Citations 2
Using a participation monitoring database to enhance recruitment in a rare cancer population
O’Rorke, Michael A. , Gryzlak, Brian , Xu, Tao, DeCook, Rhonda, Rudzianski, Nicholas J., Serrano, Kimberly , Wehrheim, Abigayle M., Schnell, Michael J., Witter, Carrie L., Gamblin, T. Clark , Kazmi, Syed Sohail, Cowell, Lindsay G., Else, Tobias, Chandaka, Sravani , Sanoff, Hanna K. , He, Fiona , Ramirez, Robert A., Liu, Mei, Lancaster, William, Mailman, Josh , Moran, Heather, Wahmann, Maryann, Gellerman, Elyse, Chrischilles, Elizabeth A., Liu, Mei , Sanoff, Hanna , O’Rorke, Michael , McDowell, Bradley D. , Rudzianski, Nicholas J , Serrano, Kimberly C., Grewal, Udhayvir Singh , Chandrasekharan, Chandrikha , Dillon, Joseph S. , Hálfdánarson, Þorvarður R. , Cowell, PhD Lindsay G., Else, Tobias , Soares, Heloisa P. , Sukrithan, Vineeth , Geller, David A. , Ramirez, Robert A., Lancaster, William, Chrischilles, Elizabeth A.
Journal of Clinical and Translational Science | February 13, 2026 | Volume: 10 | Issue: 1 | Pages: No page available
Abstract Introduction: Recruitment for rare disease studies is challenging due to small eligible populations. Traditional clinical research management systems often lack tools to track recruitment contacts prior to enrollment. The NET-PRO study, focused on neuroendocrine tumors (NETs), implemented a participation monitoring system to enhance recruitment efficiency and representativeness. Methods: NET-PRO is a multicenter cohort study of 2538 adults diagnosed with gastroenteropancreatic (GEP) or lung NETs between January 2018 and September 2024. Recruitment occurred from January 2022 to February 2025 across 14 U.S. medical centers. Sites used flexible recruitment methods (email, mail, phone, in-clinic) and tracked contacts using REDCap-based tools. Participant characteristics were analyzed by enrollment mode (online or mail) and recruitment difficulty (number of contacts required prior to enrollment) using standardized mean differences, chi-square tests, and ANOVA. Results: Of 9279 contacted patients, 2675 consented (28.8%) and 2538 enrolled (27.4%). Most enrolled online (83.2%), while 16.8% enrolled by mail. Mail respondents were older, had lower education and income, and more comorbidities. Among those enrolled, recruitment difficulty was associated with older age, lower education and income, but not comorbidity. Over half of the most difficult-to-recruit participants enrolled online. Contact methods varied by attempt, with email dominating early contacts and phone/mail used more in later attempts. Conclusions: A participation monitoring tool supported flexible, multimodal recruitment and improved sample representativeness in a rare cancer study. Tracking recruitment contacts enabled adaptive strategies and may reduce bias in observational research by enabling better outreach to harder-to-reach populations.
How common are myeloproliferative neoplasms? A systematic review and meta‐analysis
Titmarsh, Glen J., Duncombe, Andrew , O’Rorke, Michael , Vocht, Frank de , Horan, Sarah, Fritschi, Lin , Clarke, Mike, Anderson, Lesley , McMullin, Mary Frances , Mesa, Ruben A. , Clarke, Mike, Anderson, Lesley A , Fritschi, Lin
American Journal of Hematology | February 12, 2014 | Volume: 89 | Issue: 6 | Pages: 581-587
Myeloproliferative neoplasms (MPNs) are a heterogeneous group of diseases including polycythemia vera (PV), essential thrombocythemia (ET), and primary(idiopathic) myelofibrosis (PMF). In this systematic review, we provide a comprehensive report on the incidence and prevalence of MPNs across the globe. Electronic databases (PubMed, EMBASE, MEDLINE, and Web of Science) were searched from their inception to August 2012 for articles reporting MPN incidence or prevalence rates. A random effects meta‐analysis was undertaken to produce combined incidence rates for PV, ET, and PMF. Both heterogeneity and small study bias were assessed. Thirty‐four studies were included. Reported annual incidence rates ranged from 0.01 to 2.61, 0.21 to 2.27, and 0.22 to 0.99 per 100,000 for PV, ET, and PMF, respectively. The combined annual incidence rates for PV, ET, and PMF were 0.84, 1.03, and 0.47 per 100,000. There was high heterogeneity across disease entities (I2 97.1–99.8%) and evidence of publication bias for ET and PMF (Egger test, P = 0.007 and P ≤ 0.001, respectively).The pooled incidence reflects the rarity of MPNs. The calculated pooled incidence rates do not reflect MPN incidence across the globe due to the high unexplained heterogeneity. Improved, widespread registration of MPNs would provide better information for global comparison of the incidence and prevalence of MPNs. Am. J. Hematol. 89:581–587, 2014. © 2014 Wiley Periodicals, Inc.