Coeliac disease (CD) is a chronic gastrointestinal condition, the only treatment for which is a gluten-free diet (GFD). Following a GFD is restrictive, burdensome, and can impact health-related quality of life (HRQOL). People with CD can experience long delays to diagnosis and evidence suggests large variations in follow-up care, but the relationship between health care experiences and HRQOL is unknown. The main aim of this research was to develop a patient-reported outcome measure and patient experience questionnaire, and use these to investigate the relationship between adults' experiences of health care services and HRQOL in CD. The questionnaires, named the Coeliac Disease Assessment Questionnaire (CDAQ) and the Coeliac Disease Patient Experience Questionnaire (CD-PEQ), were developed following qualitative interviews with adults with CD, and refined with input from experts, and cognitive interviews. The CDAQ was also subject to a translatability assessment to assess its linguistic and cultural translatability, and a cross-sectional survey to assist with item reduction and scale generation. Members of Coeliac UK (n=267) completed the CDAQ and CD-PEQ, together with the SF-36v2 and demographic questions as part of a postal survey. Psychological health, vitality, general health, and dietary burden were found to have the greatest impact on HRQOL, with physical health and social isolation the least affected. HRQOL was found to have a strong correlation with patients' experiences of health care services. Aspects most strongly related were: the provision of information; communication with HCPs; difficulty obtaining prescriptions; and GPs' knowledge. This research has identified aspects of health care services that are strongly related to HRQOL in CD. Health care providers are recommended to focus service improvement efforts on these areas. A reliable and valid disease-specific patient-reported outcome measure and patient experience questionnaire have been developed as part of this study. The CDAQ is suitable for use in research studies, including clinical trials, to assess HRQOL in CD.

Research elucidating high rates of burnout, depression, and suicide among US clinicians has caused national concern for the sustainability of our healthcare workforce and the quality of patient care. In response, US healthcare organizations are using measures of clinician burnout in new contexts beyond their traditional use in research. Outcome measures of clinician burnout are being used to evaluate health system performance, identify demographic disparities, and educate individual clinicians regarding their own outcomes. Yet, critical gaps in the literature exist regarding the measurement properties of burnout assessments in these contexts and the relationship between clinician distress and healthcare quality. This dissertation contains three studies on measuring clinician distress and its relationship with healthcare quality. Studies 1 and 2 aim to advance what is known regarding the interpretability, reliability, and validity of a commonly used clinician burnout assessment, the Maslach Burnout Inventory-Human Services Survey for Medical Personnel (MBI-HSS). In Study 1, we aimed to improve the interpretation of the MBI-HSS by using item response theory to describe the burnout symptoms and precision associated with MBI-HSS scores in US physicians. We produced response profiles that allow health policy makers and healthcare leaders to relate actionable, qualitative meaning regarding individuals’ and groups’ burnout symptom burden to the MBI-HSS’s quantitative subscale scores. In Study 2, we examined whether demographic disparities in US physician burnout are explained by differences in the MBI-HSS’s functioning across physician age, gender, and specialty groups. Our findings revealed that differences in the MBI-HSS’s functioning across age, gender, and specialty groups did not account for observed disparities, supporting the use of the MBI-HSS as a valid tool for identifying demographic disparities in physician burnout. In Study 3, we examined the association of clinician depression, anxiety, and burnout with the inappropriate use of antibiotic prescriptions for acute respiratory tract infections (RTIs) in a retrospective cohort study of outpatient visits at Boston Medical Center. We found a significant positive association between clinician depression, anxiety, and burnout and inappropriate prescribing for acute RTIs, which depended on the visit location and diagnosis group. Our findings suggest that clinician depression, anxiety, and burnout may play an important role in the quality of routine outpatient care. As federal agencies and healthcare organizations seek to address clinician distress on local and national levels, our findings offer important implications for future assessment and intervention. / 2023-01-19T00:00:00Z

