Dutch translation and validation of the Communicative Participation Item Bank (CPIB)-short form.

BACKGROUND: Several conditions and diseases can result in speech problems that can have a negative impact on everyday functioning, referred to as communicative participation. Subjective problems with acquired speech problems are often assessed with the speech handicap index (SHI). To assess generic participation problems, the Utrecht Scale for Evaluation of Rehabilitation-Participation (USER-P) questionnaire is frequently used. The English questionnaire Communicative Participation Item Bank-short form (CPIB short form) is a 10-item valid, reliable instrument that assesses communicative participation. In the absence of a Dutch equivalent, translation and validation of the CPIB short form was required. AIMS: To translate the CPIB short form into Dutch, and to determine its psychometric properties for the group of adults with speech problems resulting from a neurological aetiology or head and neck cancer. METHODS & PROCEDURES: Translation of the CPIB short form was performed following the instructions of the European Organisation for Research and Treatment for Cancer (EORTC). In a cross-sectional multi-centre study, participants completed the Dutch CPIB short form together with the SHI and USER-P, and the CPIB a second time after 2 weeks. We assessed internal consistency and test-retest reliability of the CPIB. Construct validity was assessed based on correlations with SHI, USER-P and speech assessments. OUTCOMES & RESULTS: In the validation study, 122 participants were included: 51 with dysarthria due to different neurological disorders, 48 with speech problems due to head and neck cancer treatment and 23 healthy controls. Internal consistency of the items was high (Cronbach's alpha = 0.962), the intraclass correlation coefficient (ICC) for test-retest reliability was high 0.908 (95% CI = 0.870-0.935). Construct validity was supported by a strong correlation between the Dutch CPIB short form and the SHI total score (SHI total rs = 0.887) and a moderate correlation between the Dutch CPIB-10 and the USER-P subscales (USER-P Frequency rs = 0.365; USER-P restrictions and USER-P satisfaction rs = 0.546). A moderate correlation was found between the Dutch CPIB-10 and the speech performance assessments (degree of distortedness r = -0.0557; p ≤ 0.001; degree of intelligibility r = 0.0562). CONCLUSIONS & IMPLICATIONS: The Dutch CPIB short form provides a valid and reliable tool for clinical practice and research purposes. It allows clinicians to start using this PROM in clinical and research practice to systematically investigate the impact of the speech problems on communicative participation in a Dutch-speaking population. WHAT THIS PAPER ADDS: What is already known on the subject Communicative participation allows people to take part in life situations, but can be affected by acquired speech problems. The CPIB is a patient-reported outcome measure for the assessment of this concept. For the English language the 46-item bank and a 10-item short form is available. What this paper adds to existing knowledge This paper describes the process of translation of the CPIB short form into Dutch, and confirms its reproducibility and validity. What are the potential or actual clinical implications of this work? With this validated Dutch version of the CPIB short form available, professionals can implement this tool in clinical and research practice to systematically evaluate communicative participation.

Com-mens: a home-based logopaedic intervention program for communication problems between people with dementia and their caregivers - a single-group mixed-methods pilot study.

BACKGROUND: Communication difficulties are common in people with dementia, and often present from an early stage. However, direct treatment options for people with dementia that positively influence their daily communication are scarce. AIMS: To evaluate the potential impact and feasibility of a personalized logopaedic intervention. METHODS & PROCEDURES: A total of 40 community-dwelling persons with dementia and their caregivers were recruited. Five experienced speech and language therapists (SLTs) delivered the six-session Com-mens intervention at home. Com-mens aims to improve positive communication between people with dementia and their primary caregivers and comprises five elements: interactive history-taking, dynamic observational assessment, education about the consequences of dementia on communication, development and use of personalized communication tools, use motivational, and person-centred strategies by the SLT. We conducted a single-group mixed-methods pilot study with five measurements: baseline, directly after intervention, and at 3, 6 and 9 months follow-up. Semi-structured interviews and questionnaires for Experienced Communication in Dementia, quality of life, psychological well-being and caregiver burden were conducted. Process evaluation was performed by interviewing participants, drop-outs, SLTs and other stakeholders. OUTCOMES & RESULTS: A total of 32 dyads completed the intervention. Repeated measures analyses revealed no significant changes over time. In the interviews, participants reported a positive impact on their feelings, increased communication skills and better coping with the diagnosis. Participants would recommend the intervention to others. Facilitators were timely delivery, personalized content and adequate reimbursement. Barriers were unfamiliarity with Com-mens among referrers, an overburdened caregiver or disrupted family relationships. CONCLUSIONS & IMPLICATIONS: This newly developed logopaedic intervention is feasible and has a perceived positive impact on both people with dementia and their caregivers, which is confirmed by a stable pattern over a period of 1 year. Future comparative studies are needed to test the effectiveness of personalized interventions in this patient population. WHAT THIS PAPER ADDS: What is already known on the subject? SLTs are experts in the field of communication, but even though communication problems are common between people with dementia and their caregivers, there is a lack of logopaedic guidelines and materials for the direct treatment for this population. Interventions that are available either focus on (professional) caregivers only or aim to enhance cognitive functioning and do not target on joined communication. What this paper adds to the existing knowledge? A newly developed intervention called Com-mens can be provided by trained SLTs and takes an average of six 1-h sessions. The intervention is perceived to be valuable and feasible for people with dementia and their caregivers, by the participants themselves, as well as by healthcare professionals and other stakeholders. What are the potential or actual clinical implications of this work? Dissemination of this intervention will give SLTs skills, tools and materials to provide meaningful care to home-dwelling persons with dementia and their caregivers. Also, persons with dementia and their caregivers will receive education and materials that can help them increase their understanding of communication problems, enhance their communication skills and better cope with the communication problems that result from dementia. We consider the Com-mens intervention to be a valuable addition to the field of speech language therapy and dementia.

