title: Rare lung disease research: strategies for improving identification and recruitment of research participants.
abstract: Research in rare lung diseases faces methodologic limitations by virtue of the small number of participants available to be studied. We explored several strategies that may improve researchers' ability to identify and recruit research participants with rare lung diseases. We provide an overview of strategies based on available evidence, previously used approaches, and reasoning. First, disease detection is generally poor and may be improved through strategies targeted at primary care practitioners or directly at patients, thus increasing the pool of patients available for research studies. Next, standardization of case definitions in rare lung diseases is often lacking, hindering research recruitment efforts because of confusion over appropriate recruitment criteria. Expert consensus statements can enhance both clinical care and research recruitment by standardizing definitions. Finally, recruitment strategies using rare lung disease registries, clinical research networks, novel Internet-based direct patient recruitment approaches, and patient organizations may facilitate recruitment of patients with rare lung diseases. In summary, although several strategies for improving the identification and recruitment of research participants with rare lung diseases have been proposed, published examples are few. Objective measurement and reporting of novel recruitment methods and collaboration among researchers facing the same limitations across various rare lung diseases are required. Advancements in this area are vital to the design and performance of much-needed robust clinical studies across the spectrum of rare lung diseases.
authors: Department of Medicine, University of Toronto; Rare Lung Disease Research Program, Division of Respirology, Department of Medicine, St. Michael's Hospital, Toronto, ON, Canada; The Keenan Research Centre in the Li Ka Shing Knowledge Institute of St. Michael's Hospital, St. Michael's Hospital, Toronto, ON, Canada. Electronic address: guptas@smh.ca.
authors: Samir
authors: S
authors: Gupta
authors: Department of Medicine, University of Toronto; Department of Health Policy, Management, and Evaluation, University of Toronto; The Centre for Research on Inner City Health, St. Michael's Hospital, Toronto, ON, Canada; The Keenan Research Centre in the Li Ka Shing Knowledge Institute of St. Michael's Hospital, St. Michael's Hospital, Toronto, ON, Canada; Division of General Internal Medicine, St. Michael's Hospital, Toronto, ON, Canada.
authors: Ahmed M
authors: AM
authors: Bayoumi
authors: Department of Medicine, University of Toronto; Rare Lung Disease Research Program, Division of Respirology, Department of Medicine, St. Michael's Hospital, Toronto, ON, Canada; The Keenan Research Centre in the Li Ka Shing Knowledge Institute of St. Michael's Hospital, St. Michael's Hospital, Toronto, ON, Canada.
authors: Marie E
authors: ME
authors: Faughnan
mesh_terms: D035843:Biomedical Research / Q000458:organization & administration*; D003299:Cooperative Behavior; D006801:Humans; D020407:Internet; D008171:Lung Diseases / Q000145:classification / Q000175:diagnosis / Q000453:epidemiology; D018579:Patient Selection; D035583:Rare Diseases / Q000145:classification / Q000175:diagnosis / Q000453:epidemiology; D012042:Registries; D012955:Societies, Medical