Protocols of a diagnostic study and a randomized controlled non-inferiority trial comparing televisits vs standard in-person outpatient visits for narcolepsy diagnosis and care: TElemedicine for NARcolepsy (TENAR)

Abstract

Background: Narcolepsy is a rare chronic sleep disorder that typically begins in youth. Excessive daytime sleepiness is the main disabling symptom, but the disease is often associated with severe endocrine-metabolic and psychosocial issues, worsened by a long diagnostic delay, requiring a multidisciplinary approach. The scarcity of reference Sleep Centres forces the patient and family to travel for seeking medical consultations, increasing the economic and psychosocial burden of the disease. Growing evidence suggests that Telemedicine may facilitate patient access to sleep consultations and its non-inferiority in terms of patient satisfaction, adherence to treatment, and symptom improvement for sleep disorders. However, Telemedicine clinical and economic benefits for patients with narcolepsy are still unknown. Methods: TENAR is a two-part project, including: 1. a cross-sectional study (involving 250 children and adults with suspected narcolepsy) evaluating the accuracy of Teletriage (i.e., a synchronous live interactive sleep assessment through a Televisit) for narcolepsy diagnosis compared to the reference standard; and 2. a twoarm, parallel, open randomized controlled trial (RCT) to demonstrate the non-inferiority of the multidisciplinary care of narcolepsy through Televisits versus standard care. In this RCT, 202 adolescents (> 14 y.o.) and adults with narcolepsy will be randomly allocated (1:1 ratio) either to Televisits via videoconference or to standard in-person outpatient follow-up visits (control arm). The primary outcome is sleepiness control (according to the Epworth Sleepiness Scale). Secondary outcomes are other symptoms control, compliance with treatment, metabolic control, quality of life, feasibility, patient and family satisfaction with care, safety, and disease-related costs. At baseline and at 12 months, patients will undergo neurologic, metabolic, and psychosocial

Sleepiness in adults: An umbrella review of a complex construct

“…During the 2000s and especially the 2010s, particular attention was given to other dimensions of sleepiness, such as sleep inertia and excessive sleep, which are the main symptoms of central disorders of hypersomnolence. During this decade, new tools were mainly focused on pathological populations, since therapeutic innovations in the field of sleep disorders created a strong need for tools that are sensitive to changes and which can be used in randomized controlled trials for specific sleep disorders (60,77). Some trends, especially those emerging over the last two decades, may have escaped our analysis due to methodological limitations.…”

Narcolepsy

“…This is mandatory to understand and explore the determinants of the characteristics and comorbidities in narcolepsy, with the perspective to propose personalized medicine to patients. Identifying reference centres at National and European levels will help to guarantee to patients a high standard of cure, and to explore the usefulness of alternative screening and follow‐up methods by telemedicine (Ingravallo et al, 2020) and wearable monitoring systems (Filardi et al, 2018a,b, 2016) for screening and follow‐up. Contrasting with the important knowledge about the pathophysiology of NT1, its features, comorbidities and consequences, there is a dearth of knowledge about other central disorders of hypersomnolence such as NT2 and idiopathic hypersomnia.…”

Work productivity and activity impairment in patients with narcolepsy type 1

“…Patients were recruited from those currently attending the outpatient clinic of the Narcolepsy Centre of Bologna, Italy. The centre is located in the Emilia Romagna region but most of the patients come from other Italian regions (Ingravallo et al, 2020). Controls were recruited from among parents, children, partners, or friends who accompany patients with narcolepsy or other neurological disorders (e.g., headache, neuromuscular disorders) in the tertiary neurological outpatient clinic of the IRCCS Institute of Neurological Sciences of Bologna.…”