key: cord-0738053-azm8q8pm authors: Jaclyn, Davis; Andrew, NeSmith; Ryan, Perkins; Julianna, Bailey; Christopher, Siracusa; Nauman, Chaudary; Powers, M.; Gregory S, Sawicki; George M, Solomon title: Patient and family perceptions of telehealth as part of the cystic fibrosis care model during COVID-19 date: 2021-03-26 journal: J Cyst Fibros DOI: 10.1016/j.jcf.2021.03.009 sha: f4a2af317db555e0702b55ae62e199832b4478b5 doc_id: 738053 cord_uid: azm8q8pm BACKGROUND: Cystic Fibrosis (CF) is a chronic multi-system disease best cared for at Care centers with routine monitoring by interdisciplinary teams. Previously, remote home monitoring technology has been explored to augment in-person care. During the COVID-19 pandemic, traditional in-person care was limited and CF centers rapidly adapted to a telehealth delivery model. The purpose of this study was to understand how people with CF (PwCF) and families of PwCF experienced the shift to telehealthcare delivery. METHODS: This was a cross-sectional survey-based study conducted in 11 CF Centers. Two surveys were designed (one for adult PwCF and one for parents/guardians of PwCF) by participating CF center members with patient and family partner input. Surveys were disseminated electronically via email/text to all patients who completed a telehealth visit, and data were collected on secure Google Forms. RESULTS: Respondents rated their telehealth experiences as positive. Most were highly satisfied with their telehealth visit (77% adult, 72% pediatric) and found the visits to be highly convenient (85% for all surveyed). A majority of patients reported they had adequate time during the visit and had all questions and concerns addressed. Importantly, we also identified concerns regarding lack of in-person assessments including pulmonary function testing (PFT) and throat/sputum culture. CONCLUSION: Telehealth was a feasible and well-accepted mechanism for delivering care in a chronic CF care model during the COVID-19 pandemic and may be useful in the post-pandemic era. Further work is needed to understand the impact of telehealth on patient outcomes, healthcare utilization and associated cost. work, children missing school and costs associated with travel [7] , all of which can be especially taxing in larger US states where distance between CF centers can be considerable. Despite interest in telehealth, and small studies supporting the use of telemonitoring as an adjunct to in person care [8] [9] [10] [11] [12] [13] , regulatory, technological, and other perceived barriers had hampered broad implementation of a virtual care model. In response to the COVID-19 pandemic, unprecedented use of telehealth services was quickly enacted at multiple CF centers to ensure continued access to care [14] . To our knowledge, prior to the current COVID-19 pandemic, there has been no description in published literature of comprehensive clinical telehealth programs widely offered to a CF center's patient panel. Given the infrequent use of telehealth in the CF chronic care model prior to this time, patient experience in this virtual care paradigm is unknown. In order to better understand patient and family perceptions of telehealth in the CF population during the COVID-19 pandemic, several CF centers across the United States collaborated to design and disseminate an experience of care survey following telehealth visits. In general, we hoped to characterize the viewpoints of adult and pediatric PwCF and families of PwCF regarding integrating telehealth into their usual care. The objectives of the survey were to assess patient and family's attitudes about convenience of visit, level of concern about absence of usual in-person assessments (pulmonary function testing (PFT), weight, sputum culture, vital signs and physical exam), and their desired role of telehealth in future care. We previously assessed the viewpoints of CF providers surrounding telehealth [15] . To rapidly produce a survey focused on patient experience with telehealth, the CF Centers at the University of Alabama and Boston Children's Hospital formed a multi-center working group that met regularly via Zoom and received approval by patient/family partners. Survey development methods included cognitive mapping and brainstorming, pilot testing with patient and family partners and subsequent adjustment. Two surveys were ultimately developed in parallel, one for adults with CF receiving care at an adult CF program and a second for parents of children and young adults with CF receiving care at a pediatric CF program. The adult and pediatric surveys each consisted of 21 questions (5 multiple choice, 7 Likert scale, 5 yes/no, 3 short answer, and 1 checkbox question). Demographic data captured general age range and primary CF center. We asked participants to indicate which device (smartphone, computer or tablet) they used to access their telehealth visit and whether they used audio only. The survey focused on 3 themes to assess telehealth experience: understanding 1) the structure of and satisfaction with the visit, 2) the level of concern with absence of usual in-person assessments (PFT, weight, sputum culture, vital signs and physical exam), and 3) the desired role of telehealth in their future care. Five-point Likert scales were used to assess overall satisfaction and convenience of visits: 5 was defined as "very satisfied" or "very convenient," 1 was defined as "not satisfied" and "not convenient," and 3 was "neutral." In addition, a 5-point Likert scale was used to assess concern with lack of usual in-person assessments, where "1" was defined as "low concern" and 5 as "very concerned." We defined a score of "2" or less as a lower level of concern and "3" as moderate concern. Full surveys questionnaires are available in the online supplement. We administered the final survey to PwCF who engaged in a telehealth visit at their CF center between April 2020 and June 2020. A telehealth visit was defined as a distinct healthcare encounter with at least one medical provider, conducted at a distance by telecommunications. The survey was deployed across 11 CF programs ( Adults with CF or parents/guardians of those with CF seen in a pediatric center were invited to participate in the online survey via a link sent by email, text or electronic medical record within one week of the patient's first telehealth visit. Parents or guardians of