Editorial: Case Studies and Series: A Call for Action and Invitation for Submissions

In 1995, the associate editors of the Journal of Pediatric Psychology (JPP) published an article to encourage the submission of case reports, studies, and series (Drotar, LaGreca, Lemanek, & Kazak, 1995), which had been remarkably underrepresented in the portfolio of published articles. Apparently, this article did little to encourage submissions of case studies and series because the situation is unchanged. But being undaunted and persistent, I would like to revisit this issue in light of the continuing importance of case studies and series to the field of pediatric psychology. In its formative years, JPP published many case studies, series, and descriptions of programs (e.g., Bachara & Lamb, 1976; Davidson, Adams, Schroeder, & Tyler, 1978; O’Malley & Koocher, 1977). At that time, pediatric psychologists published such work in order to call attention to important and challenging clinical problems, as well as to share their experiences, ideas, and data concerning clinical practice. Given the state of the art of the science at that time, early case studies and series focused more on description of clinical problems and intervention methods than on empirical demonstrations of treatment efficacy, with some notable exceptions (e.g., Barnard, Christophersen, & Wolf, 1976; Linscheid, Copeland, Jacobstein, & Smith, 1981; Varni, 1980). What conclusion should we draw from the fact that recent submissions to and publications of case studies and series in JPP are so few and far between? Is it the case that the field of pediatric psychology has moved beyond the need for case studies and series to inform clinical practice and research? In fact, the standards for the evaluation of science in our field, including studies of intervention efficacy (Spirito & Kazak, 2006), are much higher now than they were in the early days of the field. Are case studies and series no longer relevant or competitive for publication in JPP? Another possibility is that pediatric psychologists have simply stopped trying to write up their case studies for JPP because they do not see it as a ready outlet for publication. Alternatively, they could be writing them, but submitting them to journals other than JPP. It's hard to know. As the current editor of JPP, I believe that case studies and series have not outlived their usefulness and that the field of pediatric psychology will continue to benefit from their publication. To address this issue, this editorial has the following goals: (1) underscore the continuing need for the publication of case studies and series in JPP; (2) clarify issues that need to be considered in preparing and reviewing case studies and series; (3) suggest ways to enhance publishability; and (4) invite authors to submit manuscripts that focus on case studies and series. Case reports and series serve several important functions that are not addressed in articles that are typically published in JPP. These include describing the following underrepresented topic areas: new clinical populations, needs, and challenges; development of new intervention models and frameworks; feasibility and preliminary efficacy of interventions; clinical effectiveness of new interventions delivered in practice settings; generalizability of empirically supported interventions to clinical practice; and the clinical utility of evidence-based assessments. Based on my experience, I would suggest that as it was true in the early years of JPP, contemporary pediatric psychologists and their trainees encounter extraordinary clinical challenges that provide very important sources of learning, professional development, and lessons learned for our field. Case studies and series can help pediatric psychologists to articulate relevant clinical issues and challenges that they face in clinical settings and bring these problems into greater focus for the field. As an example, in my work with colleagues to develop new clinical programs for the psychological care of cancer survivors (Peterson & Drotar, 2006) and medical treatment adherence problems, I have been challenged and stimulated by the complexity of clinical phenomena that we have encountered, very much as I was when I developed a pediatric psychology service more than 30 years earlier (Drotar, 1976, 1977). I do not think that my experiences are unique. Moreover, I would venture to say that at the time this editorial is being written many pediatric psychologists and their trainees are actively engaged in very important and challenging clinical work that has not been previously described in the peer-reviewed literature. A short but by no means inclusive list of new clinical challenges include those raised by the genetic testing of parents whose children are at risk for chronic illness (Hood et al., 2005; Miller, McDaniel, Rolland, & Feetham, 2006); the unmet needs of pediatric cancer survivors (Robison et al., 2005); clinical and management of pediatric sleep disorders (Palermo & Owens, 2008); unrecognized psychological needs of children and adolescents with sickle cell disease (Peterson, Palermo, Swift, Beebe, & Drotar, 2005); and the clinical and policy dilemmas raised by the transition to adulthood for adolescents with chronic health conditions (Weissberg-Benchell, Wolpert, & Anderson, 2007). Diagnostic and treatment experiences with pediatric populations are a fertile source of clinical observations and hypotheses about the development of maladaptive behavior and management of very difficult clinical problems. Such experiences can also suggest new approaches and concepts that facilitate and refine our working hypotheses and approaches to clinical management. One such example is Anderson and Coyne's (1991) concept of “miscarried helping,” defined as parents’ well-intentioned efforts to help their children manage a chronic