Journal of Pediatric Psychology Advance Access originally published online on March 1, 2006
Journal of Pediatric Psychology 2006 31(9):939-944; doi:10.1093/jpepsy/jsj105
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Commentary: Revising Behavioral Family Systems Therapy to Enhance Treatment Adherence and Metabolic Control in Adolescents with Type 1 Diabetes
Rainbow Babies and Children’s Hospital, Case Western Reserve University School of Medicine
All correspondence concerning this article should be addressed to Dennis Drotar, 11100 Euclid Avenue, Mather 230, Cleveland, Ohio 44106-6038. Email: dxd3{at}case.edu.
Received January 23, 2006; revision received January 28, 2006; accepted February 6, 2006
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Rationale for Intervention Approach |
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The quality of family relationships, which can influence critical outcomes such as adherence to medical treatment and medical management for pediatric chronic illness, is an important target of psychological interventions (Drotar, 2005
; Fiese, 2005). In a series of studies, Wysocki and colleagues found that Behavioral Family Systems Therapy (BFST; Robin & Foster, 1989) can enhance family communication for adolescents with type 1 diabetes (Wysocki et al., 1997, 1999, 2000; Wysocki, Greco, Harris, Bubb, & White, 2001). On the other hand, BFST did not enhance treatment adherence or glycemic control, which is a critical health outcome that affects the rate of onset of diabetes-related complications (DCCT Research Group, 1994). The previous failure of BFST to demonstrate a significant impact on diabetes-related treatment adherence in the context of a randomized controlled trial (RCT) stimulated Wysocki and colleagues’ most recent intervention research that is reported in this issue. This research fills an important need. Adherence to medical treatment has proven to be a very difficult outcome to change, not only in type 1 diabetes (Hampson et al., 2001), but in pediatric chronic illness in general (Rapoff, 1999). For this reason, new interventions and refinement of available models are very much needed.
In designing their research, Wysocki and colleagues faced the difficult decision concerning whether to frame the study as an "efficacy trial" (a study of treatment outcomes under relatively ideal conditions) vs. an effectiveness trial (a study of treatment outcomes under real-world conditions). Their decision to conduct an efficacy trial was based primarily on the lack of evidence for psychological intervention effects on treatment adherence and glycemic control (Hampson et al., 2001). For this reason, testing a modified effect of BFST intervention under more or less ideal circumstances was seen as a necessary next step (T. Wysocki, personal communication, January 6, 2006).
How does one account for the previous failure of BFST to improve treatment adherence and glycemic control for adolescents with type 1 diabetes? It is possible that the hypothesis that enhancing the quality of family relationships and communication would lead to enhanced treatment adherence and/or glycemic control was based on incomplete scientific data. Although variables such as family communication and conflict correlate with adherence to treatment for type 1 diabetes and glycemic control (Anderson et al., 2002), the quality of family relationships may not be causally related to treatment adherence. If this is the case, improving the quality of parent–child relationships using BFST (or any other intervention) would not necessarily result in improved adherence or glycemic control.
Another possibility is that the marked variation in the influences on and barriers to treatment adherence and glycemic control among adolescents with type 1 diabetes was very difficult to address with a single BFST intervention model. For some adolescents and their families, problematic communication and problem solving may be the primary influences on nonadherence to diabetes treatment. For others, maladaptive patterns of coping and behavior such as avoidance or skill deficits in diabetes management may be the most salient barriers to treatment adherence. Because long-term patterns of treatment adherence and glycemic control can be influenced by heterogeneous factors (Rapoff, 1999; Riekert & Drotar, 2000), a "one size fits all" model of family intervention may not have powerful effects on treatment adherence.