patients 18 years and older who were seen in a pediatric center were sent the pediatric survey. There were no repeated measures done during this study's timeframe. Survey data were collected in an anonymized fashion from April to July 2020 and analyzed using Google Forms in weekly intervals. This study was conducted under IRB approval of the parent institutions of the participating centers. GraphPad Prism 8.4 was used to create the figures and produce descriptive statistics of the survey data. Surveys were completed by 120 adult patients across 4 adult CF centers and 141 parents/guardians of patients seen in 7 pediatric centers during the study period. Total response rate was 25%. Respondents had an even distribution across the pediatric and adult age spans (Supplementary table) . Patients accessed their telehealth visits via a variety of devices, most commonly by cellphone/smartphone, and computer/laptop and less commonly by tablet/iPad ( Fig. 1 ). Most patients surveyed (72% adult, 77% pediatric) had no prior experience with telehealth and despite this, most (87% of all surveyed) denied any difficulty accessing their telehealth visit ( Fig. 2 ). Many visits were multidisciplinary, with physicians seen most frequently. Other providers most commonly seen by pediatric patients included a dietitian, social worker and registered nurse. Adults had lower rates of seeing their non-physician care team members including an advanced practice practitioner, a dietitian, and a registered nurse ( Fig. 3 ) . Most patients rated their telehealth visits the highest level of convenience (85% for all surveyed) and satisfaction (77% adult, 72% pediatric) ( Fig. 2 ) . Most indicated they saw all desired disciplines, had adequate time with their team and all concerns were addressed ( Fig. 2 ) . In terms of missing in-person assessments, significant proportions of patients reported at least moderate concern regarding lack of usual measures and testing offered in an ambulatory clinic ( Fig. 4 ) . The highest level of concern expressed was regarding lack of throat/sputum cultures in both the adult population (54% expressing at least moderate concern) and pediatric population (64% expressing at least moderate concern). Fifty-two percent of adult respondents and respondents on behalf of the pediatric population expressed concern regarding lack of pulmonary function testing. A majority of surveyed patients and parents/guardians reported low concern regarding the lack of other measures: lack of weight and lack of vital signs with physical exam ( Fig. 4 ) . Despite some concern with lack of in-person assessments, there was striking interest in using telehealth as part of future care. Most patients (53% adult, 69% pediatric) expressed an interest in having "some" visits be conducted virtually, with an additional smaller group (34% adult, 21% pediatric) preferring for "most" future visits to take place virtually ( Fig. 5 ) . Both groups of respondents (94% adult, 97% pediatric) were interested in technology that would allow for at-home assessment of pulmonary function ( Fig. 5 ) to allay concerns discussed above. This study describes the experiences of over 250 PwCF across the pediatric and adult age-span who participated in telehealth services at their CF centers across the United States during the COVID-19 pandemic. Our findings highlight the overwhelmingly positive experience reported by participants and suggest a more permanent role for telehealth in the chronic care delivery model for CF may exist. Prior studies have determined that telemonitoring as an adjunct to routine in-person care is feasible with athome technologies, though variable effects on clinical outcomes were seen. In the present study, we had the opportunity to evaluate the experience of a near-complete transformation to virtual healthcare delivery in the setting of the COVID-19 pandemic. Our group also recently published similar positive findings of the experience of providing telehealth by providers in the same multi-center group. We found similar concerns about access to remote monitoring by multi-disciplinary providers but noted agree- ment around access and convenience for patients resulting in a desire to integrate telehealth services into the CF chronic care model of C after the pandemic [15] . We recognize the uniqueness of the context allowing for this experiment, and even while interpreting our findings through this lens, we argue there are lessons to be learned about how to integrate tele-healthcare delivery into our existing care paradigms. First, this study demonstrates PwCF are willing and able to engage with care virtually. Though roughly three quarters of all respondents surveyed had never used telehealth before, almost 90% denied any technological difficulty. This finding supports the idea that existing technology allows for seamless adoption of telehealth in the CF chronic care model. Second, PwCF were satisfied with their experiences, many reported all their concerns were addressed, and could meet with all the members of their healthcare team they wished to. These findings are important because they show not only that telehealth was well-received, but respondents felt they were given adequate time to discuss their concerns and meet with members of the multidisciplinary care team, as they do during a typical in-person visit. Third, there are limitations, specifically usual in-person assessments, to what can be accomplished by telehealth and lack of sputum cultures and pulmonary function testing were causes of concern for those in this study. Despite these limitations, a majority of patients still expressed interest in continued use of telehealth in the future. Home spirometers and other remote monitoring technologies may alleviate concerns of PwCF regarding lack of in-person testing. In response to this concern, the Cystic Fibrosis Foundation has begun an initiative to provide PwCF at-home spirometry technology, which has the potential to improve the quality of remote assessment and accuracy of clinical telehealth. In the present study, an overwhelming number of both adult and pediatric respondents expressed interest in technology that would allow for pulmonary function assessment at home. Other at home assessment tools, such as scales and culture-kits, will need to be developed to allow for effective care in the telehealth arena. Going