Increasing Documentation and Disclosure of Sickle Cell Trait Status: An Implementation Science Approach
Phase: NA
Conditions: Sickle Cell Trait
Sponsor: Nemours Children's Clinic

Increasing Documentation and Disclosure of Sickle Cell Trait Status: An Implementation 
Science Approach 
 
 
NCT05387564 
 
 
 
 
Document Date: September 1, 2023 
Updated cover page: October 21, 2025 
 
 

 
Increasing Documentation and Disclosure of Sickle Cell Trait Status: An Implementation 
Science Approach: SCTalk 
 
This study will be conducted in full accordance with all applicable Nemours Research Policies 
and Procedures and all applicable Federal and state laws and regulations including 45 CFR 46.  
All episodes of noncompliance will be documented. 
The investigators will perform the study in accordance with this protocol, will obtain consent, and 
will report unanticipated problems involving risks to subjects or others in accordance with 
Nemours IRB Policies and Procedures and all federal requirements. Collection, recording, and 
reporting of data will be accurate and will ensure the privacy, health, and welfare of research 
subjects during and after the study. 
 
A two-arm, randomized, interrupted time series design will compare an “all-in” and “add-in” 
approach to a suite of intervention strategies to increase PCP’s EHR documentation and 
disclosure of NBS results and SCT status during infancy. This is a pilot study. The main goal of 
this pilot study is to establish a reliable estimate of the effect size of a suite of intervention 
strategies. 
 
Primary Objectives (Aim) 
Using an implementation framework we will assess and compare the acceptability, feasibility, 
self-efficacy, and time costs of using the intervention strategies when delivered “all-in” versus 
“add-in” by measuring the following outcomes. 
• 
Acceptability: via PCP survey for each toolkit component.  
• 
Feasibility: Percent of PCPs who use individual toolkit components.  
• 
Adoption/Self-efficacy: PCP survey. Intention and confidence to document/discuss 
NBS/SCT result.  
• 
Cost: PCP survey: Perceived time (in mins) to document and discuss NBS/SCT result, 
effect on other discussion topics during the visits.  
• 
Penetration (via interrupted time series): via EHR extraction. Rate of scanned NBS 
results available in the EHR. Rate of documentation and disclosure of NBS and SCT 
result at < 2 months of age in each of the following: the problem list, the medical history 
section, and progress note. Evaluate monthly: retrospectively (pre intervention) and 
prospectively (post intervention). 
   
Secondary Objective (Aim) 
Assess the effects of the intervention strategies when delivered “all-in” versus “add-in” by 
measuring the following outcomes. 
COMPLIANCE STATEMENT 
STUDY OBJECTIVES 

• 
Dispersion: Via EHR extraction- Proportion of children over 2 months of age who have 
NBS/SCT newly documented (as above).  
• 
Knowledge: Caregiver survey- Proportion of caregivers who accurately reported their 
child’s NBS result.  
• 
Inequities in documentation: Via chart review. Demographic data for each infant: primary 
care site, sex, race, ethnicity, and insurance type.  
• 
Satisfaction: Caregiver survey. 
 
 
 

 
General Schema of Study Design 
A two-arm, randomized, interrupted time series design will compare an “all-in” and “add-in” 
approach. In the “all-in” arm, participants receive all toolkit components at once. In the “add-in” 
arm, participants will have “multiple interruptions” with sequential addition of components in 6 
week increments (Figure – replase 3 month label with 6 week label). This will allow for 
exploration of the added benefit of certain components as compared to the whole toolkit.  
 
Toolkit Components: 
1) Intervention A: EHR “nudge” within the birth history and/or well-child progress note template 
2) Intervention B: “lunch-and-learn” sessions (in person or pre-recorded per office preference) 
3) Intervention C: written educational materials (i.e. post cards, poster, PDF files) to alert PCPs 
to the importance of SCT documentation and “talking points” to facilitate disclosure;  
 
Duration of Study Participation 
Duration of study participation for a site will last a total of approximately 5 months.  During the 5 
months, PCPs will participate in 4 surveys (15 minutes each) and 1 qualitative interview (30 
minutes).  Study duration of caregivers will be for survey completion (5-10 minutes). 
 
Total Number of Study Sites/Total Number of Subjects Projected 
INVESTIGATIONAL PLAN: Overview 

1) Sites: At least 2 sites per arm of the study with at least 10 PCPs in each arm are required.   
2) PCPs:  All PCPs from a given office will be eligible for participation.  This will result in at least 
20 Nemours PCPs at 4 or more office locations in the Delaware Valley. PCP offices will be 
purposively chosen to include a wide racial distribution within their patients. 
3) Caregivers: 500 caregivers of infants with SCT or a normal NBS who are cared for within the 
specified office location. 
  
