The Science Tooth Fairy: a Randomized Trial of a Storytelling-Based Educational Intervention to Promote Primary Tooth Donation in a Pediatric Setting

Background and SignificanceTeeth are a unique type of biospecimen that can permanently hold information about healthacross the lifespan, unlike other types of biospecimen like blood, nails, and hair.Primary or "baby" teeth specifically can contain information about early life development1‐3 because they start to form during a sensitive period of brain development9‐10. Thestudy of primary teeth can show exposure to early childhood adversity and other relevantdevelopmental markers.Teeth are becoming a more common type of biospecimen donation to research because thenatural removal of them is pain‐free, they are widely available, and they do not requirespecialized storage protocols. Although there are many benefits to teeth donation toscience, researchers have experienced mixed success in collecting teeth relative to otherbiospecimens11‐13. Further, there is research to show that tooth donation rates appeareven lower in minority and disadvantaged groups12,16. Yet, these groups may experienceearly‐life adversity at higher prevalence14‐15. Little research has been conducted toidentify why there is this disparity in tooth donation.The first step to explore the factors that determine tooth donation is to understandwillingness and attitudes for this type of biospecimen collection. Based on the currentliterature, there are multiple known factors that affect donation of non‐toothbiospecimen to research studies4‐8. Some factors that are potentially modifiable includetrust in research and security of participants' personal health information4,5,8,17‐19,knowledge about donation5‐7, and beliefs that donations benefit future generations5,8.Some potentially non‐modifiable factors that affect biospecimen donation are educationstatus6,18,20 and religious or cultural beliefs. However, there are little studies thathave investigated parents' attitudes and beliefs about donating their children's primaryteeth to science.To better understand the attitudes and willingness of parents to donate their children'sprimary teeth to science, parent and child pairs will be introduced to an educationalbook called "The Science Tooth Fairy", and their views will be assessed. This children'sbook, written by Dr. Dunn and team and being used in the STRONG Study (protocol2019P003570), highlights the importance of tooth donation to research and explains theprocess of donation. We are hoping to understand factors that influence tooth donation sowe can alter recruitment strategies and address concerns earlier in future studies toboost participation.2. Specific Aims and ObjectivesHere, we aim to investigate parental attitudes and willingness to donate tooth,specifically, children's exfoliated teeth. We would like to investigate the extent towhich a children's book about teeth donations can encourage participation in research.There is little research examining parent's attitudes and beliefs about donating theirchildren's primary teeth.We hypothesize that participants who read "The Science Tooth Fairy" book will have apositive shift in attitudes about donation primary teeth to science. After theeducational intervention, they will be more open to the idea of donating primary teeth tobiomedical research than the group that reads the control book. Further, we alsohypothesize that attitudes will be more shifted positively for participants who are moreunlikely to donate biospecimen to research (people of color and lower socioeconomicstatus12,16 ).3. General Description of Study DesignThis study is a pre‐post research design with an educational intervention. Theintervention (book or video) will be used to assess if attitudes on biospecimen donationwill change with education about the topic. This is a randomized controlled study withfour possible interventions: 1. Read printed copy of the "The Science Tooth Fairy" book (n=25) 2. Watch video of "The Science Tooth Fairy" video (n=25) 3. Watch the "The Science Tooth Fairy" video and have the option to also read the book(n=25) 4. Read "Cavities vs.Toothpaste" book about oral health (n=25). This control children'sbook does not include any information about tooth donation or research studies.We hope to recruit up to 120 participants to this study. The entire participation processwill be about 30‐45 minutes which includes the consent process, demographics survey,pre‐test, intervention, post‐test, and another demographics survey. The pre and post‐testcontain the same questions.4. Subject SelectionParticipants eligible for this research study include parents of children who are betweenthe ages of 4‐10 years old. In this study, parents will be defined as a caregiver orguardian to a child, including biological children and non‐biological children.Non‐English‐speaking parents will be excluded from this study due to language of theintervention and survey and funding limitations on translating the book into otherlanguages at this time. Eligible participants will be identified at the pediatricsclinics including at MGH Revere HealthCare Center, and other clinical spaces at MassGeneral Brigham including pediatric dental clinics, public events held such as theCambridge Science Festival and other outside MGB clinical practices as available to us.About 120 participants will be recruited.We will be recruiting in diverse spaces, including the Revere clinic, as we hope torecruit from minority populations to help us understand disparities in tooth donationfrom a diverse sample. We will be primarily recruiting at pediatric and pediatric dentalclinics because of the increased volume of parents with children within our desired agerange and our collaboration with clinicians in this clinic.In a clinic setting, the study staff will help identify eligible participants a coupledays in advance. Eligible participants will be handed a flyer by the clinic's front‐deskstaff or study staff. In the waiting room, study staff will be visible with signageintroducing the study. Providers will also present the study to eligible participantsafter their appointment and hand them a flyer. If interested, the provider will provide awarm hand‐off to the study staff in the waiting room. Our study staff will then go overconsent documents and begin participation in the study.Recruitment at public events such as the Cambridge Science Festival will involve handinga flyer to parents with children in the right age range. If interested, we will proceedwith the consent and study as outlined.5. Subject EnrollmentPotential participants will be identified by study staff and clinic front‐desk staff tosee if they meet eligibility criteria. The study staff/front‐desk staff will giveeligible participants a