Finally, as noted by Wysocki and colleagues, another possibility is that the model of BFST that was tested in previous research did not focus sufficiently on the specific behaviors that support adherence to diabetes treatment and glycemic control. It should be noted that the essential components of the BFST intervention model were developed for families of adolescents with clinically significant conduct-related problems (Robin & Foster, 1989). In order to address these adolescents’ problems, the original BFST model included several core elements, including cognitive restructuring of irrational beliefs, structural family interventions that targeted problematic family characteristics, as well as family communication and problem solving.
The original BFST model was initially adapted by Wysocki et al. (2000) for use with adolescents with type 1 diabetes and their families. However, adolescents with type 1 diabetes and their families may have very different patterns of psychological functioning than healthy adolescents with conduct problems for whom the BFST model was originally developed. Consequently, some of the elements of BFST (e.g., changing parental beliefs and family structure) that were important to modify dysfunctional patterns of family relationships among adolescents with conduct disorders (Robin & Foster, 1989) may not have been as relevant for adolescents with type 1 diabetes and their families. Moreover, the previous application of the BFST-related problem solving and communication training (Wysocki et al., 1997, 1999, 2000, 2001) did not only focus exclusively on diabetes treatment-related treatment adherence or management but also included general developmental issues such as managing curfews, chores and so on.
In order to enhance the impact of the BFST model on treatment adherence, Wysocki and colleagues developed a revised model: BFST-D, which included five components, which were based on previous research (each of these components focused on specific behaviors and the social context of diabetes treatment-related behavior). These elements were (a) targeting at least two or more diabetes problems that were identified as barriers to diabetes management or control that were identified in the first two sessions, (b) explicit training in behavioral contracting (Wysocki, Green, & Huxtable, 1989), (c) advanced education in using self-monitoring blood glucose data (Anderson, Wolf, Burkhart, Cornell, Bacon, 1989; Delamater et al., 1990), (d) parental simulation of living with diabetes (Satin, La Greca, Zigo, & Skyler, 1989), and (e) extending the intervention to other social networks that can influence diabetes care by involving peers, siblings, teachers, and conducting sessions in other locations. The BFST-D model that was tested by Wysocki and colleagues reflected a creative integration of several specific empirically supported intervention strategies. This approach, which built upon the essential principles of BFST but placed greater emphasis on specific behaviors related to diabetes treatment, was hypothesized to have more impact on treatment adherence and glycemic control than the more generic BFST intervention model.
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Contribution of the Study |
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What were the effects of the revised BFST model? When compared to either standard care or an educational support (ES) group that controlled for duration of contact, the BFST-D model resulted in lower diabetes specific family conflict and adherence to treatment, especially for those adolescents with higher, that is more problematic, levels of glycemic control at study baseline. In addition, both BFST-D and ES were associated with improved glycemic control for adolescents with higher levels of glycemic control at baseline.
Wysocki and colleagues’ findings of significant intervention effects on adherence to diabetes treatment are important, especially when considered in the context of previous research with adolescents with type 1 diabetes. For example, Hampson et al.’s (2001) meta-analysis of interventions found very small effect sizes (mean = –0.15) interventions on self-management among adolescents with type 1 diabetes compared with greater effects (M = 0.37) on psychosocial outcomes, such as family adjustment. Wysocki et al.’s findings support the value-added impact of integrating key components of specific intervention strategies that have been shown to be effective in previous research with children and adolescents with type 1 diabetes with the basic BFST model.
In contrast to previous trials of BFST, BFST-D resulted in the predicted changes in adherence to diabetes treatment, possibly reflecting the increased focus on building skills that directly affect adherence to diabetes treatment and glycemic control. These findings are potentially generalizable: treatment adherence and clinically relevant outcomes of pediatric chronic illness such as glycemic control have been shown to be complex and difficult to modify (Rapoff, 1999). However, interventions that have the most powerful effects may need to target specific outcomes using multiple components.