Study Population 
Participants will be Nemours primary care offices, Nemours PCPs, and caregivers of children 
seen for infant primary care well visits at the enrolled office. 
 
Inclusion Criteria:  
• 
Sites: Nemours primary care locations within the Delaware Valley that provide 
well child care to infants 
• 
PCPs: All Nemours PCPs (physicians and nurse practitioners) at included office 
locations. 
• 
Caregivers: English speaking caregiver of an infant seen at a participating office 
location for a well visit before 2 months of age (2 months = 60 days) with an 
email address available within the EHR.  
 
Exclusion Criteria: 
• 
Caregivers and PCPs: Unable to converse in English.  
• 
Caregivers:  Caregivers of infant seen for a first well visit at 61 days of age or 
older 
Subjects that do not meet all of the enrollment criteria may not be enrolled. Any violations of 
these criteria must be reported in accordance with IRB Policies and Procedures. 
 
Site selection:  Emails will be sent to the physician-in-charge of Nemours outpatient office 
locations within the Delaware Valley with a description of the study and an invitation to 
participate.  For those interested in participating as a site, a REDCap interest survey will be 
included asking for site specific information (i.e. location, name/contact information of physician-
in-charge (PIC), name/contact information of office manager or other person designated by the 
PIC to handle communication about the trial, Number of PCPs at site). Sites will be matched 
and paired to create dyads/triads of locations with similar racial demographics of patients (i.e. a 
dyad with the majority of patients documented as Black or Hispanic, etc).  For sites selected for 
participation in the trial, name/contact information will be collected from the PIC or office 
manager for all PCPs at the office and anyone else they would like to receive communication 
about the trial (i.e. nursing staff).  An introductory email will be sent to all staff identified by the 
PIC/office manager explaining the study. 
Randomization: Sites will then be randomized within each dyad (i.e. one clinic randomized to 
the “all-in” arm and the other to the “add-in” arm).  All PCPs from the same location will be 
assigned to the same trial arm.  
STUDY PROCEDURES 

 
Practice level data from the medical record.  For each practice location: Patient level 
demographics (based on having been seen for a well child exam in the 3 years prior to data 
extraction) will be linked with the primary practice location at the start of the trial.  Practice level 
data will be obtained from a retrospective evaluation of available EHR data and focus on. 
• 
Age and gender distribution 
• 
Race and Ethnicity distribution 
• 
Insurance make-up (Private, Medicaid, other) 
• 
Zipcode to assess median household income of patients seen at that location (census 
data), Childhood Opportunity Index (COI)  
• 
Number and portion of the patient population with SCT documented in the problem list, 
medical history section, and diagnosis codes 
 
PCP Consent and Demographic Survey:  
Toolkit components will be available to PCPs and office staff in all trial sites regardless of their 
participation in the research portion of the trial.  Use of the toolkit components is as an 
educational aid and to facilitate routine patient care.  
 
PCPs will initially be approached for consent for the research portion of the trial prior to toolkit 
initiation. For PCPs who elects to participate in the longitudinal research portion of the trial, a 
general demographic survey will be completed at time of initial consent. The demographic 
survey will be facilitated by REDCap and administered electronically.  PCPs may enroll at any 
time during the trial though first attempt at consent will be before toolkit initiation at with each 
toolkit component rollout and survey point. 
 
“Roll-out” of toolkit components:  
• 
“All-in” arm: The “all-in” trial arm will have access to and be educated regarding the 
intervention components at one time point via email, via discussion at lunch & learn, via 
office meeting, or via virtual meeting or recording (depending on site preference).  
• 
“Add-in” arm: Each PCP in the “add-in” arm will receive the intervention components in 
the same sequence (within a 1 month timeframe of other offices in the “add-in” arm) with 
1 component added every 6 weeks.  
With each toolkit component rollout an email will be sent to the office staff reminding them about 
the project, alerting them to the new component, and notifying them about a virtual Teams 
“office hours” where the study PI will be available for questions or concerns. The study PI will 
also be available for any virtual or in-person education an office site may request (ie at an office 
meeting, etc) 
 
PCP Implementation Surveys: PCPs will receive surveys by email (through REDCap) every 6 
weeks. Survey is expected to take 15 minutes to complete.  For PCPs in the “all-in” arm, the first 
survey will be sent prior to the intervention “roll-out” and every 6 weeks after that. For PCPs in 
the “add-in” arm, the first survey will be sent prior to intervention A “roll-out”, intervention B “roll-
out”, intervention C “roll-out”, and then 6 weeks after. The survey is created specifically for this 
study and focused on the acceptability of using the intervention component, feasibility of using 
the intervention component, the opportunity costs of using the intervention component (the time 
it takes to use the intervention, the perceived effect on other discussion topics with well child 
visits), and the intention and confidence (i.e. self-efficacy) to document and discuss NBS and 
SCT results. 