flyer to introduce the study and present the study‐staff in thewaiting room. In clinics, if the parent is eligible, the pediatrician will also introducethe research study through a flyer. If the participant is interested to participate inthe study, the provider or staff member will introduce the participant to a clinicalresearch coordinator. The clinical research coordinator will lead the participant to aprivate room in the clinic or remain in the waiting area if the participant iscomfortable participating in an only semi‐private space. In public event spaces, theinterviews will be done in as private a space as possible. We believe those participantswho will read the book may be able to participate in the waiting area/in public spaces.This will aid the study staff as there may not be enough private space to have twoparticipants completing the study at the same time.The clinical research coordinator will go through the consent information with theparticipant immediately before any study procedures. The clinical research coordinatorwill be available for any questions or clarifications needed. Consent forms will besigned on RedCap or using a paper copy, whichever is preferred by the participant.After consent is documented, the participant's intervention will be assigned through arandomization method. This randomization method will be done through Redcap to determinewhich of the four intervention groups the participant will be assigned. Once assignmentto one of the groups occurs, the participants will then be given the first questionnaire.6. STUDY PROCEDURESParticipation in this study will last 30‐45 minutes and will be completed in one‐sitting.During this time period, the consent document will be presented and signed, therandomized assignment to one of the four intervention groups will take place, thedemographic questionnaire will be completed, the pre‐test will be administered, theintervention will be given, and the post‐test will be administered. Study enrollment willcease when the participant leaves the clinic or if they decide to opt‐out at any timeduring the process. Participation in this study will not interfere with patient care withthe clinic.Once recruited, the study procedures are as follows: 1. Consent obtained 2. Demographics Survey: asking if participants have joined a research study before orif they have ever donated any biospecimen to science 3. Pre‐assessment: 14 items looking at attitudes towards biomedical research and teethdonation 4. Intervention 5. Post‐assessment: 14 items identical to the pre‐test 6. Demographics Survey: asking about age, race, ethnicity, educational attainment,employment status, annual household income, marital status, number of children caredfor.The pre‐ and post‐ assessments contain the same items, and the possible answers areon a 5‐point Likert scale. The order of the items will not be identical for bothassessments. If uncomfortable, participants can choose to refrain from answering anyquestions by selecting "prefer not to answer." Surveys will either be filled outdirectly into RedCap by participants on iPads or on a paper‐copy with the help of aclinical research coordinator.The items asked in the pre‐ and post‐ intervention assessments are compiled frompreviously established measures and scales used in other research studies. Theseinclude the Biobanking Attitudes and Knowledge Survey21, the Helping AttitudesScale23, and the Biomedical Research Trust Scale22. Some items from these scaleswere modified to become more easily accessible for participants in our study.Participants will be randomly assigned to one of the aforementioned interventiongroups by the randomization feature in RedCap. After assignment, the parents will begiven the intervention: either a printed‐copy book or video to be watched on theiPad or both. We expect parents will read the books to their child and/or watch thevideo with their child, however this is not necessary for the study.Participants will be compensated for their participation in the study aftercompletion of all surveys. They will have the option of a $20.00 gift card fromTarget or Stop & Shop. We will also give them a copy of The Science Tooth Fairy bookas a gift. They will be given this compensation before they leave the clinic.Participation in the study will be terminated if the participant does not want tocontinue. If a participant decides to withdraw before completion of the tasks, wewill still offer them The Science Tooth Fairy book as a gift.The primary outcome of this study is to see a difference in attitudes aboutbiospecimen donation, but specifically primary teeth donation. Using thepreestablished measures and scales, we will identify changes by comparing theintervention group with the control to see if attitudes change. A secondary outcomeof this study is to see if there are more pronounced differences within groups basedon demographic information provided or prior beliefs about tooth donation. 7. Risks and DiscomfortsParticipants will be informed that there are no foreseeable physical risks from thisresearch study.Participants may feel slightly uncomfortable completing some of the questionnaires.Participants will be reminded that they are not required to answer any questionsthat make them feel uncomfortable and are able to withdraw their participation atany time. 8. BenefitsParticipants will be informed that there may be no direct benefit from participationin this research study. However, they will be informed that the information learnedfrom this study may provide insight into different attitudes about primary teethdonation and trust in medical research. 9. Statistical AnalysisTo test our primary hypotheses, we will compare our four groups in terms of theoutcome measures to determine what level of change occurred post‐ versuspre‐intervention. For these analyses, we will calculate change scores for eachoutcome measure and use regression analysis to identify the predictors of thosechange scores, using the four groups as our predictor variables. Depending ondemographic differences in these groups at baseline (i.e., if randomization worked),we may or may not include social‐demographic factors as covariates in the analysis.This regression will allow us to quantify the direction and magnitude of changeacross groups. Further, to test our secondary hypothesis, we will additionallyexamine the role of social‐demographics as possible predictors of our analysis. Weexpect that with 30 participants per group we will have at least 80% statisticalpower to detect a change across groups (comparing all groups to each other). 10. Monitoring and Quality AssuranceWe don't foresee any adverse events as a result of taking part in this study. If anadverse event does occur, they will be documented and reported to the IRB by the PI.