Another important finding was that BFST-D did not have an effect on glycemic control for the sample as a whole. Intervention effects were much more pronounced for adolescents with more problematic glycemic control. Given their more adaptive patterns of adherence and glycemic control, it is possible that some adolescents with type 1 diabetes and their families may not have needed the full complement of the BFST intervention components. Finally, it is important to note that the BFST-D and ES interventions had comparable effects on glycemic control, both of which had greater effects than standard care. This finding supports the positive impact of nonspecific factors such as education and support on glycemic control and the need to include them in comprehensive care for adolescents with diabetes (Silverstein et al., 2005).
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Strengths of the Study |
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Wysocki and colleagues’ study has considerable strengths, which in many respects provide a model for intervention research in the field of pediatric psychology. The study design—an RCT with a control for intervention contacts and a standard diabetes care condition—reflects the state of the art for psychological interventions for pediatric chronic illness populations (Drotar, 2006
). Moreover, the evolution and testing of the BFST-D model reflected a programmatic approach to intervention development, which is also a model for the field. Wysocki and colleagues are among a handful of pediatric psychology intervention researchers who have conducted programmatic research including (a) descriptive studies of factors that relate to adherence to treatment and glycemic control that support the application of the BFST intervention model (e.g., Wysocki, 1993) and (b) RCTs of BFST and a refined model (BFST-D). Another strength of this research program includes the use of reliable and valid measures (Harris et al., 2000), which were shown to be sensitive to intervention effects. |
Challenges and Unanswered Questions |
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Wysocki and colleagues’ research also raises a number of challenges and unanswered questions for the field of psychological intervention research with pediatric chronic illness. Many of these questions center around the tension between research-based intervention studies including RCTs and their clinical applicability and significance (Drotar & Lemanek, 2001
; Drotar, 2006). For example, one such issue reflects the difference in families who enroll in psychological intervention studies vs. those who are seen for clinical care. Only 27% of families who were eligible for participation in this study actually participated. Based on previous research with pediatric populations (Zebracki et al., 2003), including children and adolescents with type 1 diabetes (Riekert & Drotar, 1999), families who participated in Wysocki et al.’s research may have had greater resources and fewer problems than nonparticipants. For this reason, Wysocki and colleagues’ findings may not generalize to other adolescents with type 1 and their families, especially adolescents with more severe problems with glycemic control than were described in this sample. For example, Harris, Harris, & Mertlich’s (2005) recent application of in-home BFST to adolescents with poorly controlled diabetes did not find durable effects on family conflict or glycemic control. However, Harris et al. (2005) did not apply the BFST-D model.
Another challenge relates to describing the nature and fidelity of the BFST-D intervention to enhance potential replication by other investigators. Although the authors included information concerning the fidelity of BFST-D, some potentially important data that are relevant to understanding of the efficacy of this intervention model were not reported. For example, the analyses of treatment integrity did not describe the specific components of diabetes-related interventions, especially those that were targeted to diabetes, including extension of the intervention to social networks, which was an optional component. In order to track the implementation and utilization of each of the specific components of this multifaceted model, it would have been useful to know what specific percentages of key intervention components were delivered to participants. Moreover, it was not clear to what extent adolescents and their families adhered to the BFST protocol. Finally, it would have also been useful to have specific data to support the authors’ assurance that the vast majority of ratings affirmed the integrity of the BFST-D intervention and also the description of procedural integrity. In this regard, the difference between what were described as legitimate differences in clinical judgment vs. violations of the written protocol was not clear. To be fair, monitoring the fidelity of psychological interventions is very difficult and burdensome but will become increasingly important in evaluating psychological interventions. See Bellg et al. (2004) for a comprehensive discussion of methodological issues in monitoring treatment fidelity in health behavior change studies.
