Guided Internet-Based Parent Training for Challenging Behavior in Children With Fetal Alcohol Spectrum Disorder (Strongest Families FASD): Study Protocol for a Randomized Controlled Trial

Background Fetal alcohol spectrum disorder (FASD) is a term used to encompass the full range of neurobehavioral and cognitive dysfunction that may occur as a consequence of prenatal alcohol exposure. There is relatively little research on intervention strategies that specifically target the behavioral problems of children with FASD. Availability and access to services are barriers to timely and effective care for families. The Strongest Families FASD intervention was recently adapted from the Strongest Families “Parenting the Active Child” program to include FASD-specific content delivered via an Internet-based application in conjunction with 11 telephone coaching sessions. Objective Our objectives are to (1) evaluate the effectiveness of Strongest Families FASD in reducing externalizing problems (primary outcome), internalizing problems, and parent distress (secondary outcomes) in children aged between 4 and 12 years diagnosed with FASD when compared to a control group with access to a static resource Web page; (2) evaluate the effectiveness of Strongest Families FASD in improving social competence (secondary outcome) in school-aged children aged between 6 and 12 diagnosed with FASD when compared with an online psychoeducation control; and (3) explore parental satisfaction with the Strongest Families FASD online parenting program. Methods Parents and caregivers (N=200) of children diagnosed with FASD who have significant behavioral challenges, ages 4-12, are being recruited into a 2-arm randomized trial. The trial is designed to evaluate the effectiveness of the Web-based Strongest Families FASD parenting intervention on child behavior and caregiver distress, compared to a control group receiving access to a static resource Web page (ie, a list of FASD-specific websites, readings, videos, and organizations). Results The primary outcome will be externalizing problems measured by the Child Behavior Checklist (CBCL). Secondary outcomes include (1) internalizing problems and (2) social competence, both measured by the CBCL; and (3) parental distress measured by the Depression Anxiety Stress Scale-21. The Client Satisfaction Questionnaire-8 (CSQ-8) and the Satisfaction Survey are completed by the intervention group at the end of session 11. Results will be reported using the standards set out in the Consolidated Standards of Reporting Trials (CONSORT) Statement. Conclusions It is hypothesized that the Strongest Families FASD intervention group will improve child behavior and parental distress. Caregiver satisfaction is anticipated to be positive. Advancing evidence on the effectiveness and acceptance of distance services can inform policy and adoption of eHealth programs. ClinicalTrial ClinicalTrials.gov NCT02210455; https://clinicaltrials.gov/ct2/show/NCT02210455 (Archived by WebCite at http://www.webcitation.org/6bbW5BSsT)

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Potential Impact
Comments: This is an important topic and this study has the potential to contribute to the body of knowledge to address a current and urgent health care problem and need. This project is a superb example of a much needed intervention for a highly neglected population. The research objectives were crafted in response to an important need for change as identified by the knowledge users. Good partnerships have been developed across multiple provinces. The knowledge exchange plan is well described and the ongoing relationship with decision makers well articulated. The study is important to those directly responsible for the coordination and provision of care for kids with FASD and their family members.

Scientific Merit
The proposal is very well written and the PI has made a convincing argument on the significance for this study. The strengths of the application include: 1)The intervention is grounded on a previously tested program (Strongest families) with kids with Oppositional Defiant Disorder (ODD) and Attention deficit hyperactively disorder (ADHD) on a different cohort (FASD population).
2)Specially trained coaches will be available to families and kids and will assist with customization and personalization of the intervention.
3)As there is diversity among families raising children with FASD, such as cultural and ethnic backgrounds, special care will be taken to develop culturally appropriate interventions for participants from First Nations, Inuit, and Metis communities. There is a very good process outlined to ensure that the Strongest families intervention is customized to a new population and the intervention is tested for its' feasibility. First stage includes surveying 100 families to make sure they are aware of all the problematic behaviors of children with FASD. Second stage includes working with 20 families to develop the text content for the intervention. 4)There is a very clear RCT trial proposed within this work and includes appropriate methods and analysis for its' evaluation.
The weaknesses of the study are the following: 1)It is not clear what the various components of the intervention are in the main proposal.
2)There is no mention of treatment fidelity checks, which is a potential flaw in the design of the project. If the intervention is so customized, how will the investigators make sure the intended dose was received and all of the components of the intervention administered?
3)There is a risk that the FASD kids and their families will not be amenable to this Strongest Families intervention as the parents are expected to make changes in their parenting styles. Given that some parents have their own mental issues, perhaps they too may need some type of treatment 4)It is not clear why an RCT trial is planned prior to a more feasibility/pilot testing trial as it is not clear if the adapted intervention will demonstrate changes in outcomes as outlined (the child's behavior and their social integration skills).

