intervention. The feasibility and acceptability of the intervention were tested using a mixed-methods single case study design. Secondary objectives included determining whether receiving this intervention was associated with changes in quality of life, fatigue, functional outcomes, and goal attainment. The intervention was tested between June 2020 and January 2021. I recruited 15 participants with MS, 3 employers and 4 healthcare professionals. On average the participants with MS received 8.36 (SD=4.48) hours of intervention and the employers received 1.94 (SD=0.38) hours. The most common topics addressed were managing cognitive problems, fatigue management, and reasonable accommodations. The intervention was only delivered remotely due to the Covid-19 pandemic. It was feasible to deliver the intervention, but it had no impact on quality of life, fatigue, and functional outcomes. However, it had a positive impact on goal attainment. Compared to baseline, the paired t-test showed a significant difference on goal attainment at the post-intervention assessment (t(14)=7.44, p=.0001, d=1.9), three (t(13)=4.81, p=.0001, d=1.28), and six (t(11)= 4.45, p=.001, d=1.28) months follow-up. Participants reported that the intervention was acceptable in the post-intervention interviews. Four themes were derived from the post-intervention interviews regarding the (1) context, (2) the employer, (3) empowerment, and (4) intervention components and attributes. Future research should focus on understanding how VR interventions can be embedded within existing healthcare services

Preventing job loss for people with Multiple Sclerosis

Multiple Sclerosis (MS) is the most common chronic neurological condition affecting young adults. Many people are diagnosed with MS while they are of working age, and many leave the workplace prematurely. Vocational Rehabilitation (VR) aims to support those with illness or disability to find new employment, remain in or return to work (RTW). The effectiveness of VR for people with MS is inconclusive. This thesis aimed to develop, implement, and evaluate a job retention VR intervention for employed people with MS. The VR intervention was developed following the Medical Research Council Framework for developing and evaluating complex interventions and the person-based approach (PBA). The first study was a systematic review to identify VR interventions implemented to support people with MS to find new employment, remain in or RTW. Fourteen studies met the inclusion criteria describing thirteen interventions. There was considerable variability across the interventions and no clear conclusion about the most effective intervention characteristics or components was reached due to the poor reporting of the interventions. The second study was a qualitative study to explore the experiences of people with MS who work, their needs for VR, and perceived barriers and facilitators to implementing the intervention. I conducted 20 semi-structured interviews with people with MS, healthcare professionals, and employers. Analysis was informed by the framework method and interviews were underpinned by theoretical frameworks. I identified nine themes reflecting the main MS symptoms (e.g., cognition, fatigue), difficulties at work, and support received (e.g., change of working hours). Providing tailored support and early intervention were seen as important attributes for the intervention. The main barrier identified to delivering VR support referred to lack of resources. Having flexibility in the intervention delivery was seen as a facilitator to receiving VR. The findings were combined following the PBA to develop the intervention’s guiding principles, logic model, and a job retention intervention. The feasibility and acceptability of the intervention were tested using a mixed-methods single case study design. Secondary objectives included determining whether receiving this intervention was associated with changes in quality of life, fatigue, functional outcomes, and goal attainment. The intervention was tested between June 2020 and January 2021. I recruited 15 participants with MS, 3 employers and 4 healthcare professionals. On average the participants with MS received 8.36 (SD=4.48) hours of intervention and the employers received 1.94 (SD=0.38) hours. The most common topics addressed were managing cognitive problems, fatigue management, and reasonable accommodations. The intervention was only delivered remotely due to the Covid-19 pandemic. It was feasible to deliver the intervention, but it had no impact on quality of life, fatigue, and functional outcomes. However, it had a positive impact on goal attainment. Compared to baseline, the paired t-test showed a significant difference on goal attainment at the post-intervention assessment (t(14)=7.44, p=.0001, d=1.9), three (t(13)=4.81, p=.0001, d=1.28), and six (t(11)= 4.45, p=.001, d=1.28) months follow-up. Participants reported that the intervention was acceptable in the post-intervention interviews. Four themes were derived from the post-intervention interviews regarding the (1) context, (2) the employer, (3) empowerment, and (4) intervention components and attributes. Future research should focus on understanding how VR interventions can be embedded within existing healthcare services