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Background and Purpose: Patients with a chronic illness, such as multiple sclerosis (MS), and their natural caregivers have a specific experience of healthcare and health services. These experiences need to be assessed to evaluate the quality of care. Our objective was to develop a French-language questionnaire to evaluate the quality of care as experienced by MS patients and their natural caregivers., Methods: Eligible patients had been diagnosed with MS according to the McDonald criteria. Eligible caregivers were individuals designated by the patients. The MusiCare questionnaire was developed in two standard phases: (i) item generation, based on interviews with patients and caregivers; and (ii) validation, consisting of validity, reliability, external validity, reproducibility, and responsiveness measures., Results: In total, 1088 patients (n = 660) and caregivers (n = 488) were recruited. The initial 64-item version of MusiCare was administered to a random subsample (n = 748). The validation process generated a 35-item questionnaire. Internal consistency and scalability were satisfactory. Testing of the external validity revealed expected associations between MusiCare scores and sociodemographic and clinical data. The questionnaire showed good reproducibility and responsiveness., Conclusions: The availability of a reliable and validated French-language self-report questionnaire probing the experience of the quality of care for MS will allow the feedback of patients and caregivers to be incorporated into a continuous healthcare quality-improvement strategy., (© 2020 European Academy of Neurology.)

Background: To enhance the use of quality of life (QoL) measures in clinical practice, it is pertinent to help clinicians interpret QoL scores., Objective: The aim of this study was to define clusters of QoL levels from a specific questionnaire (MusiQoL) for multiple sclerosis (MS) patients using a new method of interpretable clustering based on unsupervised binary trees and to test the validity regarding clinical and functional outcomes., Methods: In this international, multicenter, cross-sectional study, patients with MS were classified using a hierarchical top-down method of Clustering using Unsupervised Binary Trees. The clustering tree was built using the 9 dimension scores of the MusiQoL in 2 stages, growing and tree reduction (pruning and joining). A 3-group structure was considered, as follows: "high," "moderate," and "low" QoL levels. Clinical and QoL data were compared between the 3 clusters., Results: A total of 1361 patients were analyzed: 87 were classified with "low," 1173 with "moderate," and 101 with "high" QoL levels. The clustering showed satisfactory properties, including repeatability (using bootstrap) and discriminancy (using factor analysis). The 3 clusters consistently differentiated patients based on sociodemographic and clinical characteristics, and the QoL scores were assessed using a generic questionnaire, ensuring the clinical validity of the clustering., Conclusions: The study suggests that Clustering using Unsupervised Binary Trees is an original, innovative, and relevant classification method to define clusters of QoL levels in MS patients.

The aim was to develop a multidimensional computerized adaptive short-form questionnaire, the MusiQoL-MCAT, from a fixed-length QoL questionnaire for multiple sclerosis.A total of 1992 patients were enrolled in this international cross-sectional study. The development of the MusiQoL-MCAT was based on the assessment of between-items MIRT model fit followed by real-data simulations. The MCAT algorithm was based on Bayesian maximum a posteriori estimation of latent traits and Kullback-Leibler information item selection. We examined several simulations based on a fixed number of items. Accuracy was assessed using correlations (r) between initial IRT scores and MCAT scores. Precision was assessed using the standard error measurement (SEM) and the root mean square error (RMSE).The multidimensional graded response model was used to estimate item parameters and IRT scores. Among the MCAT simulations, the 16-item version of the MusiQoL-MCAT was selected because the accuracy and precision became stable with 16 items with satisfactory levels (r ≥ 0.9, SEM ≤ 0.55, and RMSE ≤ 0.3). External validity of the MusiQoL-MCAT was satisfactory.The MusiQoL-MCAT presents satisfactory properties and can individually tailor QoL assessment to each patient, making it less burdensome to patients and better adapted for use in clinical practice., Competing Interests: The authors have no conflicts of interest to disclose.

Objective: Quality of life (QoL) measurements are considered important outcome measures both for research on multiple sclerosis (MS) and in clinical practice. Computerized adaptive testing (CAT) can improve the precision of measurements made using QoL instruments while reducing the burden of testing on patients. Moreover, a cross-cultural approach is also necessary to guarantee the wide applicability of CAT. The aim of this preliminary study was to develop a calibrated item bank that is available in multiple languages and measures QoL related to mental health by combining one generic (SF-36) and one disease-specific questionnaire (MusiQoL)., Methods: Patients with MS were enrolled in this international, multicenter, cross-sectional study. The psychometric properties of the item bank were based on classical test and item response theories and approaches, including the evaluation of unidimensionality, item response theory model fitting, and analyses of differential item functioning (DIF). Convergent and discriminant validities of the item bank were examined according to socio-demographic, clinical, and QoL features., Results: A total of 1992 patients with MS and from 15 countries were enrolled in this study to calibrate the 22-item bank developed in this study. The strict monotonicity of the Cronbach's alpha curve, the high eigenvalue ratio estimator (5.50), and the adequate CFA model fit (RMSEA = 0.07 and CFI = 0.95) indicated that a strong assumption of unidimensionality was warranted. The infit mean square statistic ranged from 0.76 to 1.27, indicating a satisfactory item fit. DIF analyses revealed no item biases across geographical areas, confirming the cross-cultural equivalence of the item bank. External validity testing revealed that the item bank scores correlated significantly with QoL scores but also showed discriminant validity for socio-demographic and clinical characteristics., Conclusion: This work demonstrated satisfactory psychometric characteristics for a QoL item bank for MS in multiple languages. This work may offer a common measure for the assessment of QoL in different cultural contexts and for international studies conducted on MS.

