Evaluation of a prototype electronic personal health record for patients with idiopathic thrombocytopenic purpura.

BACKGROUND: Patients with rare diseases often lack information about the disease itself and appropriate health care, leading to poor quality of life. Personal health records provide health information which can then be shared between multiple health care providers. Personal health records may also offer a tool for capturing patients' reported outcomes, thus enhancing their empowerment and improving communication with health care professionals. We conducted a pilot study to evaluate the usability of Sanoia, a freely accessible personal health record, which was customized for patients with the rare disease, idiopathic thrombocytopenic purpura (ITP).
METHODS: The Sanoia interface was expanded with ITP-specific tools. A prospective study was conducted at the referent center to evaluate the usability of this new interface (referred to here as the "tool") by patients. Forty-three patients were randomized into groups to use or to not use the tool. Its use was evaluated by a specific questionnaire and by surveying individual patient adherence profiles. Evaluation of health-related quality of life using the ITP patient assessment questionnaire, was performed at baseline and after 1, 3, and 6 months via postal mail.
RESULTS: The groups were similar at inclusion in terms of characteristics, including global quality of life. During the study period, the tool was used to update the personal records of 19/28 patients (68%), with a median of two connections to the tool (range 1-12) plus access by various health care professionals (n = 22). In addition, 15/19 (78%) patients used the "personal notes" section at least once. We observed no significant changes in quality of life between patients with or without the tool during the study period.
CONCLUSION: This pilot study demonstrates the good usability of the new customized Sanoia interface for patients with ITP. Additional studies will increase its usability further, and its interface could be adapted for use with other rare chronic diseases.

RCT Entities:   Population Interventions Outcomes

Introduction

Worldwide, there are an estimated 7000 different rare chronic diseases (ie, affecting <1/2000 people).1 In addition to their frequent severity and/or absence of validated treatments, patients with rare diseases often lack information about the disease itself and its appropriate care, which ideally should combine many different spheres of expertise.

Adult immune thrombocytopenic purpura (ITP) is a rare chronic autoimmune disorder characterized by antibody-mediated platelet destruction, often leading to bleeding symptoms that may range from mild bruising and mucosal bleeding to severe hemorrhage.2 Patients also often complain of fatigue and report poor health-related quality of life.3 The treatment-related side effects and quality of life for these patients, including limitations to occupation and daily activities, have only recently become a focus of clinical research.

Personal health records and their potential application in rare diseases have rarely been evaluated. Yet, these can provide health information and allow information to be shared between multiple health care providers, which is particularly important when patients are away from their referent center. Also, personal health records offer tools for capturing patient-reported outcomes (eg, pain, symptoms, self-scoring), which may enhance patient empowerment as well as improve communication with health care professionals.

In France, Sanoia has recently developed a freely accessible personal health record interface that fully addresses privacy concerns and is already being used by >120,000 patients.4 We designed a prototype specifically customized for patients with ITP and conducted a pilot 6-month study to evaluate its usability and potential impact on quality of life.

Materials and methods

Creation of the tool

This study was conducted at the referent center, the Hospital Conception (Centre de Compétence, Marseille, France), which diagnoses and manages ITP. Sanoia has developed a web tool integrated with electronic personal health records, which offers full privacy protection using an innovative anonymity technique.4 The tool already includes numerous factors related to chronic disease, ie, vaccination records, allergies, prescription history, previous symptoms ( Figure 1A), and medical test results (Figure 1B). Once the patient has created his/her personal health record, they receive a unique personal identifier online that they can keep with them and communicate to a physician or health care provider (Figure 1C). The latter can then access the patient’s medical information in four languages (Figure 1D). Notably, only the patient has the personal password needed to modify and update the content of their personal health record.

Figure 1

Sanoia interface. The Sanoia interface already includes various information corresponding to vaccination records, allergies, prescription history, and previous symptoms (A), and medical test results (B). Once the personal health record is created, the patient receives a personal unique identifier online that he/she can keep with them (C). This can be communicated to any health care provider, who can then access the medical information in four languages (D).

