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Conventional approaches to participant recruitment are often inadequate in rare disease investigation. Social networking sites such as Facebook may provide a vehicle to circumvent common research limitations and pitfalls. We report our preliminary experience with Facebook-based methodology for participant recruitment and participation into an ongoing study of autoimmune hepatitis (AIH).
The goal of our research was to conduct a pilot study to assess whether a Facebook-based methodology is capable of recruiting geographically widespread participants into AIH patient-oriented research and obtaining quality phenotypic data.
We established a Facebook community, the Autoimmune Hepatitis Research Network (AHRN), in 2014 to provide a secure and reputable distillation of current literature and AIH research opportunities. Quarterly advertisements for our ongoing observational AIH study were posted on the AHRN over 2 years. Interested and self-reported AIH participants were subsequently enrolled after review of study materials and completion of an informed consent by our study coordinator. Participants returned completed study materials, including epidemiologic questionnaires and genetic material, to our facility via mail. Outside medical records were obtained and reviewed by a study physician.
We successfully obtained all study materials from 29 participants with self-reported AIH within 2 years from 20 different states. Liver biopsy results were available for 90% (26/29) of participants, of which 81% (21/29) had findings consistent with AIH, 15% (4/29) were suggestive of AIH with features of primary biliary cholangitis (PBC), and 4% (1/29) had PBC alone. A total of 83% (24/29) had at least 2 of 3 proposed criteria: positive autoimmune markers, consistent histologic findings of AIH on liver biopsy, and reported treatment with immunosuppressant medications. Self-reported and physician records were discrepant for immunosuppressant medications or for AIH/PBC diagnoses in 4 patients.
Facebook can be an effective ancillary tool for facilitating patient-oriented research in rare diseases. A social media-based approach transcends established limitations in rare disease research and can further develop research communities.
Challenges in recruiting patients into rare disease clinical studies contribute to wide knowledge gaps in understanding disease pathogenesis, natural history, and optimal therapeutic approaches. In the United States, rare diseases are defined as affecting fewer than 200,000 people at a given time. Despite these seemingly small numbers, there are close to 7000 different rare diseases that impact more than 25 million Americans (7% of the population) [
The digital age has created new tools for rare disease research, as many proactive patients connect with each other and investigators through online support groups on social networking sites (SNS) such as Facebook and Twitter [
Considering the established barriers to AIH research and the abundance of AIH patient support groups on SNS, we formed the physician-led Autoimmune Hepatitis Research Network (AHRN) Facebook group to foster patient support, share current AIH information, and promote AIH research efforts. In 2014, we initiated an observational study of patients with AIH (Genetic Repository of Autoimmune Liver Diseases and Contributing Exposures, GRACE Study) for investigating genetic and environmental underpinnings in disease development and clinical outcomes. We conducted a pilot study to assess whether a Facebook-based methodology is capable of recruiting geographically widespread participants into AIH patient-oriented research and obtaining quality phenotypic data.
We established the AHRN on Facebook in 2014 to provide an online repository of summarized AIH literature, discussion of study results and application to patient care, and opportunities to participate in our ongoing AIH research studies. As of May 2017, the AHRN Facebook group has 1640 members and is led and moderated by a hepatologist (CL) from Indiana University (IU). New membership has grown by individuals finding the page via Facebook search results, referrals shared in other AIH Facebook groups by our members, and direct physician-to-clinical-patient marketing. The AHRN is a private Facebook group; individual users wishing to join must be granted approval from the group moderator. Once approved, members are able to view and share content with other group members. A private group setting has the benefit of protecting the privacy of members by hiding their group affiliation from nonmember friends and allows group moderators to screen potential members for illegitimate accounts.
We posted a GRACE study recruitment advertisement, consisting of a recruitment message, study summary sheet, and representative study image, on the AHRN Facebook group after obtaining institutional review board approval. The study summary sheet outlined study aims, procedures, inclusion criteria, and the study coordinator’s contact information (phone and email). Inclusion criteria consisted of individuals 18 years and older, diagnosis of AIH or AIH with features of overlap disease (primary biliary cholangitis [PBC] or primary sclerosing cholangitis [PSC]) established by a medical doctor, and no provision of medical care at IU or surrounding affiliate institutions. The study advertisement was posted on the AHRN Facebook group at quarterly intervals over 24 months by the group moderator. The study advertisement directed interested individuals to contact the study coordinator to discuss study objectives and subsequently give consent by phone.
After verbal consent, a study packet containing epidemiologic surveys [
Once received, outside medical records were reviewed and tabulated into the local AIH database by study personnel. Missing clinical and laboratory support data for AIH diagnosis from outside records were identified and subsequently targeted with repeat release of information requests. Collected results (including saliva kit, epidemiologic surveys, and outside diagnostic reports) were stored in a secure location.