Muscle ultrasound is a sensitive biomarker in oculopharyngeal muscular dystrophy.

INTRODUCTION/AIMS: Oculopharyngeal muscular dystrophy (OPMD) is a late-onset, progressive muscle disease. Quantitative muscle ultrasound (QMUS) assesses structural changes in muscles and is a sensitive biomarker in neuromuscular disorders. Our aim of this study was to determine whether QMUS can detect muscle pathology and can be used as longitudinal imaging biomarker in OPMD. METHODS: Genetically confirmed OPMD patients, recruited by their treating physicians or from the national neuromuscular database, were examined twice, 20 months apart, using QMUS of orofacial and limb muscles, and measurements of functional capacity and muscle strength. Absolute echo intensity (AEI) and muscle thickness of all muscles were analyzed and correlated with clinical data. RESULTS: The tongue, deltoid, iliopsoas, rectus femoris, and soleus muscles showed increased AEI at baseline compared with normal values in 43 OPMD patients, with the rectus femoris being most often affected (51%).The AEI and muscle thickness of 9 of 11 muscles correlated significantly with the motor function measure, 10-step stair test, swallowing capacity, dynamometry, Medical Research Council grade, tongue strength, and bite force (r = 0.302 to -0.711). Between baseline and follow-up, deterioration in AEI was found for the temporalis, tongue, and deltoid muscles, and decreased muscle thickness was detected for the temporalis, masseter, digastric, tongue, deltoid, iliopsoas, and soleus muscles (P < .05). No relation was found between the change in AEI and repeat length or disease duration. DISCUSSION: QMUS detected muscle pathology and disease progression in OPMD over 20 months. We conclude that QMUS should be considered as a biomarker in treatment trials.

Age-Associated Salivary MicroRNA Biomarkers for Oculopharyngeal Muscular Dystrophy.

Small non-coding microRNAs (miRNAs) are involved in the regulation of mRNA stability. Their features, including high stability and secretion to biofluids, make them attractive as potential biomarkers for diverse pathologies. This is the first study reporting miRNA as potential biomarkers for oculopharyngeal muscular dystrophy (OPMD), an adult-onset myopathy. We hypothesized that miRNA that is differentially expressed in affected muscles from OPMD patients is secreted to biofluids and those miRNAs could be used as biomarkers for OPMD. We first identified candidate miRNAs from OPMD-affected muscles and from muscles from an OPMD mouse model using RNA sequencing. We then compared the OPMD-deregulated miRNAs to the literature and, subsequently, we selected a few candidates for expression studies in serum and saliva biofluids using qRT-PCR. We identified 126 miRNAs OPMD-deregulated in human muscles, but 36 deregulated miRNAs in mice only (pFDR < 0.05). Only 15 OPMD-deregulated miRNAs overlapped between the in humans and mouse studies. The majority of the OPMD-deregulated miRNAs showed opposite deregulation direction compared with known muscular dystrophies miRNAs (myoMirs), which are associated. In contrast, similar dysregulation direction was found for 13 miRNAs that are common between OPMD and aging muscles. A significant age-association (p < 0.05) was found for 17 OPMD-deregulated miRNAs (13.4%), whereas in controls, only six miRNAs (1.4%) showed a significant age-association, suggesting that miRNA expression in OPMD is highly age-associated. miRNA expression in biofluids revealed that OPMD-associated deregulation in saliva was similar to that in muscles, but not in serum. The same as in muscle, miRNA expression levels in saliva were also found to be associated with age (p < 0.05). Moreover, the majority of OPMD-miRNAs were found to be associated with dysphagia as an initial symptom. We suggest that levels of specific miRNAs in saliva can mark muscle degeneration in general and dysphagia in OPMD.

