Severe perineal trauma with injury to the anal sphincter at childbirth can have a profound effect on the physical and psychological wellbeing of women. This thesis describes literature examining resulting outcomes including effects on body image. It describes patient-based outcome measures used to capture this information, and evaluation of their psychometric properties. Body Image can be defined as an individual's perceptions and feelings about their own body. There is a growing interest in how this concept can influence quality of life and psychosocial dysfunction in medical disorders. This thesis aimed to examine relationships between severity of perineal trauma, general and genital specific body image and potentially influences such as symptoms of pelvic floor dysfunction. No patient-reported outcome measure validated for use in women after anal sphincter injury exists and this thesis also aimed to psychometrically evaluate an existing electronic questionnaire, ePAQ (electronic personal assessment questionnaire), for this application. In the thesis, a retrospective review of body image and physical outcomes attending a perineal clinic shows over half of women report perceived changes in body image after anal sphincter injury, with negative effects on self-esteem. A prospective observational cohort study explored genital and general body image in primiparous women grouped according to degree of perineal trauma or caesarean delivery. Women completed the Female Genital Self Image Score, the modified Body Image Score, ePAQ and the Edinburgh Postnatal Depression Scale a mean of 15.5 weeks (standard deviation 1.6) after delivery. There were significant differences in genital body image scores between the groups, but not in general body image, with regression analysis showing the greatest influence on genital body image to be the anatomical extent of the trauma. Embedded into this study was the evaluation of reliability (internal consistency and test-retest) and validity (face, content and construct) of ePAQ in the group of women with anal sphincter tears. This thesis presents the first research to quantify issues surrounding severe perineal trauma and body image and demonstrates that more severe trauma leads to a poorer genital body image. It also reports psychometric evaluation of ePAQ in women after anal sphincter injury providing the first single instrument with validity and reliability for use in this context.

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.

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)

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.

no / Background Improving cognition is service users' top research priority for life after stroke, and future research should include outcomes that they deem important. Patient perspectives on outcomes are collected using patient-reported outcome measures (PROMs). There is currently no patient-centred PROM specific for cognitive rehabilitation trials. Objective Inform PROM development by exploring stroke survivor perspectives on the important, measurable impacts of persisting post-stroke cognitive problems. Design Qualitative semi-structured interviews in participants' homes. Participants Purposive sample of 16 cognitively impaired stroke survivors at least six months post-stroke. Methods Interviews used a schedule and communication aids developed through patient consultation. Interviews were transcribed verbatim with non-verbal communication recorded using field notes. Data were analysed using a framework approach to find commonalities to shape the focus and content of an outcome measure. Results Participants identified important impacts of their ‘invisible’ cognitive problems, outside of other stroke-related impairments. Cognitive problems exacerbated emotional issues and vice versa. Changes in self-identity and social participation were prominent. Impact was not spoken about in terms of frequency but rather in terms of the negative affect associated with problems; terms like ‘bothered’ and ‘frustration’ were often used. Conclusions The results support the development of a PROM specifically designed to address the impact of cognitive problems. It should: include items addressing a comprehensive range of cognitive skills; ask questions about mood, self-identity and social participation; use accessible wording that respondents understand and endorse; measure impact rather than frequency; and explore perceived impact on carers.

Cognitive difficulties can persist for months and years after stroke and adversely impact confidence, mood and functional recovery. Stroke survivors, carers and healthcare professionals collectively agree that improving cognition is the number one research priority for life after stroke. Future research should include measurements of outcome that service users deem important. Patient reported outcome measures (PROMs) are a means of gaining patient perspectives that can be standardised for use in a trial. PROMs should be developed with service users to incorporate their priorities but people with cognitive difficulties are often systematically excluded from the development and use of PROMs. Study 1 used qualitative interviews (N=16) to explore stroke survivor perspectives on the important and measureable impacts of persisting cognitive problems. The results of this study generated requirements for a PROM that related to conceptual underpinning and face validity of a measurement tool. Study 2 was a systematic review of existing PROMs related to cognition. 20 Identified PROMs were critically appraised against the requirements generated in the qualitative study. No existing PROMs were identified that met all of the qualitative study review criteria. The next stage described in chapter 3, was to develop a new PROM that: utilised the strengths of existing tools; met qualitative study requirements; and was refined through consultation with different stakeholders, prioritising feedback of stroke survivors with cognitive difficulties. The result of this work was the Patient Reported Evaluation of Cognitive State (PRECiS) scale. Study 3 was a psychometric study with stroke survivors (N=164) to test PRECiS in a large sample. Quantitative and qualitative data were collected on acceptability, feasibility and other psychometric properties of validity and reliability. PRECiS demonstrated good acceptability to stroke survivors and performed well psychometrically. Future validation work required for PRECiS is described in discussion chapter 4. Subject to further validation work, PRECiS may be particularly useful for pragmatic trials of cognitive rehabilitation after stroke.