In collaboration with physicians from the center, the existing interface of the Sanoia was expanded with ITP-specific tools, which included a link to ITP-related web resources that had been validated by physicians (Figure 2A), and an online emergency protocol, created from updated national recommendations. 7 The Sanoia interface has also been modified to allow patients to take personal (bloc) notes of symptoms, medications, and any intercurrent events (Figure 2B). The new interface is hereafter referred to as the “tool”.

Figure 2

Sanoia options in the customized interface for patients with ITP (the tool). The existing interface of Sanoia was expanded with ITP-specific tools, including addition of a link to ITP-related web resources validated by physicians (A), and a section for personal (bloc) notes for symptoms, medications, or intercurrent events (B).

Abbreviation: ITP, immune thrombocytopenic purpura.

Prototype evaluation

The principal aim of this 6-month prospective study was to evaluate the usability of the tool by patients with ITP. The secondary aim was to evaluate its potential impact on quality of life. To avoid any bias caused by selection of patients who might be more confident with the use of computers, patients with ITP hospitalized at the center between 2000 and 2010 were randomly and directly contacted by telephone to participate in the study. For this pilot study, we planned to enroll between 10 and 15 patients per group (see below). Eligible patients were men or women aged between 18 and 75 years with a diagnosis of chronic ITP according to the American Society of Hematology guidelines,3 permanent web access, and no neuropsychiatric conditions. After providing their written consent, the patients enrolled were randomized into three groups.

Groups 2 and 3 had personal health records initiated at the center during their first visit. Basic instructions concerning the use of the study tool were provided by resource personnel, and all patients received a printed manual (see Supplementary data). Those assigned to group 3 were also able to contact resource personnel (by phone and/or email) throughout the study period. The usability of the tool was evaluated by a specific questionnaire (including use of the tool by the patient alone or with a health care professional) and, more objectively, by surveying the number of individual times the patients connected with the tool, either to update personal records or to use it with a health care professional. After 3 months, the usage profiles were determined as previously reported.5

Briefly, patients were classified as nonadherent, adherent, or very adherent if they connected on <2, 2, or ≥3 occasions with the tool, respectively (not including access by professionals). Demographic and medical data, as well as evaluation of health-related quality of life, using the ITP patient assessment questionnaire4 were recorded at inclusion and at 1, 3, and 6 months via postal mail (see questionnaire in the Supplementary data). Group 1 comprised controls and had no access to the tool, and these patients were only evaluated for quality of life.

Results

Forty-three of 72 patients who were contacted (15 were not interested and 14 did not fulfill the inclusion criteria), were enrolled and randomized into three groups (n = 15, n = 14, and n = 14, respectively, in groups 1, 2, and 3). At inclusion, clinical and biological characteristics, as well as global quality of life, were similar between the groups (Tables 1 and 2).

Table 1

Patient characteristics at enrolment

	Group 1 (n = 15)	Groups 2 and 3 (n = 28)
Age (years)	44 (19–67)	49 (21–73)
Gender (female), %	73.3	75
Mean duration of ITP, (years)	13	9
Splenectomy, %	33.3	28.6
Chronic treatments,* n	7	8
Steroids	3	5
TPO agonists	1	2
Other**	3	1
Platelets (Giga/L)	120 (4–450)	160 (16–453)

Notes: Unless otherwise specified, results are given as their medians (range).

Intercurrent treatments (ie, short corticosteroid therapy initiated for a flareup of the disease) were not taken into account.

disulone, hydroxychloroquine.

Abbreviations: ITP, immune thrombocytopenic purpura; TPO, thrombopoietin.

