Hemophilia is a rare X-linked recessive hemorrhagic disorder that affects mostly men. Hemophilia A and B are caused by deficiencies of coagulation factors VIII and IX, respectively [1]. One common severe complication of this congenital disorder is spontaneous and repetitive bleeding, particularly in the synovial joints [1]. Poor management can eventually lead to permanent joint deformity and chronic hemarthropathy, a severe type of arthritis caused by bleeding into the joints. In addition to impaired physical functioning, patients may show reduced psychosocial functioning and occupational outcomes owing to frequent hospitalization and absenteeism from work or school [2].

Currently, coagulation factor replacement therapy is the most common and effective treatment for hemophilia [3]. Patients with moderate to severe hemophilia undergo regular infusion of plasma-derived or recombinant coagulation factors to prevent spontaneous bleeding. In addition, coagulation factor replacement therapy may be administrated in the event of break-through bleeding. The introduction of home infusion therapy has also empowered patients and their families to manage the disease in a more independent manner. However, significant barriers and perceived limitations have led to a lack of adherence to treatment among patients. Patients with a poor perception of the consequences of their illness and of the necessity of treatment may reduce their adherence to prophylactic infusions [4]. Individuals with lifelong chronic conditions may present with anxiety and stress, particularly those with hemophilia, who must acquire considerable knowledge and independent management skills at a young age [5].

To address the needs of patients with hemophilia, platforms that involve various types of technology have been implemented to promote health education and good protective health behavior [6]. In July 2019, we published a review in the Journal of Medical Internet Research to summarize the literature on the effectiveness of telehealth interventions in improving health outcomes in patients with hemophilia [7]. This review included 16 trials and observational studies and showed that mobile technology seemed to improve patients’ adherence and accuracy in recording infusion logs and bleeding events. Studies on the provision of disease-related information and practical skills regarding the management of hemophilia yielded promising outcomes, especially among adolescent and young adult patients [7]. Patients generally reported improvements in self-efficacy in managing hemophilia after the implementation of telehealth technology, but the sustainability of the intervention depends largely on its usability and the patients’ receptivity.

Emerging studies in the literature are demonstrating the importance of understanding factors that affect the acceptance of health care technology, especially in patients with chronic diseases [8-12]. The concept of technology acceptance broadly refers to perceived usefulness or user satisfaction with a technology and often encompasses other constructs such as system usability, user feedback, perceived ease of use, attitude toward using, intention to use, and actual usage [8]. Studies have shown that affordability and accessibility are important factors influencing technology acceptance and uptake [9-12]. One Chinese study reported that individuals who were younger and had higher education attainment, higher income, and better family support were more likely to use a smartphone [11]. Other behavioral factors such as technology anxiety, resistance to change, and a lack of trust in the use of devices for self-management are associated with resistance with using mobile health apps among patients with diabetes [12]. It is important to identify these barriers during the preimplementation phase so that users’ acceptance and adoption of mobile health technology can be enhanced to maximize the success of the intervention or program.

Approximately 200 patients in Hong Kong currently have mild to severe hemophilia [13,14]. Since January 2020, a team of patient advocates, clinicians, and academic researchers has obtained sponsorship from a local philanthropic organization to establish a community program for patients with hemophilia in Hong Kong. One goal of this program is to harness mobile technology to (1) promote adherence to prophylactic infusion and recording of bleeding events, (2) facilitate timely sharing of self-documented information with clinicians to formulate or modify the treatment plans, (3) promote knowledge about self-management (eg, infusion techniques), and (4) improve social interaction in the patient community.

Before the inception of this program, we conducted a cross-sectional survey to evaluate the patients’ level of technology acceptance and identify their expectations regarding the use of mobile technology for self-management of hemophilia.

Between June and December 2019, participants were recruited using consecutive sampling through the Hong Kong Hemophilia Society, the only active nongovernmental organization in Hong Kong that provides services to patients with hemophilia. All participants had received a diagnosis of hemophilia A or B from a hematologist and were able to read Chinese or English. The pediatric patient (<18 years of age) surveys were completed by their parents. The Chinese University of Hong Kong Survey and Behavioral Research Ethics Committee approved this study before its inception (Ref SBRE-18-052), and written consent was obtained from all participants.

The participants completed a structured questionnaire that comprised 3 sections. The first section collected the participant’s demographic and socioeconomic information, and the second section evaluated his/her level of technology acceptance. Eleven questions were developed on the basis of the Technology Acceptance Model, one of the most widely applied models to describe consumer acceptability of information technology [15]. The model posits that perceived usefulness and perceived ease of use are important factors determining whether a newly introduced technology would be accepted by its potential users [8,15]. The participants rated their perceived confidence and acceptance in using the new mobile technology on a 5-point Likert scale (1=strongly disagree, 5=strongly agree). The score for each item was summed to yield a total score ranging 11-55, with a higher score indicating better acceptance of the technology. In the last section, the participants were asked to identify the top 6 features (from a list of 10) that they perceived to be the most important components of a mobile app for self-management of hemophilia. These are the 10 most common features according to a review of the existing literature on the use of mobile technology among patients with hemophilia [6,7,16-18]. The participants had the option to provide additional factors and justifications. The questionnaire was developed in traditional Chinese and was piloted with 5 patients. The questionnaire was disseminated in both paper-based and electronic formats. Self-administration of the questionnaire required approximately 10 minutes.

Descriptive statistics were used to summarize the data. The Mann–Whitney U test was used to identify differences in the technology acceptance score observed across clinically relevant subgroups: disease severity (mild to moderate versus severe), treatment type (prophylaxis versus on-demand therapy), housing type (public versus private housing, which is a surrogate marker for low versus high socioeconomic status, respectively, in Hong Kong), and highest educational attainment (for adult patients only). An exploratory analysis was conducted to evaluate the reliability (Cronbach α) of the scale. All statistical analyses were performed using SAS (version 9.4, The SAS Institute) and the tests were 2-tailed.

In total, 56 participants completed the study (Table 1) (response rate=100%). Their average age was 37.2 (SD 14.5) years for adult patients (n=42, 75%) and 10.0 (SD 2.8) years for pediatric patients (n=14, 25%). Most of the patients (n=42, 81%) had a diagnosis of hemophilia A with a moderate to severe condition (n=49, 88%). Approximately half of the cohort (n=26, 46%) lived in public housing.

The mean technology acceptance score was 42.3 (95% CI 40.1-44.4; range 27.0-55.0). In general, the participants considered themselves skilled in using mobile apps (mean 4.3, 95% CI 4.1-4.5; Figure 1). They were willing to learn to use the new mobile app to organize their bleeding records (mean 4.0, 95% CI 3.7-4.3) and to manage their health (mean 4.2, 95% CI 4.1-4.5). Cronbach α values of the scale were .89, .90, and .87 for the overall cohort, adult patients, and parents of pediatric patients, respectively, which indicate high internal consistency.

Participants who lived in public housing (mean 39.9, 95% CI 37.1-42.9) reported lower technology acceptance than those who lived in private housing (mean 44.4, 95% CI 41.3-47.3; P=.04). No significant association was identified between the technology acceptance level and disease severity (P=.17), treatment type (P=.91), or education level (P=.75, adult patients only).

The most important features identified by the participants concerned documenting of infusion logs (n=49, 88%), bleeding events (n=48, 86%), and secure delivery of the bleeding information to health care professionals (n=40, 71%; Figure 2). One participant proposed health care appointment reminders to be considered as an additional feature as he struggled with “remembering his scheduled appointments with the hematology, physiotherapy, and orthopedics specialists.”