illness that are perceived as intrusive and result in maladaptive parent–child relationships, including conflict that can disrupt chronic illness management. This concept, which was based on clinical experience, has been of significant heuristic value in helping practitioners understand the family relationships of children with chronic illnesses, such as Type 1 diabetes, and suggesting new research and intervention strategies (Harris et al., 2008). Case studies and series also provide useful opportunities to describe and document the feasibility and preliminary efficacy of new intervention models. In particular, a well-documented, programmatic case series (e.g., Stark, Bowen, Tyc, Evans, & Passero, 1990) provide useful preliminary studies that facilitate the development of controlled intervention studies, including randomized controlled trials (RCTs). Comprehensive interventions that are designed to address difficult clinical problems may not be either feasible to implement or effective as they were initially designed and need to be modified. For example, intervention models that seemed like cogent ideas may not turn out to be acceptable to children and families. Case studies and series provide invaluable data and experiences to describe the feasibility and efficacy of interventions that are eventually tested in clinical trials. Available criteria for establishing empirical support for psychological interventions focus entirely on statistical significance (Chambless et al., 1998). Information concerning clinical significance is often a key missing ingredient in published studies of psychological interventions with pediatric populations (Drotar, 2002; Drotar & Lemanek, 2001). Case studies and series that describe interventions conducted on clinical populations provide important opportunities to document the clinical significance of interventions using a variety of methods, such as the Reliable Change Index (Jacobson, Roberts, Burns, & McGlinchey, 1999; Jacobson & Traux, 1991), normative comparisons (Kendall, Marrs-Garcia, Nath, & Sheldrick, 1999); or analyses of the functional impact of change (Kazdin, 1999, 2000, 2008). Pediatric psychologists who work in cost and outcome-focused hospital settings have unique opportunities to report case studies and series that demonstrate the efficacy of psychological interventions for changing health outcomes (e.g., symptoms, illness, control, and/or complications), reducing costs of care (e.g., by reducing frequency of pediatric hospitalizations for pediatric chronic illness), or lessening the level of functional impairment associated with problems such as chronic pediatric pain (Palermo & Scher, 2001). Hospital administrators, physicians, and other healthcare professionals may not be aware of the content, utility, and potential impact of psychological interventions. The combination of a compelling narrative or “story” of an individual child and family along with data that document the efficacy of an intervention, which comprise the essence of case studies, is especially informative to a range of professional audiences. Another key function of case studies and series is to document the generalizability or effectiveness of empirically supported interventions for a wide range of clinical populations and clinical practice situations. Owing to the constraints of research designs and their implementation, children, adolescents, and families who participate in research on the efficacy of psychological interventions, including RCTs, conducted in pediatric populations are not usually the same individuals who are referred to and seen by practicing pediatric psychologists. In fact, children, adolescents, and families who are selected and recruited for intervention studies are likely to be very different from those who are referred to pediatric psychologists in practice settings (Drotar, 2002, 2006; Drotar & Lemanek, 2001; Kahana, Drotar, & Frazier, 2008). For example, children and adolescents with complex clinical problems and comorbidities who are typically seen by practitioners in pediatric settings are generally excluded from research-based intervention protocols. In addition, only a subset of eligible children and their families volunteer to participate in intervention studies. This is one reason why research on psychological interventions does not typically generalize to practice, which has been observed for many years in studies of child psychotherapy (Weisz & Weiss, 1993). In addition, and most importantly for effective dissemination of research into practice, even well-documented empirically supported interventions are not equally effective for all children and adolescents and need to be individualized or tailored to specific clinical issues and populations. However, the specific practical strategies that facilitate tailoring empirically supported interventions to various populations need to be developed. For this reason, practicing pediatric psychologists are in an excellent position to test methods of individualizing empirically supported interventions to challenging clinical populations, documenting these approaches, and evaluating relevant outcomes (Kazdin, 2008). For all the above reasons, case studies and series drawn from the clinical practice of pediatric psychology can provide critical data concerning the generalizability of empirically supported interventions (e.g., specific clinical populations for whom empirically supported interventions do or do not work). Because of the heterogeneity of clinical populations in the field of pediatric psychology, it will be impossible to use large-scale intervention research, especially RCTs, to document the generalizability of empirically supported interventions for the broad range of clinical problems that are seen in practice. For this reason, case studies and series that document the clinical effectiveness of empirically