The Shared Decision-Making Tool for Caregivers of Children with Prenatal Opioid Exposure: A Pilot and Acceptability Study

The purpose of this pilot study was to report initial �ndings about developmental concerns expressed by caregivers of children with prenatal opioid exposure using the Shared Decision-Making Tool, and to assess provider acceptability of incorporating the tool into their work�ow. Data were collected from 83 caregivers and �ve providers from a medical clinic and an early intervention service center. Communication was the most frequently cited concern in four consecutive age categories. Inattention/impulsivity was the second most frequently cited concern in three consecutive age categories. Problem behavior was the third most frequently cited concern in three age categories. All providers ‘strongly agreed’ or ‘agreed’ with all statements on the provider feedback survey, except two instances. Data from this pilot study will guide our next research steps, focusing on psychometric testing and usability feedback from caregivers, before expanding trials of the Shared Decision-Making Tool to a wider geographic area. Findings from this pilot study will also inform the development of caregiver resources for managing areas identi�ed as top concerns through the �rst �ve years of childhood.


Introduction
Prenatal Opioid Exposure (POE) has been associated with adverse childhood health and developmental outcomes (Liu et al., 2022;Skumlien et al., 2020;Yeoh et al., 2019).Although the immediate effects of prenatal exposure, such as low birth weight and withdrawal, typically subside within the rst few months after birth, longer-term consequences may continue well into childhood and adolescence (Arter et al., 2021; Oei et al., 2017).While information related to long-term health and developmental outcomes of this population is growing, the current body of literature is primarily composed of studies focusing on the birth to three-year age range.Despite this limitation, identi ed risks for developmental challenges (Azuine et al., 2019;Rubenstein et al., 2018) validate the need to support caregivers to engage in developmental monitoring as their children grow.For all children, regularly scheduled well-child visits is one way to provide developmental surveillance and screening that is essential for the early identi cation and intervention of developmental disorders.Well-child visits occur shortly after birth and extend throughout childhood.These visits can screen for medical conditions, motor skills, behavioral and socio-emotional concerns, and hearing and visual impairments (Lipkin, et al., 2020).Additionally, Lipkin et al. (2020) advise close monitoring of children in the 4 to 5-year age range in particular as they begin their elementary education.However, there is some evidence to suggest that children with POE may be less likely to attend well-child visits (Goyal, et. al., 2020;Jarlenski et al., 2020).
Children with POE may present with a wide range of developmental concerns, including behavioral, cognitive, motor, sensory, socio-emotional, or speech/language.These potential concerns can vary in severity and become more apparent as the child ages.The unpredictable trajectory of development proves challenging for caregivers and providers to anticipate the needs of this population.Therefore, developmental surveillance in educational settings as well as well-child appointments provide crucial opportunities for caregivers to communicate pressing concerns and identify developmental delays in collaboration with their child's medical and educational providers (henceforth jointly referred to as providers).
Effective communication is fundamental to high-quality, patient-and family-centered care, impacting patient and family satisfaction, treatment adherence, and the creation of a non-judgmental environment where relevant information may be freely shared (Leonard, Graham, & Bonacum, 2004).For caregivers with opioid use disorders, stigma is a well-documented barrier to effective communication.This likely contributes to the increased risk of children with POE missing well-child visits during their rst two years of life compared to non-exposed peers (Goyal, et. al., 2020).Screening tools, such as a shared decisionmaking tool, can be helpful in standardizing interactions with caregivers, and ensuring visits meet patient and caregiver needs.
Shared decision-making is a process of deliberation between providers, patients, and families, where the best available evidence for each treatment option is shared, and patients and families are supported to consider these options to achieve individual preferences (Elwyn et al., 2010).Shared decision-making becomes even more important when treating complex conditions, such as behavioral disorders, considering the numerous treatment modalities available and treatment preference can be highly in uenced by family, cultural, or societal values (Lipstein et al., 2016).Shared decision-making has been recognized as vital to positive health outcomes, and is part of the Maternal Child Health Bureau's System of Care for Children with Special Health Care Needs Core Outcomes (Lipstein et al., 2016).Yet, there is a gap in the literature exploring the use of shared decision-making in the care of children with POE.This is a pressing issue, given the increasing incidence of prenatal opioid exposure in children as well as the growing body of literature suggesting children with POE are at risk for complex developmental conditions.
The Shared Decision-Making Tool (SDMT) was developed to facilitate conversation between caregivers of children birth to ve years with a history of POE and the provider, directed by the prioritized areas of need of caregivers to ensure all relevant areas of development are addressed.This tool prompts caregivers to identify concerns in different developmental domains while also establishing a hierarchy of those concerns.The completed tool then serves as the impetus for focused assessments and conversations between caregivers and providers regarding necessary monitoring or interventions to promote the child's unique needs.The purpose of this pilot study is to report initial ndings regarding developmental concerns expressed by caregivers of children with POE using the SDMT and to assess provider acceptability of incorporating the tool into their work ow.Results from this study will highlight areas of development with which caregivers express frequent concern and those that are of top priority, as well as provide valuable feedback with which to improve the usefulness of the tool for providers.

