Many healthcare innovations are not sustained over the long term, wasting costly implementation efforts and often desperately-needed initial improvements. Although there have been advances in knowledge about innovation implementation, there has been considerably less attention focused on understanding what happens following the early stages of change. Research is needed to determine how to improve the ‘staying power’ of healthcare innovations. As almost no empirical knowledge exists about innovation sustainability in nursing, the purpose of our study was to understand how a nursing best practice guidelines (BPG) program was sustained over a long-term period in an acute healthcare centre.We conducted a qualitative descriptive case study to examine the program’s sustainability at the nursing department level of the organization. The organization was a large, urban, multi-site acute care centre in Canada. The patient safety-oriented BPG program, initiated in 2004, consisted of an organization-wide implementation of three BPGs: falls prevention, pressure ulcer prevention, and pain management. Data were collected eight years following program initiation through 14 key informant interviews, document reviews, and observations. We developed a framework for the sustainability of healthcare innovations to guide data collection and content analysis.Program sustainability entailed a combination of three essential characteristics: benefits, institutionalization, and development. A constellation of 11 factors most influenced the long-term sustainability of the program. These factors were innovation-, context-, leadership-, and process-related. Three key interactions between factors influencing program sustainability and characteristics of program sustainability accounted for how the program had been sustained. These interactions were between: leadership commitment and benefits; complementarity of leadership actions and both institutionalization and development; and a reflection-and-course-correction strategy and development.Study findings indicate that the successful initial implementation of an organizational program does not automatically lead to longer-term program sustainability. The persistent, complementary, and aligned actions of committed leaders, in a variety of roles across a health centre department, seem necessary. Organizational leaders should consider a broad conceptualization of sustainability that extends beyond program institutionalization and/or program benefits. The development of an organizational program may be necessary for its long-term survival. In healthcare, the issue of sustaining organizational and practice changes is of great concern because many beneficial innovations are not maintained over the long term , 2. InnoThe literature about the sustainability of healthcare innovations varies significantly in definitions and conceptualizations of the phenomenon , 6. SustReviews of the literature about innovation sustainability in health and social service domains have underscored that conceptualizations of sustainability and related factors differ according to the type of innovation and context –3, 6, 10There is a specific need for knowledge about the sustainability of healthcare innovations in the field of nursing, particularly in acute care settings. The majority of nurses work in acute care, where they represent the biggest provider group , 14. ExpIn the context of nursing, we found only a few studies –31 conduAlthough not nursing-specific, other studies have been conducted on the sustainability of organization-wide, multidisciplinary care improvement initiatives in acute care centers –42. The Further research on organizational program sustainability is justified as the few studies that have identified factors influencing program persistence were not based on existing theory and used narrow conceptualizations of sustainability. Perhaps due to the typically shorter-term timeframes they studied , there was no consideration of how programs may have developed over time. The studies also centered on the perspective of one type of participant , neglecting the viewpoints of leaders at different levels of organizational management. Moreover, they provided minimal description about the nature of what was sustained and at what level of the organization (department versus unit), with no exploration of the complex relationships between the innovation, the influencing factors, and the circumstances studied. This makes it difficult to determine how programs come to be sustained. These gaps related to sustaining organizational programs are consistent with the general calls for more in-depth, contextually-based, process-oriented studies that consider broader conceptualizations of sustainability and longer-term trajectories of innovations , 10, 43.The overall study purpose was to understand how a nursing best practice guidelines (BPG) program was sustained over a long-term period in an acute healthcare centre. The specific research questions for this paper were: at the nursing department level of the organization, 1. How was program sustainability characterized?; 2. What were the factors that most influenced program sustainability?; and 3. How was the program sustained over the long-term?In preparation for this study, we developed a framework for the sustainability of healthcare innovations based on a broad literature review and concept analysis . We defiThe two central components of the framework are “characteristics” (attributes) of