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Tina S Gambling1 †, Andrew F Long2 1School of Healthcare Sciences, Cardiff University, Cardiff, UK; 2School of Health Care, University of Leeds, Leeds, UK†Tina S Gambling passed away on November 23, 2020.Correspondence: Andrew F Long Email a.f.long@btinternet.comAbstract: Hip dysplasia is a significant public health issue, representing the single largest cause for total hip arthroplasty in young adults. To gain insight into patient concerns and the impact of hip dysplasia on young adults, we developed a patient-centred outcome measure, the ‘Quality of Life Impact and Concerns’ (QoLI&C) measure. This short report provides additional evidence on test–retest reliability and suggests refinements to the measure to enhance its research and clinical utility.Keywords: acetabular dysplasia, developmental dysplasia of the hip, paediatric hip conditions, test–retest reliability, psychometric validation

Andrew F Long,1 Tina Gambling21School of Healthcare, Faculty of Health and Social Care, University of Leeds, Leeds, UK; 2School of Health Sciences, College of Biomedical and Life Sciences, Cardiff University, Cardiff, Wales, UKCorrespondence: Andrew F LongSchool of Healthcare, Health Systems Research, University of Leeds, Baines Wing, Leeds LS2 9UT, UKEmail a.f.long@btinternet.comPurpose: To explore the question of ‘how to evaluate a qualitative patient-centred outcome measure’, comprising predominantly open-ended items, including perhaps emojis, story writing and/or pictures, in a way that does not compromise the strictures of the qualitative paradigm, doing so in a credible and authoritative manner. The paper aims to promote debate and discussion in the measurement validation community.Methods: Comprehensive literature review of three electronic databases (PubMed; SCOPUS; Web of Science/Knowledge) and searches of three outcome-focused journals.Results: The vast majority (>90%) of the papers only used qualitative methods in the initial, in particular, content validation of a measure and then used (quantitative) psychometric validation procedures. The remaining papers comprised articles that were either methodologically or methods focused and the role of qualitative research. A number of key issues are raised, inter alia: giving primacy to the patient’s perspective; exploring the meaning and interpretation respondents place on the concept and possible items in a measure; prioritising maximising meaningful discrimination from the respondent’s perspective; ensuring face and content validity and relevance of items in the item content pool; and using appropriate qualitative methods, for example, concept elicitation, “think-aloud” and cognitive interviews and expert respondent panels/judges. This approach is applied to validate a child-friendly outcome measure for children with Perthes disease, a paediatric hip condition presenting primarily amongst male children aged 5-8 years.Conclusions: The core messages are to: (i) not force validation of a qualitative outcome measure into psychometric validation; but (ii) retain full adherence to the principles of the qualitative paradigm and employ procedures drawn from that paradigm. In this manner, primary emphasis would lie on issues of meaningfulness, face and content validity, the meaning of item and measure scores to respondents and, for a child-friendly measure, the child-friendliness of the measure.Keywords: qualitative validation, outcome measure, psychometric validation, qualitative paradigm, Perthes disease, child-friendly measure