Study Design
A cross-sectional pilot study was conducted to assess the acceptability and preliminary child outcomes using the SDMT.Ethics approval was granted by the hospital's IRB Committee.

Participants
Convenience sampling was used to recruit health care and early intervention providers who care for children 0-5 years with a history of POE to pilot the SDMT.Providers who volunteered to participate were trained to identify appropriate caregiver/child dyads with whom to utilize the tool using the following inclusion criteria: caregiver aged 18 years or older, providing care for a child ages 0-5 years old with a history of or suspected POE, and English is primary language.

Outcome Measures
Shared Decision-Making Tool Caregivers were asked to rate their level of concern for each area of their child's development using a ve-point Likert-type verbal scale ranging from 'no concern' to 'extreme concern.'A visual analog scale employing faces showing increasing levels of concern was also included alongside the Likert-type verbal scale to provide clarity in answer choices.Caregivers were instructed to rank their top three areas of concern to communicate their priorities to the provider.See Fig. 1 for an image of the tool.SDMTs were lled out using pen and paper.Investigators collected the completed tools from the study sites and manually entered the data into a study database.

Provider Acceptability Questionnaire
Provider acceptability of the SDMT was measured using a questionnaire developed by the investigators.
The questionnaire consisted of nine statements with which providers were asked to rate their level of agreement using a ve-point Likert-type scale ranging from 'strongly disagree' to 'strongly agree.' Providers could add free-text comments at the end of the survey for additional feedback.Provider acceptability data were collected and managed using Research Electronic Data Capture (REDCap) tools hosted at Cincinnati Children's Hospital Medical Center.REDCap is a secure, web-based software platform designed to support data capture for research studies, providing an intuitive interface for validated data capture, audit trails for tracking data manipulation and export procedures, automated export procedures for seamless data downloads to common statistical packages and procedures for data integration and interoperability with external sources (Harris et al., 2019).

Data Analysis
SDMT data were anonymous and only included the child's age, scoring of the caregiver's concern in each area of development, and ranking of the caregiver's top three areas of concern.Descriptive statistics were used to identify areas of developmental concern and prioritization of concerns categorized by age: 0-1 year, 1-2 years, 2-3 years, 3-4 years, and 4-5 years.To identify areas of concern, we examined response frequency and mean level of concern.Response frequency was determined by assigning a point value of 0 to 'no concern' responses and a point value of 1 for any response above 'no concern.'Response values were then summed for each developmental area.Mean level of concern was determined by assigning each item response option a point value ranging from 0 ('no concern') to 4 ('extreme concern').Item response values were summed then divided by the number of responses above 'no concern.'Regarding the caregiver's prioritization of concerns, point values were assigned according to the caregiver's designated rst, second, and third areas of concern.First priority concerns were assigned three points, second priority concerns were assigned two points, and third priority concerns were assigned one point.Rankings were used to ensure that the top priorities of the caregiver were addressed during the visit even if the provider's concerns did not align with the caregiver's priorities Provider acceptability data were also anonymous and included years of experience as a provider, years of experience caring for children with POE, and scoring of the acceptability questionnaire.Descriptive statistics were used to summarize provider feedback data.All study data can be made available upon request to the authors.

