Patient Experience in Home Respiratory Therapies: Where We Are and Where to Go

The increasing number of patients receiving home respiratory therapy (HRT) is imposing a major impact on routine clinical care and healthcare system sustainability. The current challenge is to continue to guarantee access to HRT while maintaining the quality of care. The patient experience is a cornerstone of high-quality healthcare and an emergent area of clinical research. This review approaches the assessment of the patient experience in the context of HRT while highlighting the European contribution to this body of knowledge. This review demonstrates that research in this area is still limited, with no example of a prescription model that incorporates the patient experience as an outcome and no specific patient-reported experience measures (PREMs) available. This work also shows that Europe is leading the research on HRT provision. The development of a specific PREM and the integration of PREMs into the assessment of prescription models should be clinical research priorities in the next several years.


Introduction
Long-term oxygen therapy (LTOT) and/or home mechanical ventilation (HMV) are well-established therapies for patients with chronic respiratory failure, such as those with chronic obstructive pulmonary disease (COPD), neuromuscular diseases, and obstructive sleep apnea (OSA), among others. These
Three studies reported the assessment of HRT prescription at a regional level (Catalan, Spain; Hong Kong, China; Tasmania, Australia), eight at a national level (Sweden, Canada, Poland, Denmark, England, Australia, France, Spain), and four at an international level (two countries, seven countries, 13 European countries, 16 European countries). Survey content: provider characteristics, including services and education provided; user characteristics (age, ventilation type, primary disorder, duration of ventilation); criteria for initiation and monitoring ventilation effectiveness; equipment (ventilators and interfaces used, ventilator servicing arrangements and backup); training and education (audience, structure, topics, ongoing competency assessment); liaisons and transitions (referral, barriers to transition); follow-up (structure, frequency, location).
Response rate 152/171 (89%). 4334 ventilator-assisted individuals: an estimated prevalence of 12.9/100,000 population. 73% receiving NIV and 18% receiving intermittent mandatory ventilation (9% not reported). Services were delivered by 39 institutional providers and 113 community providers. Various models of ventilator servicing were reported. 64% of providers stated that caregiver competency was a prerequisite for home discharge, but repeated competency assessment and retraining were offered by 45%. Barriers to home transition: insufficient funding for paid caregivers, equipment, and supplies; a shortage of paid caregivers; negotiating public funding arrangements.   Most of the COPD patients were prescribed oxygen therapy by a hospital doctor immediately after an acute hospitalization, and the number of prescriptions from general practitioners was continuously declining toward zero during the study period. An increasing number of the COPD patients were prescribed oxygen at least 15 h daily and had delivered oxygen concentrator and mobile oxygen, whereas, in general, the oxygen flow remained low (≤1.5 L/minute). Compared with men, women started LTOT more often in connection with hospitalization and more often stopped LTOT within the first 6 months. Women were prescribed a lower oxygen flow than men and the treatment was more often specified to take place for 15-24 h per day. If centralized data were not available, regional departments administering LTOT services were contacted.
Data: Costs were defined as "equipment only" (fees paid to oxygen companies) or "equipment and administrative" (wages and non-labor costs of administering programs included).       Most studies included both children and adult patients in their analysis. Only one of the studies specifically focused on a pediatric population [18]. Questionnaires, having been used in 10 studies, were the preferred method of data collection. In five studies, existing databases from HRT registries or health services were used. Irrespective of the data collection method used, data on users (age, sex, and diagnosis), type and duration of respiratory therapy, and equipment and interfaces were the most commonly recorded. None of the 15 studies reported the patient's experience with HRT.

Assessment of Patient Experience
Assessing the patient experience has become a common approach to describing healthcare from the patient's point of view, evaluating the process of care, and measuring the outcome of care [25][26][27]. Both quantitative and qualitative methods are being used to assess patients' perception. Self-reported questionnaires, individual interviews, and focus groups are among the most frequently used methods of collecting data.