sustainability and “factors” (preconditions) that influence sustainability. Over a period of time, the effect of the relationships between the characteristics of and configurations of factors determines the level of sustainability of an innovation.The three characteristics of innovation sustainability are: benefits, routinization or institutionalization, and development. “Benefits” pertains to consistent achievement of goals and lasting improvement in positive results. “Routinization” or “institutionalization” (the term used henceforth) is the embedded structures and processes of the innovation that are part of the regular or habitual function of individuals, organizations, and systems. “Development” signifies the evolution of the innovation and/or adaptation of the context within which the innovation is embedded.Four categories of factors proposed to influence sustainability are: innovation, context, leadership, and process. Innovation factors pertain to the nature of the product, practice, policy, or program that is new to the organization or to a group of individuals at the time of adoption. Contextual influences are due to the environment, setting, situation, or conditions within which the innovation is implemented. Leadership is the formal or informal manager(s) or organizer(s) of a group, with certain authorities, attributes, and actions that influence other people. Process factors refer to series of events, strategies, or activities that lead to a particular result.To explore the complex phenomenon of program sustainability in its natural, organizational setting, we used a qualitative descriptive case study approach –49. We eThe organization was a large, tertiary/quaternary urban academic acute health centre in Canada. It had six hospital sites, eight clinical missions/programs, 47 inpatient units, approximately 1000 staffed beds, and more than 3000 nurses. In 2004, the nursing department launched a patient safety-oriented BPG program. Overall program goals were to improve nursing care and patient outcomes; harmonize practices; expand knowledge translation and performance measurement capacities; and develop nursing leadership competence across the organization. The program consisted of the organization-wide implementation of three Registered Nurses’ Association of Ontario1” program status, and securing financial support from numerous sources. The main implementation activities at the department level were related to education , equipment , and evaluation . At the nursing unit level, the main focus of the program was to improve staff nurses’ performance of patient care and documentation practices based on the BPGs. Department-level BPG task force members and change advocates worked with individual nursing units over two to three month-long periods to support the initial implementations of each of the three BPGs. Those externally-facilitated implementation “start-ups” used a combination of educational sessions, nurse BPG champion support, and audit and feedback activities to help nursing units integrate BPG standards into daily practice.At the nursing department level, multiple changes were made to establish the BPG program across the health centre. These included designating program co-chairs, creating a steering committee, formalizing BPG-specific task forces, training volunteer practice change advocates, employing a part-time program coordinator, achieving RNAO “Best Practice Spotlight OrganizationWe selected this organization’s nursing BPG program as the focus of our investigation to ascertain how and why the measureable improvements in program-related patient outcomes had been sustained. Administrative data demonstrated that between 2004 and 2012, the organization as a whole achieved maximal reductions in rates of more than 60 % for pressure ulcers, 30 % for falls, and 30 % for patient reports of moderate to severe pain . There wData collection occurred between August 2011 and June 2012, eight years after program initiation. Fourteen individual, semi-structured, framework-guided interviews were conducted with organizational key informants who were all female Registered Nurses with department-wide leadership positions. We purposefully selected the informants, seeking representation from the variety of roles in the program ; emailed potential participants a study information letter; and followed-up with them by telephone or email. All individuals who were invited agreed to participate in the study. Questions during each interview moved from open queries  to more specific queries  if informants did not spontaneously address some of the main elements of the conceptual framework or to pursue additional detail about important emerging topics. We also reviewed program-related documents and completed observational experiences within the nursing department. Additional characteristics of the data sources are summarized in Table Data collection and the preliminary phase of analysis occurred concurrently. The main “unit of analysis” was the BPG program at the departmental level of the organization . We usedNVivo 9/10 software was used to organize, code, and query data. Qualitative content analysis guided our data coding and interpretation , 57, 58.We used Guba and Lincoln’s  criteriaThe Research Ethics Board (MUHC-11-036-PSY) of McGill University Health Centre, the participating institution, granted approval of the study protocol. Informants provided informed consent in writing prior to participation. Informants’ participation was voluntary and confidential. Complete anonymity for a few participants could not be ensured because of the unique, identifiable nature of their job roles.We have organized our findings to answer each of the three research questions. The questions also correspond with central elements of the proposed framework. First, we report on how program sustainability was characterized by describing what elements of the program were sustained. Next, we summarize the factors that most influenced program sustainability over time. Finally, we provide a detailed examination of the salient relationships between characteristics and factors, as these interactions most accounted for how the program was sustained.Informant descriptions reflected that program sustainability consisted of a combination of three essential characteristics: benefits, institutionalization, and development. Informants and documents described substantial program benefit and institutionalization, and provided evidence that, over time, development had become the most prominent feature of program sustainability at the departmental level. We have displayed these three characteristics graphically in Fig. The notion of BPG program benefits was described primarily as the improvement in patient outcomes for the organization as a whole, which corresponded to the program’s main objective. Informants cited the administrative data that objectively demonstrated the maintenance of the program’s initial improvements in patient outcomes. However, the majority of informants also contended that the improvements had leveled off over time. As a participant summarized, “the bad news is that we’ve just sustained [results], as opposed to driven the improvements deeper. That’s what we are trying to address now. It’s not good enough to hold gains.” O13.Most informants also described the importance of viewing program benefits broadly and across multiple levels of the system. They described other advantages that had occurred beyond improved patient outcomes. These were consistent with other program goals and included increased standardization of the BPG practices across the organization’s nursing units, development of departmental leadership capacity, reinforcement of the organization’s evidence-informed practice culture, and improvement of the department’s performance measurement skills.Institutionalization was viewed in terms of the program’s enduring processes and structures. Examples of BPG program institutionalization at the organizational level were: program management infrastructure ; the annual BPG-based, organization-wide prevalence survey (for pressure ulcers and pain) and regular reporting procedure for incidents ; a BPG-specific component integrated within the central nursing orientation program; regular topic-specific education sessions for organizational and unit-based nursing staff ; integration of BPG-based documentation items and tools into the health centre’s newly implemented electronic patient record; formalization of BPG-specific organizational policies; and ongoing communication about program activities . The majority of such changes had been initially instituted in the first few years of the program. Informants perceived these established or recurring program elements as representing the integration of the program into the regular organization and function of the nursing department.Most of the informant and documentary data characterizing program sustainability pertained to development. Development, mainly described in terms of “evolution,” had occurred increasingly in two major ways over the eight years since the start of the initiative. It entailed both the adaptation of the original program as well as the implementation of subsequent related innovation. We provide several illustrations of development in the last section of the findings, as program adaptation and subsequent innovation were central to explaining how the program had been sustained within the nursing department.Informants described that the influences on program sustainability were “multi-factorial.” Although a variety of factors were discussed, there was consistency in informants’ reports about which innovation, context, leadership, and process factors had been most influential in sustaining the BPG program at the departmental level over time. These 11 factors, sorted into our framework’s four categories of factors, are displayed in Fig. Informants perceived that two main innovation-related factors had positively influenced program sustainability. First, they affirmed that the program had remained relevant to a fundamental mandate of the health centre–to improve the quality of basic patient care. Second, they suggested that the evidence-informed nature of the program  had continued to be valued by nursing department members.There were five main context-related factors that influenced program sustainability. Two factors had positive influences. First, informants reported that the formal extra-organizational partnership with RNAO brought prestige to the initiative and encouraged some accountability for program activities and results. Second, in later years of the program, there was increased availability and exchange of patient safety-related data from other local and international centers. This, combined with accreditation bodies’ increased expectations for safety performance, provided external pressure on organizational leaders to ensure program continuation.The remaining