Potential Impact
Comments:

Brief Summary
This 3 year research proposal seeks to develop and evaluate a family-centered intervention the families (parents or caregivers) of children with fetal alcohol spectrum disorder (FASD) between the ages of 4 and 12 years. The intervention will be an Internet-based parent training program specifically adapted for the parents, families or caregivers of children with FASD who present significant behavioral challenges. The evaluation will include a randomized clinical trial (RCT) component whereby the feasibility and outcomes of those receiving the intervention will be compared to those of a control group not receiving the intervention.

Potential Benefit
Children with FASD suffer from neuro-behavioral deficits and usually present significant challenges to those responsible for their care and safety. The parents or guardians who care for these children, particularly those in rural and remote communities, often do not have access to the necessary support and detailed advice on how to mange with these symptoms. Strong letters of support for this initiative have been provided by such organizations as:

Strengths
Strength of the proposed research is that it builds on a successful program in place at the IWK Health Centre in Halifax, NS. The program is called Strongest Families. The Strongest Families program has already developed modules for attention deficit hyperactivity disorder (ADHD) and oppositional defiance disorder (ODD). Another strength of the Strongest Families program compared to care delivered in a clinical setting or office practice is its flexibility and convenience. This educational program for parents or guardians uses trained coaches to work with parents or caregivers-at times that are convenient for the family.
Potential Areas for Improvement The scope of the proposed intervention is limited to FASD children aged 4 to 12 years; however FASD is a lifetime condition. The study team may wish to include a component which will help parents and caregivers deal with the challenges of their FASD children as they undergo the transition into adolescence and young adulthood.

Knowledge Transfer
The KT plan is aimed at diverse audiences and is very well described. KT activities will include linkage with current programs and initiatives of the Public Health Agency of Canada -such as the Community Acton Plan for Children and the Canada Prenatal Nutrition Program. Other linkages include a variety of FASD support networks and the Canadian Association of Pediatric Health Centers. Partnerships for Health System Improvement Comité: Partenariats pour l'amélioration du système de santé

Scientific Merit
Comments:

Study Team
The study team is strong and diversified. It is led by a professor of psychology at Dalhousie University who also holds faculty appointments in pediatrics and psychiatry as well as the Canada Research Chair at Dalhousie. Other members of the study team have professional and research backgrounds in a number of areas pertaining to FASD. They include psychiatry, behavioral sciences, mental health, addictions, pharmacology, toxicology and epidemiology. The study team appears to have the experience and skills needed to successfully complete this research and development project.

Methods and Approach
The research plan consists of three consecutive studies. Study 1 will examine the behavioral and emotional problems of children diagnosed with FASD and their parents or guardians. Participants to be enrolled in the study will be identified using information obtained from the clinical records of organizations providing care to FASD patients and families. Participants will be selected so as to ensure at least 30% representation from First Nations, Métis and Inuit communities. Information will be gathered through telephone surveys (n=100) using a survey instrument developed by the principal investigator. Study 2 will use the findings of the telephone surveys to develop text content for illustrative teaching materials (30 video clips and 75 audio clips). These materials will be used in a progressive series of individualized interactive learning modules. A number of families (n=20) will be recruited to try out the modules and provide feedback as to their value and usability. Study 3 will consist of a randomized clinical trial (RCT) of 17 months duration. A total of 200 families with an FASD child will be recruited and assigned randomly to the intervention group or to the control group. The intervention group will receive the training program over the Internet via a personalized website -plus weekly verbal support from a trained "coach" over the telephone. The control group will not receive the intervention, but will be referred to available sources of information about the care of FASD children. The outcome variable to be analyzed in the RCT will be changes in the behavior of the FASD child assessed according to a scale called the Child Behavior Checklist (CBCL).
The use of a single variable to compare the different results achieved by the intervention group and the control group may not be sufficient to demonstrate conclusively that the educational intervention alone is capable of achieving significantly better outcomes in terms of child behavior. Overall, however, the methods and approach described in this study appear likely to yield findings and results that could be used in the improvement of other support programs for the parents or caaregivers of FASD children.
Potential Areas for Improvement Since some parents of FASD children may still have alcohol related problems which impair their ability to provide good care, the study team may wish to consider adding an educational component to the study that would provide additional support and guidance to those who need it.

Budget
The total proposed budget for this 3 year study Is $567K of which $400K is being sought from CIHR. The balance of $167K will be provided in the form of cash and in-kind contributions from study partners such as the Saskatchewan Population Health and Research Unit, NeuroDevNet (a network of centres of excellence in developmental neuroscience, and the NS Center for Research in Family Health. Major