While the importance of assessing the occupational consequences of insomnia and other sleep disorders is emphasised in clinical nosologies and research guidelines, there is little consensus on which aspects of occupational performance should be assessed, how such impairment should be measured, and how outcomes should be reported. The research programme described in this thesis aimed to address this issue. Chapter 1 presents a systematic review and methodical critique of studies reporting those aspects of occupational performance most impacted by (or most frequently associated with) insomnia symptoms and degraded sleep quality. Equivocal results, wide variations in reporting conventions, and the overall lack of comparability among studies, strongly indicated the need to develop a standardised metric able to quantify sleep related occupational performance and serve as an assessment and outcome instrument suitable for use in research and clinical settings. Informed by the literature review, Chapters 2-4 describe the development and validation of the Loughborough Occupational Impact of Sleep Scale ( LOISS ), a unidimensional 19 item questionnaire that captures sleep-related occupational impairment across a number of workplace domains over a 4-week reference period. Chapters 5-7 describe LOISS outcomes from: i) surveys in a random population sample; ii) a representative sample of the UK workforce; and iii) a clinical sample of patients with obstructive sleep apnoea (before and after treatment with CPAP). Overall, the scale showed strong internal consistency (Cronbach s alpha range=0.84-0.94) and test-retest reliability (r=0.77, r2=0.59, p<0.001), high levels of criterion validity (significantly discriminating between good and poor sleepers), and proved an effective outcome measure in OSA. From the survey data reported in Chapters 2-7, LOISS score distributions showed no consistent gender difference but did show a significant ageing gradient, with sleep-related occupational impairment declining with increasing age. In conclusion, the work presented here supports the usability, validity and reliability of the LOISS as an assessment and outcome instrument, and also demonstrates the utility of this instrument in exploring the dynamics of sleep-related occupational performance

Context: Medical management for head and neck cancer (HNC) often includes neck dissection surgery, a side effect of which is shoulder dysfunction. There is no consensus for which patient-reported outcome measure (PRO) is most appropriate to quantify shoulder dysfunction in this population. Objective: The aims of this research study were to: (1) use Rasch methodologies to assess construct validity and overall appropriateness of test score interpretation of Disability of the Arm, Shoulder and Hand (DASH), QuickDASH, Shoulder Pain and Disability Index (SPADI) and Neck Dissection Impairment Index (NDII) in the HNC population; (2) determine appropriateness of use of University of Washington Quality of Life (UW-QoL) shoulder subscale as a screening tool for shoulder impairment; (3) recommend a new PRO, or combination of PROs, that more accurately portrays the construct of shoulder dysfunction in the HNC population. Design: One hundred and eight-two individuals who had received a neck dissection procedure within the past 2 weeks to 18 months completed the PROs. Rasch methodologies were utilized to address the primary aim of the study through consideration of scale dimensionality [principal components analysis, item and person fit, differential item functioning (DIF)], scale hierarchy (gaps/redundancies, floor/ceiling effects, coverage of ability levels), response scale structure, and reliability (person and item reliability and separation statistics). The secondary aim was addressed through correlational analysis of the UW-QoL (shoulder subscale), DASH, QuickDASH, SPADI and NDII. Results: The DASH did not meet criteria for unidimensionality, and was deemed inappropriate for utilization in this sample. The QuickDASH, SPADI and NDII were all determined to be unidimensional. All scales had varying issues with person and item misfit, DIF, coverage of ability levels, gaps/redundancies, and optimal rating scale requirements. The NDII meets most requirements. All measures were found to meet thresholds for person and item separation and reliability statistics. The third aim of this study was not addressed because the NDII was determined to be appropriate for this population. Conclusions: Rasch analysis indicates the NDII is the most appropriate measure studied for this population. The QuickDASH and SPADI are recommended with reservation. The DASH and the UW-QoL (shoulder subscale) are not recommended.

The rate of Emergency Department (ED) visits due to mental health and drug abuse among children and youth has been increasing for more than a decade and is projected to become the leading cause of ED visits. Identifying high-risk patients well before an ED visit will enable mental health care providers to better predict ED resource utilization, improve their service, and ultimately reduce the risk of a future ED visit. Many studies in the literature utilized medical history to predict future hospitalization. However, in mental health care, the medical history of new patients is not always available from the first visit and it is crucial to identify high risk patients from the beginning as the rate of drop-out is very high in mental health treatment. In this study, a new approach of creating a text representation of questionnaire data for deep learning analysis is proposed. Employing this new text representation has enabled us to use transfer learning and develop a deep Natural Language Processing (NLP) model that estimates the possibility of 6-month ED visit among children and youth using mental health patient reported outcome measures (PROM). The proposed method achieved an Area Under Receiver Operating Characteristic Curve of 0.75 for classification of 6-month ED visit. In addition, a novel method was proposed to identify the words that carry the highest amount of information related to the outcome of the deep NLP models. This measurement of word information using Entropy Gain increases the explainability of the model by providing insight to the model attention. Finally, the results of this method were analyzed to explain how the deep NLP model achieved a high classification performance. / Dissertation / Master of Applied Science (MASc) / In this document, an Artificial Intelligence (AI) approach for predicting 6-month Emergency Department (ED) visits is proposed. In this approach, the questionnaires gathered from children and youth admitted to an outpatient or inpatient clinic are converted to a text representation called Textionnaire. Next, AI is utilized to analyze the Textionnaire and predict the possibility of a future ED visit. This method was successful in about 75% of the time. In addition to the AI solution, an explainability component is introduced to explain how the natural language processing algorithm identifies the high risk patients.