Table 2

Usability of the study tool and impact on quality of life

	Group 1 (n = 15)	Group 2 (n = 14)	Group 3 (n = 14)
Usability of the tool
Users*	–	19 (68%)
Connections#	–	2 (1–12)
Personal notes**	–	15 (78%)
Impact on QoL##
Overall QoL at baseline (M0)	89 (27, 100)	98 (36, 100)	92 (16, 100)
Change in QoL, baseline to M1	0 (−27, 16)	−3 (−47, 24)	−2 (−18, 27)
Change in QoL, baseline to M3	0 (−27, 33)	−20 (−42, 24)	−2 (−18, 20)
Change overall QoL, baseline to M6	−7 (−49, 7)	−13 (−56, −1)	−9 (−20, 2)

Notes:

At least one connection to the tool during the study period;

Number of connections by users;

at least one use of the “personal notes” section by users;

QoL (quality of life) evaluated using the immune thrombocytopenic purpura patient assessment questionnaire (maximum score of 100).3 Results are shown as the median and range.

Results concerning the usability of the tool during the study period are reported in Table 2. Data reported by patients in the questionnaires were in complete agreement with the number of connections to the tool recorded during the study period. The tool was used to update personal records by 19/28 patients (68%), with each of the 19 patients making a median of two connections (range 1–12). In month 3, heterogeneous usage profiles were observed between patients from groups 2 and 3, which were classified as nonadherent (less than two connections), adherent (two connections), or very adherent (at least three connections) in 28%, 40%, and 32%, respectively. Except for being older, no other differences were observed between very adherent and nonadherent patients (mean age 57 versus 44 years respectively, P = 0.04) with regards to their characteristics at inclusion.

During the study period, patients used the tool with various health care professionals: general practitioners (n = 11), dentists (n = 2), nurses (n = 1), specialists (cardiologist, internist, or ophthalmologist, n = 5), or physiotherapists (n = 3). Notably, patients did not report any refusal from health care professionals to use information from the tool during a consultation. In addition, 15/19 (78%) used the personal notes section at least once for various reasons (to report symptoms or medical events, to write questions to ask their doctor).

Of note, patients from group 3 had no contact with resource personnel, likely because they received adequate information through the instructions provided during initiation of the tool and in its dedicated manual. During the study period, we observed no significant changes in quality of life between patients from group 2 and 3, or in patients from group 1 who did not use the tool (Table 2).

Discussion

The use of electronic personal health records has recently increased in spite of concerns regarding the privacy of recorded data. This first pilot study confirms the usability of a self-managed personal health record interface which was specifically customized for patients with a specific rare disease, ie, ITP. Indeed, 68% of patients used the tool effectively, and 72% of these met criteria for good and very good adherence. Also, the tool was not only used for updating the personal health record, but also during consultations with various health care professionals and to make personal notes.

The usefulness of this tool relies on patients’ understanding and using their records appropriately. The help provided directly by the hospital during initiation of the tool, as well as the creation of a specific manual to guide first users, were probably key factors in enabling its good adoption. However, we observed varying levels of usage of the tool, so additional research is needed to identify possible barriers to adoption. Interestingly, a previous study has reported increased usability of a similar tool by patients with asthma who used mobile phones instead of computers.6 Sanoia have recently implemented an interface for mobile phones. Thus, future studies involving the tool could also evaluate the impact of this parameter on the usability by patients with ITP.

During the study period, we did not observe a significant impact on quality of life. This negative result may be linked to our small sample size, which precluded any statistical per protocol analyses (ie, studying the impact of quality of life only in those who could effectively use the tool). Alternatively, the impaired quality of life at inclusion of our patients was not as severe as that reported in other studies.7 During the study period, none of the enrolled patients presented with a flare-up of their disease. Thus, the actual impact of the tool on patient health outcomes and well-being should be evaluated further in more patients, with other severe/active diseases, and for a longer period.

In conclusion, we found that patients with ITP, a rare disease, adapted satisfactorily to using the electronic personal health record with the Sanoia interface. Even though this study did not show a significant improvement in health-related quality of life, it has demonstrated the usability of this customized and freely accessible Sanoia interface, which could potentially be adapted for use by patients with other rare diseases.