supported interventions with clinical populations have a critical role to play in the future of pediatric psychology. In addition, descriptions of the failure of empirically supported interventions to generalize to specific populations together with suggestions to enhance intervention effectiveness are valuable contributions of case studies. Another important role of case studies and series is to describe the application and utility of evidence-based psychological assessments in clinical practice. One of the hallmarks of the clinical practice of pediatric psychology is the frequent use of standardized, validated psychological assessment instruments to describe the psychological needs of children and adolescents, diagnose clinical problems, conduct treatment planning, target interventions, and, in some instances, evaluate the effectiveness of clinical interventions. Cohen and colleagues (2008) published a landmark issue of JPP that summarized reviews of evidence-based psychological assessments in several critical domains (e.g., pediatric pain, family functioning, psychological adjustment, quality of life, treatment adherence, etc.) in the field of pediatric psychology. One consistent recommendation from these reviews was to integrate psychological assessments into clinical care and demonstrate their clinical applications in practice settings (Alderfer et al., 2008; McQuaid, 2008; Palermo et al., 2008). For this reason, case studies and series that describe the role and utility of evidence-based psychological assessments in planning interventions, targeting goals for clinical management, and evaluating the clinical effectiveness of interventions in practice settings (see previous section) are critical to the future clinical application of evidence-based assessments. By now, I hope I have convinced you that case series and reports are important to retain and expand in the portfolio of JPP publications, not merely to continue the initial tradition of case description in our field in deference to the past but as a way to integrate research and practice and develop the clinical significance of research, which have been identified as priorities for the future of pediatric psychology (Brown & Roberts, 2000). That said, authors face significant challenges in their quest to develop well-reasoned and documented reports of case studies or series that can successfully run the gauntlet of the editorial process. Prospective authors of case studies struggle with the question of whether their case material is significant enough to warrant publication and with the related issue of selecting a focus or theme for their manuscript. As noted earlier, case studies and series have several different purposes, each with various implications for the field. For this reason, prospective authors need to determine the most compelling and significant focus that best captures the potential contribution of their case material to the field. In this regard, potential questions for authors to consider include the following: What is the significance of the case report or series and in suggesting new research? Why is it important or significant? What is most novel and interesting? How specifically does it extend clinical knowledge or understanding? Authors also face the challenge of documenting their clinical observations, interventions, and outcomes in a clear and convincing manner. Prospective authors may find it difficult to know what kind of information is necessary to include in a case study or series, especially if a single subject design is not used. Key descriptive information in case studies and series include history, presenting problem, baseline data (e.g., symptoms, level of functional impairment), description of interventions that are conducted including target goals and methods, description of the child or family's progress over time, length of follow-up, measures of outcome, description and analysis of outcomes, clinical significance of outcomes, and follow-up data, as relevant. In order to maximize their opportunities to obtain and retrieve the data that are necessary for case studies and series, prospective authors need to be thoughtful and proactive in planning practice-based data collection, targeting measures to assess specific presenting problems, symptoms, and impairment, and choosing standardized outcome measures that yield data that describe the clinical significance of changes from pre- to posttreatment. Owing to the infrequent submissions of case studies and series, JPP editorial board members generally have had little experience evaluating manuscripts that describe case studies and series, which have a different purpose and format than typical manuscript submissions. Consequently, reviewers need help from authors to appreciate the purpose, significance, and value of their case studies and series and the relevant implications for the science and practice of pediatric psychology. In this section, I present specific suggestions that I hope will help authors to improve the clarity and impact of their case studies and reviewers to evaluate the quality of authors’ manuscript submissions (Drotar et al., 1995). In order to facilitate preparation of their manuscripts, authors may wish to consult published descriptions of case studies in JPP (e.g., Palermo & Scher, 2001; Bernard, Cohen, & Moffett, 2009) and other journals (e.g., the Journal of Clinical Child and Adolescent Psychology's new series on case studies in evidence-based practice) (Gill, Hyde, Shaw, Dishion, & Wilson, 2008; Lock, Hirsch, Greif, & Hilderbrandt, 2009). Authors of case studies and series need to establish the significance of their observations and data and show how their manuscript makes a new contribution that transcends relevant published work (Drotar, 2008). For this reason, clear description of what is known based on previous publications and an