Background: The extent to which MS patients with cognitive dysfunction can accurately self-report outcomes has been a crucial issue. The aim of this study was to quantify and compare the relevance of the quality of life (QoL) assessment between two populations with a high occurrence of cognitive dysfunction, specifically in individuals with multiple sclerosis (MS) and in individuals suffering from schizophrenia (SCZ)., Design: A cross-sectional study was performed using the following inclusion criteria: MS and SCZ patients were diagnosed according to the McDonald criteria and DSM-IV criteria, respectively. Data on sociodemographic (age, gender, education level) and clinical (disease severity, disease duration) factors, QoL (disease-specific questionnaires, MusiQoL and SQoL) and cognitive performance (executive, memory, and attention functions) were collected. Non-impaired and impaired populations were defined according to the French norms. Psychometric properties were compared to those reported in reference populations, which were assessed in the respective validation studies. Suitability indices were provided used to quantitatively compare how the structures in the different populations matched with the initial structure of the questionnaires (reference populations)., Results: One hundred and twenty-four MS patients and 113 SCZ patients were enrolled. Factor analysis was performed on the impaired populations and revealed that the questionnaire structure adequately matched the initial structure of the disease-specific QoL questionnaires. All of the suitability indices of construct and external validity in the non-impaired populations ranged from 70 to 100%., Conclusions: Our study suggested that cognitive dysfunction did not compromise the reliability or validity of the self-reported QoL questionnaires among subjects with cognitive dysfunction, such as MS and SCZ. Thus, this report may clarify the relevance of using self-reported QoL assessments in clinical practice.

Background: Responsiveness, defined as the ability to detect a meaningful change, is a core psychometric property of an instrument measuring quality of life (QoL) rarely reported in multiple sclerosis (MS) studies., Objective: To assess the responsiveness of the Multiple Sclerosis International Quality of Life (MusiQoL) questionnaire to change in disability over 24 months, defined by change in the Expanded Disability Status Scale (EDSS) score., Methods: Patients with MS were enrolled into a multicenter, longitudinal observational study. QoL was assessed using both the MusiQoL and the 36-Item Short-Form (SF-36) instruments at baseline and every 6 months thereafter up to month 24; neurological assessments, including EDSS score, were performed at each evaluation., Results: The 24-month EDSS was available for 524 patients. In the 107 worsened patients, two specific dimensions of MusiQoL, the sentimental and sexual life and the relationships with health care system dimensions, and 'physical' scores of SF-36 showed responsiveness., Conclusions: Whereas specific dimensions of MusiQoL identified EDSS changes, the MusiQoL index did not detect disability changes in worsened MS patients in a 24-month observational study. Future responsiveness validation studies should include longer follow-up and more representative samples.

Background: Attention disturbances are frequently observed in multiple sclerosis (MS) patients. The aim of this study was to provide new evidence regarding the suitability of using self-reported QoL information in this impaired population by exploring the construct validity, reliability, and external validity of a MS-specific quality of life (QoL) instrument., Design: cross-sectional study., Inclusion Criteria: MS patients of any disease subtype., Data Collection: sociodemographic (age, gender, marital status, education level, and occupational activity) and clinical data (Expanded Disability Status Scale, disease duration); QoL (MusiQoL and SF36); and attention performance (Wechsler Memory Scale and PASAT). According to the French norms, non-impaired and impaired populations were defined. For each population, suitability indices were provided to quantify how the structures matched with the initial structure of the reference population assessed in the validation study., Findings: One hundred and twenty-four consecutive patients were enrolled. The factor analysis performed in the impaired populations showed that the questionnaire structure adequately matched the initial structure of the MusiQoL. The unidimensionality of the dimensions was preserved, and the internal/external validity indices were close to those of the reference population., Conclusions: Our study suggests that attention impairment dysfunction did not compromise the reliability and validity of the self-reported QoL questionnaires.