 
PCP Qualitative Interviews: At completion of the Intervention period, PCP participants will be 
contacted to schedule the qualitative interview.  Interviews will be expected to last ~30 minutes. 
Interviews will be completed over the telephone or using a video conferencing platform.  If video 
conferencing is used, participants will be instructed to keep their camera turned off, so their 
image will not be recorded.  The participant will be notified verbally when the recording has 
started and when it has stopped.   
 
The qualitative interviews will assess their experience with the components and intention to 
document and disclose NBS/SCT results in the future. 
 
Infant Identification:  
Infants seen at a participating site during the retrospective and intervention period for a well 
child visit before 2 months of age will be identified through EHR data extraction on a rolling 
basis (i.e. once a month during the trial) based on ICD10 code for a new patient/well child exam 
(ICD 10: Z00.110, Z00.111, Z00.121, Z00.129, Z00.00, Z00.01 or CPT: 99381, 99382, 99383, 
99384, 99385, 99391, 99392, 99393, 99394, 99395).     
 
Documentation and Discussion of NBS/SCT results for all infants identified: via chart review. 
For each infant identified, the following data will be collected via EHR extraction and manual 
chart review:  
• 
Email 
• 
Preferred language 
• 
MRN (to facilitate manual chart review) 
• 
Gender 
• 
Race, ethnicity 
• 
Insurance 
• 
Zipcode (to assess Childhood Opportunity Index) 
• 
Phase of study they were first seen for a WCC (retrospective, “all-in” A/B/C, Intervention 
A, Intervention B, Intervention C) 
• 
Presence/absence of NBS 
• 
Result of hemoglobinopathy NBS 
• 
Presence/absence of NBS result documented in 1) problem list, 2) medical history, 3) 
progress notes 
• 
Presence/absence of a disclosure conversation about NBS result in progress notes 
(including topics discussed: inheritance patterns, medical implications, reproductive 
implications, hematology/genetics consultation) 
• 
Presence/absence of NBS results in After Visit Summary communication 
 
Caregiver Survey from retrospective period: 
Caregivers of three groups of infants will be surveyed if they are identified as speaking English 
(through EHR demographic data) and with an email address in the EHR:  
• 
Group 1: All infants with SCT seen before 2 months of age within the 6 month 
retrospective period 
• 
Group 2: All infants identified as Black or Hispanic seen before 2 months of age within 
the 6 month retrospective period 

• 
Group 3: Infants seen before 2 months of age within the 3 month retrospective period 
and not included in group 1 or 2 
 
Caregiver Survey during Intervention Period:   
Caregivers from the participating office locations of three groups of infants will be surveyed if 
they are identified as speaking English (through EHR demographic data) and with an email 
address in the EHR:  
• 
Group 1: All infants with SCT  
• 
Group 2: All infants identified as Black or Hispanic 
• 
Group 3: Infants seen in the first half of the month and not included in group 1 or 2 
Caregivers will receive a survey at 2 and 6 months of age (+/- 1 month) delivered by email 
(through REDCap).  Survey is expected to take less than 10 minutes to complete.  Consent and 
survey responses will be obtained electronically. The survey will inquire about results of the 
NBS, preferences for information receipt, and satisfaction with well child visit. 
 
 
• 
See site selection under Study Procedures 
 
• 
PCP Recruitment.  An introductory email (through REDCap) will be sent to all staff 
identified by the PIC/office manager for any site participating in the trial explaining the 
study before the Intervention period. The PI will be available to attend any office 
meetings or question and answer sessions that the PIC or PCPs request.  PCPs will be 
informed that participation in the research components is voluntary, will not affect their 
employment, and that they will receive access to the toolkit components without 
participation. The study coordinator and/or PI will follow-up with PCPs by telephone, 
email, virtual meeting, or in person to answer additional questions.  
 