Bakgrund: Bröstcancer-relaterat lymfödem (BCRL) är en vanlig komplikation efter bröstcancerbehandling. Tidigare forskning visar varierande förekomst av BCRL, tillståndet tros vara underdiagnostiserat. Tidiga tecken kan beskrivas som en vag tyngdkänsla och/eller spänningskänsla. Detta kan leda till stora negativa konsekvenser vilket motiverar vikten att detektera BCRL i ett tidigt stadie då tidig behandling förbättrar prognosen.  Syfte: Undersöka förekomst av tidiga besvär/symtom samt undersöka sambandet mellan patientrapporterade tidiga besvär/symtom och läkarens bedömning av besvär vid samma tillfälle.  Metod: Kvinnor som genomgått kurativ behandling mot bröstcancer deltog i studien. Datainsamlingen skedde vid återbesöket på onkologmottagningen 3 månader efter avslutad behandling. Data samlades in med patientenkät (avseende besvär/symtom från armen) och läkarbedömning, inom ramen för ett kliniskt utvecklingsprojekt.   Resultat: 61 enkäter ingick i studien. 22 deltagare upplevde besvär från armen på den opererade sidan. 70% av de 43 deltagarna som skattade symtom angav förekomst av ≥1 symtom. I 76% av de 55 läkarbedömningarna hade bedömningen inga besvär gjorts. Samband kunde påvisas gällande frekvensen av patientrapporterade besvär och läkarens bedömning av armsvullnad (p=0,005) och handödem (p=0,003) samt mellan patientrapporterade symtom och läkarens bedömning av armsvullnad (p=0,008). I de fall patienten skattat besvär/symtom fanns en signifikant andel där läkaren bedömt att det inte förekom ödem/svullnad. I de fall patienten inte rapporterade besvär hade läkaren också bedömt frånvaro av ödem/svullnad. Konklusion: Det är vanligt att patienten upplever besvär/symtom från armen på den opererade sidan, och att patienter upplever symtom i större utsträckning än att läkaren gjort bedömningen att handödem, subcutant ödem eller armsvullnad förekommer. / Background: Breast cancer-related lymphedema (BCRL) is a complication following breast cancer treatment, it is probably underdiagnosed and the prevalence varies in previous studies. Early signs can be subtle sensations of heaviness and/or tightness in limbs. BCRL can have several negative impact, this motivates the importance of early detection because early intervention improves the prognosis.   Aim: Investigate the prevalence of early signs/symtoms of BCRL and investigate the association between patient reported outcomes and the physician assessment of BCRL.  Method: Women who underwent curative treatment of breast cancer was included. Data was collected at the follow-up 3 months after finished treatment in purpose for a clinical project. A questionnaire was used for the patient (regarding signs/symtoms from the arm) and physician rescpectively. Results: 61 participants were included. 22 participants reported signs from the arm on the affected side. 70% of those who reported symptoms, reported a presence of ≥1 symtoms. The physicians estimated no signs in 76% of 55 participants. There was an association between the frequency of patient reported signs from the arm and the physicians assessment of armswelling (p=0,005), hand edema (p=0,003) and between patient reported symtoms and the physicians assessment of armswelling (p=0,008). When the patient reported signs/symptoms, there was a significant proportion where the physician estimated no edema/swelling. In cases when the patient reported no signs/symptoms, the physician also assessed absence of edema/swelling. Conclusion: Patients often experience signs/symptoms from the arm on the affected side, and patients reports symptoms more frequent than physicians assess presence of early signs of BCRL.

Tradition measures of recovery, such as patient-reported symptoms, objective measures such as balance, specific dimensions such as depression, fatigue, cognitive status, and exercise tolerance do not fully capture the impact of the concussion on performing individual activities and participating in life situations as experienced by the patient. No concussion-specific measure of functional status currently exists. Objectives The overarching purpose of this dissertation was to develop a concussion-specific measure of functional status. There were two specific objectives: 1. To examine the concept of functioning post-concussion; 2. To generate questionnaire items based on a conceptual model of functioning. Methods This dissertation follows the recommendations of the Association for Medical Education in Europe as a framework with which to meet the objectives. The first objective was addressed by (1) generating a list of concussion-specific concepts through a systematic review (Chapter 3), and (2) qualitative interviews with individuals with persistent post-concussion symptoms and clinicians with concussion expertise (Chapter 4). The relationships between the concepts that emerged from those studies are presented graphically in a conceptual model to meet the second objective. The concepts were then transformed into questionnaire items and pretested through cognitive interviews with individuals with PPCS and clinicians with concussion expertise. Finally, the questionnaire items were critically evaluated for proportion of shared content against existing measures used in concussion clinical trials by coding all items to the International Classification of Functioning, Disability and Health. Results Objective 1 Three main themes emerged from the qualitative findings: (1) functioning at the level of the individual and society; (2) environmental barriers and facilitators; and (3) capacity, defined as the length of time one could perform a task before the onset of symptoms, and the length of time it took to recovery from those symptoms. Objective 2 The final questionnaire is presented as the CORE-Q, which is comprised of 53 items over three complimentary subscales, namely the Post-Concussion Functional Scale, the Concussion Modifiers Scale, and the Global Functional Recovery Scale. Each subscale corresponds to one of the three main themes. No existing outcome measure contained more than 40% of the content within the CORE-Q, or 55% of any subscale. Conclusions The CORE-Q is a unique measure of functional status post-concussion that considers functioning from a biopsychosocial perspective. Further studies are needed to assess the psychometric properties of the CORE-Q before it is adopted into clinical practice and intervention trials.