Developmental Concerns
One medical clinic and one community-based service center participated in this pilot study with a total of 117 completed Shared Decision-Making Tools collected.Duplicate questionnaires were eliminated, leading to a sample size of 83 questionnaires lled out by caregivers of children with POE.All of the Shared Decision-Making Tool data categorized by age group can be found in the supplemental le.Below, the top developmental concerns and prioritization of concerns for the age groups are highlighted in Table 1.Only one caregiver ranked developmental concerns in the 0-1 year age group, so ranked group data on caregiver priority concerns in that age group is not available.

Provider Acceptability
Data were collected from ve providers from both study sites related to their feedback on use of the SDMT.Three were medical providers (physicians or advanced practice registered nurses) and two were early intervention specialists.Experience working as a provider ranged from 2-11 years (mean 8.6 years).Experience working with children with POE ranged from 3 months-12 years (mean 8.1 years).Provider feedback results can be found in Table 2.All providers either 'strongly agreed' or 'agreed' that the SDMT helped the caregiver with de ning or explaining their concerns and helped the provider identify concerns and make referrals for the patient.Four providers either 'strongly agreed' or 'agreed' that the SDMT helped them share professional knowledge and/or recommendations with the caregiver, and one provider was 'neutral.'All providers either 'strongly agreed' or 'agreed' that the SDMT helped check their understanding of the caregiver's concerns, and helped with identifying caregiver priorities regarding their child's development.All providers either 'strongly agreed' or 'agreed' that the SDMT was easy to and worth integrating into their current work ow.Four providers either 'strongly agreed' or 'agreed' that the caregiver understood how to ll out the SDMT, but one provider 'disagreed.'As a result of using the SDMT, all providers 'strongly agreed' or 'agreed' that they feel con dent in their ability to provide referrals and/or recommendations to caregivers of this patient population.

Survey Question Strongly
Agree Agree Neutral Disagree The SDMT helped the caregiver with de ning or explaining their concerns 3 2 0 0 The SDMT helped me identify concerns and make referrals for the patient 3 2 0 0 The SDMT helped me share professional knowledge and/or recommendations with the caregiver 2 2 1 0 The SDMT helped check my understanding of the caregiver's concerns 2 3 0 0 The SDMT helped with identifying caregiver priorities regarding their child's development The SDMT was easy to integrate into my current work ow The SDMT is worth integrating into my current work ow 4 1 0 0 The caregiver understood how to ll out the SDMT 2 2 0 1 As a result of using the SDMT, I feel con dent in my ability to provide referrals and/or recommendations to caregivers of this patient population 2 3 0 0