They will be given the contact information for the PI and study coordinator for any 
questions or withdrawal from participation. Three follow-up requests will be sent to non-
responders at ~1 week intervals and then at 6 week intervals (ie when interventions are 
rolled out and surveys distributed) during study time period.   
 
• 
PCP Retention.  Trial procedures will include every 6 week on-line REDCap facilitated 
surveys and a final qualitative interview. Surveys will include an introductory expression 
of appreciation for their continued participation and reminder of reason for the trial. 
Incentive compensation will be given after each study procedure (survey or qualitative 
interview) is completed. Three reminder e-mails and/or telephone calls will be provided 
for each survey. For surveys, we will also include the option of completing the survey by 
phone or on paper if the participant prefers. We will provide flexibility in timing for the 
final qualitative interview to accommodate their work schedule and maximize 
participation.  PCPs will be compensated $25 per survey they complete and $100 for the 
qualitative interview. PCPs will receive an additional $25 if all four surveys are 
completed. 
 
• 
Caregiver Survey Recruitment: Infants seen at a participating site for a well child visit 
before 2 months of age will be identified through EHR data extraction on a rolling basis 
Recruitment of Participants 

(i.e. at start of the Intervention phase and then every month during the intervention 
phase) and NBS result reviewed if available in the EHR.  Caregivers identified as 
speaking English (through EHR demographic data) and with an email address in the 
EHR will receive a survey at the start of the intervention phase if seen during the 
retrospective phase or at 2 and 6 months of age (+/- 1 month) if seen during the 
intervention phase.  Email will be delivered through REDCap. Consent and survey 
responses will be obtained electronically.  
 
• 
They will be told that participation is voluntary. They will be given the contact information 
for the PI and study coordinator for any questions or withdrawal from participation. The 
on-line survey will be ~5 minutes in length. Three follow-up requests will be sent to non-
responders at ~1 week intervals. Responses will be collected within REDCap.  
Participants be informed that completion of each survey will enter them into a lottery for 
a $100 gift card. Twenty lottery gift cards will be given in total; 10 at 4 month intervals 
after initiation of the trial. Each caregiver that completes a survey will also receive a $10 
gift card. 
 
• 
Caregiver Survey Retention: The only study procedure is completion of a 2 and 6 
month survey. Each time the caregiver completes a survey they will be entered into a 
lottery for a $100 gift card. Each caregiver that completes a survey will also receive a 
$10 gift card. 
 
 
 
For PCPs: One consent form, called an information sheet, has been developed for this study 
that incorporates consent and HIPAA information.  It has been designed to not require a 
signature. We are applying for a waiver of documentation of informed consent and HIPAA and 
alteration of consent.  The phone numbers of the PI, research coordinator, and Nemours IRB 
are provided on the information sheet for potential participants to call or email with any 
questions that might arise regarding the research. This information sheet will be included in the 
REDCap survey link from the study team and attached to the recruitment email.   
An individual REDCap survey link will be emailed to potential participants.  The information 
sheet will be the first item to appear once clicked on.  After reading the page, the participant will 
be instructed to select an “I Agree” or “I Disagree” button as acknowledgement of consent.  
Participants that consent will then click a “submit” button to continue to the first questions of the 
survey. Those that select “I Disagree” will be thanked for their time and the survey window will 
close.   
 
As informed consent is an on-going process, prior to the qualitative interview, all participants will 
be asked to consent to use of a recording device and will be asked to reaffirm their 
consent/assent to participate in the study.  These statements will be documented on the 
interview recording.   
 
For caregiver surveys:  One consent form, called an information sheet, has been developed 
for this study that incorporates consent and HIPAA information.  It has been designed to not 
require a signature. We are applying for a waiver of documentation of informed consent and 
HIPAA and alteration of consent. The phone numbers of the PI, research coordinator, and 
Informed Consent/HIPAA Authorization 

Nemours IRB are provided on the information sheet for potential participants to call or email with 
any questions that might arise regarding the research. This information sheet will be included in 
the REDCap survey link from the study team and attached to the recruitment email.   
An individual REDCap survey link will be emailed to potential participants.  The information 
sheet will be the first item to appear once clicked on.  After reading the page, the participant will 
be instructed to select an “I Agree” or “I Disagree” button as acknowledgement of consent.  
Participants that consent will then click a “submit” button to continue to the first questions of the 
survey. Those that select “I Disagree” will be thanked for their time and the survey window will 
close.   
 