Tina Gambling,1 Andrew F Long21School of Healthcare Sciences, College of Biomedical and Life Sciences, Cardiff University, Cardiff, UK; 2School of Healthcare, University of Leeds, Leeds, UKPurpose: To develop and validate a patient-centered, evaluative outcome measure to assess patient-significant and identified impacts of the three pediatric hip conditions (the “Quality of Life, Concerns and Impact Measure” (QoLC&I)), for use by patients and clinicians in discussions over treatment options and the evaluation of treatment and post-operative rehabilitation.Patients and methods: The measure was developed through a qualitative study, via two web-based forums (patient narratives, n=84) and one specialist orthopedic adult hip clinic (conducting interviews, n=38). The draft (1) measure was piloted in an asynchronous web-based discussion group forum; following revision, it was piloted with a group of clinicians and patients to assess its patient and clinical utility, face and content validity. The final, refined prototype measure (QoLC&I, draft 3) was subjected to psychometric evaluation.Results: A total of 230 patients provided useable data for the psychometric analysis: 70% (160) had a confirmed diagnosis of Developmental Dysplasia of the Hip, 15% (35) Perthes, 11% (26) Slipped Upper Femoral Epiphyses; 4% (9) PHC not stated. The scale showed good acceptability (few missing items, good spread, low floor/ceiling effects), relevance (76% stating they would find the measure useful in their discussions with clinicians), and good internal consistency (Cronbach’s α=0.98; average ICC=0.98). Hypotheses on convergent validity (with the General Health Questionnaire, to measure depression, and the International Hip Outcome Tool Short Form, to measure quality of life) and divergent validity (with the General Self-Efficacy Scale, to measure coping) were confirmed.Conclusion: The 64-item QoL&CI measure is a practical and valid measure addressing areas of clinical and patient significance and has potential value to assist patients and clinicians in discussions about treatment choices and treatment progress. Future research will address further psychometric testing (test–retest validity and responsiveness to change), in additional sites, and embedding the measure into clinical practice.Keywords: Developmental Dysplasia of the Hip, Perthes disease, Slipped Upper Femoral Epiphyses, psychology, osteoarthritis

Objectives:\ud\udSymptomatic developmental dysplasia of the hip can present in the young, active adult patient and can lead to severe pain and disability at a young age. The overall aim of the study was to deepen the understanding of the impact of developmental dysplasia of the hip on young adults’ quality of life and psycho-social well-being, focusing on how differential access to early diagnosis impacted the trajectory of the disease and treatment options.\ud\ud\ud\udMethods:\ud\udWe conducted semi-structured interviews and gathered online stories from 97 participants. A narrative and thematic analysis was used to integrate the dataset producing a multi-dimensional view.\ud\ud\ud\udResults:\ud\udA narrative plot showing how events unfolded over time was identified for each participant. Two common plots were identified: Plot 1 focused around those participants who received a prompt diagnosis and were treated within 12 months of diagnosis (n = 22) and Plot 2 focused around those who experienced a late/delayed diagnosis (mean = 8 years; range = 12–364 months) (n = 75) and thus delayed treatment. Participants in Plot 2 became more debilitated over time and experienced chronic hip pain for a prolonged period before an accurate diagnosis and/or intervention was made. It was clear from the narratives when pain persists over time, participants’ quality of life and psycho-social well-being were affected.\ud\ud\ud\udConclusions:\ud\udDevelopmental dysplasia of the hip is an under-recognised condition; more research is needed to develop a clear clinical picture that can be used to alert frontline health professionals to the potential for developmental dysplasia of the hip so that they can promptly diagnose patients and refer them to specialist centres. Patients can also experience a profound impact on their quality of life and psycho-social well-being. Patients actively seek information from support groups about all aspects of their condition. Further research is required to determine the long-term impact of developmental dysplasia of the hip to develop evidence-based information for clinicians and patients.

Aim. To determine the social, physical, and emotional impact of living with Perthes’ disease on affected children and their family (caregivers). Patients and Methods. Through a mixed methods approach, we interviewed 18 parents and explored the perspectives of 12 children affected by Perthes’ disease (mean = 7.1 years, SD = ±4.1 years) using a survey tool. Thematic analysis of parents’ interviews provided an insight into disease-specific factors influencing patients and family’s daily life activities. Using the childhood survey tool, good and bad day scores were analyzed using MANOVA (multivariate analysis of variance). Results. Thematic analysis of the parent interviews (main themes n = 4) identified a marked effect of the disease on many facets of the child’s life, particularly pain and the impact on sleep, play, and school attendance. In addition, the interviews identified a negative effect on the family life of the parents and siblings. Children indicated that activities of daily living were affected even during “good days” (P < .05), but pain was the key limiting factor. Conclusion. Perthes’ disease negatively affects the social, physical, and emotional well-being of children and their family. These findings provide outcome domains that are important to measure in day-to-day care and add in-depth insight into the challenges caused by this disease for health care professionals involved in clinical management.