three context-related factors had negative influences. First, informants described that in the absence of a designated operating budget to support the program, it had been a constant challenge to secure sufficient funding for ongoing activities. Second, informants described that issues related to organizational culture had made it difficult to instill a sense of ownership of the program across the nursing department. They indicated that the lack of a unified identity , as well as a weak philosophy of accountability (due to in part to the lack of enforced performance expectations), were entrenched cultural challenges. Third, because collaborations with other professions and departments had not been preserved or expanded over time, the program had remained largely limited to nursing, despite the necessity for multidisciplinary involvement.There were three main leadership-related factors that all had positive influences on program sustainability. The first was that the program had been led by co-directors. Informants emphasized how the same two active and persevering nurse leaders, who held top organizational positions, had chaired the program steering committee from the beginning. Informants reported that the main advantage of the partnership had been that each individual possessed a distinct expertise as well as a unique perspective on the program. One leader had a background in practice and administration; the other was seasoned in research and evidence-informed change. The second positive leadership-related factor was the long-standing commitment of several nursing leaders to the program. The third factor pertained to the complementary nature of those leaders’ actions across the departmental levels.Informants and documents described a variety of strategies and events that had occurred over the years since the beginning of the program. However, the data clearly revealed one dominant process that had become a strong positive influence on program sustainability. The persisting strategy of “reflection and course-correction” emerged as the main process underlying the use of several sustainability-oriented activities. As the expression suggests, this process entailed iterations over time of leaders’ deliberate efforts to learn from program experiences and, in response, to try to implement continued improvements to the program.Informants consistently described that the last three factors mentioned above: commitment of leadership, complementarity of leadership actions, and strategy of reflection-and-course-correction, had the greatest positive influences on program sustainability over the long term. We elaborate on these three factors in the next section of the findings, as their influences were central to explaining how the program was sustained.Although we have summarized separately the characteristics of and the factors that influenced BPG program sustainability, it was descriptions about the complex relationships between characteristics and factors that provided the explanations for how the program had been sustained within the nursing department. Informant accounts of how the program had been sustained converged on three important interactions between characteristics and factors. These three interactions are displayed in Fig. A key positive leadership factor for program sustainability was long-time commitment of a core group of nursing department leaders to the program. Several leaders, including co-directors, task force members, change advocates, and the BPG program coordinator had “stuck with” the program since early implementation. Informants explained that the leaders had remained dedicated largely because of the multiplicity of benefits that had resulted from the program. Several informants made clear that, as one expressed, “the positive things that we can see have driven the sustainability of us continuing to be involved. If we didn’t feel like we were making progress, the willingness to devote time [to the program] would be zilch” (O11). There was a reinforcing and reciprocal interaction between this sustainability characteristic and factor as leadership commitment contributed to program benefits, and multiple benefits then motivated further leadership commitment.Another key positive leadership factor for program sustainability was the complementarity of several leaders’ actions across the different departmental levels from the executive to the nursing units. Informants cited the concerted work of a core group of leaders from three particular types of roles–program co-directors, task force members, and clinical program nursing directors. The co-directors led the program from an executive-level perspective. Some task force members, who knew the clinical content of the BPGs, were regularly present on units to observe BPG practices and serve as expert resources for staff. These members then used their unit-based experiences to inform their task force’s strategic departmental work. The impact of some of the clinical program nursing directors was related to their function as the main linkage between the department executive and the nursing unit management teams under their supervision. We have summarized, in Table Several informants explained that the significance of the set of actions was that they were interdependently related to one another and orchestrated to operationalize common objectives across the different departmental levels. They were also coherently aligned to address the various sustainability-related