Increasing Documentation and Disclosure of Sickle Cell Trait Status: An Implementation Science Approach

The hemoglobinopathy newborn screen (NBS) performed on all neonates in the U.S. allows for early life-saving medical care for infants with sickle cell disease (SCD), an autosomal recessive genetic disorder. Because of its detection method, the NBS incidentally reveals hemoglobinopathy traits including sickle cell trait (SCT). In an effort to uphold the rights of the newborn to their medical data and preserve autonomy in medical decision making, pediatric and genetic society guidelines recommend disclosure and documentation of SCT results during infancy. Despite this guidance, a large guideline-to-practice gap exists: SCT status is grossly under-documented in the pediatric electronic health record and few adults report knowing their SCT status despite universal screening. We plan to evaluate the effect of a toolkit of SCT Documentation and Disclosure (SCT-DD) strategies on documentation and disclosure of SCT by pediatric primary care providers in a 2-arm randomized interrupted time series trial.

Study Overview

• Status: Recruiting
• Conditions: Sickle Cell Trait
• Intervention / Treatment: Behavioral: SCT Documentation and Disclosure Toolkit (SCT-DD)

• Study Type: Interventional
• Enrollment (Estimated): 500
• Phase: Not Applicable

Contacts and Locations

• Study Contact: Name: Corinna Schultz, MD, MSHP; Phone Number: 3023313088; Email: corinna.schultz@nemours.org

Study Locations

• United States
  • Delaware
    • Wilmington, Delaware, United States, 19803
      • Recruiting
      • Nemours Children's Hospital, Delware
      • Contact: Corinna Schultz; Phone Number: 302-651-5500; Email: corinna.schultz@nemours.org

Participation Criteria

Eligibility Criteria

• Ages Eligible for Study: Child; Adult; Older Adult
• Accepts Healthy Volunteers: No

Description

Inclusion Criteria:

• Outpatient pediatric primary care providers within Nemours and their patients

Exclusion Criteria:

• none

Study Plan

How is the study designed?

Design Details

• Primary Purpose: Health Services Research
• Allocation: Randomized
• Interventional Model: Sequential Assignment
• Masking: None (Open Label)

Number of Arms

2

Arms and Interventions

Participant Group / Arm	Intervention / Treatment
Active Comparator: "All-in"; In the "all-in" arm, pediatric primary care physicians receive all toolkit components at once.	Behavioral: SCT Documentation and Disclosure Toolkit (SCT-DD); A toolkit of implementation strategies
Active Comparator: "Add-in"; In the "add-in" arm, pediatric primary care physicians will have sequential addition of toolkit components in 6 week increments	Behavioral: SCT Documentation and Disclosure Toolkit (SCT-DD); A toolkit of implementation strategies

What is the study measuring?

Primary Outcome Measures

Outcome Measure	Measure Description	Time Frame
Penetration	Rate of documentation and disclosure of NBS and SCT by 2 months of age within the medical history section of the electronic health record by chart review.	Every 1 month through study completion, on average 1 year
Acceptability	Acceptability of toolkit components by pediatric primary care providers by survey	Every 6 weeks through study completion, on average 1 year
Self-efficacy	Increase in the intention and confidence to document/discuss SCT result by pediatric primary care providers by survey	Every 6 weeks through study completion, on average 1 year
Feasibility of using toolkit components	Percent of pediatric primary care providers who use individual toolkit components by survey	Every 6 weeks through study completion, on average 1 year

Secondary Outcome Measures

Outcome Measure	Measure Description	Time Frame
Dispersion	Proportion of children over 2 months of age who have SCT newly documented within the electronic health record via chart review	Every 1 month through study completion, on average 1 year
Knowledge	Proportion of caregivers who accurately reported their child's SCT status via survey	Every 1 months through study completion, on average 1 year

Collaborators and Investigators

• Sponsor: Nemours Children's Clinic
• Investigators: Principal Investigator: Corinna Schultz, MD, MSHP, Nemours

Study record dates

Study Major Dates

• Study Start (Actual): January 18, 2024
• Primary Completion (Estimated): July 1, 2024
• Study Completion (Estimated): December 1, 2024

Study Registration Dates

• First Submitted: May 17, 2022
• First Submitted That Met QC Criteria: May 19, 2022
• First Posted (Actual): May 24, 2022

Study Record Updates

• Last Update Posted (Actual): April 9, 2024
• Last Update Submitted That Met QC Criteria: April 8, 2024
• Last Verified: April 1, 2024

More Information

Terms related to this study

• Keywords: pediatric; implementation; electronic health record; newborn screen
• Additional Relevant MeSH Terms: Hematologic Diseases; Genetic Diseases, Inborn; Anemia; Anemia, Hemolytic, Congenital; Anemia, Hemolytic; Hemoglobinopathies; Anemia, Sickle Cell; Sickle Cell Trait
• Other Study ID Numbers: 348506

Plan for Individual participant data (IPD)

• Plan to Share Individual Participant Data (IPD)?: NO

Drug and device information, study documents

• Studies a U.S. FDA-regulated drug product: No
• Studies a U.S. FDA-regulated device product: No