Making sense of narcolepsy: A qualitative exploration of how persons with narcolepsy perceive symptoms and their illness experience.

INTRODUCTION: Understanding how persons with narcolepsy conceptualize symptoms, daily impact and illness experience is key to facilitating dialogue between patients and healthcare professionals. These concepts are usually explored from the perspective of healthcare professionals/researchers and rarely from the perspective of those with narcolepsy. METHODS: 127 self-reported persons with narcolepsy were recruited from an Australian patient support group. A short demographic survey was completed. All agreed to participate in a subsequent 1:1 semi-structured interview. Saturation was reached after 24 interviews (mean age = 33 years (SD 11) with 44% reporting cataplexy). A multidisciplinary team of researchers/clinicians analyzed interview transcripts using thematic analysis. RESULTS: Participants perceived physical fatigue, sleepiness, and two separate experiences of 'falling asleep/sleep attacks' as distinct symptoms rather than a multidimensional construct (i.e. excessive daytime sleepiness). We also identified two experiences of cataplexy, one triggered by acute emotion and another by a stressor. Participants determined their narcolepsy to be 'well-managed' by the level of functional impairment rather than the frequency of any symptom. Almost all participants described experiencing anticipated stigma and internalized or 'self-' stigma, likely stemming from societal devaluation of sleep and the conflation of sleepiness with laziness. CONCLUSION: Descriptions of common symptoms often differed between participants and the existing literature. These differences likely impact patient-physician communication, with both parties utilizing the same terminology to communicate different concepts. The characterization of stigma in narcolepsy presents opportunities for future research exploring the impact and possible development of interventions to reduce the substantial psychological comorbidity in persons with narcolepsy.

A qualitative exploration of the lived experience of mothers caring for a child with narcolepsy.

STUDY OBJECTIVES: Parents/carers of a child with narcolepsy (CwN) are often required to become an expert in narcolepsy and navigate healthcare, education, and welfare systems on behalf of their child. Managing pediatric narcolepsy is complex and challenges the child and the entire family, yet few studies have explored carers' experiences. METHODS: Twenty mothers (50% had a CwN <18 years at the time of interview; 85% NT1) participated in a 1:1 semi-structured interview. Participation from fathers was sought; however, none were recruited. A multidisciplinary team of researchers/clinicians analyzed interview transcripts using thematic analysis. RESULTS: Mothers perceived that most people misunderstood the whole-person impact of narcolepsy, including their child's peers, teachers, and support networks. Narcolepsy had a substantial psychological impact on both the child and the whole family, yet was largely unaddressed by healthcare professionals, leaving mothers unsure of where to turn for help. Most parents described negative experiences with their child's specialist, often perceiving the specialists to lack knowledge specific to narcolepsy. Information about illness trajectory and support services was limited or inaccessible, fuelling many mothers' hopes and fears for their child's future. Mothers also frequently described feelings of abandonment by the healthcare system. CONCLUSIONS: Our results contextualize the whole-person impact of narcolepsy from the perspective of parents and carers, highlighting the need for proactive inclusion of parents/carers in developing healthcare policy and practice. It calls for developing tools and resources to capture 'well-managed' narcolepsy from the perspective of parents/carers for use in research and clinical practice.

A systematic review of the print media representation of ketamine treatments for psychiatric disorders.

