This is an open-access article distributed under the terms of the Creative Commons Attribution License (http://creativecommons.org/licenses/by/2.0/), which permits unrestricted use, distribution, and reproduction in any medium, provided the original work, first published in the Journal of Medical Internet Research, is properly cited. The complete bibliographic information, a link to the original publication on http://www.jmir.org/, as well as this copyright and license information must be included.
Recruiting pediatric samples for research may be challenging due to parental mistrust of the research process, privacy concerns, and family time constraints. Recruitment of children with chronic and genetic conditions may further complicate the enrollment process.
In this paper, we describe the methodological challenges of recruiting children for research and provide an exemplar of how the use of information technology (IT) strategies with social networking may improve access to difficult-to-reach pediatric research participants.
We conducted a cross-sectional descriptive study of boys between the ages of 8 and 18 years with Klinefelter syndrome. This study presented unique challenges for recruitment of pediatric participants. These challenges are illustrated by the report of recruitment activities developed for the study. We reviewed the literature to explore the issues of recruiting children for research using conventional and IT approaches. Success rates of conventional recruitment approaches, such as brochures, flyers in medical offices, and physician referrals, are compared with IT-based outreach. The IT approaches included teleconferencing via a Klinefelter syndrome support group, services of a Web-based commercial recruitment-matching company, and the development of a university-affiliated research recruitment website with the use of paid advertising on a social networking website (Facebook).
Over a 3-month period, dissemination of over 150 recruitment brochures and flyers placed in a large urban hospital and hospital-affiliated clinical offices, with 850 letters to physicians and patients were not successful. Within the same period, face-to-face recruitment in the clinical setting yielded 4 (9%) participants. Using Web-based and social networking approaches, 39 (91%) agreed to participate in the study. With these approaches, 5 (12%) were recruited from the national Klinefelter syndrome advocacy group, 8 (19%) from local and teleconference support groups, 10 (23%) from a Web-based research recruitment program, and 16 (37%) from the university-affiliated recruitment website. For the initial 6 months, the university website was viewed approximately 2 to 3 times per day on average. An advertisement placed on a social networking site for 1 week increased website viewing to approximately 63 visits per day. Out of 112 families approached using all of these methods, 43 (38%) agreed to participate. Families who declined cited either travel distance to the study site (15, 22%) or unwillingness to disclose the Klinefelter syndrome diagnosis to their sons (54, 78%) as the reasons for nonparticipation.
Use of Web-based technologies enhances the recruitment of difficult-to-reach populations. Of the many approaches employed in this study, the university-affiliated recruitment website supported by a Facebook advertisement appeared to be the most successful. Research grant budgets should include expenses for website registration and maintenance fees as well as online advertisements on social networking websites. Tracking of recruitment referral sources may be helpful in planning future recruitment campaigns.
Recruiting children for research can present many challenges due to parental mistrust of the research process, privacy concerns, and family time constraints [
Prior to 1996, medical treatment of children was based on clinical trials and the testing of products and medications that were conducted in adults [
The general public increasingly uses information technology (IT) as a source of health information. Approximately 80% of the American public search for health information using Internet sources [
The Internet offers many opportunities for informing potential research participants about a study. These include email [
On the one hand, exposure of information to vast numbers of Internet users creates an enormous opportunity for visibility and communication with potential research participants. At the same time, the recruitment process can be sabotaged by problems on the Internet, such as emails sent to spam folders [
The purpose of this paper is to describe methodological challenges associated with the recruitment of children as volunteers in research and to discuss how IT may improve access and enrollment of children in research. A case study of our experience in recruiting boys with Klinefelter syndrome for an exploratory cross-sectional study is used to illustrate specific challenges encountered with a difficult-to-recruit pediatric research population and how IT was used to support the enrollment of participants in the study.