Improving Communication between Persons with Mild Dementia and Their Caregivers: Qualitative Analysis of a Practice-Based Logopaedic Intervention.

OBJECTIVE: To identify the essential elements of a newly developed, practice-based logopaedic intervention, which focuses on communication between persons with dementia (PwDs) and their caregivers. METHODS: The intervention of 6 one-hour sessions was conducted and evaluated with 4 PwD-caregiver dyads. Eighteen therapy sessions were video recorded and semi-structured interviews with all dyads and an interview with the speech-language therapist (SLT) were audio recorded. Framework analysis and triangulation were used to analyse the data. RESULTS: Five elements were found, which were systematically applied in the treatment of all dyads: interactive history taking, dynamic assessment, education about consequences of dementia for communicative effectiveness, development and use of two communication tools, and specific didactic strategies of the SLT. Regarding the outcome of the treatment, the dyads valued the focus on the interaction between PwD and caregiver, the usefulness of the received pieces of advice, and the empowering attitude of the SLT, which improved their self-confidence. The SLT added another element for an efficient approach: the ability to deliver treatment in people's home environment. CONCLUSION: A short pragmatic but consistent approach for communication problems caused by dementia seems promising for improving daily communication and reducing stress and frustration. Further research will explore the feasibility and efficacy of this approach.

The Radboud Dysarthria Assessment: Development and Clinimetric Evaluation.

OBJECTIVE: In the absence of an adequate dysarthria assessment in the Netherlands, we developed the Radboud Dysarthria Assessment (RDA). This article describes its development and clinimetric evaluation. PATIENTS AND METHODS: Forty-three patients were assessed with the RDA. The recording forms were subjected to exploratory factor analysis and estimation of internal consistency. The self-evaluation questionnaire was tested for internal consistency and the severity scale for intra- and inter-rater reliability. Construct validity of the severity scale and questionnaire was determined by relating them to the Speech Handicap Index (SHI), Dutch sentence intelligibility assessment (NSVO-Z), and category fluency task. RESULTS: Exploratory factor analysis extracted 4 factors (articulation, resonance, phonation, respiration/prosody) yielding an explained variance of 70.3%. Each factor showed good internal consistency (Cronbach's α: 0.89-0.91). The self-evaluation questionnaire showed excellent internal consistency (Cronbach's α: 0.90). Intra-class correlation coefficients of the severity scale (0.85-0.86) showed good reliability. The severity scores and self-evaluation questionnaire correlated substantially to strongly with the SHI (rs = 0.40 and 0.80) and substantially with the NSVO-Z (rs = -0.65 and -0.52). CONCLUSIONS: The RDA is a valid and reliable tool, but further investigation is needed to demonstrate whether this instrument can successfully support speech-language therapists in correctly diagnosing the type of dysarthria.

Effectiveness of remotely delivered speech therapy in persons with Parkinson's disease - a randomised controlled trial.

Background: Increasing evidence supports the merits of speech therapy in Parkinson's disease, but the current practice of multiple in-house treatments is demanding for patients. We therefore assessed the effectiveness of remotely delivered and personalised speech therapy on improving quality of life and speech quality in persons with Parkinson's disease. Methods: We performed a single blinded randomised controlled trial (the PERSPECTIVE study), comparing 8 weeks of personalised remote speech therapy to no intervention (waiting list design). Patients with reduced speech intelligibility were included, regardless of disease stage or dysarthria severity. Patients were assigned randomly (1:1) to the intervention or control group. Measurements took place at baseline and after 8 weeks (both groups), and after 32 weeks (intervention group only). Patients were treated remotely by 20 experienced speech therapists. The primary outcome was disease-related quality of life at 8 weeks, assessed with the Parkinson's Disease Questionnaire 39 (PDQ-39). Data were analysed using analysis of covariance based on the intention-to-treat principle. This trial is registered in ClinicalTrials.gov, NCT03963388. Findings: Between March 1, 2019, and March 27, 2021, 214 patients were enrolled in the intervention group (n = 109) or control group (n = 105). At the primary timepoint, the adjusted mean difference in PDQ-39 was -2.0 in favour of the intervention group (95% CI -4.0 to 0.1); p = 0.056). The intervention group scored better on the communication index score of the PDQ-39 (post hoc analysis), with an adjusted mean difference of -5.3 (95% CI -9.4 to -1.2; p = 0.011). We found no between-group differences on any other PDQ-39 domain. Follow-up measurements showed a significant reduction of the PDQ-39 compared to the primary timepoint with a difference of 2.40 (95% CI 0.77-4.02; p = 0.004). Interpretation: Personalised remote speech therapy improved communication-related quality of life, but not overall quality of life. Funding: Michael J. Fox Foundation, Gatsby Foundation, and Health∼Holland.

