FormalPara Key Summary Points

Why carry out this study?

Hemophilia demands continuous management to prevent bleeds, posing economic challenges and a need for innovative management solutions.

The florio® HAEMO app, with activity tracking and wearable support, seeks to improve daily life for patients with hemophilia, potentially easing the disease's burden.

Satisfaction and practical aspects of hemophilia treatment monitoring using the florio HAEMO app are maintained even with long-term experience.

Pairing a wearable device with the florio HAEMO app might support everyday activities for people with hemophilia through easier access to app features and increased motivation for data entry.

What was learned from the study?

Adherence to the florio HAEMO app is maintained over time, and pairing the app with a wearable can make the app easier to use, motivate users to enter data more frequently and provide greater certainty in managing daily activities for people with hemophilia.

The results of this study could encourage the development of more user-friendly and effective treatment monitoring solutions in hemophilia care.

Introduction

The management of hemophilia has seen significant progress in recent years with the introduction of several novel pharmacological treatments, including extended half-life recombinant factors, non-factor therapy and the production of endogenous factors with gene therapy [1]. Yet, despite adequate prophylaxis and advanced treatment options, managing hemophilia continues to disrupt everyday routines and significantly impact the quality of life (QoL) of individuals living with the disease [1]. Routine factor administration also causes scarring and damage to veins, potentially resulting in poor medication adherence [1].

Although most people with hemophilia can participate in and enjoy various physical activities, including contact sports, prophylaxis foreplanning is essential because of a higher risk of bleeding and injury [1]. Furthermore, long-term complications such as joint arthropathy still occur in individuals on prophylaxis treatment, representing a significant source of morbidity [1]. Smartphone hemophilia monitoring applications (apps) have been developed to help people with hemophilia track bleeds, injections and factor supply as well as foreplan their daily physical activities [2,3,4,5]. The latest mobile app, florio® HAEMO (Florio GmbH; https://www.florio.com), can be used with any factor product and has the option to be paired with a wearable device (wearable) [2]. florio HAEMO uses the centralized McMaster population pharmacokinetic (PopPK) database based on Web Accessible Population Pharmacokinetics Service-Hemophilia (WAPPs-Hemo) to securely input PopPK-powered real-time factor level estimations [2, 6]. Knowledge of PopPK estimations in daily life helps people with hemophilia self-manage their treatment, avoid unexpected bleeding episodes and foreplan their daily activities. Wearable technologies in health research, such as fitness trackers, smartwatches, electrocardiogram monitors, blood pressure monitors and biosensors, have offered much potential, with demand increasing in recent years [7, 8]. How the use of wearable technology affects healthcare providers’ decisions and patient behavior remains, as yet, unclear [8]. Moreover, to the best of our knowledge, no data on pairing a wearable with a hemophilia monitoring app have been published to date. Emerging evidence from the literature suggests that using wearables in healthcare can empower individual patients by assisting with diagnosis, behavior change, treatment self-monitoring and management, and QoL parameters such as supporting daily physical activities [9].

Previously, we conducted the first user satisfaction survey of the florio HAEMO app in Central Europe [2]. Users of this hemophilia monitoring app were invited by their treating physician to complete a baseline survey after a median of 3–4 weeks using the app [2]. Initial results from part one of the survey indicated that 89.4% of 66 users were very satisfied or rather satisfied with the florio HAEMO app [2]. In addition, 97.0% of florio HAEMO users reported that the app was very easy or rather easy to use, with 94.0% of users reporting that the app was intuitive to use [2]. During the first part of the survey, participants had the option to test a wearable with the florio HAEMO app, with the aim that it may further assist them in tracking physical activity more precisely, among other benefits [2]. A pre-planned follow-up questionnaire was designed to be completed at least 6 months after the baseline survey [2]. We report the results of the second part of the florio HAEMO app survey, which evaluates additional data on user satisfaction, longer-term adherence and functionality using a wearable with the app. This study aims to evaluate user satisfaction and long-term usage with the florio HAEMO hemophilia monitoring app. In addition, we aimed to determine the impact on data entry in a subgroup of patients who chose to test a wearable paired with the app.