issues that arose as the program continued. For example, the program co-directors advanced an agenda of expectations related to BPGs. This led towards task force members updating organizational policies and procedures with BPG content. Accordingly, clinical program directors included BPG-related criteria in their evaluations of managers’ and units’ overall performance. Another example included co-directors prioritizing program activities, resulting in clinical program directors relieving managers from other less essential dossiers. Informants perceived that these coordinated leadership actions promoted aspects of both institutionalization and development. In turn, further institutionalization and development allowed for subsequent iterations of these complements of leadership actions.Interview and documentary data clearly and consistently pointed towards one fundamental sustainability-oriented process that emerged over time. The strategy of “reflection and course-correction” meant that leaders continually assessed the “lessons learned” from their work on the program. Leaders then used these lessons to inform their modifications to the program. This was described as: “you never finish. We got to here, we’ve learned this, now let’s make it better. That is what helps in keeping [the program] alive. It’s evolving” (O12). Although departmental leaders had never overtly planned or named this as a strategy, informants described how several leaders had been deliberate in encouraging a reflective and responsive approach towards the work of sustaining the program.Informants described several examples of how this process factor  interacted with the development characteristic of sustainability . This interaction was perceived as having been strong and critical to program survival. Below are two commonly-cited illustrations of the relationships between the reflection-and-course-correction strategy and program adaptation. These are followed by the most prominent example related to subsequent innovation. These descriptions depict development at different time periods of the program and show the back-and-forth effects between reflection-and-course-correction and development.The first example pertained to the adaptation of the initial implementation phase of the program. It took six years for the department to implement the BPGs across almost all of its nursing units. Most informants described how the protracted time period had offered leaders the opportunity to reflect on their experiences of facilitating change with the first cohorts of units and to use the reflections to improve implementation with the later cohorts. One course-correcting change was that the support provided by external facilitators to nursing units became more structured . Another course-correcting change was related to the adaptation of the BPGs to the unique circumstances of units . Eventually, instead of using standardized implementation formats for all units, “mini implementations” were done on units  where the BPGs were less relevant. “Tailored implementations” were done on units where a common type of patient sub-population  prevented direct adherence to the standard BPG recommendations .The second example pertained to the adaptation of approaches to monitoring performance and improving quality. The department’s annual prevalence survey (pressure ulcers and pain) and the reporting system for incidents  were the program’s regular performance monitoring activities. Given the leaders’ intents to learn from previous efforts, measurement activities were modified over time to become more comprehensive and sophisticated. Informants and documents noted three course-correcting changes that reflected this. First, leaders made patient outcome performance targets increasingly specific over time. Initially the objective was simply “to improve.” Then leaders set internal benchmarks that felt reasonable for the institution. Leaders eventually drew upon emerging nationally and internationally reported research and administrative publications as standard-setting sources. Second, survey methods were modified over time. Initially, upper-level nursing leaders were the main surveyors. Then increasing numbers of staff nurses were engaged to collect data on units that were not their own. Eventually, in an effort to increase accountability for the BPG practices, staff nurses were required to survey their own units. The information collected in the survey was also changed. It grew from a focus on the three BPGs to also including data about related patient safety practices , in parallel with the department’s expanding safety agenda. Third, leaders tried to improve the communication about performance data. Early on, survey results were emailed to clinical program nursing directors and unit managers, under the assumption that the data would be acted on. Eventually, the program co-directors and coordinator scheduled meetings with management teams to discuss results. Informants described that unit teams were going to have to formally report back to their supervisor about how they would respond to their results.Informants described how the refinement of performance monitoring methods had allowed departmental leaders to identify units with lower levels of sustainability of nursing practices changes and of patient outcomes. This stimulated individualized, departmentally-supported quality improvement initiatives for those particular units. For example, externally-facilitated “BPG