Specialized Allied Health Care for Parkinson's Disease: State of the Art and Future Directions.

People with Parkinson's disease (PD) experience a range of progressive motor and non-motor symptoms, that negatively affect their daily functioning, social participation and quality of life. Allied health therapies have emerged as an effective treatment approach-complementary to pharmacological and neurosurgical treatments-which reduces the impact of PD in daily life. In this article, we propose criteria for what constitutes specialized allied health care for PD, and we review allied health research in PD in terms of meeting these criteria and its outcomes for monodisciplinary approaches as well as multi- or interdisciplinary allied health interventions. We focus on the three most studied allied health disciplines in PD: physical therapy, occupational therapy and speech-language therapy. Overall, the available evidence underscores the importance and potential benefits of specialized allied health care for people with PD. Our proposed criteria and recommendations for future research might help in further delineating specialized allied health care.

Muscle MRI in Patients With Oculopharyngeal Muscular Dystrophy: A Longitudinal Study

BACKGROUND AND OBJECTIVES: Oculopharyngeal muscular dystrophy (OPMD) is a rare progressive neuromuscular disease. MRI is one of the techniques that is used in neuromuscular disorders to evaluate muscle alterations. The aim of this study was to describe the pattern of fatty infiltration of orofacial and leg muscles using quantitative muscle MRI in a large national cohort and to determine whether MRI can be used as an imaging biomarker of disease progression in OPMD. METHODS: Patients with OPMD (18 years or older) were invited from the national neuromuscular database or by their treating physicians and were examined twice with an interval of 20 months, with quantitative MRI of orofacial and leg muscles to assess fatty infiltration which were compared with clinical measures. RESULTS: In 43 patients with genetically confirmed OPMD, the muscles that were affected most severely were the tongue (mean fat fraction: 37.0%, SD 16.6), adductor magnus (31.9%; 27.1), and soleus (27.9%; 21.5) muscles. The rectus femoris and tibialis anterior muscles were least severely affected (mean fat fractions: 6.8%; SD 4.7, 7.5%; 5.9). Eleven of 14 significant correlations were found between fat fraction and a clinical task in the corresponding muscles (r = -0.312 to -0.769, CI = -0.874 to -0.005). At follow-up, fat fractions had increased significantly in 17 of the 26 muscles: mean 1.7% in the upper leg muscles (CI = 0.8-2.4), 1.7% (1.0-2.3) in the lower leg muscles, and 1.9% (0.6-3.3) in the orofacial muscles (p < 0.05). The largest increase was seen for the soleus (3.8%, CI = 2.5-5.1). Correlations were found between disease duration and repeat length vs increased fat fraction in 7 leg muscles (r = 0.323 to -0.412, p < 0.05). DISCUSSION: According to quantitative muscle MRI, the tongue, adductor magnus and soleus show the largest fat infiltration levels in patients with OPMD. Fat fractions increased in several orofacial and leg muscles over 20 months, with the largest fat fraction increase seen in the soleus. This study supports that this technique is sensitive enough to show worsening in fat fractions of orofacial and leg muscles and therefore a responsive biomarker for future clinical trials.

One-year evaluation of people recovering from COVID-19 receiving allied primary healthcare: A nationwide prospective cohort study.

BACKGROUND: A Dutch nationwide prospective cohort study was initiated to investigate recovery trajectories of people recovering from coronavirus disease 2019 (COVID-19) and costs of treatment by primary care allied health professionals. OBJECTIVES: The study described recovery trajectories over a period of 12 months and associated baseline characteristics of participants recovering from COVID-19 who visited a primary care allied health professional. It also aimed to provide insight into the associated healthcare and societal costs. METHODS: Participants completed participant-reported standardized outcomes on participation, health-related quality of life, fatigue, physical functioning, and costs at baseline (ie, start of the treatment), 3, 6, 9 and 12 months. RESULTS: A total of 1451 participants (64 % women, 76 % mild/moderate severity) with a mean (SD) age of 49 (12) years were included. Linear mixed models showed significant and clinically relevant improvements over time in all outcome measures between baseline and 12 months. Between 6 and 12 months, we found significant but not clinically relevant improvements in most outcome measures. Having a worse baseline score was the only baseline factor that was consistently associated with greater improvement over time on that outcome. Total allied healthcare costs (mean €1921; SEM €48) made up about 3% of total societal costs (mean €64,584; SEM €3149) for the average participant in the cohort. CONCLUSIONS: The health status of participants recovering from COVID-19 who visited an allied health professional improved significantly over a 12-month follow-up period, but nearly the improvement occurred between baseline and 6 months. Most participants still reported severe impairments in their daily lives, and generated substantial societal costs. These issues, combined with the fact that baseline characteristics explained little of the variance in recovery over time, underscore the importance of continued attention for the management of people recovering from COVID-19. TRIAL REGISTRATION: clinicaltrials.gov (NCT04735744).

