Symbol Digit Modalities Test Variant in a Smartphone App for Persons With Multiple Sclerosis:Validation Study

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A two-minute walking test with a smartphone app for persons with multiple sclerosis:Validation study

BACKGROUND: Walking disturbances are a common dysfunction in persons with multiple sclerosis (MS). The 2-Minute Walking Test (2MWT) is widely used to quantify walking speed. We implemented a smartphone-based 2MWT (s2MWT) in MS sherpa, an app for persons with MS. When performing the s2MWT, users of the app are instructed to walk as fast as safely possible for 2 minutes in the open air, while the app records their movement and calculates the distance walked. OBJECTIVE: The aim of this study is to investigate the concurrent validity and test-retest reliability of the MS sherpa s2MWT. METHODS: We performed a validation study on 25 persons with relapsing-remitting MS and 79 healthy control (HC) participants. In the HC group, 21 participants were matched to the persons with MS based on age, gender, and education and these followed the same assessment schedule as the persons with MS (the HC-matched group), whereas 58 participants had a less intense assessment schedule to determine reference values (the HC-normative group). Intraclass correlation coefficients (ICCs) were determined between the distance measured by the s2MWT and the distance measured using distance markers on the pavement during these s2MWT assessments. ICCs were also determined for test-retest reliability and derived from 10 smartphone tests per study participant, with 3 days in between each test. We interviewed 7 study participants with MS regarding their experiences with the s2MWT. RESULTS: In total, 755 s2MWTs were completed. The adherence rate for the persons with MS and the participants in the HC-matched group was 92.4% (425/460). The calculated distance walked on the s2MWT was, on average, 8.43 m or 5% (SD 18.9 m or 11%) higher than the distance measured using distance markers (n=43). An ICC of 0.817 was found for the concurrent validity of the s2MWT in the combined analysis of persons with MS and HC participants. Average ICCs of 9 test-retest reliability analyses of the s2MWT for persons with MS and the participants in the HC-matched group were 0.648 (SD 0.150) and 0.600 (SD 0.090), respectively, whereas the average ICC of 2 test-retest reliability analyses of the s2MWT for the participants in the HC-normative group was 0.700 (SD 0.029). The interviewed study participants found the s2MWT easy to perform, but they also expressed that the test results can be confronting and that a pressure to reach a certain distance can be experienced. CONCLUSIONS: The high correlation between s2MWT distance and the conventional 2MWT distance indicates a good concurrent validity. Similarly, high correlations underpin a good test-retest reliability of the s2MWT. We conclude that the s2MWT can be used to measure the distance that the persons with MS walk in 2 minutes outdoors near their home, from which both clinical studies and clinical practice can benefit

A prospective web-based patient-centred interactive study of long-term disabilities, disabilities perception and health-related quality of life in patients with multiple sclerosis in The Netherlands:The Dutch Multiple Sclerosis Study protocol

BACKGROUND: In the past two decades the widespread use of disease modifying drugs with moderate to strong efficacy has changed the natural course of multiple sclerosis (MS). Health care professionals, researchers, patient organizations and health authorities are in need of recent information about the objectified and subjective long-term clinical outcomes in MS patients. Such information is scarce. METHODS/DESIGN: We started a prospective, web-based, patient-centred, interactive study of long-term disabilities, disabilities perception and health-related quality of life (HRQoL) in MS patients in The Netherlands (Dutch Multiple Sclerosis Study). The study has an on online patient-driven inclusion and online acquisition of patient-reported outcomes (PROs). At six-months intervals participants complete the Multiple Sclerosis Impact Profile (MSIP) (disabilities and disabilities perception in seven domains and four symptoms), the Multiple Sclerosis Quality of Life-54 items (MSQoL-54), the Modified Fatigue Impact Scale-5 items (MFIS-5) and the Leeds Multiple Sclerosis Quality of Life-8 items (LMSQoL) questionnaires, and a Medication and Adherence Inventory. Every three years the Expanded Disability Status Scale (EDSS) score is assessed by phone. The monthly completion of the MFIS-5, LMSQoL and Medication and Adherence Inventory is optional. Completed questionnaires and inventories, and automatically generated scores are made available online to patients for self-monitoring and self-management purposes, and to authorized health care professionals for the evaluation of disease activity and of the effectiveness of treatments. Study duration is planned to be 15 years. Results will be analyzed periodically using means and standard deviations for continuous variables, and frequencies for categorical variables. Relations between time points, variables, patient and treatment characteristics will be evaluated in random effects repeated measures models. DISCUSSION: The Dutch Multiple Sclerosis Study is characterized by online patient-driven inclusion; online data acquisition; the use of PROs; the optional monthly completion of short questionnaires; the interactive use of personal study data by patients and authorized health care professionals for self-monitoring, self-management and multidisciplinary care; the expected representativeness of the study sample; and a long-term time horizon. The study will provide valuable data on long-term disabilities, disabilities perceptions and HRQoL in MS patients in The Netherlands