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Implications and Future Directions |
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Wysocki and colleagues’ study have outlined several important directions for future research that presage critical directions for the "final frontier" of research on psychological interventions with pediatric populations: clinical significance. One important but as yet unanswered question relates to the efficiency of psychological intervention, defined as the power of an intervention relative to the number of sessions or time involved. To have any chance of widespread clinical application, psychological interventions with children and adolescents with chronic illness will need to be implemented as efficiently as possible. Wysocki and colleagues’ findings suggest that the efficiency of a BFST-inspired intervention concerning diabetes treatment adherence will be enhanced by an increased emphasis on adherence to treatment and illness management, especially glycemic control. However, it should also be recognized that the BFST-D model’s emphasis on family communication and problem solving may have facilitated adolescents’ and their families’ abilities to utilize the diabetes-specific components of the model.
As the authors have noted, another important issue will be to document the durability of BFST-D treatment effects over time. Assuming these findings are durable, a significant future challenge will be to determine the potential generalizability and clinical significance of BFST-related intervention effects to a wider range of populations of adolescents with type 1 diabetes. In order to accomplish this formidable task, future studies will need to emphasize flexibility, convenience, and cost containment with a wide range of possible strategies such as reducing the number of sessions, supplementing sessions with online content and resources, using telemedicine and so on (T. Wysocki, personal communication, January 6, 2006). One example of a family-centered intervention model that is potentially applicable to clinical care is the office-based family team work model that has shown positive effects on glycemic control in youth with type 1 diabetes (Laffel et al., 2003).
Wysocki and colleagues’ finding that the efficacy of BFST-D varied as a function of glycemic control (e.g., greater effects with adolescents with poorer glycemic control) also has potentially important clinical and research implications. One of these is that investigators might wish to target adolescents with more problematic patterns of glycemic control than what is normative for a particular clinic population. Practitioners may also consider targeting this at-risk group for psychological intervention.
Systematic targeting of adolescents with type 1 diabetes for interventions based on empirical data concerning their levels of adherence and glycemic control will require an individualized, preventive approach to ongoing diabetes care that involves different levels of care. For example, comprehensive care is recommended for all adolescents with type 1 diabetes (Silverstein et al., 2005). A second level of care could include more intensive psychological intervention for those adolescents who demonstrate greater than average problems in adherence and/or glycemic control. Wysocki et al.’s findings suggest that practitioners might refer adolescents with type 1 diabetes who demonstrate higher than average levels of glycemic control for additional psychological intervention, assuming there are resources for such intervention. In this approach, such adolescents would receive psychological intervention sooner than is often the case in some diabetes centers where adolescents with the most severe family and illness management problems receive a disproportionate share of psychological services. The fact that more problematic levels of glycemic control predict the onset of diabetes-related complications supports the need to develop such targeted preventive, psychological interventions (DCCT, 1994). Similar preventive models have been proposed by Rapoff (2000) for juvenile rheumatoid arthritis and Kazak et al. (2003) for pediatric cancer.
Finally, as was discussed by the authors, their use of a multifaceted intervention model could not clarify the specific components of the intervention that accounted for intervention change. However, given the early stages of intervention research in the field of pediatric adherence with chronic conditions, the first research priority was to demonstrate the efficacy of specific intervention models with specific conditions (Drotar, 2006). Nevertheless, dismantling studies would help to determine the key ingredients of the BFST-D intervention (T. Wysocki, personal communication, January 6, 2006). On the other hand, it may be that the complex menu of BFST-D intervention components is necessary to facilitate tailoring the intervention to each family’s skills, attributes, and presenting problems. In other words, each of the elements of BFST-D may prove to be critical to enhancing the progress and outcomes of a subset of adolescents and their families (T. Wysocki, personal communication, January 6, 2006). For this reason, studies of the clinical effectiveness of matching therapeutic structure to individual families’ circumstances and resources could prove to be a valuable contribution to future research on interventions to promote adherence to diabetes treatment (T. Wysocki, personal communication, January 6, 2006).
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Acknowledgments |
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TopRationale for Intervention...Contribution of the StudyStrengths of the StudyChallenges and Unanswered...Implications and Future...AcknowledgmentsReferences |
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The excellent work of Susan Wood in word processing this manuscript is gratefully acknowledged.
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