The Complexity of Nutritional Problems in Persons with Dementia: Expanding a Theoretical Model.

BACKGROUND: Persons with dementia are at risk of developing nutritional problems. Theoretical models on nutritional problems have been developed, but have not been evaluated with healthcare professionals. OBJECTIVE: This study aimed to explore the comprehensiveness and applicability of a theoretical model of nutritional problems in persons with dementia for daily nursing home practice. METHODS: A qualitative design employing a combined deductive and inductive approach was used. Healthcare professionals were eligible to participate if they 1) had expert knowledge of and experience with nutritional problems related to dementia, and 2) worked in a nursing home affiliated with an academic network covering the east and south of the Netherlands. Three focus group interviews with 20 healthcare professionals from seven professions were held. We conducted thematic analysis and we compared themes with existing theoretical models from the literature. RESULTS: We identified six themes, four of which corresponded with the existing models (observing and analysing nutritional problems; consequences of nutritional problems; functioning of the person with dementia; environmental factors). Interprofessional collaboration and ethical factors were identified as new themes. The analyses indicated interactions within each theme, between themes, and a bidirectional connection between themes. CONCLUSIONS: This study demonstrated the relevance of interprofessional collaboration and ethical considerations in nutritional problems related to dementia. It uncovered complex bidirectional relations within and between factors regarding nutritional problems. All aspects should be taken into account to minimize the consequences of nutritional problems for persons with dementia.

Clinimetric Evaluation of the Experienced Communication in Dementia Questionnaire.

BACKGROUND AND OBJECTIVES: Tools to measure self-perceived communication between persons with early-stage dementia and their caregivers are lacking. Therefore, we developed a questionnaire for Experienced Communication in Dementia (ECD) with a patient version (ECD-P) and a caregiver version (ECD-C), which contains items on (a) caregiver competence, (b) social communication, (c) communication difficulties, and (d) experienced emotions. This article describes the feasibility and clinimetric evaluation of this instrument. RESEARCH DESIGN AND METHODS: A prospective observational cohort study was conducted with 57 dyads (community-dwelling person with dementia and primary caregiver). ECD-P, ECD-C, and measures on quality of life, caregiver burden, cognitive functioning, physical functioning, and functional independence were administered. After 2 weeks, the dyads filled out the ECD again. Feasibility (completion time and missing values per item), internal consistency (Cronbach's α), test-retest reliability (intraclass correlation coefficients [ICCs]), and construct validity (hypotheses testing with Spearman's r) were evaluated. RESULTS: Mean completion time was 10 min per questionnaire. ICCs for test-retest reliability ranged from 0.67 to 0.78, except for ECD-P2 (ICC = 0.31). Internal consistency ranged from α = 0.75 to 0.82 for ECD-P1 and all parts of ECD-C, except for ECD-P2 (α = 0.66). Correlation coefficients for convergent validity ranged from r = 0.31 to 0.69 and correlation coefficients for divergent validity were r < 0.20 and statistically insignificant. DISCUSSION AND IMPLICATIONS: Pending future research, the ECD, except part ECD-P2, seems to be a promising tool to measure experienced communication between persons with early-stage dementia and their caregivers.

Evaluation of Primary Allied Health Care in Patients Recovering From COVID-19 at 6-Month Follow-up: Dutch Nationwide Prospective Cohort Study.