 
The study procedures within this application meet the definition of Behavioral Human Subjects 
research. Human subjects approval from the Institutional Review Board of Nemours will be 
obtained prior to recruitment or any study procedures occurring. All study procedures will take 
place at Nemours. All study personnel will have undergone human subjects training for the 
protection of human subjects. Every effort will be made to assure confidentiality. 
 
For all procedures: The PI and co-investigators will be responsible for data management and 
accuracy of records. The PI may assign designated qualified individuals to perform screening, 
facilitate surveys, conduct interviews, and communicate with study sites. Only the PI and 
assigned research staff under direct supervision from the PI will have access to study data. All 
information collected in this study will be kept confidential as required by law. Upon enrollment, 
subjects will be designated a unique 3-digit identification (ID) study number. Any written 
demographics, recordings, and transcripts will be labeled with the subject’s unique identifier 
number. A master list of participant name or MRN and ID study number will be kept in a secure 
Nemours OneDrive or Sharedrive location only accessible by study investigators and purged 
upon completion of the study. Demographic data of PCPs will be collected directly into REDCap 
and then saved on a secure Nemours OneDrive or Sharedrive location for analysis. Any paper 
based originals will be kept in a locked office. Survey requests will be sent directly via email 
through REDCap or from the study coordinator to the PCP’s Nemours email address or to the 
caregivers’ email provided with the Nemours’ electronic health record. Data extracted from the 
EHR and chart review will be kept on a secure Nemours OneDrive or Sharedrive location 
accessible only by study staff. All data and records generated during this study will be kept 
confidential in accordance with Institutional policies and HIPAA on subject privacy.  The 
Investigator and other site personnel will not use such data and records for any purpose other 
than conducting the study. No identifiable data will be used for future study without first 
obtaining IRB approval. Data from this study will be used for research purposes only. 
 
For qualitative interviews: During qualitative interviews, a semi-structured format with the 
interviewer employing a discussion guide composed of open-ended questions will be used. The 
PI may review selected audiotapes and transcripts of the interviews to monitor the interviewers’ 
performance and to provide further guidance as needed. All interviewers will be trained to 
handle any emergencies that may arise and to be sensitive to the participants. Each semi-
structured interview will be digitally audio recorded and stored on a secure Nemours OneDrive 
or Sharedrive location only accessible to study staff. Recordings will be labeled with the 
participant’s 3-digit identification code. In the event a name or other identifying information is 
mentioned during the interview, it will be removed during the transcription process. Digital audio 
Protection against risk  

recordings will be kept until transcription is complete, checked for accuracy and verified, and will 
then be destroyed.   
 
Control of Bias and Confounding 
We anticipate that responses could vary based on social desirability.  We will test our survey 
and interview scripts beforehand in attempt to limit any leading questions.  Additionally, we will 
utilize two coders for qualitative data during analysis to further reduce any reporting or 
interpretation bias. 
 
 
We are asking for an alteration of the consent process.  Study procedures are brief and 
minimal/no risk. Requiring inperson communication of busy physicians to provide consent would 
be distruptive and time intenstive during patient care hours (ie business hours) and would be 
equally burdensome to parents of young infants.  To decrease the burden of participation, a 
virtual, asynchronous consenting process will allow participants to consider study participation 
while away from patient care hours or child care duties and leisurely consideration of consent 
documents.  All potential participants will have the option to speak with a study team member 
(coordinator or PI) before consent if desired. 
 
We are requesting to waive a signature for consent, as the participants will be asked to 
document consent/assent via REDCap survey, and reaffirm consent/assent verbally prior to the 
qualitative interview. The surveys and qualitative interview presents no more than minimal risk 
to the participants.  Additionally, we are not asking for any additional personal health information 
or identifiers apart from Greenphire information. The consent document would be the only other 
document connecting the survey responses and the PCP.  The participants will be provided with 
an information sheet in recruitment emails and prior to starting the REDCap survey. 
 
We are asking for a waiver of HIPAA authorization.  The survey study involves no more than 
minimal risk to the participant.  Additionally, the HIPAA form would be the only other document 
connecting survey response and the participant.  Because we want the participants to be as 
honest as possible, we do not want the participant to feel that we can connect them to their 
responses and that connection could include the HIPAA documentation.   
PHI will be used 1) to ensure eligibility of participants; 2) to contact for recruitment; and 3) to 
appropriately describe our sample. We will collect identifiable information (i.e. DOB and 
address) for those who receive a gift card to process study payment through Greenphire. This 
data will not be provided to or used for anything aside from payment.  As this study will be 
conducted entirely remotely, it cannot be conducted without access to and use of PHI such as 
contact information.   
Subjects may withdraw from the study at any time without prejudice to their care or employment.  
The PI may withdraw a participant from the study if it is learned that they did not fulfill eligibility 
criteria.  It will be documented whether or not each subject completes the study.  
Alteration of Consent, Waiver of Documentation of Consent and HIPAA 
Authorization 
Subject Completion/Withdrawal 