Patient-Reported Experience Measures
The development of self-reported questionnaires, namely, patient-reported experience measures (PREMs) and patient-reported outcome measures (PROMs), has exponentially increased in the last several years. These two types of questionnaires collect information about the patient's perspective but with distinct purposes. A PREM evaluates patients' perception of their personal experience of the healthcare received, while a PROM assesses the perception of their health status and health-related quality of life [10,28]. A combination of PROMs and PREMs is essential to fully understand the performance of healthcare systems. Moreover, both measures are useful to provide a patient-centered perspective of healthcare, but PREMs are more adequate to assess experience with healthcare.
Distinct instruments to assess the patient's experience with healthcare are available. Table 2 summarizes 14 instruments designed to assess the patient's experience with the provision of care in different clinical settings [29][30][31][32][33][34], hospital [35][36][37][38], primary care [39,40], intermediate care [41], and community [33,41]. The majority of such instruments are generic and designed to be used for a diverse range of health conditions. However, two of the described questionnaires were specifically developed for patients with chronic diseases [29,34], and one was intended particularly for patients with COPD [30]. The majority of PREMs were developed to target adult patients and tested in patients who were at least 15 years old. Only two developed instruments were tested with the carers of children [31,39]. English is the most common language used, with some instruments also in Norwegian [31,38,39], Italian [35,41], and Spanish [29]. Most instruments already had some of their psychometric properties explored, namely, their reliability and validity.
None of the instruments above were specifically designed to assess the patient's experience with HRT. However, a recent European Respiratory Society (ERS)/European Lung Foundation (ELF) survey was conducted across 11 European countries and assessed the attitudes and preferences of 687 patients on HMV and those of 100 carers [42]. A questionnaire was specifically developed for this study in eight languages (English, German, Dutch, Spanish, Italian, Portuguese, Greek, and French) and explored four areas: (1) patients' demographic and clinical characteristics; (2) issues influencing compliance, such as interface comfort, abilities to travel, sleep, and socialize with a ventilator, type and technical functioning of the ventilator (e.g., alarms, ability to operate and change settings, on/off switches, and electricity consumption); (3) support, training, and education; and (4) requests for improved devices and support.
Today, it is possible to evaluate a patient's perception of the HRT received using one of the described PREMs. Nevertheless, in the near future, the aim should be to develop a specific PREM to assess patients' personal experience with HRT.  [32,43] Tested in 828 patients in an orthopedic pre-operative assessment clinic [32] and in 90 adult patients (≥18 years) from general practices (10 with COPD) [43].
Generic, applicable without change across all patient categories and care settings, including primary, secondary, community, emergency, domiciliary, and social care.

English Dutch
Clinical care (kindness and communication), Organization of care (promptness and organization).
Four items scored using a four-point scale from 0 (poor) to 3 (excellent). Tested in 60 patients (≥15 years) undergoing a magnetic resonance scan.

Individual Interviews and Focus Groups
Qualitative studies that explore the experience of patients receiving HRT are still limited in the literature. Nevertheless, the literature review revealed some studies that explored the experience of patients living with COPD, pulmonary fibrosis, and OSA. These studies specifically focused on patients' needs and the adaptation process to respiratory therapies. Two studies explored the patient's experience with LTOT [45,46], and the others assessed the patient's experience with non-invasive ventilation [47][48][49][50][51]. These studies were conducted in the United States of America [45,47], New Zealand [48,49], the United Kingdom [50], Sweden [51], and Spain [46] and included both adult patients and carers. Two reviews were also found on the needs of patients with COPD and were also used in the present analysis [52,53].
From the analysis of these studies, it was possible to clearly identify education, training, support, and carer involvement as important key-points in facilitating a patient's treatment experience and subsequent adherence. Below, each one of these four key-points is described in detail.
Education: on the basis of the perspectives of patients, it is apparent that education is crucial for defining clear expectations about the treatment and motivating patient adherence. The main education topics raised by patients receiving respiratory therapies are related to disease self-management (e.g., COPD, OSA); physical effects and potential clinical benefits of the respiratory therapy; risks of not using the respiratory therapy; guidance on the use and function of equipment (e.g., continuous positive airway pressure (CPAP) devices, oxygen concentrators, how to use pulse oximeters and adjust flow with exertion); side effects and guidance on its management (skin protection, dry mouth, nasal congestion, irritated eyes); traveling with equipment; follow-up appointments; and assistance with financial elements (e.g., how to claim electricity costs) [45,46,49,50].
Training: formal training on appropriate equipment use has been suggested to be an important strategy for improving adherence [46][47][48][49][50][51]. Healthcare professionals need to introduce the device, explore possible practical problems, and give advice/help to solve these problems. In their initial experiences with respiratory therapy, patients should have a hands-on demonstration for setting up the device, trialing different masks/pressures, making mask adjustments, conquering different side-effects, and finding the best position for the tubing or machine (also considering the loudness of the device). Regular follow up visits or phone calls are important to assess practical problems being experienced (e.g., pressure from the mask, mask leakage, disturbing noise, and difficulties changing sleeping positions) and to discuss effective strategies to address them. Support: establishing a trustworthy relationship with healthcare professionals after the initiation of respiratory therapy is perceived as helpful by patients, and these relationships positively influence their adherence [46]. Healthcare professionals need to foster a non-judgmental environment in which patients have opportunities to ask questions, share concerns and feelings, feel listened to, and feel understood. This is particularly important following the initiation of therapy [47], as questions or concerns are more likely to arise during the first days or weeks of treatment [49,52]. These opportunities can arise during regular follow-up visits, scheduled follow-up phone calls, and through access to a 24-h hotline [47].
Carer involvement: carers provide substantial care (emotional, physical) to the individual on a daily basis and, most of the time, live in the same house as the patient. On the basis of their important role in patients' lives, carer involvement has been found to be essential to patients receiving HRT [45][46][47][48][50][51][52][53]. Patients recognize that carers play a major role in their treatment by helping them manage the disease and adapt to the equipment (e.g., verbal reminders, encouragement, setting up the machine, making mask adjustments, reassurance of therapy benefits). Carers themselves recognize their need for information regarding aspects of the disease and benefits of the HRT [47]. Carer involvement is thus perceived by all stakeholders as an essential component of education and training from the beginning of treatment [45,47,48,[50][51][52][53], and it is generally associated with positive results, namely, the patients' adoption and adherence to HRT [47,53].