BACKGROUND: Public and patient expectations of treatment influence health behaviours and decision-making. AIMS: We aimed to understand how the media has portrayed the therapeutic use of ketamine in psychiatry. METHOD: We systematically searched electronic databases for print and online news articles about ketamine for psychiatric disorders. The top ten UK, USA, Canadian and Australian newspapers by circulation and any trade and consumer magazines indexed in the databases were searched from 2015 to 2020. Article content was quantitatively coded with a framework encompassing treatment indication, descriptions of prior use, references to research, benefits and harms, treatment access and process, patient and professional testimony, tone and factual basis. RESULTS: We found 119 articles, peaking in March 2019 when the United States Food and Drug Administration approved esketamine. Ketamine treatment was portrayed in an extremely positive light (n = 82, 68.9%), with significant contributions of positive testimony from key opinion leaders (e.g. clinicians). Positive research results and ketamine's rapid antidepressant effect (n = 87, 73.1%) were frequently emphasised, with little reference to longer-term safety and efficacy. Side-effects were frequently reported (n = 96, 80.7%), predominantly ketamine's acute psychotomimetic effects and the potential for addiction and misuse, and rarely cardiovascular and bladder effects. Not infrequently, key opinion leaders were quoted as being overly optimistic compared with the existing evidence base. CONCLUSIONS: Information pertinent to patient help-seeking and treatment expectations is being communicated through the media and supported by key opinion leaders, although some quotes go well beyond the evidence base. Clinicians should be aware of this and may need to address their patients' beliefs directly.

Evaluation of psychometric properties of patient-reported outcome measures frequently used in narcolepsy randomized controlled trials: a systematic review.

STUDY OBJECTIVES: To systematically determine subjective and objective outcome measures used to measure the efficacy of narcolepsy interventions in randomized controlled trials (RCTs) in adults and children and assess psychometric properties of patient-reported outcome measures (PROMs) used. METHODS: We searched bibliographical databases and clinical trial registries for narcolepsy RCTs and extracted objective and subjective outcome measures. If PROMs were used, we searched for psychometric studies conducted in a narcolepsy population using bibliographical databases and appraised using Consensus-based Standards for the Selection of Health Measurement Instruments (COSMIN) guidelines. RESULTS: In total, 80 different outcome measures were used across 100 RCTs. Epworth Sleepiness Scale (ESS) (n = 49) and Maintenance of Wakefulness Test (n = 47) were the most frequently used outcome measures. We found 19 validation studies of 10 PROMs in narcolepsy populations. There was limited evidence for validity or responsiveness of the ESS; yet sufficient reliability (pooled ICC: 0.81-0.87). Narcolepsy Severity Scale (NSS) had sufficient reliability (pooled ICC: 0.71-0.92) and both adult and pediatric versions had sufficient discriminant validity (treated/untreated). Content validity was only evaluated in pediatric populations for ESS-CHAD and NSS-P and rated inconclusive. Quality of evidence of the psychometric studies for all scales ranged from very low to low. CONCLUSIONS: Although recognized by regulatory bodies and widely used as primary outcome measures in trials, there is surprisingly little evidence for the validity, reliability, and responsiveness of PROMs frequently used to assess treatment efficacy in narcolepsy. The field needs to establish patient-centered minimal clinically important differences for the PROMs used in these trials.

How patient centric is health policy development? The case of the Parliamentary Inquiry into Sleep Health Awareness in Australia.

Objective Parliamentary inquiries into health-related issues empower everyday Australians to contribute to the development and reform of health policy. We explored how patient and family/carers concerns are translated by this process, using a less well-known disease, narcolepsy, as an example. Methods Written submissions made to the Parliamentary Inquiry into Sleep Health Awareness in Australia 2018 by self-identified patients or family/carers with narcolepsy (n = 13) were extracted and thematically analysed using the Framework Approach. Each submission was systematically coded and abstracted into emergent themes before being evaluated against the final policy recommendations. Results Although patients and their family/carers prioritised issues that affected their daily lives (i.e. mental health sequela, workplace accommodations), the policy recommendations in the report focused mainly on issues of healthcare infrastructure, funding and engagement. Our analysis highlighted several barriers that patients and their family/carers face when contributing to this part of healthcare policy formation. Conclusions Our findings suggest that the parliamentary inquiry process in its current form is not an ideal vehicle by which patients and family/carers can contribute or influence healthcare policy. Despite calling for submissions from patients and their family/carers, the final report and subsequent health policy recommendations made by the inquiry do not appear to be patient-centric or reflective of the submissions written by these stakeholders. Increased transparency, development of processes to balance stakeholder priorities and improved accessibility for stakeholders to participate are needed if health-related parliamentary inquiries are to produce healthcare policy that ultimately meets the needs of patients and family/carers.

