Barriers and facilitators to research participation among adults, and parents of children with sickle cell disease: A trans‐regional survey
Authors: Robert M. Cronin, Jane S. Hankins, Patricia Adams‐Graves, Alexis A. Thompson, Karen Kalinyak, Jeannie Byrd, Brandi McClain, Adetola Kassim, Michael DeBaun, Marsha Treadwell
Journal: American Journal of Hematology (2016)

To the Editor: Patient recruitment into sickle cell disease (SCD) clinical trials has not always been robust, leading to delayed results or even failure to trial completion 1. Disparities impacting participation in research include race, lower socioeconomic status and lower health literacy 2. We sought to extend previous research by surveying adults with SCD and parents of children with SCD across geographically diverse regions in the U.S. We hypothesized that demographic variables such as lower income and education would present barriers to interest in research participation, as would psychosocial variables (depression symptoms and lower health literacy). We also evaluated patient/family preferences in use of technology for being contacted about research. We surveyed a convenience sample of adults, and parents of children with SCD, all genotypes. We used the Mid-South Clinical Data Research Network 3 survey to gather socio-demographic information, depression symptoms (Patient Health Questionnaire—PHQ-2) and health literacy (Brief Health Literacy Screening). We assessed participant interest in varying research types and asked if they would want to be contacted for research using electronic communication—e-mail, text messaging, and social media—as well as traditional face-to-face conversations in clinic or phone calls. We examined differences in demographic and psychosocial variables between participants who responded they were interested in each research type versus those that were not (study outcome) using Fisher's exact and Wilcoxon tests, and created logistic regression models for each research type, analyses performed in R version 3.2.2. Sample characteristics included: 211 adults and 331 parents of children with SCD; ages 1–70 years for individuals with SCD; over 50% female; 97% Black/African American race, 2% Hispanic ethnicity. Median education for adults with SCD and parents was some college education, and 75% reported "good" health literacy. However, 45% reported it was "somewhat" to "very difficult" to pay monthly bills. Twenty-two percent of adults obtained scores of ≥3 on the PHQ-2 (vs. 14% for pediatrics). Most had participated in research previously, with adults (63%) participating more frequently than pediatrics (56.5%). Most participants (95%) expressed interest in research, preferring text messaging (52.1% of adults) and e-mail (58.9% of parents) as modes of contact. Participants were most interested in completing research surveys (83%), followed by studies involving phone/internet contact or community meetings (76%); and giving blood samples (74%). Adults and parents were least interested in participating in studies where they would have to take medication (58%) or where they/their child would have to stay in the hospital (52%). Adults, and parents of children with SCD were more likely to express interest in several types of research if they had previously participated in research (Table I in Supporting Information). Higher health literacy increased the odds of interest in giving a blood sample for parents. Adults from the Mid-South were less likely to express interest in studies involving medications, compared with the Midwest and West. Adults with depressive symptoms were more likely to express interest in medication studies, while parents of children with more symptoms of depression were less interested in medication studies. Parents were more likely to express interest in studies requiring hospital admission if they had difficulty paying bills and higher scores on the PHQ-2. In regression analyses, when other variables were in the model, female gender predicted interest in completing surveys, and depressive symptoms predicted interest in medication studies and studies requiring hospitalization for adults. For parents, lower health literacy predicted interest in research involving hospitalization; depression symptoms and higher health literacy predicted interest in giving a blood sample, and male gender, younger age of child and depression symptoms predicted interest in phone/internet studies. Our hypotheses were partially supported, as we found previous research participation was associated with interest in subsequent research participation, for adults and parents 4, and lower health literacy was associated with less interest in some research types, for parents. Contrary to expectations, parents expressed greater interest in studies requiring hospital admissions if they had difficulty paying bills, and in studies involving giving blood samples if they had more depressive symptoms; and adults with more depression symptoms expressed greater interest in studies requiring hospitalization or taking medication. The only regional variation we found was that adults from the Mid-South expressed less interest in studies involving medications, possibly related to lower prior participation compared with the other two regions. Our findings are critical to consider, as exploration of new pharmacotherapeutic approaches is growing rapidly, yet more than half of adults, and parents of children with SCD, were not interested in participating in studies involving taking medication or hospital admission. Our results may reflect poor communication between providers/researchers and patients or historic mistrust 5. Our findings that depressive symptoms in both adults and parents of children with SCD, and difficulty paying bills for parents were associated with greater interest in more invasive research may highlight some of the cognitive challenges 6 and vulnerability of the SCD population. We recognize the limitations with our survey that relied on self-report about future events and that did not directly evaluate the influence of trust, concerns about harm, issues in relation to time commitments and cognitive impairment. Our convenience sample may not be representative of the broader population of patients with SCD although we demonstrated that adults with SCD and parents in several regions across the U.S. are interested in research participation. Our findings support the need for clear, detailed information about research, at appropriate literacy levels, delivered through channels that are attractive to potential research participants (i.e., email and text messaging). When designing educational materials or approaching patients, researchers must be vigilant those individuals with SCD and cognitive impairment, and/or those with lower income and depressive symptoms are not being subtly coerced into research participation, and that negative perceptions are addressed, given past research misuses. By understanding barriers to research participation for individuals with SCD with different characteristics and across the lifespan, we can create approaches that improve recruitment rates in clinical trials, thereby improving health outcomes in SCD. This work was supported by PCORI Grant Number CDRN 1501-26498. The authors thank the members of the Vanderbilt-Meharry Center of Excellence in Sickle Cell Disease for their thoughtful and helpful comments in reviewing the manuscript; Bertha Davis for regulatory matters; Brittany L. Myers, DNP, RN for support with patient recruitment; and Natasha Dean for assistance with preparation of this manuscript. Robert M. Cronin,1,2,3* Jane S. Hankins,4 Patricia Adams-Graves,5 Alexis A. Thompson,6 Karen Kalinyak,7 Jeannie Byrd,8 Brandi McClain,8 Adetola Kassim,9 Michael DeBaun,8 and Marsha Treadwell10 1Department of Biomedical Informatics, Vanderbilt University Medical Center, Nashville, Tennessee; 2Department of Internal Medicine, Vanderbilt University Medical Center, Nashville, Tennessee; 3Department of Pediatrics, Vanderbilt University Medical Center, Nashville, Tennessee; 4Department of Hematology, St Jude Children's Research Hospital, Memphis, Tennessee; 5Department of General Internal Medicine, University of Tennessee Health Science Center, Memphis, Tennessee; 6Department of Pediatrics, Department of Medicine, Northwestern University, Chicago, Illinois; 7Division of Hematology in Cancer and Blood Disease Institute, Cincinnati Children's Hospital, Cincinnati, Ohio; 8Department of Pediatrics, Division of Hematology/Oncology, Vanderbilt-Meharry Center for Excellence in Sickle Cell Disease, Vanderbilt University Medical Center, Nashville, Tennessee; 9Department of Medicine, Division of Hematology/Oncology, Vanderbilt-Meharry Center for Excellence in Sickle Cell Disease, Vanderbilt University Medical Center, Nashville, Tennessee; 10Department of Hematology/Oncology, UCSF Benioff Children's Hospital Oakland, Oakland, California Additional Supporting Information may be found in the online version of this article. Supporting Information Table 1. Please note: The publisher is not responsible for the content or functionality of any supporting information supplied by the authors. Any queries (other than missing content) should be directed to the corresponding author for the article.