Successful enrollment of clinical research participants is both a science and an art. A number of factors, including patients, health care professionals and researchers, structural and organizational entities, and history, interact to compromise or undermine successful enrollment of patient volunteers into a clinical study [
Health care providers in nonacademic settings may have a limited understanding or interest in clinical trials or may have misgivings about academic institutions [
Other barriers facing researchers include lack of attention to the mistrust of the population to be recruited, failure to demonstrate cultural sensitivity, and lack of training in understanding health care disparities in underserved populations [
Communities may also be sensitive about allowing researchers entry into their environment, especially when they perceive that their participation in the scientific efforts does not result in any return or reward at the community level [
The history of disreputably negative research practices persists in the minds of the public and these perceptions may influence the attitudes of potential research volunteers. The awareness of inhumane treatment by Nazi researchers during World War II and the infamous Tuskegee Syphilis Project conducted by the US Public Health Service from 1932 to 1972 [
In the United States, several guidelines offer protection to the public with regard to personal health care and participation in research. Public Law 104-191, also known as the Health Insurance Portability and Accountability Act (HIPAA), was enacted to protect the privacy and personal health information of the public [
Although pediatric researchers are charged with the responsibility of recruiting children for research, several challenges exist in such efforts. Because parents are legally responsible for their children, it is the parent who must be approached for permission for their child to participate. Parents’ willingness to have their child participate in a study may be influenced by their perception of benefits, risks, and barriers to participation [
The exemplar case illustrates our recent experience with recruiting boys with Klinefelter syndrome for participation in a cross-sectional study. Traditional approaches to recruitment fell short of obtaining the desired sample and expanding the approach with IT resulted in a significant gain in enrollment.
Klinefelter syndrome is a genetic condition caused by the presence of an extra X chromosome (karyotype 47, XXY). This condition occurs in an estimated 1 in 450-500 male births [
Klinefelter syndrome is a misunderstood condition owing to a paucity of research in children, lack of clear clinical guidelines for treatment during life stages, and unfortunate conclusion errors made by early researchers that suggested men with Klinefelter syndrome were at increased risk for criminal behavior [
We conducted an exploratory descriptive study to better understand phenotype, biomarkers, and psychosocial health parameters of boys with Klinefelter syndrome between the ages of 8 and 18 years [
New recruitment strategies included the development of a study website, in-person information sessions, Web-links, teleconferences, and email access to members of a national and several regional Klinefelter syndrome support organizations, as well as registration with a computer platform clinical recruitment-matching service. Each strategy is briefly described subsequently.
A study information and recruitment website [
We contacted a national Klinefelter syndrome advocacy association, Knowledge Support & Action (KS&A) [
RecruitSource is a search engine and computer platform for matching clinical research participants with researchers [
Patients who might be interested in research participation register their health information via PrivateAccess [
Social networking is often defined by Web-based platforms, such as Facebook and others. Social networking, however, may also include face-to-face and teleconference transactions with groups, audiences, researcher-participant, and participant-participant networking. Participant-participant networking is the central component to the recruitment strategy known as
Of 112 families approached, 43 (38%) agreed to participate. The most frequent reasons for families declining participation was nondisclosure of the diagnosis to their sons (54/112, 78%) and geographic distance from the study site (15/112, 22%). Most parents who had not disclosed the diagnosis to their sons feared that their sons would learn of the diagnosis through participation.
Recruitment approaches for the participants in the Klinefelter syndrome study are summarized in
Of the 69 families who declined, over one-fifth (15/69, 22%) came from direct clinical contact; almost twice that number (29/69, 42%) declined during support group presentations, and one-sixth (10/69, 16%) declined during the national KS&A meeting. The most frequent reason for decline was parents not wanting their boys to learn of their diagnosis (n=54, 78%) and travel distance to study site (15/69, 22%).
In an effort to boost activity from general Web users, we placed an ad on Facebook. The ad ran for 1 week in June of 2010, targeting a general audience. Impressions are the raw number of times an ad is shown to different Facebook users. The Facebook ad was shown a total of 2,522,169 times. Social impressions reflect the number of times the ad was shown with social context who visited the study webpage. There were 2835 social impressions for this ad resulting in 509 clicks directly to the study website’s home page. At a total cost of $311 for the week’s ad, this represents the researcher’s cost of $0.61 per visit. Prior to placing the ad on Facebook, the study website received 2 to 3 visits per day. During the week of Facebook advertising, website visits climbed to an average of 63 visits per day. The Klinefelter study website activity increase in response to the Facebook ad can be seen in
Because multiple techniques were employed to attract this difficult-to-reach population, it is difficult to attribute any one recruitment approach to increasing the number of participants in this study.
Number of participants using traditional and information technology with social networking recruitment approaches to the Klinefelter syndrome study (N=43).