Whole Exome Sequencing in Multi-Incident Families Identifies Novel Candidate Genes for Multiple Sclerosis

Multiple sclerosis (MS) is a degenerative disease of the central nervous system in which auto-immunity-induced demyelination occurs. MS is thought to be caused by a complex interplay of environmental and genetic risk factors. While most genetic studies have focused on identifying common genetic variants for MS through genome-wide association studies, the objective of the present study was to identify rare genetic variants contributing to MS susceptibility. We used whole exome sequencing (WES) followed by co-segregation analyses in nine multi-incident families with two to four affected individuals. WES was performed in 31 family members with and without MS. After applying a suite of selection criteria, co-segregation analyses for a number of rare variants selected from the WES results were performed, adding 24 family members. This approach resulted in 12 exonic rare variants that showed acceptable co-segregation with MS within the nine families, implicating the genes MBP, PLK1, MECP2, MTMR7, TOX3, CPT1A, SORCS1, TRIM66, ITPR3, TTC28, CACNA1F, and PRAM1. Of these, three genes (MBP, MECP2, and CPT1A) have been previously reported as carrying MS-related rare variants. Six additional genes (MTMR7, TOX3, SORCS1, ITPR3, TTC28, and PRAM1) have also been implicated in MS through common genetic variants. The proteins encoded by all twelve genes containing rare variants interact in a molecular framework that points to biological processes involved in (de-/re-)myelination and auto-immunity. Our approach provides clues to possible molecular mechanisms underlying MS that should be studied further in cellular and/or animal models

Risk knowledge of people with relapsing-remitting multiple sclerosis:Results of an international survey

Background Adequate disease and treatment-related risk knowledge of people with Multiple Sclerosis (pwMS) is a prerequisite for informed choices in medical encounters. Previous work showed that MS risk knowledge is low among pwMS and role preferences are different in Italy and Germany. Objective We investigated the level of risk knowledge and role preferences in 8 countries and assessed putative variables associated with risk knowledge. Methods An online-survey was performed based on the Risk knowledge questionnaire for people with relapsing-remitting MS (RIKNO 2.0), the electronic Control Preference Scale (eCPS), and other patient questionnaires. Inclusion criteria of participants were: (1) age 18 years, (2) a diagnosis of relapsing-remitting MS (RRMS), (3) being in a decision making process for a disease modifying drug. Results Of 1939 participants from Germany, Italy, the Netherlands, Serbia, Spain and Turkey, 986 (51%) (mean age 38.6 years [range 18–67], 77% women, 7.8 years of disease duration) completed the RIKNO 2.0, with a mean of 41% correct answers. There were less than 50 participants in the UK and Estonia and data were not analysed. Risk knowledge differed across countries (p &lt; 0.001). Variables significantly associated with higher risk knowledge were higher education (p &lt; 0.001), previous experience with disease modifying drugs (p = 0.001), correct answer to a medical data interpretation question (p &lt; 0.001), while higher fear for wheelchair dependency was negatively associated to risk knowledge (p = 0.001). Conclusion MS risk knowledge was overall low and differed across participating countries. These data indicate that information is an unmet need of most pwMS.</p

The MS@Work study:a 3-year prospective observational study on factors involved with work participation in patients with relapsing-remitting Multiple Sclerosis