Discussion
Despite the small sample size (n = 83) in this study, patterns emerged in the areas of caregivers' developmental concerns as the children aged.Communication was the most frequently cited concern in four consecutive age categories (1-5 years).Inattention/impulsivity were the second most frequently cited concern in three consecutive age categories (2-5 years).Problem behavior was the third most frequently cited concern in three age categories (1-2 and 3-5 years).Patterns that emerged in the ranking of caregivers' priority concerns aligned with the patterns observed in the frequency results.
Communication was ranked as a priority concern in four consecutive age categories (1-5 years).Problem behavior and inattention and impulsivity were both ranked as priority concerns in three age categories (1-2, 3-5 years; and 2-5 years respectively).Although this was a pilot study without a power analysis, results support the hypothesis that developmental concerns for children with POE do persist at least through the rst ve years of life.Developmental concerns reported by caregivers using the Shared Decision-Making Tool were consistent with previously published literature nding lower language scores (Hunt et  ndings provides initial evidence that use of the Shared Decision-Making Tool can assist with the identi cation and communication of developmental concerns relevant to this population, although o cial psychometric testing will be required before wider implementation.
Given the similarity in frequency and ranking results, ranking data may not add any more useful information for the provider above the frequency results.Additionally, only one caregiver provided ranking data for the 0-1 year age group, indicating that instructions may not have been clear, or possibly that the tool was lled out by the provider in consultation with the caregiver during the appointment.Further investigation is required to determine how the tool was administered and the degree to which providers adhered to the guidance on use of the tool.Caregiver feedback will also be necessary to better inform future improvements made to the tool, such as better highlighting ranking instructions, or determining whether ranking should be removed and replaced with something more bene cial, such as the top three areas caregivers want to cover during the visit with the provider.
Provider feedback was generally positive for all questions asked.All providers 'strongly agreed' or 'agreed' with all statements on the provider feedback survey, except in two instances.One provider was 'neutral' that the Shared Decision-Making Tool helped them share professional knowledge and/or recommendations with the caregiver.This could be because the provider is already con dent in his/her abilities to share recommendations with caregivers, in which case the tool does not add any further bene t.Additionally, one provider 'disagreed' that the caregivers understood how to ll out the Shared Decision-Making Tool.This provider's response in addition to the lack of caregiver prioritization of developmental concerns for children in the 0-1 year age group indicate that there is room for improvement to ensure all caregivers understand how to complete the Shared Decision-Making Tool.
Moving forward, caregiver feedback will be collected and used to improve the tool before implementing on a larger scale.

Limitations
These preliminary study outcomes should be interpreted considering study limitations.Given the pilot nature of this study, a power analysis was not completed.We were able to recruit a sample size of 83, but convenience sampling was used, limiting our ability to generalize results of this study to all children with POE or to make comparisons with caregiver concerns across groups of same-aged children without POE.Shared Decision-Making Tools were administered using pen and paper.This administration method requires subsequent manual data entry into an excel spreadsheet for analysis, increasing the risk of human error related to reading participant handwriting or transferring data into the spreadsheet.We attempted to minimize transfer error through random data checks by two researchers.It is important to note that ndings from this study do not imply that developmental concerns are the result of POE.There are a vast number of factors that in uence child development and behavior.What this study adds to the existing literature is a preliminary report of results using a newly developed tool intended to facilitate communication between caregivers and providers related to development of children with POE.

Summary
This cross-sectional pilot study examined the acceptability and preliminary outcomes of caregivers' developmental concerns of children with POE in the rst ve years of life using the Shared Decision-Making Tool.The results indicated high acceptability of the tool with a small number of providers in medical and early childhood settings and demonstrated patterns of developmental concerns across age groups.The most frequently cited concerns: communication, problem behavior, and inattention and impulsivity, mirrored caregivers' top ranked concerns in most age groups.Data from this pilot study will guide our next research steps, focusing on psychometric testing and usability feedback from caregivers, before expanding trials of the Shared Decision-Making Tool to early childhood and medical clinics in a wider geographic area.Findings from this pilot study will also inform the development of caregiver resources for managing areas identi ed as top concerns through the rst ve years of childhood.

Declarations
The did not receive support from any organization for the submitted work.
All authors certify that they have no a liations with or involvement in any organization or entity with any nancial interest or non-nancial interest in the subject matter or materials discussed in this manuscript.
The Ethics Committee of Cincinnati Children's Hospital Medical Center has determined the research activities in this study to be EXEMPT from IRB review in accordance with applicable regulations and The SDMT was developed based on a literature review of developmental interventions and outcomes of children with POE during the rst ve years of life, and qualitative interviews with community partners (adoptive parent, kinship care, neonatologist, developmental psychologist, occupational therapist) to identify current needs of caregivers of children with POE.The resulting SDMT assesses eleven areas of developmental function, including: Sleep, Feeding, Bonding with Caregivers, Sensory Avoiding, Sensory Seeking, Problem Behavior, Communication, Motor Skills, Child's Mood, Play Skills, Inattention and Impulsivity, and Learning.
(11, 21pts)    *Priority Concerns n = # of respondents who ranked the developmental areas as top priority concerns

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