CHAPTER 1: Introduction: The most common craniofacial congenital anomaly is the cleft lip and/or palate (CLP). The CLEFT-Q is the first condition-specific comprehensive patient reported outcome instrument (PROM) for patients with CLP. Other measures used in assessment of patients with CLP are Child Oral Health Impact Profile (COHIP) and Cleft Hearing, Appearance and Speech Questionnaire (CHASQ). The development and validation of the CLEFT-Q have been completed in three phases. In phase I, 138 patients with CLP from six countries were interviewed, and data were used to form 13 scales measuring appearance, facial function and health-related quality of life (HR-QOL). In phase II, scales were field-tested internationally with 2434 patients to examine reliability and validity as well as develop a common scoring algorithm for international use. Phase III, the focus of this thesis, aimed to examine further construct validity and responsiveness of the CLEFT-Q scales. CHAPTER 2: Methods: Patients were recruited at six cleft centres in Canada, USA and UK between January 2018 and October 2019. The sample included patients aged 8-29 seeking rhinoplasty, orthognathic, cleft lip scar revision and alveolar bone graft (ABG) operations. Before and six months after surgery, participants were asked to complete the CLEFT-Q scales relevant to their operation and two other PROMs frequently used in cleft research, i.e., COHIP and CHASQ. Cross-sectional construct validity was examined by testing prespecified hypotheses about expected relationships between CLEFT-Q, CHASQ and COHIP instruments. Internal responsiveness was examined using the distribution-based method. Data were analysed using paired sample t-tests and calculation of effect sizes (ESs) and minimally important differences (MIDs). CHAPTER 3: Results: Examination of cross-sectional construct validity of the CLEFT-Q scales using the COHIP and CHASQ subscale resulted in 39/53 (74%) hypotheses having been supported by the results. The required sample size to examine responsiveness using the anchor-based approach was not reached. Assessment of internal responsiveness using the distribution-based approach demonstrated that the appearance scales were highly responsive to change following cleft-specific surgeries, with statistically significant results and ESs ranging from 0.4 (small) to 1.8 (large). Change on the CLEFT-Q HR-QOL scales was not statistically significant. As predicted, the ESs on scales measuring facial aspects most affected by rhinoplasty and orthognathic surgeries were larger than the ESs on scales measuring facial aspects least affected by these surgeries. MIDs for each scale in each operation were determined. CHAPTER 4: Discussion: Assessment of cross-sectional construct validity demonstrated that CLEFT-Q performs as it was intended when compared with other similar measures (i.e., CHASQ and COHIP). The CLEFT-Q appearance scales were responsive to change following rhinoplasty, orthognathic and cleft lip scar revision operations. As predicted, the CLEFT-Q appearance scales did not detect change following the ABG operation as this operation does not result in visible difference. As hypothesized, the CLEFT-Q HR-QOL scales were less responsive to change than appearance scales as HR-QOL is a more distal construct than appearance in relation to the cleft-related surgeries performed. The preliminary MIDs estimated by the distribution-based approach should be confirmed in studies with diverse CLP populations and larger sample sizes using the anchor-based approach. The findings of this phase III study build on the results of another CLEFT-Q validation study, which demonstrated the ability of the CLEFT-Q scales to detect differences between groups with varying surgical status, i.e., need surgery, have had surgery and never needed surgery. CHAPTER 5: Conclusion: Cross-sectional construct validity of the CLEFT-Q scales was supported by most prespecified hypotheses. The CLEFT-Q scales were found to be responsive to change. MIDs were determined. The results of this phase III study should be confirmed in a larger and more culturally diverse patient population. Future studies to examine reproducibility and measurement error as well as external responsiveness of the CLEFT-Q scales may be beneficial to add to the psychometric evaluation process. / Thesis / Master of Science (MSc)