Recruitment approach | n | (%) | |
|
4 | 9 | |
|
Physician letters | 0 | 0 |
|
Patient letters | 0 | 0 |
|
Brochures | 0 | 0 |
|
Clinic referral | 4 | 9 |
|
39 | 91 | |
|
Advocacy group | 5 | 12 |
|
Support groups and teleconference | 8 | 19 |
|
RecruitSource | 10 | 23 |
|
Study website with Facebook ad | 16 | 37 |
Response (visits per day to the Klinefelter syndrome study website) during Facebook advertisement period showing increase in activity during Facebook advertising.
Recruitment to the Klinefelter syndrome study by source timeline.
The Internet represents an increasingly valuable resource for researchers, especially for those who wish to understand the social and cultural context of the populations they are attempting to reach [
Since the advent of IT and social networking in the scientific community, there has been a steady evolution of its use for recruitment and Internet-based interventions. Even within the past 5 to 7 years, much has been learned about the limitations of Internet-based approaches and how such problems might be mitigated.
Although early experience with the use of IT-based recruitment for clinical research, as reported by Koo and Skinner [
Our recent experience in recruiting boys for the Klinefelter syndrome study can be described as a multilayered strategy of communication using IT. The process of communication began with a traditional print exposure that proved to be ineffective. Adding the various IT communication approaches, including the study website, a computer-based research recruitment website, social networking on Facebook, exposure via support groups online, and by teleconference, offered parents multiple exposures to study information. Although our original sample size calculation called for 46 participants based upon an effect size of 0.40, the effect size from our study proved to be larger (–0.47). We believe that the overall number of participants (43 boys) did not negatively affect the study.
Recruiting boys for a study on Klinefelter syndrome proved to be a challenging endeavor that was best accomplished using IT-based techniques. Important lessons were learned as we dealt with early recruitment challenges. The first lesson is that multiple exposures to the study information and personal contact with the researcher may be helpful in fostering parental trust. Parents must believe that the study, the institution, and the researcher are trustworthy before they will agree to have their child take part. These acts of communication, presented in multiple ways, were central to the success of our recruitment effort and are distinct advantages offered by IT-based strategies. Expenses related to website creation, registration of a domain name, website maintenance, and planning for social networking advertising were not initially anticipated by us, but should be considered by future researchers in the planning process when study budgets are developed. A limitation of our reported recruitment observations is that an in-depth recruitment analysis was not conducted to determine how multiple recruitment exposures occurred. Future IT-based recruitment efforts should preplan the collection of profile data, including IP addresses and tracking of how, when, and how many times a recruitment website was visited. This type of data may assist in the planning of customized approaches for the creation of more effective social networking banner advertising. Nevertheless, the observations from this study may advance the understanding of how difficult-to-recruit participants, like children, might be reached and have parental communication needs met with a view to obtaining their consent to participate in a study. It is noteworthy to mention that there has been inadequate representation of children in Klinefelter syndrome research and in other genetic conditions.
Researchers need to expand their knowledge of how potential recruits might be encouraged to participate in studies by understanding the utility of traditional approaches versus IT and other social networking approaches for recruitment. By offering multiple opportunities for exposure, parents have the opportunity to digest and think about the idea of having their child participate in a study. Because IT and social networking have become well-accepted modes of communication, these tools enable the researcher to layer the recruitment message in order to optimize the likelihood that recruitment efforts will be successful.
Screenshot of Columbia University Klinefelter syndrome study website.
Screenshot of Knowledge Support & Action (KS&A) Klinefelter syndrome advocacy association website.
Screenshot of Klinefelter Syndrome Global Support Group website.
Screenshot of RecruitSource website.
Screenshot of PrivateAccess website.
Screenshot of Klinefelter syndrome study Facebook advertisement.
Internet protocol
information technology
Klinefelter syndrome
This research was supported by The Pediatric Endocrinology Nursing Society, the National Association of Pediatric Nurse Practitioners, the Alpha Zeta Chapter of Sigma Theta Tau, Columbia University’s CTSA grant No. UL1 RR024156 from NCATS-NCRR/NIH and Yale School of Nursing T32 Post-Doctoral Fellowship Training Grant, No. 5T32NR008346-08. The content is solely the responsibility of the authors and does not necessarily represent the official views of the NIH.
None declared.