Effect of interventions for the well-being, satisfaction and flourishing of general practitioners-a systematic review.

OBJECTIVES: Clinician well-being has been recognised as an important pillar of healthcare. However, research mainly addresses mitigating the negative aspects of stress or burnout, rather than enabling positive aspects. With the added strain of a pandemic, identifying how best to maintain and support the well-being, satisfaction and flourishing of general practitioners (GPs) is now more important than ever. DESIGN: Systematic review. DATA SOURCES: We searched MEDLINE, PsycINFO, Embase, CINAHL and Scopus from 2000 to 2020. STUDY SELECTION: Intervention studies with more than 50% GPs in the sample evaluating self-reported well-being, satisfaction and related positive outcomes were included. The Cochrane Risk of Bias 2 tool was applied. RESULTS: We retrieved 14 792 records, 94 studies underwent full-text review. We included 19 studies in total. Six randomised controlled trials, three non-randomised, controlled trials, eight non-controlled studies of individual or organisational interventions with a total of 1141 participants. There were two quasi-experimental articles evaluating health system policy change. Quantitative and qualitative positive outcomes were extracted and analysed. Individual mindfulness interventions were the most common (k=9) with medium to large within-group (0.37-1.05) and between-group (0.5-1.5) effect sizes for mindfulness outcomes, and small-to-medium effect sizes for other positive outcomes including resilience, compassion and empathy. Studies assessing other intervention foci or other positive outcomes (including well-being, satisfaction) were of limited size and quality. CONCLUSIONS: There is remarkably little evidence on how to improve GPs well-being beyond using mindfulness interventions, particularly for interventions addressing organisational or system factors. This was further undermined by inconsistent reporting, and overall high risk of bias. We need to conduct research in this space with the same rigour with which we approach clinical intervention studies in patients. PROSPERO REGISTRATION NUMBER: CRD42020164699. FUNDING SOURCE: Dr Diana Naehrig is funded through the Raymond Seidler PhD scholarship.

Agreement between subjective and objective measures of sleep duration in a low-middle income country setting.

OBJECTIVES: Describe sleep duration in adult Sri Lankans and determine the bias and agreement of self-report and actigraphic assessments. DESIGN: Validation sub-study nested within the Colombo Twin and Singleton Study (2012-2015). SETTING: Colombo, Sri Lanka. PARTICIPANTS: 175 adults with actigraphy, randomly selected from 3497 participants with self-reported sleep assessed in a population-based cohort. MEASUREMENTS: Self-reported sleep duration, ascertained by the Pittsburgh Sleep Quality Index (PSQI), was compared to a minimum of four days of actigraphy. Bias and agreement were assessed using the Bland-Altman method and a novel application of criterion cut-point analysis. Objective measurements of wake after sleep onset (WASO) and sleep efficiency were evaluated. RESULTS: Sri Lankans have short sleep duration; averaging 6.4h (SD 1.5) self-reported and 6.0h (SD 0.9) actigraphically. Poor sleep quality was prevalent with an average WASO of 49 min., and sleep efficiency <85%. Bias was observed, with self-report consistently over-reporting sleep on average by 27.6 min (95% CI: -0.68, -0.24) compared to objective measures, but wide individual variation in disagreement, ranging from over-reporting by 3.34h to under-reporting by 2.42h. A criterion cut-point method also failed to define agreed definitions of short and long sleep duration. CONCLUSIONS: Sleep in Sri Lankan adults, whether measured subjectively or objectively, is of short duration and suboptimal objective quality by High Income Country consensus standards. Given the high cardiometabolic morbidity in Sri Lanka and poor measurement agreement observed, this warrants further investigation and supports the need for culturally appropriate, reliable, and valid assessment for analytic epidemiology in non-Western settings.