 
Evaluation of the Toolkit will focus on Proctor’s framework of IS outcomes:47  
Primary Endpoint 
• 
Penetration (via interrupted time series): via EHR extraction. Rate of scanned NBS 
results available in the EHR. Rate of documentation and disclosure of NBS and SCT 
result at < 2 months of age in each of the following: the problem list, the medical history 
section, and progress note. Evaluate monthly: retrospectively (pre intervention) and 
prospectively (post intervention). 
 
Secondary Endpoint 
• 
Acceptability: via PCP survey for each toolkit component.  
• 
Feasibility: Percent of PCPs who use individual toolkit components.  
• 
Adoption/Self-efficacy: PCP survey. Intention and confidence to document/discuss 
NBS/SCT result.  
• 
Cost: PCP survey: Perceived time (in mins) to document and discuss NBS/SCT result, 
effect on other discussion topics during the visits.  
• 
Dispersion: Via EHR extraction- Proportion of children over 2 months of age who have 
NBS/SCT newly documented (as above).  
• 
Knowledge: Caregiver survey- Proportion of caregivers who accurately reported their 
child’s NBS result.  
• 
Inequities in documentation: Via chart review. Demographic data for each infant: primary 
care site, sex, race, ethnicity, and insurance type.  
• 
Satisfaction: Caregiver survey. 
 
Statistical Methods 
Unit of analysis will be the individual PCP (i.e. pre/post) and by trial arm with the primary end 
point being the rate of documentation and disclosure of NBS and SCT (penetration).  
Data will be summarized in numeric and graphic forms. Mean and standard deviation or median 
and interquartile range will be used for summarizing numerical variables and frequencies and 
percentages will be used for categorical variables. PCP’s EHR documentation and disclosure of 
newborn screen (NBS) and sickle cell trait (SCT) carrier results will be presented by pre-post 
intervention as well as by intervention groups. The primary end point of the study is to compare 
the proportion of PCP’s/ EHR documentation and disclosure of NBS and SCT between pre- and 
post- intervention periods.  
 
Generalized linear mixed effects model will be used for the analysis of the primary end-point. 
Odds ratio with confidence interval will be provided for the documentation and disclosure. 
Generalized linear mixed effects model will also be used to compare the proportion of post-
intervention documentation and disclosure over time (monthly) between two intervention groups. 
Mean score of acceptability for each intervention (A, B, C) as well as mean total score for all 
three interventions will be compared between two intervention groups. Similarly, other survey 
results such as Self-Efficacy of PCP to document NBS and time cost will be compared between 
two intervention groups. Also, dispersion, caregiver knowledge and caregiver satisfaction will be 
STATISTICAL CONSIDERATIONS 

compared between two groups. Two sample t-test, Mann-Whitney U-test, Chi-square test, 
analysis of variance (ANCOVA), liner mixed effects model, logistic regression, and mixed effects 
logistic regression will be used, for the above comparisons, as appropriate. PCP nested within 
clinic will be used as the random effects in the model as needed. All tests will be two-tailed with 
an overall level of significance of 0.05. Benjamini-Hochberg method will be used to adjust the 
level of significance for multiple comparisons.  Statistical software packages SPSS, SAS 
(version 9.4) or latest version of R will be used for the analysis.   
 