Saliva was collected from the subjects using an Oragene collection kit and stored according to kit instructions. DNA was isolated from 500 µL of saliva in PrepIT-L2P Purifier Reagent (Lot PT150219-A, DNA Genotek Inc), and remaining collected saliva sample was saved for future study.
We enrolled 29 participants in 2 years between June 2014 and June 2016. Participants were from 20 different states, with 27 (93%) indicating a primary residence outside of Indiana and a median distance of 727 miles from our center. Of 29 participants, 28 (97%) successfully completed and returned study instruments within 3 months of the coordinator-led phone consent. All participants were female, 90% (26/29) were white, and median age at enrollment was 52 years.
Of 28 participants, 20 (71%) subjectively reported a diagnosis of AIH alone, while 29% (8/28) reported AIH with features of PBC (
All participants recorded their current medication list within an epidemiologic survey provided at enrollment (
Appropriate outside medical records were received after the first request along with the medical information release form for 79% (23/29) of participants (
Three-quarters of the participants (22/29, 76%) had available autoimmune markers for the simplified score assessment. However, there were a number of missing values for the simplified criteria given the nature of data collection. Therefore, in order to enroll AIH patients into the GRACE study, we sought to categorize the participants most likely to have AIH based on meeting at least 2 of 3 lab- or clinician-reported criteria: positive autoimmune markers (simplified criteria), liver biopsy with typical findings of AIH, and/or reported treatment with immunosuppressant medications. Of 29 participants, 24 (83%) met at least 2 of 3 criteria, with 19 of these participants (79%) satisfying all 3.
Saliva samples were obtained from all 29 study participants. All collected samples were at least 2 mL in volume. DNA isolation from representative saliva samples yielded sufficient quantity and quality of DNA for genetic investigations. Median value of DNA yield was 41 µg per 500 µL saliva sample (interquartile range [IQR] 41.86) and 260/280 ratio was 1.84 (IQR 0.125).
Objective participant data collected from outside medical records.
Simplified AIHa score parameters | |||||||||
IgGb score | |||||||||
1 | 1 | 0 | 2 | 2 | 2 | 6 | Yes | AIH | |
2 | 3 | 2 | — | 2 | 0 | — | No | PBCd | |
3 | 3 | 2 | — | — | 2 | — | Yes | AIH | |
4 | 3 | — | — | 2 | 2 | — | Yes | AIH | |
5 | 1 | 2 | 2 | 2 | 2 | 8 | Yes | AIH | |
6 | 3 | 2 | — | — | 2 | — | Yes | AIH | |
7e | 3 | — | — | — | — | — | Yes | — | |
8 | 1 | 2 | — | 2 | 1 | — | Yes | AIH | |
9 | 2 | 2 | 2 | 2 | 2 | 8 | Yes | AIH | |
10 | 1 | 2 | 0 | 2 | 2 | 6 | Yes | AIH/PBC | |
11 | 3 | — | — | — | 2 | — | Yes | AIH | |
12 | 3 | — | — | — | 1 | — | Yes | AIH | |
13 | 3 | 0 | — | 2 | 1 | — | Yes | AIH | |
14 | 1 | 2 | 2 | 2 | 2 | 8 | Yes | AIH/PBC | |
15 | 2 | 2 | 0 | 2 | 2 | 6 | Yes | AIH | |
16 | 1 | — | — | 0 | 1 | — | — | AIH | |
17 | 1 | 0 | 0 | 2 | 1 | 3 | Yes | AIH | |
18 | 1 | 1 | 2 | 2 | 1 | 6 | Yes | AIH/PBC | |
19 | 1 | 2 | 0 | 2 | 2 | 6 | Yes | AIH | |
20 | 1 | — | — | 2 | 2 | — | Yes | AIH/PBC | |
21 | 1 | 2 | 0 | 2 | 1 | 5 | Yes | AIH | |
22 | 1 | 2 | 1 | 2 | 2 | 7 | Yes | AIH/PBC | |
23 | 1 | 2 | — | — | 2 | — | Yes | AIH | |
24e | 1 | — | — | — | — | — | — | AIH | |
25 | 1 | 0 | 0 | 0 | 1 | 1 | Yes | AIH | |
26 | 1 | 2 | 2 | 2 | 2 | 8 | Yes | AIH | |
27 | 1 | 2 | 0 | 2 | 2 | 6 | Yes | AIH or AIH/PBC | |
28 | 1 | 1 | — | 2 | 1 | — | Yes | AIH | |
29 | 1 | 2 | — | 2 | 1 | — | Yes | AIH |
aAIH: autoimmune hepatitis.
bIgG: immune globulin.
cMD: medical doctor.
dPBC: primary biliary cholangitis.
eParticipants without biopsy results available for review.
Subjective participant data collected from epidemiologic survey.