explicit statement of the manuscript's specific new contribution are just as important in case studies and series as they are in any other article. In order to determine and document the significance of their work, I would strongly encourage prospective authors of case studies and series to conduct a comprehensive literature review to determine what has been published in the area of their manuscript. In addition, I would also suggest that prospective authors discuss their ideas for case studies, relevant case material, and the focus or theme of their case studies with colleagues in their own and other clinical settings. Relevant questions to discuss with colleagues include the following: Do other practitioners find the case material and focus of the case to be of clinical interest? Does the case material reflect a challenging problem or issue that is typically encountered in different clinical settings? Do the findings suggest interesting, useful, and/or new ways of understanding or managing clinical problems? The central purpose and theme of the case report/series are focal points for the organization of manuscripts. the functions of case reports described earlier, authors have any of to as the theme of their case studies. For example, Drotar et the following (1) and important clinical that and practice with specific (2) hypotheses concerning the development of (3) new clinical and research (4) novel methods or treatment and feasibility and the efficacy of new of potential include the following: (1) empirically supported interventions that generalize to new clinical (2) empirically supported that do not generalize to specific populations and need to be and (3) of evidence-based assessments to new populations (e.g., clinical different or The quality and of the data that are in case studies and series are just as important as they are in any other manuscript. However, on the purpose of their case authors will need to provide different and For example, if the purpose of their manuscript is to describe an (e.g., or clinical problem, authors will need to reviewers that the clinical problem is relevant and the other if the purpose of the case report is to describe the clinical effectiveness of treatment in a practice empirical of change will be The most convincing data to demonstrate intervention effectiveness the design (see & (2008) designs are the for evaluating clinical and I would encourage authors to to this & Stark, 2008). At the very of an intervention effectiveness based on a case study or series should a description of baseline data to the intervention for the target outcome of and of a significant change following the case studies or series (e.g., those that do not report the of are also important to the field of pediatric psychology. However, such manuscripts challenges for authors and reviewers because they often presenting and very different of information (e.g., history, and (see 1993). For this reason, authors need to consider what of information and data should be in their case reports and describe the for their In addition, it is very important that authors develop a of so that can the of all the information that is with the initial theme or question considered by the case and with implications and 1993). The data and that are drawn from case studies and series may be subject to and (Kazdin, 1980). For this reason, authors need to consider and discuss of data that they and of their methods, data, and (Drotar et al., 1995). The generalizability of drawn from case studies and series is potential However, on the purpose of the case the of information and the that are of generalizability may not be a Authors can enhance the generalizability of the data from their case studies in several by describing including seen in different settings; interventions with or by data from with that of a of children in a relevant (see & A key potential value of case studies and series the of novel ideas about clinical hypotheses for research, new methods of intervention, or applications of psychological assessments. To clarify the implications of their case studies, authors may wish to consider some of the following How specifically does their case study or series clarify important clinical What clinical practice lessons can be drawn from the case study or What are the implications for clinical What new of research and specific research questions are by this What new or hypotheses about clinical issues are by their What new clinical needs are by their How should they be What in empirically supported interventions are (Drotar et al., 1995). In order to submissions of case studies and series, JPP will develop a new that will the current on studies. I invite pediatric psychologists to submit manuscripts that report case studies and series that describe their work and facilitate the development of our field. such work is is a that the knowledge and of individual practitioners will be and not benefit the field as a (Kazdin, 2008). I also strongly encourage with trainees in preparing case reports and series for publication because this can provide an excellent In preparing case reports and series, practitioners and trainees have valuable opportunities to novel clinical observations that inform the development of new methods of assessment and/or to submissions. Prospective authors can of potential case studies and series for my of the editorial and reviewers who have in reviewing case studies and series should also know. are in reviewers who have had experience in and reviewing case studies and series. prospective authors and reviewers should consult the of for authors and reviewers that are based on this article and are on the JPP The hard and excellent work of in this manuscript is The ideas and writing in this editorial were and by the from the current associate editors and editors of JPP and colleagues in the at Hospital