BACKGROUND: Patients recovering from COVID-19 often experience persistent problems in their daily activities related to limitations in physical, nutritional, cognitive, and mental functioning. To date, it is unknown what treatment is needed to support patients in their recovery from COVID-19. OBJECTIVE: This study aimed to evaluate the primary allied health care of patients recovering from COVID-19 at 6-month follow-up and to explore which baseline characteristics are associated with changes in the scores of outcomes between baseline and 6-month follow-up. METHODS: This Dutch nationwide prospective cohort study evaluated the recovery of patients receiving primary allied health care (ie, dietitians, exercise therapists, occupational therapists, physical therapists, and speech and language therapists) after COVID-19. All treatments offered by primary allied health professionals in daily practice were part of usual care. Patient-reported outcome measures on participation, health-related quality of life, fatigue, physical functioning, and psychological well-being were assessed at baseline and at 3- and 6-month follow-up. Linear mixed model analyses were used to evaluate recovery over time, and uni- and multivariable linear regression analyses were used to examine the association between baseline characteristics and recovery. RESULTS: A total of 1451 adult patients recovering from COVID-19 and receiving treatment from 1 or more primary allied health professionals were included. For participation (Utrecht Scale for Evaluation of Rehabilitation-Participation range 0-100), estimated mean differences of at least 2.3 points were observed at all time points. For the health-related quality of life (EuroQol Visual Analog Scale, range 0-100), the mean increase was 12.3 (95% CI 11.1-13.6) points at 6 months. Significant improvements were found for fatigue (Fatigue Severity Scale, range 1-7): the mean decrease was -0.7 (95% CI -0.8 to -0.6) points at 6 months. However, severe fatigue was reported by 742/929 (79.9%) patients after 6 months. For physical functioning (Patient-Reported Outcomes Measurement Information System-Physical Function Short Form 10b, range 13.8-61.3), the mean increase was 5.9 (95% CI 5.9-6.4) points at 6 months. Mean differences of -0.8 (95% CI -1.0 to -0.5) points for anxiety (Hospital Anxiety and Depression Scale range 0-21) and -1.6 (95% CI -1.8 to -1.3) points for depression were found after 6 months. A worse baseline score, hospital admission, and male sex were associated with greater improvement between baseline and 6-month follow-up, whereas age, the BMI, comorbidities, and smoking status were not associated with mean changes in any outcome measures. CONCLUSIONS: Patients recovering from COVID-19 who receive primary allied health care make progress in recovery but still experience many limitations in their daily activities after 6 months. Our findings provide reference values to health care providers and health care policy makers regarding what to expect from the recovery of patients who receive health care from 1 or more primary allied health professionals. TRIAL REGISTRATION: ClinicalTrials.gov NCT04735744; https://tinyurl.com/3vf337pn. INTERNATIONAL REGISTERED REPORT IDENTIFIER (IRRID): RR2-10.2340/jrm.v54.2506.

Time trends in demographic characteristics of participants and outcome measures in Parkinson's disease research: A 19-year single-center experience.

Background: Females, people with young-onset PD and older individuals, and non-white populations are historically underrepresented in clinical Parkinson's disease (PD) research. Furthermore, research traditionally focused predominantly on motor symptoms of PD. Including a representative and diverse group of people with PD and also studying non-motor symptoms is warranted to better understand heterogeneity in PD and to generalize research findings. Objective: This project aimed to determine whether, within a consecutive series of PD studies performed within a single center in the Netherlands: (1) the proportion of included females, mean age and proportion of native Dutch people changed over time; and 2) reports of the ethnicity of participants and the proportion of studies with non-motor outcomes changed over time. Methods: Characteristics of participants and non-motor outcomes were analyzed using a unique dataset of summary statistics of studies with a large number of participants conducted at a single center during a 19-year period (2003-2021). Results: Results indicate no relationship between calendar time and proportion of females (mean 39 %), mean age (66 years), proportion of studies that reported ethnicity, and proportion of native Dutch people in studies (range 97-100 %). The proportion of participants in whom non-motor symptoms were assessed increased, but this difference was consistent with chance. Conclusion: Study participants in this center reflect the PD population in the Netherlands in terms of sex, but older individuals and non-native Dutch individuals are under-represented. We have still a lot to do in ensuring adequate representation and diversity in PD patients within our research.

Validation of the pediatric Radboud Dysarthria Assessment.

PURPOSE: The Radboud Dysarthria Assessment (RDA) was published in 2014. Adaptation into a pediatric version (p-RDA) was required because of relevant differences between children and adults. The purpose of this study was to assess the feasibility of the p-RDA and to test intra-rater and inter-rater reliability as well as the validity of the two severity scales (function and activity level). METHODS: Video recordings were made of 35 participants with (suspected) dysarthria (age 4 to 17 years) while being assessed using the p-RDA. Intra-rater reliability was assessed by one, and inter-rater reliability by two experiments using the Intraclass Correlation Coefficient (ICC). Validity of the severity scales was tested by correlating the consensus scores with the independently rated scores on four communication scales, three mobility scales, and one self-care scale using Spearman correlation coefficients (rs). RESULTS: The assessment was applicable for 89% of the tested sample, with good intra-rater and inter-rater reliability (ICC = 0.88-0.98 and 0.83-0.93). The p-RDA severity scales (function and activity level) correlated from substantially to strongly with the communication scales (rs = 0.69-0.82 and 0.77-0.92) and self-care scale (rs = 0.76-0.71) and correlated substantially with the mobility scales (rs = 0.49-0.60). CONCLUSION: The feasibility, reliability and validity of the p-RDA are sufficient for clinical use.