Methods

Study Design

The design of this two-part survey has been previously fully described [2]. After about 24–26 weeks of florio HAEMO usage, participants in the first patient satisfaction questionnaire completed a follow-up questionnaire focusing on long-term usage (approximately 6 months or more) and patient satisfaction with the florio HAEMO app. Wearable functionality was also assessed, and participants > 12 years of age were invited by their physicians to trial a wearable during the second part of the study. Participants could choose to test a loaned Garmin Vivoactive 3 smartwatch or an Apple Watch Series 3 smartwatch for the purposes of this study. Participants could only test a loaned smartwatch. The second online questionnaire consisted of 16 questions about overall satisfaction, ease of use, intuitiveness and type of prophylaxis therapy using a 5-point Likert scale and open-ended questions. Participants were invited by their physicians to complete part two of the survey at least 4 months after starting to use the florio HAEMO app (i.e., 4 months after part one of the survey was completed).

The online survey platform (Typeform™) was administered by Seesame s.r.o (Bratislava, Slovakia) for the follow-up questionnaire (questionnaire 2), as for the first online questionnaire conducted at baseline (questionnaire 1). Responses were collected and analyzed by Seesame s.r.o in compliance with European General Data Protection Regulation 2016/679, local legislations and Ethics Committee approval (University Hospital Bratislava Ethics Committee) in accordance with the Helsinki Declaration of 1964 and its later amendments. All respondents (people with hemophilia and caregivers/parents of people with hemophilia) provided informed consent for partaking in the study and any subsequent publication before completing the two questionnaires as part of the survey initiation. No internet protocol addresses or other identifiable information was stored. The Central European florio HAEMO expert panel interpreted the anonymized results.

Participants

Adults and children with either hemophilia A or B of any severity on regular prophylaxis with any factor concentrate (e.g., plasma-derived or recombinant [r] standard half-life [SHL] or extended half-life [EHL]) participated in this survey. Other hemophilia therapies (e.g., non-factor prophylaxis, on-demand factor treatment, etc.) were not supported by the florio HAEMO app at the time of the survey. A total of 106 people with hemophilia or caregivers of people with hemophilia using florio HAEMO in Central Europe were initially invited to complete the online survey via an electronic link sent by the treating physician. A total of 66 people with hemophilia or caregivers of people with hemophilia using florio HAEMO in Central Europe completed the first online questionnaire as described previously. Participants who completed the baseline questionnaire (questionnaire 1) and continued using florio HAEMO were eligible for inclusion in the follow-up analysis presented herein. Eligible participants were invited to complete the follow-up questionnaire (questionnaire 2) online between 15 June and 16 December 2021.

Statistical Analysis

Results from the second part of the survey are reported using descriptive statistics. No formal statistical testing was performed.

Results

Participants

Of the 66 participants completing the first part of the florio HAEMO survey (questionnaire 1), 50 (75.8%) completed the second part of the survey (questionnaire 2). The respondents of questionnaire 2 represented people with either hemophilia A (n = 43; 86.0%) or hemophilia B (n = 7; 14.0%). Table 1 shows that most respondents (32.0%) were people with hemophilia aged 26–49 years. In questionnaire 2, 14 respondents used a wearable with the florio HAEMO app, 8 (57.1%) of which were people with hemophilia aged between 13 and 25 years. SHL or EHL rFactor VIII (rFVIII) products were the most frequently administered type of prophylaxis (36.0% and 44.0%, respectively) by participants of the survey. A total of 30 respondents (60.0%) had used the florio HAEMO app for at least 7 months, and 20 respondents (40.0%) had used the app for 6 months or less before completing questionnaire 2. An Android operating system was used in conjunction with the florio HAEMO app by most respondents (76.0%), including those using a wearable (92.9%) (Table 1).