Background: Multiple Sclerosis (MS) is the most common cause of neurological disability in young and middle-aged adults. At this stage in life most people are in the midst of their working career. The majority of MS patients are unable to retain employment within 10 years from disease onset. Leading up to unemployment, many may experience a reduction in hours or work responsibilities and increased time missed from work. The MS@Work study examines various factors that may influence work participation in relapsing-remitting MS patients, including disease-related factors, the working environment and personal factors. Methods/design: The MS@Work study is a multicenter, 3-year prospective observational study on work participation in patients with relapsing-remitting MS. We aim to include 350 patients through 15-18 MS outpatient clinics in the Netherlands. Eligible participants are 18 years and older, and either currently employed or within three years since their last employment. At baseline and after 1, 2 and 3 years, the participants are asked to complete online questionnaires (including questions on work participation, work problems and accommodations, cognitive and physical ability, anxiety, depression, psychosocial stress, quality of life, fatigue, empathy, personality traits and coping strategies) and undergo cognitive and neurological examinations. After six months, patients are requested to only complete online questionnaires. Patient perspectives on maintaining and improving work participation and reasons to stop working are gathered through semi-structured interviews in a sub-group of patients. Discussion: Prospective studies with long-term follow-up on work participation in MS are rare, or take into account a limited number of factors. The MS@Work study provides a 3-year follow-up on various factors that may influence work participation in patients with relapsing-remitting MS. We aim to identify factors that relate to job loss and to provide information about preventative measures for physicians, psychologists and other professionals working in the field of occupational health

Patient-reported adverse effects of high-dose intravenous methylprednisolone treatment:a prospective web-based multi-center study in multiple sclerosis patients with a relapse

In a prospective multi-center observational study, we evaluated the frequency, severity, and impact on activities of daily living (ADL) of adverse effects (AEs) of high-dose intravenous methylprednisolone (IVMP) in relapsing remitting multiple sclerosis (MS) patients with a relapse. Online self-report questionnaires stating IVMP's most common AEs were completed at baseline, the 2nd day of treatment, and 1 day and 1 week after treatment. Eighty-five patients were included, 66 completed the baseline questionnaire, and 59 completed at least one post-baseline questionnaire. Patients reported on average 4 (median) AEs; two (3.4 %) reported no AE. Most frequent was change in taste (61 %), facial flushing (61 %), sick/stomach pain (53 %), sleep disturbance (44 %), appetite change (37 %), agitation (36 %), and behavioral changes (36 %). Of all AEs, 34.3 % were severe and 37.9 % impacted on ADL. A 3-day course resulted in 4 (median) AEs and a 5-day course in 7. All patients with high disease impact had two or more AEs, compared with 79 % of those with low impact (p <0.01). Of patients with high disability, 45 % had severe AEs, compared with 16 % of those with low disability. Severe central nervous system (CNS)-related AEs occurred two times more frequently in patients with high disease impact, and two-and-a-half times more frequently in patients with high disability. Therefore, in virtually all patients, high-dose IVMP leads to AEs, with about one of three AEs being severe with impact on ADL. Patients with high disease impact or high disability may experience more (severe) AEs, due to a higher occurrence of severe CNS-related AEs

The interactive web-based program MSmonitor for self-management and multidisciplinary care in multiple sclerosis:utilization and valuation by patients

Background: MSmonitor is an interactive web-based program for self-management and integrated, multidisciplinary care in multiple sclerosis. Methods: To assess the utilization and valuation by persons with multiple sclerosis, we held an online survey among those who had used the program for at least 1 year. We evaluated the utilization and meaningfulness of the program's elements, perceived use of data by neurologists and nurses, and appreciation of care, self-management, and satisfaction. Results: Fifty-five persons completed the questionnaire (estimated response rate 40%). The Multiple Sclerosis Impact Profile (MSIP), Medication and Adherence Inventory, Activities Diary, and electronic consultation (e-consult) were used by 40%, 55%, 47%, and 44% of respondents and were considered meaningful by 83%, 81%, 54%, and 88%, respectively. During out-patient consultations, nurses reportedly used the MSmonitor data three to six times more frequently than neurologists. As to nursing care, more symptoms were dealt with (according to 54% of respondents), symptoms were better discussed (69%), and the overall quality of care had improved (60%) since the use of the program. As to neurological care, these figures were 24%, 31%, and 27%, respectively. In 46% of the respondents, the insight into their symptoms and disabilities had increased since the use of the program; the MSIP, Activities Diary, and e-consult had contributed most to this improvement. The overall satisfaction with the program was 3.5 out of 5, and 73% of the respondents would recommend the program to other persons with multiple sclerosis. Conclusion: A survey among persons with multiple sclerosis using the MSmonitor program showed that the MSIP, Medication and Adherence Inventory, Activities Diary, and e-consult were frequently used and that the MSIP, Medication and Adherence Inventory, and e-consult were appreciated the most. Moreover, the quality of nursing care, but not so neurological care, had improved, which may relate to nurses making more frequent use of the MSmonitor data than neurologists