forward, it is reasonable to plan for telehealth to play some role in the care model for PwCF. There are a number of different ways to incorporate telehealth into the current care paradigm, including replacing one or more routine in-person visit with a telehealth appointment in clinically stable PwCF, conducting some part of the care team's assessment virtually (i.e. nutrition assessment, nursing coordination or social work check-in), providing hospital or home IV follow-up, or possibly triaging sick calls from the nursing line. A reduction in the number of in-person visits could decrease the travel burden experienced by some PwCF, possibly reduce missed clinic appointments and minimize infection risks. Future care plans integrating telehealth will need to remain flexible and considered individually for each patient, and their results warrant continued study. Our study has noted strengths and some limitations. Strengths to this study include its novel and timely description of perceptions of telehealth in the CF population. We surveyed an array of PwCF and their family members, across both the pediatric and adult age span, and across multiple CF centers in the United States, including the opinions of over 250 respondents. As with any survey design study, our data depends upon those who choose to respond, rather than a random sample of participants. We minimized recall bias by sending the surveys shortly after the visit was completed. Given the perceptions elicited in this study are colored by the specific situation of using telehealth during the early months of the COVID-19 pandemic, these results may not necessarily be generalizable to a time when social distancing is no longer mandated. Further, our results capture one point in time per respondent and recognize that as the programs developed during these early months, experiences could also have changed. We do not have demographic data beyond age, limiting our ability to study factors related to social determinants of health, including the potential digital divide based on socioeconomic status that limits internet availability. Telehealth visits with interpreter services were offered to those who do not use English as a primary language; as our survey was offered only in English, we were unable to capture responses from the small proportion of CF patients who are non-English speakers. Future studies should be designed to understand and optimize the experience for this subpopulation. Respondents on behalf of patients at pediatric centers aged 18 years and greater were caregivers, whose contact information is primary; this focuses on the patient family experience, rather than that of these adult-aged patients. Finally, as the survey period spanned just several months, our study cannot answer whether telehealth meets the standards of CFF recommended care. While at this time, PwCF show interest in continuing telehealth visits in the future, it will be important to reassess attitudes and study outcomes in a longitudinal fashion and in the post-pandemic era. Telehealth is a powerful tool that has enabled continued access to care for chronically ill patients during an unprecedented time for our healthcare system. It will remain an important aspect of care for PwCF as long as social distancing recommendations remain in place and we should consider how to integrate this promising technology into standards of care going forward. By capitalizing on its merits of convenience and ability to connect entire care teams to patients remotely, we can more easily reach those geographically or logistically distanced from major care centers. By reducing the burden on clinical space, we may reduce health resource utilization and its associated costs. When implementing more permanent programs, we must ensure all have access to telehealth in the CF community and should seek solutions for issues of limited internet access, digital literacy, or language barriers. Future studies should aim to both optimize effectiveness of telehealth programs and evaluate the effects on CF outcomes including rates of pulmonary exacerbations, need for inpatient admissions and other patient-reported outcomes such as quality of life. Continued collaboration across centers will be essential to achieve these goals. Telehealth, in combination with remote monitoring devices, has the promise to drastically impact our current approach to healthcare delivery for those with CF, bringing convenience, patient satisfaction and possibly even improved value. Funding provided by NIH ( 1K08HL138153-01A1 and 2P30DK072482-12 ) and CFF (Solomon 20Y0). Cystic fibrosis. Lancet Clinical outcome in relation to care in centres specialising in cystic fibrosis: cross sectional study Early referral to cystic fibrosis specialist centre impacts on respiratory outcome Overview of telehealth and its application to cardiopulmonary physical therapy Technology and its role in respiratory care Telehealth clinics increase access to care for adults with cystic fibrosis living in rural and remote Western Australia The use of telehealth (text messaging and video communications) in patients with cystic fibrosis: a pilot study A smartphone application for reporting symptoms in adults with cystic fibrosis improves the detection of exacerbations: results of a randomised controlled trial JCF [m5G A feasibility study of home telemedicine for patients with cystic fibrosis awaiting transplantation Designing home telecare: a case study in monitoring cystic fibrosis Evaluation of a home telemonitoring service for adult patients with cystic fibrosis: a pilot study Five years of telemedicine in cystic fibrosis disease Telemonitoring in chronic obstructive airway disease and adult patients with cystic fibrosis A feasibility study of urgent implementation of cystic fibrosis multidisciplinary telemedicine clinic in the face of COVID-19 pandemic: single-center experience Favorable clinician acceptability of telehealth as part of the cystic fibrosis care model during the COVID-19 pandemic Ann Am Thorac Soc The authors would like to acknowledge all members of the multi-center collaboration that facilitated survey development and broad data collection, our patients and patient families who participated in telehealth, and the CFLN. Supplementary material associated with this article can be found, in the online version, at doi: 10.1016/j.jcf.2021.03.009 .