Table 1 Demographics and characteristics of respondents
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Overall User Satisfaction, Compliance and Patient Preference

Most respondents (92.0%) remained very satisfied or rather satisfied using the florio HAEMO app throughout this study (Fig. 1). Of the people with hemophilia using a wearable in part 2 of the survey, most indicated that they were very satisfied with florio HAEMO. Most respondents (78.0%) completing questionnaire 2 indicated that they managed to enter all relevant data for all infusions over the past 2 weeks before completing questionnaire 2 (Supplementary Material Figure S1); however, 22.0% reported that they had skipped data entry for at least one infusion. Of the seven respondents (14.0%) on prophylaxis who had experienced pain over the past 2 weeks prior to completion of questionnaire 2, three respondents (42.9%) reported using the modality of florio HAEMO app to enter their pain level compared to four (57.1%) who did not enter their pain level when it was present at least once. Numerically, the use of a wearable indicated a trend for increased data entry. Of the 17 (34.0%) respondents who indicated in the survey they had experienced a breakthrough bleed during the study period, most (82.4%, 14 of 17 users) entered the bleeding episode into the florio HAEMO app. The remaining three respondents who indicated in the survey they had experienced a breakthrough bleed (17.6%) missed inputting data about their bleeding episode into florio HAEMO at least once (Fig. 2). The most frequently used and the least frequently used app function indicated by respondents did not change between the first and second questionnaires, with 90.0% of respondents in questionnaire 2 indicating they used the app to monitor their factor levels and only 2.0% indicating they discussed their florio HAEMO data with their doctor, respectively (Fig. 3).

Fig. 1
figure 1

Overall satisfaction using florio HAEMO

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Fig. 2
figure 2

Bleeding episodes since starting to use florio HAEMO

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Fig. 3
figure 3

Florio HAEMO functions used in the last 12 weeks

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Ease of Use and Intuitiveness

Florio HAEMO users were asked to assess how easy it was to use the app and how intuitive its features were. Overall, the results provided by responders in questionnaire 2 were similar to those provided in the first questionnaire (Supplementary Material Figure S2). For most responders completing questionnaire 2 (92.0%), florio HAEMO remained very easy or rather easy to use, especially for people with hemophilia using a wearable (Supplementary Material Figure S2). Similarly, 44 of the 50 respondents (88.0%) indicated that the app is very intuitive or rather intuitive to use (Fig. 4). A total of 42 respondents (82.4%) did not experience any difficulties using the app (Fig. 5).

Fig. 4
figure 4

Intuitiveness of the florio HAEMO app

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Fig. 5
figure 5

Difficulties experienced using florio HAEMO in the last 12 weeks

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Preference for Wearables and Impact of Florio HAEMO on Daily Activities

When asked how essential the florio HAEMO app was in bringing more certainty to daily activities for people with hemophilia or their parents/caregivers, 34 of 50 responders (68.0%) in questionnaire 2 indicated that the app was very important or rather important to them (Fig. 6). In particular, most people with hemophilia testing a wearable (85.7%) indicated that florio HAEMO was very or rather important in bringing certainty to daily activities. Of the 14 people with hemophilia using florio HAEMO with a wearable, 13 users tested the Garmin Vivoactive 3 smartwatch and 1 user tested the Apple Watch Series 3 smartwatch. A total of ten wearable users (71.4%) indicated that they would strongly prefer or prefer to use a wearable in the future. Notably, of the 36 respondents who had not used a wearable with the florio HAEMO app, 14 (38.9%) indicated that they would also strongly prefer or prefer to use a wearable in the future (Fig. 7).

Fig. 6
figure 6

Importance of florio HAEMO in bringing certainty to daily activities

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Fig. 7
figure 7

Preference to use a wearable device linked to florio HAEMO in the future

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Discussion

Successful management of chronic diseases such as hemophilia often requires people with the condition to develop self-management skills (i.e., bleed recognition, self-infusion, medicines management, pain and risk management and conceptualization of preventative therapy). In addition, to improve health outcomes, QoL and well-being, people with hemophilia must adhere to their treatment regimen and maintain positive health behaviors from a young age [10]. Recent evidence from the literature suggests that using innovative interventions delivered by telehealth routes, such as mobile apps, can help improve adherence and self-care (e.g., using electronic logs and reminder systems) [2, 11, 12]. In the first part of our survey in Central Europe, we previously reported that the majority of first users of the mobile hemophilia monitoring app, florio HAEMO, found it to be very important or rather important in bringing more certainty to everyday activities for people with hemophilia and/or their caregivers [2]. In the second part of the florio HAEMO survey, 75.7% of participants who completed the first part also completed the follow-up questionnaire after approximately 6 months, indicating a continuously high level of adherence to the florio HAEMO app. Overall, most respondents completing the second questionnaire indicated a high level of satisfaction (92.0%), ease of use (92.0%) and intuitiveness (88.0%) with using the app, consistent with the findings from the first part of our survey. These updated findings support the hypothesis that an efficiently designed hemophilia monitoring app can support the needs of patients with hemophilia on prophylaxis and/or their caregivers with the potential for long-term adherence.