Sample Size Justification: This is a pilot study and there is no reported effect size on the 
intervention of this study in the literature. Therefore, the main goal of this study is to explore 
reliable effect size that would assist designing future studies with formal hypothesis testing.  The 
primary aim of the study is to increase PCP’s EHR documentation and disclosure of NBS and 
SCT carrier results during infancy (age less than 1 month). It has been found in a chart review 
that 71% of newborns over the last 3 years had a newborn screen result scanned into their 
chart. The goal of this study is to raise this to 90%. A sample size of 74 in each of the pre- and 
post-intervention period can detect the targeted difference in newborn screen of 19% with 86% 
power at the level of significance of 0.05.  PCP documentation has been reported as 20% in the 
literature. We aim to raise this 50%. Again, a sample size of 43 in each of the pre- and post- 
intervention period would be adequate to detect the targeted difference in the PCP 
documentation with a power of 85% at the level of significance of 0.05. Currently 53% sickle cell 
traits has been documented. Once again, the goal is to raise this to 90%. A sample size of 21 
infants with sickle cell traits (SCT) in each of pre- and post-intervention periods would be 
adequate to detect this difference in this documentation with a power of 80% at the level of 
significance of 0.05. In DE, the prevalence of SCT is 2.1% in overall children and about 7% in 
African Americans. Considering 2.1% SCT in overall children, we need 1000 infants to screen in 
each of the pre-and post-intervention periods, to detect the targeted difference with a power of 
80% at the level of significance of 0.05. The study plans to recruit 10 PCPs in each of the 
intervention arm (20 PCP in both arms). About 6-10 infants are expected to be seen by a PCP 
in a month and 54-90 new infants are expected to be seen by a physician during 9 months of 
post intervention period. We expect that minimum of 540 x 2 = 1080 patients (540 for each 
intervention) to be seen by 20 PCPs. Therefore, a minimum sample size of 1080 infants is 
planned for each of the pre-and post-intervention periods for this study. The planned minimum 
sample size of 1080 in the post intervention period would also be adequate to determine the 
effect size for survey results and documentation and disclosures for each intervention arm 
separately.        
 
Analysis of Qualitative Data: 
Interviews will be digitally recorded and transcribed by GMR Transcription, a company vetted 
through Nemours that specializes in transcription of medical data, or via Teams. Transcripts will 
be analyzed by two reviewers. We will develop an a priori coding scheme and dictionary prior to 
initiation of coding based on relevant literature and the researcher’s clinical expertise.  The 
coding scheme will be revised over the first several transcripts and as needed after that.  We 
will utilize a grounded theory approach to build a theoretical framework for understanding the 
interview data.  We will utilize a constant comparative analysis to assess and develop themes.    
We will assess inter-rater reliability.  Any differences in coding will be discussed and negotiated.  
We will utilize Dedoose (an on-line qualitative data software) to assist our analysis.  Content 
analysis will begin shortly after interviews are initiated and continue throughout accrual.   
 

 
Clinical Adverse Events 
Some information collected from the study participants is sensitive information (attitudes, 
information giving preferences, etc) and may cause the subjects emotional discomfort. This risk 
is considered minimal and no more than what is normally associated with being a physician or 
nurse practitioner or routine well child care. Should the participant experience psychological 
distress during or after participation in this study, they will have contact information for the PI 
and study coordinator to connect with appropriate services.  
Adverse event reporting 
Since the study procedures are not greater than minimal risk, significant adverse events (SAE) 
are not expected. If any unanticipated problems related to the research involving risks to 
subjects or others happen during the course of this study (including SAEs) these will be 
reported to the IRB. AEs that are not serious but that are notable and could involve risks to 
subjects will be summarized in narrative or other format and submitted to the IRB at the time of 
continuing review.  
 
Data Collection and Management 
All study related documentation will be accessible only to research team members.  All study 
material will be collected solely for the purpose of this investigation and will be kept in secured 
Nemours OneDrive or ShareDrive locations only accessible by the study staff.  Only the PI and 
other necessary team members will have access to the human subjects’ data and identifying 
information.  All data will be identified only be Subject ID number and the master list linking the 
subjects’ identities to the ID numbers will kept in a separate file.  
A master list of subjects who participate and who refuse will be kept within Excel for study 
duration to assess participation rate and prevent re-contact.  Names/email addresses will not be 
attached to actual study responses.   
The Primary and co-investigators will be responsible for data management and accuracy of 
records.  The PI may assign designated qualified individuals to perform screening and initiate 
survey distribution. Only the principal investigator on this protocol and assigned research staff 
under direct supervision from the principal investigator will have access to study data.  
All information collected in this study will be kept confidential as required by law.  Upon 
enrollment, subjects will be designated a unique study number.  All evaluable data will be 
identified only by Participant ID number and the master list linking the participants’ identities to 
the code numbers will be kept in an Excel/Word file separate from where the data is stored.   
A master list of subjects who participate and who refuse will be kept within Excel and REDCap 
for study duration to assess response rate and assure appropriate compensation.  Names and 
email addresses will not be attached to actual study responses within the evaluable dataset.   
 
Demographic data and survey responses will be collected via participant self-report directly into 
the Nemours’ REDCap[29] secure password-protected server unless requested differently. If a 
participant wished to answer questions verbally, their responses will be directly entered into 
STUDY ADMINISTRATION 
SAFETY MANAGEMENT 

REDCap.  The database will be designed with validation rules to facilitate entry of data and 
completion of all questions but will allow for skipping of questions per the respondents choice. 
 