re-implementations” were done to help revitalize faded practices. “Targeted support” initiatives, such as assistance with audit and feedback activities, were also provided to help build units’ capacities to more effectively sustain the practice changes.The third example involved subsequent innovation. Several years into the BPG program, leaders’ reflections led them to seek innovative ways to further its initial successes. They chose to implement “Transforming Care at the BedsideInformants reported that TCAB had become the new focus for several reasons. Leaders had recognized the eventual need for many units to address larger quality issues, further than just the particulars of the BPGs. Leaders also recognized a need for units to work more on unit-specific issues, not just on organizationally-imposed mandates; and for units to build local quality improvement skills, instead of depending on outside support from the department. Most informants explained how TCAB, as one informant articulated, was “centered on broader areas than just nursing practice. It was stepping up the change a few levels…” However, it was a very clear message from informants that they “would not have seen TCAB if BPG hadn’t been here first” (O03). Several described that the development of the department  that had occurred as a result of the BPG program had “paved the way” for the subsequent, ambitious nursing initiative to be launched. TCAB was viewed by many as the next chapter in the development of the nursing department.Overall, the findings of this study support our conceptual framework. The framework proposed three characteristics of sustainability , influences from four categories of factors , and relationships between characteristics and factors. As our findings suggest, some aspects of the framework were more prominent than others in accounting for the sustainability of a BPG program at the nursing department level of the organization. Our discussion is therefore focused on these.First, with respect to the characterization of program sustainability, it was evident that all three characteristics were present, in different proportions and in different relationships to one another over the eight-year period. This substantiates a conceptualization of innovation sustainability that is broader than just the continuation of initial program activities. The eventual dominance of development thus characterized long-term program sustainability largely in terms of ongoing change. As there has been little examination of the nature and extent of the developmental aspect of sustained healthcare innovations , our finSecond, with respect to factors, we identified 11 factors that had most influenced the sustainability of the BPG program at the nursing department level of the organization. These factors are similar to those previously identified in studies about sustaining organizational programs in acute care –42. WhilLeadership, although represented in a range of ways , is one of the most commonly cited influences across sustainability studies from different fields , 3, 11. Strategic organizational processes, such as the notion of “navigating competing priorities,” have been identified as important for sustaining healthcare innovations . HoweverFinally, although sustainability characteristics and factors have typically been presented as discrete and distinct e.g., , 11, 71), 7111, 7Although our study provided an in-depth, contextualized, theory-informed illustration of one healthcare organization’s experience of sustaining a program over an eight-year-long term, the investigation would have been strengthened by comparisons with other similar programs within or outside of the organization. It would have been valuable to include the viewpoints of other types of program stakeholders, such as patients, families, or other professionals and administrators, beyond those of a diversity of informants from all levels of the nursing department. Although we used multiple data types, especially history-tracing documentation and long-involved informants, the retrospective approach misses observations attainable only through prospective methods.We have provided a theoretical illustration of our findings that is represented by the layered series of Figs. and the discontinuation of different types of innovations in a variety of organizational nursing settings. This could include a focus on organizational changes that are externally mandated, but not perceived to be a local priority; initiatives that are more “bottom-up” instead of organizationally-driven; and programs that have less departmental leadership infrastructure and support. Such investigations could be considered among institutions with varied governance approaches, organizational cultures, and types of healthcare service. We recommend that attention be directed to better understanding the mechanisms underlying the relationship between the three fundamental characteristics of sustainability, as well as between the characteristics and the factors that occur over long term periods–especially those between the development of innovations and organizational learning processes. We recommend further research using qualitative and mixed methodologies including in-depth, longitudinal, comparative case studies and ethnographies. This is appropriate given the complex and evolving nature of the phenomenon of healthcare innovation sustainability, in addition to the early state of knowledge about innovation sustainability in nursing.Future