Based on our survey design and the first questionnaire results completed by 66 first-time users of florio HAEMO, we speculated that using a wearable might increase the precision capabilities of florio HAEMO and further support hemophilia treatment and daily physical activities. While pain is an inherently subjective experience, inadequate assessment can lead to undertreatment. In recent years, focus has shifted toward develo** and utilizing self-reported pain in clinical practice and clinical trials [13,14,15,16]. We propose that florio HAEMO can be used by people with hemophilia, their caregivers and physicians to better assess how, when and for how long reported pain interferes with daily life activities.

Wearable data are increasingly being used in clinical decision-making [17]. In our second questionnaire, 14 participants opted to test a wearable with the app. The reasons why the remaining 36 participants did not choose to test a wearable in part two of this survey are unknown. However, one possible explanation could be that some people may not consider themselves to be ‘tech savvy’ and may feel uncomfortable analyzing this novel data source [18]. Although the wearables were loaned to participants for the duration of this study, it is understandable that individuals from various socioeconomic backgrounds might view smartwatches as a non-essential item, potentially causing hesitation in borrowing one. In addition, some individuals with hemophilia, such as the elderly or less educated, may not have the capacity to understand how to use wearables [18]. Our survey also included young people and children who might not be familiar with using a wearable daily. Unsurprisingly, a recent qualitative survey of smartphone users among people with hypertension showed that younger age, higher income and higher education were key predictors for a higher level use of smartphone health apps [19]. Moreover, participants were able to recognize that self-monitoring of blood pressure could lead to empowerment in terms of their understanding of their condition and interactions with their general practitioners [19]. In our modern society, as the population continues to age, we should expect the proportion of people who consider themselves to be uncomfortable with such technologies is likely to decrease [18]. Among respondents who were engaged in testing a wearable in the second part of our survey, there was a numerically higher trend for data entry in the florio HAEMO app (e.g., for infusions and pain when indicated) but not for defining factor levels for future physical activity, suggesting that pairing the app to a wearable may be associated with increased motivation for data entry into the app. Of those who completed the second part of our survey, 66.0% indicated that they might consider or would prefer to use a wearable in the future with florio HAEMO, which is encouraging.

To the best of our knowledge, our survey is the first to evaluate the functionality of pairing a wearable with a hemophilia monitoring app. However, we acknowledge that the number of participants testing wearables is very small, and not all Central European countries were represented. Other limitations are similar to those identified for the first part of our survey, including the lack of standardized validity assessment for the questionnaire, no formal statistical comparison and self-reported study design, as described previously [2]. The flora HAEMO app currently does not support on-demand therapy or non-factor treatment. In addition, we cannot rule out selection bias of the treating physician when inviting participants to participate in the online survey. Therefore, the authors acknowledge that the results presented in this paper should be interpreted cautiously.

Conclusions

In summary, results from our follow-up questionnaire indicate that users of florio HAEMO maintain a high level of adherence to the smartphone app. Even after a more extended period of use, users find the app platform readily accessible and user-friendly, providing an easy-to-use solution for people with hemophilia and/or their caregivers on prophylactic treatment. In general, the concurrent use of a wearable device may be associated with increased motivation for data entry in the app. Furthermore, pairing a wearable in the future with the florio HAEMO app was considered a practical and/or preferable option for most participants who completed the study. Ultimately, our survey results suggest that the florio HAEMO hemophilia monitoring app is easy to use and appropriate for long-term use by people with hemophilia and/or their parents/caregivers. The option to pair the app with a wearable might also increase sports activity tracking, enable easier access to the app and provide more certainty about daily activities.