A Sense of Being Needed: A Phenomenological Analysis of Hospital-Based Rehabilitation Professionals' Experiences During the COVID-19 Pandemic.

OBJECTIVE: The purpose of this study was to explore lived experiences of rehabilitation professionals working in hospitals during the COVID-19 pandemic, including the ethical issues and moral distress that these professionals might have encountered. METHODS: An interpretative phenomenological study was performed. First-person experiences of rehabilitation professionals (dieticians, occupational therapists, physical therapists, and speech-language therapists) were collected with semi-structured interviews and analyzed with interpretative phenomenological analysis. RESULTS: The data of 39 hospital-based rehabilitation professionals revealed 4 themes: a disease with great impact, personal health and safety, staying human in chaotic times, and solidarity and changing roles. Participant experiences show that the virus and COVID-19 measures had a significant impact on the in-hospital working environment due to the massive downscaling of regular care, due to infection prevention measures, and due to unknown risks to rehabilitation professionals' personal health. At the same time, participants experienced a certain freedom, which made room for authentic motives, connection, and solidarity. Participants felt welcomed and appreciated at the COVID-19 wards and intensive care units and were proud that they were able to fulfill their roles. The findings reflect a wide range of situations that were morally complex and led to moral distress. CONCLUSION: To diminish the long-lasting negative impact of the COVID-19 pandemic and moral distress, employers should empathize with the experiences of hospital-based rehabilitation professionals and create conditions for ethical reflection. Our data show that hospital-based rehabilitation professionals value professional autonomy. Creating room for professional autonomy helps them feel needed, connected, and energized. However, the needs of hospital-based rehabilitation professionals may conflict with organizational rules and structures. IMPACT: Hospital-based rehabilitation professionals were involved in situations they considered morally undesirable, and they inevitably faced moral distress during the COVID-19 crisis. This study offers rationale and guidance to employers regarding how to reduce the long-term negative impact of the COVID-19 pandemic on rehabilitation professionals.

Experienced Respiratory Symptoms and the Impact on Daily Life from the Perspective of People with Parkinson's Disease: A Grounded Theory.

BACKGROUND: Abnormal respiratory function tests can be observed early in the course of Parkinson's disease (PD). A better understanding of the impact of respiratory dysfunction on daily life in PD is needed to prevent later occurring complications as a (aspiration) pneumonia. OBJECTIVE: To explain which respiratory symptoms people with PD or a form of atypical parkinsonism experience and how these symptoms impact on their daily lives. METHODS: This qualitative study used a grounded theory approach. A purposeful sample strategy was used to capture information-rich cases. Data were collected in semi-structured interviews with participants diagnosed with either PD (n = 11) or atypical parkinsonism (n = 3), all of whom had confirmed respiratory symptoms. Data were analyzed using grounded theory analysis by creating codes, categories, theoretical themes, and, ultimately, a conceptual model. RESULTS: Four respiratory profiles emerged, describing different types of respiratory dysfunction, with various positive and negative influencing factors. First, a loss of breathing automatism was experienced. Second, episodes of breathlessness or a rapid, shallow breathing pattern were triggered by either physical exertion, fatigue, or postural deformities. Third, stress and anxiety also triggered episodes of breathlessness. Fourth, a decreased cough strength and frequent coughing. Based on these findings, we constructed a conceptual model that visualizes the relations between these four types of respiratory dysfunction and their impact on daily life, with 'discomfort' and 'avoidance of social activities' as crucial elements. CONCLUSION: A tailored approach for each profile of respiratory dysfunction is recommended to improve respiratory dysfunction and to reduce its social impact in people with PD.

Pathophysiology of diurnal drooling in Parkinson's disease.

