Evaluation of participant recruitment methods to a rare disease online registry

Johnson, Kimberly; Mueller, Nancy; Williams, Karel; Gutmann, David H.

doi:10.1002/ajmg.a.36530

Cited by 38 publications

(52 citation statements)

References 25 publications

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“…This approach has the additional benefit of minimizing the time clinical and researcher staff spend on recruitment and data entry, improving recruitment and retention. Feedback from patients has shown that they find the dynamic record keeping and the ability to print off their data for personal use particularly valuable, which supports the findings from other internet-based registries [28]. We propose that if patients are aware of exactly what they are contributing to, and are updated with how it is progressing the evidence base for their disease, they will see themselves as partners in the project, and thus may feel more empowered…”

Section: Discussionsupporting

confidence: 70%

The RUDY study platform – a novel approach to patient driven research in rare musculoskeletal diseases

Javaid

Forestier-Zhang

Watts

et al. 2016

Orphanet J Rare Dis

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BackgroundResearch into rare diseases is becoming more common, with recognition of the significant diagnostic and therapeutic care gaps. Registries are considered a key research methodology to address rare diseases. This report describes the structure of the Rare UK Diseases Study (RUDY) platform that aims to improve research processes and address many of the challenges of carrying out rare musculoskeletal disease research.RUDY is an internet-based platform with online registration, initial verbal consent, online capture of patient reported outcome measures and events within a dynamic consent framework. The database structure, security and governance framework are described.ResultsThere have been 380 participants recruited into RUDY with completed questionnaire rates in excess of 50 %. There has been one withdrawal and two participants have amended their consent options.ConclusionsThe strengths of RUDY include low burden for the clinical team, low research administration costs with high participant recruitment and ease of data collection and access. This platform has the potential to be used as the model for other rare diseases globally.

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Section: Discussionsupporting

confidence: 70%

The RUDY study platform – a novel approach to patient driven research in rare musculoskeletal diseases

Javaid

Forestier-Zhang

Watts

et al. 2016

Orphanet J Rare Dis

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“…Of the 12 studies that found social media to be the best method of recruitment, 8 were observational studies [14,16,17,26,28,31,35,41] and the remaining 4 were interventional studies [15,32,36,42], as shown in Multimedia Appendix 2. In addition, 6 of these studies targeted populations deemed hard to reach [16,17,28,31,32,36], and 6 studies targeted specific conditions or disorders [14,16,17,28,31,32].…”

Section: Resultsmentioning

confidence: 99%

“…In addition, 6 of these studies targeted populations deemed hard to reach [16,17,28,31,32,36], and 6 studies targeted specific conditions or disorders [14,16,17,28,31,32]. Furthermore, 1 study targeted only young adults (aged 23-30 years) [14].…”

Section: Resultsmentioning

confidence: 99%

The Use of Social Media in Recruitment for Medical Research Studies: A Scoping Review

Topolovec-Vranic¹,

Natarajan²

2016

J Med Internet Res

390

328

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BackgroundRecruiting an adequate number of participants into medical research studies is challenging for many researchers. Over the past 10 years, the use of social media websites has increased in the general population. Consequently, social media websites are a new, powerful method for recruiting participants into such studies.ObjectiveThe objective was to answer the following questions: (1) Is the use of social media more effective at research participant recruitment than traditional methods? (2) Does social media recruit a sample of research participants comparable to that recruited via other methods? (3) Is social media more cost-effective at research participant recruitment than traditional methods?MethodsUsing the MEDLINE, PsycINFO, and EMBASE databases, all medical research studies that used social media and at least one other method for recruitment were identified. These studies were then categorized as either interventional studies or observational studies. For each study, the effectiveness of recruitment, demographic characteristics of the participants, and cost-effectiveness of recruitment using social media were evaluated and compared with that of the other methods used. The social media sites used in recruitment were identified, and if a study stated that the target population was “difficult to reach” as identified by the authors of the study, this was noted.ResultsOut of 30 studies, 12 found social media to be the most effective recruitment method, 15 did not, and 3 found social media to be equally effective as another recruitment method. Of the 12 studies that found social media to be the best recruitment method, 8 were observational studies while 4 were interventional studies. Of the 15 studies that did not find social media to be the best recruitment method, 7 were interventional studies while 8 were observational studies. In total, 8 studies stated that the target population was “hard-to-reach,” and 6 of these studies found social media to be the most effective recruitment method. Out of 14 studies that reported demographic data for participants, 2 studies found that social media recruited a sample comparable to that recruited via traditional methods and 12 did not. Out of 13 studies that reported cost-effectiveness, 5 studies found social media to be the most cost-effective recruitment method, 7 did not, and 1 study found social media equally cost-effective as compared with other methods.ConclusionsOnly 12 studies out of 30 found social media to be the most effective recruitment method. There is evidence that social media can be the best recruitment method for hard-to-reach populations and observational studies. With only 30 studies having compared recruitment through social media with other methods, more studies need to be done that report the effectiveness of recruitment for each strategy, demographics of participants recruited, and cost-effectiveness of each method.

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“…Detailed methods on the registry have been previously published [15–17]. Briefly, following online consent, registrants select an adult or minor questionnaire version, with parents/legal guardians instructed to complete the minor questionnaire version for registrants <18 years of age.…”

Section: Methodsmentioning

confidence: 99%

Peri-gestational risk factors for pediatric brain tumors in Neurofibromatosis Type 1

Johnson

Zoellner

Gutmann

2016

Cancer Epidemiology

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Background Individuals with Neurofibromatosis Type 1 (NF1) are strongly predisposed to developing pediatric brain tumors (PBTs), especially optic pathway gliomas (OPGs). Although developmental factors have been implicated in the origins of PBTs in both human and animal studies, associations between early-life factors and PBTs have not been evaluated in individuals with NF1. Our objective was to evaluate associations between peri-gestational characteristics and PBTs in this population. Methods We conducted a cross-sectional study, ascertaining questionnaire and medical record data for 606 individuals <18 years old who enrolled in the NF1 Patient Registry Initiative (NPRI) from 6/9/2011-6/29/2015. One hundred eighty-four individuals had reported PBT diagnoses, including 65 who were identified with OPG diagnoses. Cox proportional hazards regression was used to calculate hazard ratios (HRs) and 95% confidence intervals (CIs) for associations between PBT and OPG diagnoses and peri-gestational characteristics (prematurity, birth weight, parental age, plurality, family history of NF1, assisted reproductive technology, maternal vitamin supplementation, and parental smoking). Results We observed no significant associations between any of the assessed characteristics and PBTs overall or OPGs with the exception of birth weight. After controlling for potential confounding variables, we observed a significant positive association between birth weight quartile and OPGs with a HR of 3.32 (95% CI 1.39–7.94) for the fourth (≥3915.5 g) compared to the first (≤3020 g) quartile (p for trend = 0.001). Conclusions Consistent with results for PBTs in the general population, these results suggest that higher birth weights increase OPG risk in individuals with NF1.

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Evaluation of participant recruitment methods to a rare disease online registry

Cited by 38 publications

References 25 publications

The RUDY study platform – a novel approach to patient driven research in rare musculoskeletal diseases

The RUDY study platform – a novel approach to patient driven research in rare musculoskeletal diseases

The Use of Social Media in Recruitment for Medical Research Studies: A Scoping Review

Peri-gestational risk factors for pediatric brain tumors in Neurofibromatosis Type 1

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