Qualitative Interviews: Qualitative interviews will be conducted over the telephone or using a 
video conferencing platform such as Teams.  Qualitative data will consist of recordings that will 
be downloaded to the secure Nemours server, labeled with a study ID number, and deleted 
from the recording device. Recordings will then be transferred electronically via a secure 
method to GMR Transcription for processing. The recordings will be transcribed verbatim into 
Microsoft Word and stripped of any identifying information by the transcribing service, which is 
knowledgeable about qualitative data preparation techniques and emphasizes privacy and 
confidentiality. The transcripts will then be sent securely back to the study team, stored on a 
secure Nemours server, and checked against the recordings to ensure accuracy.  The 
recordings will then be deleted.   The PI may review selected audio files and transcripts of the 
interviews to monitor the interviewers’ performance and to provide further guidance as needed.  
All interviewers will be trained to handle any emergencies that may arise and to be sensitive to 
the participants.   
 
Confidentiality 
All data and records generated during this study will be kept confidential in accordance with 
Institutional policies and HIPAA on subject privacy.  The Investigator and other site personnel 
will not use such data and records for any purpose other than conducting the study.  No 
identifiable data will be used for future studies, without first obtaining IRB approval.   
The Investigator and other site personnel will not use such data and records for any purpose 
other than conducting the study.  Name and email address will not be included in the final 
identifiable dataset and will only be used to recruit participants and provide compensation. 
 
Data and Safety Monitoring Plan 
As this study poses no more than minimal risk to participants, the Principal Investigator will 
monitor study progress, ensure the accuracy and security of the data and ensure subject safety.  
Additionally, the Data Safety Monitoring Board of the Sickle Cell COBRE grant will be available. 
Safeguards to maintain subject confidentiality and data security are described in “Data 
Collection and Management” above.      
Risk Assessment 
This project is not greater than minimal risk. There is no physical risk to the PCP, study staff, or 
caregiver participant in the surveys or interviews.  The knowledge gained through the interviews 
and surveys will help assure we are providing NBS and SCT results in ways PCPs and 
caregivers prefer. The major risk to this study is the discussion of sensitive subject matter.  We 
will conduct interviews virtually via telephone or Teams at a time that is convenient to the 
participant. In addition, each interview script will be reviewed by the Stakeholder groups to 
minimize any confrontational or judgmental terminology and assess for face validity and 
comprehension.  
 
REGULATORY AND ETHICAL CONSIDERATIONS 

Since the study procedures are not greater than minimal risk, significant adverse events (SAE) 
are not expected. If any unanticipated problems related to the research involving risks to 
subjects or others happen during the course of this study (including SAEs) these will be 
reported to the IRB in accordance with Nemours IRB policies. AEs that are not serious but that 
are notable and could involve risks to subjects will be summarized in narrative or other format 
and submitted to the IRB at the time of continuing review.  
 
Potential Benefits of Study Participation 
The PCPs within the implementation science pilot trial may receive the following direct benefits: 
improved knowledge of the importance of NBS and SCT results, enhanced self-efficacy (i.e. 
confidence) in the disclosure of NBS and SCT results to their patients, improved rates of 
documentation of NBS and SCT results. If the implementation strategies are found to be 
acceptable, feasible, and enhance penetration of NBS and SCT result documentation and 
disclosure they will be further tested within a multi-site implementation trial. 
 
The caregiver participants may not receive any direct benefit to completing the survey. They 
may receive direct benefit because their PCP is a participant since they may receive additional 
education from their PCP regarding their child’s NBS results. Results from the caregiver 
participants will help to improve the delivery of care for others in the future. 
 
Payment to Subjects/Families 
All compensation will be provided via Greenphire gift card: Name, address, and date of birth will 
be required to facilitate gift cards. 
• 
Primary Care Provider (PCP) Survey Incentives (1 every 6 weeks during the intervention 
phase x 4). $25 per survey. An additional $25 if all four surveys are completed. 
• 
PCP Qualitative Interviews: $100 per interview  
• 
Caregiver Participant Survey Incentives: Each caregiver who completes a survey will be 
entered into a drawing for a $100 gift card for each survey they complete. There will be 
20 lottery gift cards given. Each caregiver that completes the survey will also receive a 
$10 gift card. 
PUBLICATION 
The results of this study may be presented at professional meetings or published in a 
professional journal.  Any personal identifying information will not be included in these 
presentations or publications.