AIHa | PBCb | UDCAc | ||
1 | Yes | Yes | 2 | No |
2 | Yes | Yes | 0 | Yes |
3 | Yes | No | 0 | Yes |
4 | Yes | No | 2 | Yes |
5 | Yes | No | 2 | Yes |
6 | Yes | No | 2 | No |
7 | Yes | Yes | 2 | No |
8 | Yes | No | 1 | No |
9 | Yes | No | 1 | No |
10 | Yes | Yes | 1 | Yes |
11 | Yes | No | 2 | No |
12 | Yes | No | 0 | No |
13 | Yes | No | 0 | No |
14 | Yes | Yes | 0 | Yes |
15 | Yes | No | 1 | Yes |
16 | Yes | No | 0 | No |
17 | Yes | No | 1 | No |
18 | Yes | Yes | 1 | Yes |
19 | Yes | No | 1 | Yes |
20 | Yes | Yes | — | Yes |
21 | Yes | No | 0 | No |
22 | Yes | Yes | 1 | Yes |
23 | Yes | No | 2 | No |
24 | Yes | No | 2 | No |
25 | Yes | No | 2 | No |
26 | Yes | No | 2 | No |
27 | Yes | Yes | 2 | No |
28 | Yes | No | 1 | No |
29 | Yes | No | 1 | No |
aAIH: autoimmune hepatitis.
bPBC: primary biliary cholangitis.
cUDCA: ursodeoxycholic acid.
Sustained AIH research advances are continually impeded by low participant enrollment numbers, limited and disconnected investigators, and poor engagement of geographically spread patients wanting to contribute to scientific progress [
The retrieval of supporting outside records and eventual clinical diagnosis of AIH can be challenging for participants recruited both locally and remotely. Remote access was impeded most frequently by delayed response from corresponding medical record departments. Despite the obstacles inherently imposed by the study design, we were able to successfully collect epidemiologic questionnaires and biologic samples from all 29 participants using Facebook. While the effort spent on retrieval of outside records was time intensive, the passive approach to recruitment required less time spent identifying and recruiting patients enrolled locally through traditional methods. The successful recruitment of 29 subjects was substantial for the GRACE study, as our traditional approach at a large academic center provided only 120 participants (24% more cases obtained with social media) in the same duration of study.
SNS have evolved rapidly over the past decade to support our need for social connectedness and instant digital knowledge. Application of these tools in rare disease research is important, as they transcend well-established limitations such as cost, prolonged study courses, and geographic barriers. Our study was cost- and time-efficient and effective at recruiting patients up to 2200 miles away from our medical center. Furthermore, the cohort was similar to prior work according to gender (female predominant), ethnicity, concurrent autoimmune illnesses, fatigue, and itch [
In our study, prospective participants quickly engaged our study coordinator for recruitment. However, self-attestation of AIH diagnosis and current treatment strategies were not always congruent with their physician-composed medical records. Furthermore, we found certain medical records had insufficient documentation or lacked specific workup components. Our assessment of at least 2 of 3 key criteria, as outlined above, suggests AIH diagnosis in 24 out of 29 study participants (83%). Other studies have shown similar levels of agreement between self-reported diagnosis data and medical record documentation using traditional research methods, suggesting this challenge is not unique to social media–based research [
Successful implementation of a social media–based methodology comes with its own unique set of challenges. While SNS can supplement traditional recruitment, the process of sending and receiving medical records and study materials from affiliated institutions and participants proved to be time-intensive for the study team. Nonetheless, the time cost associated with this methodology is considerably less compared to the effort required to initiate recruitment at multiple study sites to achieve a similar geographical reach. Additionally, the demographics of AIH patients support a social media approach as they align well with the SNS user demographics [
Finally, a notable challenge of implementing SNS in medical research is simply its acceptability by providers and health systems. The medical community has been hesitant to embrace the potential of this approach to research recruitment, in part due to the risk of Health Insurance Portability and Accountability Act violations and the potential for demographic biases [
We foresee opportunities for social media to help connect like-minded investigators for research collaborations [
In summary, SNS can be an effective tool for facilitating patient-oriented research in AIH. We have successfully implemented a Facebook-based methodology to engage individuals unable to participate in research through traditional methods due to geographic or financial barriers. Mindful use of SNS can transcend established limitations in rare disease research and further cultivate powerful and engaged research communities.
Autoimmune Hepatitis Research Network
autoimmune hepatitis
antimitochondrial antibody
Genetic Repository of Autoimmune Liver Diseases and Contributing Exposures
interquartile range
Indiana University
primary biliary cholangitis
primary sclerosing cholangitis
social networking sites
ursodeoxycholic acid
We would like to express our appreciation to Tiebing Liang for DNA preparation and quantification as well as the Social Media Residency with the Mayo Clinic Social Media Network for providing the educational foundation of our digital research approaches. This publication was made possible with support provided to CL from grant numbers KL2TR001106 and UL1TR001108 (A Shekhar, PI) from the National Institutes of Health, National Center for Advancing Translational Sciences, Clinical and Translational Sciences Award.
None declared.