Discussion
This comprehensive review is a first critical step toward the assessment of the patient experience in the clinical context of HRT. It demonstrates that research in this area is still limited, with no example of an HRT prescription model that incorporates the patient experience as an outcome and with no specific PREM available. This review also shows that European countries have been involved in HRT provision research from an early stage.
Most of the research on the assessment of HRT prescription models has been conducted within the last decade and mainly in European countries, highlighting the emergent interest and Europe's leading position in this area of health research. In addition, HMV has attracted more attention from the scientific community in comparison with LTOT. Questionnaires were found to be the preferred method for data collection, however, existing databases from HRT registries or health services have also been used. Databases in comparison with questionnaires have the advantage of generating more representative data and may be a method of choice in future studies. The patient experience has not been examined in the assessment of the prescription models presented. While this reality was expected from the oldest studies, it was quite a surprising result for those from the last decade. These results show that, until now, the assessment of patients' perceptions has not been seen as a priority in the assessment of prescription models. Unfortunately, this is also a reality in other health contexts and settings [10]. The Organisation for Economic Co-operation and Development (OECD) and Europe in "Health at a Glance: Europe 2018" reported critical gaps in the data on patient-reported experience, and they recommended collecting data on the patient experience from any doctor in ambulatory care settings [10]. Thus, future studies on the provision of HRT should address this important gap in the literature.
To address this gap, we need to be aware of the current methods being used to assess the patient experience. Different instruments used at distinct levels of healthcare are available and described in this review. These instruments were developed to be completed by adult patients and, in some cases, by carers of children. In our opinion, although the carers' perspective is, of course, incredibly valuable, it should do not replace the children's experience. The development of PREMs for pediatric populations is crucial to the collection of information on the experience and outcome of children's care. Additionally, as previously mentioned, none of the instruments have been specifically designed to assess the patient's experience with HRT. The development of a specific PREM for this health context should be a research priority in the upcoming years. The most commonly assessed domains in the described instruments, including the ERS/ELF survey, together with the key facilitators of the patient's treatment experience, can be used as important sources of data to inform the development of a comprehensive instrument. Access to information and support, implementation of effective and clear communication, active participation in shared decision making, enhanced accessibility and navigability across the healthcare system for patients and families, particularly across transitional care, and management of polypharmacy are known to influence the patient experience in other healthcare settings and could be topics of interest to be included in future PREMs for patients on HRT [54]. Future studies should explore which of these raised topics are indeed meaningful for patients and carers.
On the basis of qualitative studies, it was found that education, training, support, and carer involvement were important key-points in facilitating the patient's treatment experience and adherence. This knowledge comes mainly from the perspective of adult patients with COPD, pulmonary fibrosis, and OSA receiving CPAP and from their carers. These studies were conducted in five countries (three from Europe) [45][46][47][48][49][50][51][52][53]. Thus, this evidence may not completely apply to the experience of younger patients (including children) and that of their carers or to patients with other diseases and other treatment modalities (e.g., Bilevel Positive Pressure Airway, LTOT) and from other countries/continents. Considering these identified gaps, the experience of other patients receiving HRT could be explored in future studies. The identified key-points may inform the development process of semi-structured guides of focus groups or individual interviews to be used in these exploratory studies.

Conclusions
To the authors' best knowledge, this is the first published work to review the emerging topic of the patient experience in the clinical context of HRT and give important insights into the status of this clinical research area while also pointing out possible directions in which to move to realize patient-centered care. The assessment of the patient experience is in its early stages, and further research is needed to integrate these measures with routine healthcare delivery and the core set of healthcare quality indicators, as well as and to drive quality improvements in HRT.