Background: Memory disturbances, in particular episodic verbal memory dysfunction, are the most frequent cognitive impairment observed in multiple sclerosis (MS) patients. The use of self-reported outcomes for evaluating treatment and managing care of these subjects has been questioned. The aim of this study was to provide new evidence about the suitability of self-reported outcomes for use in this impaired population by exploring the internal structure, reliability and external validity of a specific quality of life (QoL) instrument, the Multiple Sclerosis International Quality of Life questionnaire (MusiQoL)., Design: cross-sectional study., Inclusion Criteria: MS patients of any disease subtype., Data Collection: sociodemographic (age, gender, marital status, education level, and occupational activity) and clinical data (MS subtype, Expanded Disability Status Scale, disease duration); QoL (MusiQoL and SF36); and memory performance (Grober and Buschke test). In accordance with the French norms of the memory test, non-impaired and impaired populations were defined for short- and long-delay free composites and for short- and long-delay total composites. For the 8 populations, psychometric properties were compared to those reported from the reference population assessed in the validation study., Principal Findings: One hundred and twenty-four consecutive patients were enrolled. The analysis performed in the impaired populations showed that the questionnaire structure adequately matched the initial structure of the MusiQoL. The unidimensionality of the dimensions was preserved, and the internal/external validity indices were close to those of the reference population., Conclusions/significance: Our study suggests that memory dysfunction did not compromise the reliability or validity of the self-reported QoL questionnaires.

Background: Cognitive impairment occurs in about 50% of multiple sclerosis (MS) patients, and the use of self-reported outcomes for evaluating treatment and managing care among subjects with cognitive dysfunction has been questioned. The aim of this study was to provide new evidence about the suitability of self-reported outcomes for use in this specific population by exploring the internal structure, reliability and external validity of a specific quality of life (QoL) instrument, the Multiple Sclerosis International Quality of Life questionnaire (MusiQoL)., Design: cross-sectional study., Inclusion Criteria: MS patients of any disease subtype., Data Collection: sociodemographic (age, gender, marital status, education level, and occupational activity) and clinical data (MS subtype, Expanded Disability Status Scale, disease duration); QoL (MusiQoL and SF36); and neuropsychological performance (Stroop color-word test)., Statistical Analysis: confirmatory factor analysis, item-dimension correlations, Cronbach's alpha coefficients, Rasch statistics, relationships between MusiQoL dimensions and other parameters., Principal Findings: One hundred and twenty-four consecutive patients were enrolled. QoL scores did not differ between the 69 cognitively non-impaired patients and the 55 cognitively impaired patients, except for the symptoms dimension. The confirmatory factor analysis performed among the impaired subjects showed that the structure of the questionnaire matched with the initial structure of the MusiQoL. The unidimensionality of the MusiQoL dimensions was preserved, and the internal validity indices were satisfactory and close to those of the reference population., Conclusions/significance: Our study suggests that executive dysfunction did not compromise the reliability and the validity of the self-reported QoL questionnaires.

Background: The aim of this study was to propose an alternative approach to item response theory (IRT) in the development of computerized adaptive testing (CAT) in quality of life (QoL) for patients with multiple sclerosis (MS). This approach relied on decision regression trees (DRTs). A comparison with IRT was undertaken based on precision and validity properties. Materials and methods: DRT- and IRT-based CATs were applied on items from a unidimensional item bank measuring QoL related to mental health in MS. The DRT-based approach consisted of CAT simulations based on a minsplit parameter that defines the minimal size of nodes in a tree. The IRT-based approach consisted of CAT simulations based on a specified level of measurement precision. The best CAT simulation showed the lowest number of items and the best levels of precision. Validity of the CAT was examined using sociodemographic, clinical and QoL data. Results: CAT simulations were performed using the responses of 1,992 MS patients. The DRT-based CAT algorithm with minsplit = 10 was the most satisfactory model, superior to the best IRT-based CAT algorithm. This CAT administered an average of nine items and showed satisfactory precision indicators (R = 0.98, root mean square error [RMSE] = 0.18). The DRT-based CAT showed convergent validity as its score correlated significantly with other QoL scores and showed satisfactory discriminant validity. Conclusion: We presented a new adaptive testing algorithm based on DRT, which has equivalent level of performance to IRT-based approach. The use of DRT is a natural and intuitive way to develop CAT, and this approach may be an alternative to IRT. [ABSTRACT FROM AUTHOR]

Background: Multiple sclerosis (MS), a common neurodegenerative disease, has well-described associations with quality of life (QoL) impairment. QoL changes found in longitudinal studies are difficult to interpret due to the potential response shift (RS) corresponding to respondents' changing standards, values, and conceptualization of QoL. This study proposes to test the capacity of Random Forest (RF) for detecting RS reprioritization as the relative importance of QoL domains' changes over time. Methods: This was a longitudinal observational study. The main inclusion criteria were patients 18 years old or more with relapsing-remitting multiple sclerosis. Every 6 months up to month 24, QoL was recorded using generic and MS-specific questionnaires (MusiQoL and SF-36). At 24 months, individuals were divided into two 'disability change' groups: worsened and not-worsened patients. The RF method was performed based on Breiman's description. Analyses were performed to determine which QoL scores of SF-36 predicted the MusiQoL index. The average variable importance (AVI) was estimated. Results: A total of 417 (79.6%) patients were defined as not-worsened and 107 (20.4%) as worsened. A clear RS was identified in worsened patients. While the mental score AVI was almost one third higher than the physical score AVI at 12 months, it was 1.5 times lower at 24 months. Conclusion: This work confirms that the RF method offers a useful statistical approach for RS detection. How to integrate the RS in the interpretation of QoL scores remains a challenge for future research. [ABSTRACT FROM AUTHOR]