Purpose This study aimed to explore the associations between dose-volume parameters of localized breast irradiation, longitudinal interleukin-6 soluble receptor (sIL-6R), and leukocyte counts as markers of an immune-mediated response and fatigue as a centrally-driven behavior. Methods and Materials This prospective cohort study recruited 100 women who were diagnosed with stage 0-IIIa breast cancer, prescribed 40 Gy in 15 fractions over 3 weeks adjuvant radiation therapy, and had no prior or concurrent chemotherapy. Dose-volume parameters were derived from treatment plans and related to serum sIL-6R concentrations, leukocyte counts, and a validated measure of self-reported fatigue at baseline, after 10 and 15 fractions, and 4 weeks after radiation therapy. Results sIL-6R concertation was significantly higher in patients with a total volume of tissue irradiated within the 50% isodose >2040 cm3 (P = .003). When controlling for body mass index, this result only remained significant after treatment. The volume of liver irradiated within the 10% isodose correlated with the sIL-6R concentration during and after radiation therapy (ρ = .3-.4; P = .03-.007). The 38% of the cohort that was classified as fatigued had a higher mean sIL-6sR concentration at all observation points, but the differences were only statistically significant during radiation therapy: Mean (standard deviation [SD]) after 15 fractions for fatigued patients was 47.6 ng/dL (11.2 SD) versus 41.6 ng/dL (11.4 SD) for nonfatigued patients (P = .01). Cohort leukocyte counts and leukocyte subsets decreased consistently from baseline and the values for the fatigued group were 4% lower at baseline and between 7% and 9% lower during and after treatment compared with those of the nonfatigued group but the differences were not statistically significant. Conclusions This is the first study to show that localized irradiation induces increased systemic sIL-6R during treatment in participants who reported elevated levels of fatigue before, during, and after treatment. This behavioral response appears to reflect a variation in innate host immunity, which then mediates the cellular and/or psychological stress of radiation therapy.

Appendix S2. Children’s booklet. (DOCX 242 kb)

Appendix S1. Systematic review data extraction form. (DOCX 20 kb)

Background Behavioural change interventions for persons with chronic illness draw on a variety of theoretical approaches including motivational interviewing and shared decision making. Health literacy provides an additional, potentially powerful explanatory framework to guide research and practice.\ud \ud Objective To examine the changes in the depth and detail of diabetes-related knowledge and confidence for persons with type 2 diabetes.\ud \ud Design Two-year, prospective, observational study, using questionnaire data at two time points (baseline and 2 years later) and in-depth interviews with a theoretically selected subsample.\ud \ud Setting and participants A total of 319 patients initially recruited from a deprived urban area in north-west England\ud \ud Intervention Dedicated tele-carer education and support, tailored to the individual circumstances of the patient.\ud \ud Main outcome measures Perceptions of confidence, levels of empowerment, learning for self-care and most helpful aspects of the intervention.\ud \ud Results Over 90% expressed confidence in keeping their blood sugar controlled, and high levels of perceived empowerment (mean = 4.25; 95% CI, 4.17–4.33) were found. Changes in the depth and detail of diabetes-related knowledge and confidence, from the specific to the more general, were observed and enhanced competence in translating knowledge into practice.\ud \ud Discussion and conclusions The intervention, built within a developed working partnership between tele-carer and patient, operated at two levels: health literacy, enhancing knowledge, developing personal skills and enabling self-control; and socio-psychological behavioural change, tailored to individuals within their socio-economic environments, enabling increased motivation and supportive problem-solving. Both approaches find reflection in the findings and provide powerful explanatory lenses to interrogate the data.