京都大学 / 新制・課程博士 / 博士(医学) / 甲第23790号 / 医博第4836号 / 新制||医||1057(附属図書館) / 京都大学大学院医学研究科医学専攻 / (主査)教授 山本 洋介, 教授 佐藤 俊哉, 教授 柳田 素子 / 学位規則第4条第1項該当 / Doctor of Medical Science / Kyoto University / DFAM

Background: Amyotrophic Lateral Sclerosis (ALS) is a fatal neurodegenerative disease characterized by the loss of motor neurons. Preference-based measures (PBMs) of health-related quality of life (HRQL) can be utilized for cost-effectiveness analyses of interventions in individuals with ALS. However, current measures are generic (GPBMs) and the psychometric properties of these measures have not yet been evaluated in ALS. Purpose: The purpose of this thesis was to evaluate the psychometric properties of GPBMs in ALS by 1) conducting a systematic review of the psychometric properties of GPBMs, and 2) assessing the content and convergent validity of GPBMs in ALS. Methods: Two studies were conducted. First, a systematic review was performed, and four databases were searched to identify studies that used and reported on the psychometric properties of GPBMs in ALS. Second, participants were recruited from three clinical sites across Canada and outcome measures were administered through an online or hardcopy survey. Areas of importance to the HRQL of individuals with ALS were identified using the Patient Generated Index (PGI), mapped against GPBMs to determine their coverage and scores were compared to determine convergent validity. Results: For the first study, the EQ-5D-3L was found to be the most commonly used GPBMs in ALS. It demonstrated convergent and known-groups validity however, significant floor effects were observed. For the second study, results indicated that the majority of GPBMs identified approximately half of the areas impacted by ALS. In addition, there were several domains not identified by GPBMs. Conclusion: This thesis highlights the importance of complete psychometric evaluation of measures in ALS. There is the need for the development of an ALS specific preference-based measures that reflects the health concerns of individuals with ALS; as GPBMs used in ALS were evaluated and deemed to be lacking in support for their usage in ALS. / Thesis / Master of Science Rehabilitation Science (MSc) / Amyotrophic Lateral Sclerosis (ALS) is a fatal disease that causes individuals to lose their strength and eventually the ability to speak, eat, move and breathe. Questionnaires can be used to understand the health-related quality of life (HRQL) of individuals with ALS however these measures do not always reflect the experiences of these individuals. The goal of this dissertation was to identify whether measures truly capture areas important to individuals with ALS. In our studies, we found that there is little proof in the accuracy of measures used. In addition, the measures do not fully capture the areas of life important to individuals with ALS. This is important to help researchers and health care professionals understand the effects of ALS on HRQL. These results will help them determine which treatments are worthwhile and the best to use in practice and provide recommendations for future research.

Measurement Based Care (MBC) is considered to be an evidence-based practice. Despite its well-documented efficacy, it is underutilized in the clinical community for various reasons, including clinician and patient buy-in. A key component to the successful implementation of MBC is the routine administration of Patient Reported Outcome Measures (PROMs). There is a lack of research describing the utilization of MBC in a telehealth setting. As technological innovations continue, a greater number of clinics are offering telemental health services. Additionally, the COVID-19 pandemic caused a majority of underprepared clinics to begin implementing telehealth. The present study sought to evaluate patient compliance with PROMs in MBC after an abrupt shift to telehealth due to the COVID-19 pandemic. Participants were collected from a clinical population at a community based psychological training clinic. The participants were separated into groups: modality 1 (in-person services, n = 17), modality 2 (telehealth services, n = 17), and modality 3 (hybrid of modalities 1 and 2, n = 10), to assess the effect of modality on mean PROM compliance. The participants were separated into groups adult (≥18 years of age, n = 23) and child (<18 years of age, n = 17), to assess the effect of maturity on mean PROM compliance. Results showed that mean PROM compliance was significantly higher in the in-person modality than the telehealth modality. Results also showed that PROM compliance was significantly higher in adults than in children. There was not a significant interaction effect of modality and maturity on PROM compliance. Additionally, results showed that PROM compliance decreased significantly after the switch from in-person services to telehealth services in the hybrid modality. These findings show that clinics may face significant barriers to the implementation of MBC after a sudden shift to telehealth caused by an unforeseen stressful event. / M.S. / Measurement-Based Care (MBC) is an effective practice for the treatment of patients in psychological practices. It is a collaborative process that involves the clinician and patient tracking treatment progress and outcomes through the use of consistently administered measures known as Patient Reported Outcome Measures (PROMs). Although MBC has been shown to be effective, there is little literature regarding its use in a telehealth setting. As a greater number of clinics begin offering telehealth services, questions regarding patient adherence to interventions have arisen. Furthermore, the global COVID-19 pandemic forced a majority of underprepared clinics to offer telehealth services. The current study sought to better understand potential barriers to the implementation of MBC in a telehealth setting. Participants were collected from patients receiving therapy at a community based psychological training clinic. The participants were separated into the groups: in-person services (n = 17), telehealth services (n = 17), and hybrid of in-person and telehealth (n = 10), to assess the effect of modality on mean PROM compliance. The participants were separated into groups adult (≥18 years of age, n = 23) and child (<18 years of age, n = 17), to assess the effect of maturity on mean PROM compliance. Results showed that patients in the in-person therapy group were more likely to complete their measures than patients in the telehealth therapy group, regardless of their maturity. Additionally, adult patients were more likely to complete their measures than child patients, regardless of the modality. Results also showed that patients who experienced both in-person and telehealth services were more likely to complete their measures before the switch to telehealth. These findings show that clinics may face significant barriers to the implementation of MBC after a sudden shift to telehealth caused by an unforeseen stressful event.