Drooling is an incapacitating feature of Parkinson's disease. Better pathophysiological insights are needed to improve treatment. In this study, we tested the hypothesis that the cause of drooling is multifactorial. We examined 15 patients with Parkinson's disease with distinct diurnal saliva loss ("droolers") and 15 patients with Parkinson's disease without drooling complaints ("nondroolers"). We evaluated all factors that could potentially contribute to drooling: swallowing capacity (maximum volume), functional swallowing (assessed with the dysphagia subscale of the Therapy Outcome Measures for rehabilitation specialists), unintentional mouth opening due to hypomimia (Unified Parkinson's Disease Rating Scale item), posture (quantified from sagittal photographs), and nose-breathing ability. We also quantified the frequency of spontaneous swallowing during 45 minutes of quiet sitting, using polygraphy. Droolers had more advanced Parkinson's disease than nondroolers (Unified Parkinson's Disease Rating Scale motor score 31 vs 22; P=.014). Droolers also scored significantly worse on all recorded variables except for nose breathing. Swallowing frequency tended to be higher, possibly to compensate for less efficient swallowing. Logistic regression with adjustment for age and disease severity showed that hypomimia correlated best with drooling. Linear regression with hypomimia as the dependent variable identified disease severity, dysphagia, and male sex as significant explanatory factors. Drooling in Parkinson's disease results from multiple risk factors, with hypomimia being the most prominent. When monitored, patients appear to compensate by increasing their swallowing frequency, much like the increased cadence that is used to compensate for stepping akinesia. These findings can provide a rationale for behavioral approaches to treat drooling.

Reproducibility and validity of patient-rated assessment of speech, swallowing, and saliva control in Parkinson's disease.

OBJECTIVE: To report on the development and psychometric evaluation of the Radboud Oral Motor Inventory for Parkinson's Disease (ROMP), a newly developed patient-rated assessment of speech, swallowing, and saliva control in patients with Parkinson's disease (PD). DESIGN: Reliability and validity study. SETTING: Tertiary-care Parkinson center for multidisciplinary assessment. PARTICIPANTS: Consecutive community-dwelling patients with PD (n=129) or atypical parkinsonism (AP; n=49; mean ± SD age, 64±9.8y; mean ± SD disease duration, 7y; median Hoehn and Yahr [HY] stage, 2.5). INTERVENTIONS: Not applicable. MAIN OUTCOME MEASURES: To evaluate reproducibility, 60 patients completed the ROMP twice within a mean of 24±12 days. To study validity, another cohort of 118 patients who had completed the ROMP was assessed by both a neurologist (HY stage, Unified Parkinson's Disease Rating Scale III) and speech-language pathologist (severity of dysarthria, dysphagia, drooling) who were blinded to ROMP scores. RESULTS: Confirmatory factor analysis identified the 3 a priori-designed ROMP domains of speech, swallowing, and saliva control. Internal consistency was .95 for the total ROMP and .87 to .94 for the 3 domains or subscales. Intraclass correlation coefficients for reproducibility were .94 and .83 to .92 for the subscales. Construct validity was substantial to good with correlations ranging from .36 to .82. The ROMP differentiated significantly (P<.001) between patients indicated for speech therapy (based on independent assessment) and those who were not and between mild, moderate, and severe PD according to HY stage. CONCLUSIONS: The ROMP provides a reliable and valid instrument to evaluate patient-perceived problems with speech, swallowing, and saliva control in patients with PD or AP.

Longitudinal Assessment of Strength, Functional Capacity, Oropharyngeal Function, and Quality of Life in Oculopharyngeal Muscular Dystrophy.

ObjectiveOculopharyngeal muscular dystrophy (OPMD) is a late-onset, progressive muscle disease. Disease progression is known to be slow, but details on the natural history remain unknown. We aimed to examine the natural history of OPMD in a large nationwide cohort to determine clinical outcome measures that capture disease progression and can be used in future clinical trials.MethodsPatients, invited by their treating physicians or from the national neuromuscular database, and invited family members, were examined twice, 20 months apart, using fixed dynamometry, MRC grading, maximum bite force and isometric tongue strength, Motor Function Measure (MFM), 10-step stair test, maximum swallowing-, chewing-, and speech-tasks and quality of life assessments.ResultsDisease progression was captured by 8 out of 18 measures over 20 months in forty-three genetically confirmed OPMD patients. The largest deterioration was seen in deltoid muscle strength (-27% (range -17 - -37%)), followed by the quadriceps (-14% (range -6 - -23%)), iliopsoas (-12.2%), tongue (-9.9%) and MRC sum-score (-2.5%). The 10-step stair test (-12.5%), MFM part D1 (-7.1%), and maximum repetition rate of /pa/ (-5.3%) showed a significant decrease as well (all p<0.05). Domain 'Physical functioning' of the SF-36 Health Survey significantly deteriorated (p=0.044). No relationship was found between disease progression and genotype or disease duration (p>0.05).ConclusionsDespite the slow disease progression of OPMD, this study showed that several outcome measures detected progression within 20 months. The deltoid muscle strength, measured by fixed dynamometry, showed the greatest decline. This longitudinal data provides clinical outcome measures that can be used as biomarkers in future clinical trials.