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      Engaging Caregivers and Providers of Children With Sickle Cell Anemia in Shared Decision Making for Hydroxyurea: Protocol for a Multicenter Randomized Controlled Trial

      , BA, MA, PhD 1 , , , PhD 2 , , BA 3 , , MS 2 , , PhD 2 , , PhD 2 , , DHPE, MCHES 4 , 5 , , MD 4 , 6 , , MD 4 , 6 , , MD 4 , 6 , , BS 7 , , MD 8 , 9 , , MD 8 , 9 , , MD 10 , , MD 11 , , MD 12 , 13 , , MD 14 , 15 , , MD 15 , , MD 16 , , MD 17 , , PhD 18 , 19 , , PhD 18 , 19 , , MD 20 , 21 , 22 , , MD 23 , , MD 24 , , MD 25 , , MD 4 , 26 , 27 , , MD 28 , , PhD 29 , , MD 4 , 30 , , MD 4 , 5 , , PsyD 2 , 4 , 27
      JMIR Research Protocols
      JMIR Publications
      dissemination, decisional uncertainty, quality of care, child health, NHLBI guidelines

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          Sickle cell anemia (SCA) is a genetic blood disorder that puts children at a risk of serious medical complications, early morbidity and mortality, and high health care utilization. Until recently, hydroxyurea was the only disease-modifying treatment for this life-threatening disease and has remained the only option for children younger than 5 years. Evidence-based guidelines recommend using a shared decision-making (SDM) approach for offering hydroxyurea to children with SCA (HbSS or HbS/β0 thalassemia) aged as early as 9 months. However, the uptake remains suboptimal, likely because caregivers lack information about hydroxyurea and have concerns about its safety and potential long-term side effects. Moreover, clinicians do not routinely receive training or tools, especially those that provide medical evidence and consider caregivers’ preferences and values, to facilitate a shared discussion with caregivers.


          The aim of this study is to understand how best to help parents of young children with sickle cell disease and their clinicians have a shared discussion about hydroxyurea (one that considers medical evidence and parent values and preferences).


          We designed our study to compare the effectiveness of two methods for disseminating hydroxyurea guidelines to facilitate SDM: a clinician pocket guide (ie, usual care) and a clinician hydroxyurea SDM toolkit (H-SDM toolkit). Our primary outcomes are caregiver reports of decisional uncertainty and knowledge of hydroxyurea. The study also assesses the number of children (aged 0-5 years) who were offered and prescribed hydroxyurea and the resultant health outcomes.


          The Ethics Committee of the Cincinnati Children’s Hospital Medical Center approved this study in November 2017. As of February 2021, we have enrolled 120 caregiver participants.


          The long-term objective of this study is to improve the quality of care for children with SCA. Using multicomponent dissemination methods developed in partnership with key stakeholders and designed to address barriers to high-quality care, caregivers of patients with SCA can make informed and shared decisions about their health.

          Trial Registration

          ClinicalTrials.gov NCT03442114; https://clinicaltrials.gov/ct2/show/NCT03442114

          International Registered Report Identifier (IRRID)


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          Most cited references53

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          Controlling the False Discovery Rate: A Practical and Powerful Approach to Multiple Testing

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            Research electronic data capture (REDCap)--a metadata-driven methodology and workflow process for providing translational research informatics support.

            Research electronic data capture (REDCap) is a novel workflow methodology and software solution designed for rapid development and deployment of electronic data capture tools to support clinical and translational research. We present: (1) a brief description of the REDCap metadata-driven software toolset; (2) detail concerning the capture and use of study-related metadata from scientific research teams; (3) measures of impact for REDCap; (4) details concerning a consortium network of domestic and international institutions collaborating on the project; and (5) strengths and limitations of the REDCap system. REDCap is currently supporting 286 translational research projects in a growing collaborative network including 27 active partner institutions.
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              Centering predictor variables in cross-sectional multilevel models: a new look at an old issue.

              Appropriately centering Level 1 predictors is vital to the interpretation of intercept and slope parameters in multilevel models (MLMs). The issue of centering has been discussed in the literature, but it is still widely misunderstood. The purpose of this article is to provide a detailed overview of grand mean centering and group mean centering in the context of 2-level MLMs. The authors begin with a basic overview of centering and explore the differences between grand and group mean centering in the context of some prototypical research questions. Empirical analyses of artificial data sets are used to illustrate key points throughout. The article provides a number of practical recommendations designed to facilitate centering decisions in MLM applications. Copyright 2007 APA, all rights reserved.