Introduction: Conversation treatment for individuals with aphasia (IwAs) aims to enhance language and communication skills within naturalistic settings. Group dynamics, including psychosocial support (PSS) and vicarious learning (VL), are important components of this treatment. However, the lack of established aphasia-friendly methods for quantifying group dynamics impedes understanding of its contribution to treatment efficacy. This study addresses three key research goals: (1) Can group dynamics be reliably quantified? (2) Do measures of group dynamics vary with group size or time? (3) Are group dynamics linked to changes in self-reported psychosocial health measures? Methods: IwAs were assigned to participate in either a large group or dyadic condition as part of a larger study. Participants completed multiple standardized assessments before and after receiving conversation treatment for one hour twice a week for ten weeks. A coding system was developed to track PSS and VL during sessions. Video recordings of sessions at the start (Session 2) and end (Session 19) of the treatment period were analyzed for five large groups and six dyads, with seven additional dyads coded for Session 19 after reliability was confirmed. Intrarater and interrater reliability were assessed by recoding 21% of the videos randomly. Results: To address the first research goal, group dynamics were defined and then a code was created to represent occurrences of PSS and VL. After establishing a reliable coding system, instances of PSS and VL were compared across size conditions to address the second research goal. The results suggested that PSS differed between conditions later in treatment, but VL did not. For research goal three, the data suggest that large groups experience larger gains in quality of life measures, possibly due to increased exposure to PSS. Conclusion: Findings indicate that group dynamics can be reliably tracked and used for quantitative analysis. PSS was more common in groups than dyads later in the 10 week treatment period, but VL did not vary across conditions or at the start or end of treatment. Further, there is some evidence that PSS in larger groups contributes to improvements in quality of life measures. / Communication Sciences

Yes / Purpose: To develop and validate the first patient-reported outcome measure (PROM) to quantify vision-related dizziness. Dizziness is a common, multifactorial syndrome that causes reductions in quality of life and is a major risk factor for falls, but the role of vision is not well understood. Methods: Potential domains and items were identified by literature review and discussions with experts and patients to form a pilot PROM, which was completed by 335 patients with dizziness. Rasch analysis was used to determine the items with good psychometric properties to include in a final PROM, to check undimensionality, differential item functioning, and to convert ordinal questionnaire data into continuous interval data. Validation of the final 25-item instrument was determined by its convergent validity, patient, and item-separation reliability and unidimensionality using data from 223 patients plus test–retest repeatability from 79 patients. results: 120 items were originally identified, then subsequently reduced to 46 to form a pilot PROM. Rasch analysis was used to reduce the number of items to 25 to produce the vision-related dizziness or VRD-25. Two subscales of VRD-12-frequency and VRD-13-severity were shown to be unidimensional, with good psychometric properties. Convergent validity was shown by moderately good correlations with the Dizziness Handicap Inventory (r = 0.75) and good test–retest repeatability with intra-class correlation coefficients of 0.88. conclusion: VRD-25 is the only PROM developed to date to assess vision-related dizziness. It has been developed using Rasch analysis and provides a PROM for this under-researched area and for clinical trials of interventions to reduce vision-related dizziness. / College of Optometrists (UK) research studentship.