                Author and article information

                JMIR Res Protoc
                JMIR Res Protoc
                JMIR Research Protocols
                JMIR Publications (Toronto, Canada )
                May 2021
                21 May 2021
                : 10
                : 5
                : e27650
                [1 ] Developmental Neurosciences Institute of Child Health University College London London United Kingdom
                [2 ] Behavioral Medicine and Clinical Psychology Cincinnati Children's Hospital Medical Center Cincinnati, OH United States
                [3 ] Department of Psychology Oklahoma State University Stillwater, OK United States
                [4 ] Department of Pediatrics University of Cincinnati College of Medicine Cincinnati, OH United States
                [5 ] Division of Hematology Cincinnati Children's Hospital Medical Center Cincinnati, OH United States
                [6 ] Division of General and Community Pediatrics Cincinnati Children's Hospital Medical Center University of Cincinnati College of Medicine Cincinnati, OH United States
                [7 ] Cincinnati Children's Hospital Medical Center Partner Cincinnati, OH United States
                [8 ] Department of Pediatrics Feinberg School of Medicine Northwestern University Chicago, IL United States
                [9 ] Division of Hematology, Oncology and Stem Cell Transplant Ann & Robert Lurie Children's Hospital of Chicago Chicago, IL United States
                [10 ] Center for Child Health Policy and Advocacy Baylor College of Medicine Houston, TX United States
                [11 ] Department of Pediatrics Division of Hematology/Oncology Baylor College of Medicine Houston, TX United States
                [12 ] Children’s Hospital of Philadelphia Philadelphia, PA United States
                [13 ] Perelman School of Medicine Philadelphia, PA United States
                [14 ] Program in Occupational Therapy and Pediatrics Division of Hematology and Oncology Washington University School of Medicine and St. Louis Children’s Hospital St. Louis, MO United States
                [15 ] Division of Pediatric Hematology/Oncology Washington University School of Medicine St. Louis, MO United States
                [16 ] Center for Innovation in Pediatric Practice Division of Pediatric Hematology/Oncology/BMT Nationwide Children's Hospital Columbus, OH United States
                [17 ] University Hospitals Rainbow Babies and Children's Hospital Cleveland, OH United States
                [18 ] Center for Healthcare Delivery Science Nemours Children’s Health System Wilmington, DE United States
                [19 ] Division of Behavioral Health Nemours/ Alfred I duPont Hospital for Children Wilmington, DE United States
                [20 ] Department of Hematology/Oncology Children's Hospital Oakland Oakland, CA United States
                [21 ] AbbVie North Chicago, IL United States
                [22 ] University of California San Francisco San Francisco, CA United States
                [23 ] Pediatrics, Pediatric Hematology/Oncology Indiana Hemophilia and Thrombosis Center Indianapolis, IN United States
                [24 ] Pediatric Hematology and Oncology Boston Medical Center Boston, MA United States
                [25 ] Pediatrics and Child Health College of Medicine Howard University Washington, DC United States
                [26 ] Adolescent and Transition Medicine Cincinnati Children’s Hospital Medical Center Cincinnati, OH United States
                [27 ] James M. Anderson Center for Health Systems Excellence Cincinnati Children’s Hospital Medical Center Cincinnati, OH United States
                [28 ] Department of Pediatrics University of Illinois College of Medicine Peoria, IL United States
                [29 ] Department of Hematology/Oncology University of California San Francisco Benioff Children’s Hospital Oakland, CA United States
                [30 ] Cancer and Blood Diseases Institute Division of Hematology Cincinnati Children’s Hospital Medical Center Cincinnati, OH United States
                Author notes
                Corresponding Author: Anna M Hood a.hood@ 123456ucl.ac.uk
                Author information
                ©Anna M Hood, Heather Strong, Cara Nwankwo, Yolanda Johnson, James Peugh, Constance A Mara, Lisa M Shook, William B Brinkman, Francis J Real, Melissa D Klein, Rogelle Hackworth, Sherif M Badawy, Alexis A Thompson, Jean L Raphael, Amber M Yates, Kim Smith-Whitley, Allison A King, Cecelia Calhoun, Susan E Creary, Connie M Piccone, Aimee K Hildenbrand, Steven K Reader, Lynne Neumayr, Emily R Meier, Amy E Sobota, Sohail Rana, Maria Britto, Kay L Saving, Marsha Treadwell, Charles T Quinn, Russell E Ware, Lori E Crosby. Originally published in JMIR Research Protocols (https://www.researchprotocols.org), 21.05.2021.

                This is an open-access article distributed under the terms of the Creative Commons Attribution License ( https://creativecommons.org/licenses/by/4.0/), which permits unrestricted use, distribution, and reproduction in any medium, provided the original work, first published in JMIR Research Protocols, is properly cited. The complete bibliographic information, a link to the original publication on http://www.researchprotocols.org, as well as this copyright and license information must be included.

                : 4 February 2021
                : 5 February 2021
                : 17 February 2021
                : 22 February 2021
                Custom metadata
                This paper was peer reviewed by the Patient-Centered Outcomes Research Institute (PCORI). See the Multimedia Appendix for the peer-review report;

                dissemination,decisional uncertainty,quality of care,child health,nhlbi guidelines


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