research should further explore the characteristics of and the factors related to both the sustainability Sustainability requires considerable attention and continual effort, which should be undertaken as an integrated part of improving overall institutional performance. The persistent, complementary, and shared actions of committed leaders, in a variety of roles across the department, seem necessary. Leaders may need to continually reflect on the successes and failures of their sustainability-oriented work, remaining attentive to the multiplicity of factors that accentuate or attenuate program sustainability, and to use those reflections as a basis for continued evolution and improvement. Structured facilitation and contextually-sensitive program implementation efforts, performance monitoring, and targeted quality improvement efforts with individual units, appear to contribute to program sustainability. Leaders could implement subsequent innovations that build upon, are consistent with, and extend the initial program, in order to respond to changing organizational needs, circumstances, and developmental stage.This study is a first to provide an in-depth description of the longevity of an evidence-informed program at the nursing department level of an acute healthcare centre. Our findings suggest that the successful initial implementation of an organizational program does not automatically lead to longer-term program sustainability. Organizational leaders should consider a broad conceptualization of sustainability, beyond just program institutionalization and/or program benefits, because the development of a departmental program may be necessary for its long-term survival.


RCSD1 gene has recently been identified as a novel gene fusion partner of the ABL1 gene in cases of B-cell Acute Lymphoblastic Leukemia (B-ALL). The RCSD1 gene is located at 1q23 and ABL1 is located at 9q34, so that the RCSD1-ABL1 fusion typically arises through a rare reciprocal translocation t. Only a small number of RCSD1-ABL1 positive cases of B-ALL have been described in the literature, and the full spectrum of clinical, morphological, immunophenotypic, and molecular features associated with this genetic abnormality has not been defined. We describe extensive genetic characterization of a case of B-ALL with RCSD1-ABL1 fusion, by using conventional cytogenetic analysis, Fluorescence In Situ Hybridization (FISH) studies, and Chromosomal Microarray Analysis (CMA). The use of CMA resulted in detection of an approximately 70 kb deletion at 7p12.2, which caused a disruption of the IKZF1 gene. Deletions and mutations of IKZF1 are recurring abnormalities in B-ALL and are associated with a poor prognosis. Our findings highlight the association of the deletion of IKZF1 gene with the t and illustrate the importance of comprehensive cytogenetic and molecular evaluation for accurate prediction of prognosis in patients with B-cell ALL.The  ABL1 gene at 9q34 are a well-known example of chromosomal abnormalities of major importance for diagnosis, risk stratification, and treatment selection in hematologic malignancies. ABL1 codes for the protein with tyrosine kinase activity. Its major fusion partner is the BCR gene, involved in the t which results in a formation of the so-called Philadelphia chromosome (derivative chromosome 22). The t results in the formation of a chimeric BCR-ABL1 fusion transcript, which encodes a constitutively active ABL1 tyrosine kinase. The BCR/ABL1 fusion is a well-studied entity, present in all the cases of Chronic Myeloid Leukemia (CML) and approximately 20% cases of B-cell ALL. In addition to BCR, six other genes have been reported to form rearrangements with ABL1: ETV6 at 12p13, RCSD1 at 1q24, SFPQ at 1p34, ZMIZ1 at 10q22.3, NUP214 at 9q34.13, FOXP1 at 3p12, SNX2 at 5q23, and EML1 at 14q32 /46,XX[14]. The patient was enrolled on AALL-1131 and received a 4-drug induction therapy with Vincristine/Daunorubicin/Pegaspargase/Prednisone. Her day 8 peripheral blood minimal residual disease (MRD) testing showed 18–25% blasts and her day 29 bone marrow evaluation revealed induction failure. She continued chemotherapy per the very high-risk control arm of AALL-1131 with the addition of dasatinib due to the RCSD1-ABL1 fusion. Her consolidation course was complicated due to presumed fungal infection treated with Ambisome 500 mg Q 24 hours IV and C. difficile colitis treated with Metronidazole and Vancomycin PO, but she was able to complete the consolidation treatment. The last bone marrow done on November 2014 to see the remission status was hypocellular (20–25% cellularity) with regenerative trilineage hematopoiesis, mild dyserythropoiesis, and no evidence of persistent/recurrent leukemia. The patient had a bone marrow transplant from a sibling in November 2014, but a few weeks after transplant she developed severe sepsis as well as capillary leak syndrome. She remained admitted to the intensive care unit where she unfortunately died on March 12, 2015. A 15-year-old female was admitted in June 2014 because of the 4-week history of sore throat and 4-day history of left thigh pain. On examination she had no hepatosplenomegaly or lymphadenopathy. Peripheral blood counts revealed hemoglobin of 9.2 g/dL, white cell count of 25100/Cytogenetic studies done on the bone marrow sample at the time of diagnosis revealed the karyotype 46,XX,t[10]/46,XX[14] . The sec MLL gene rearrangement, and trisomy for chromosomes 4, 10, and 17. BCR/ABL1 FISH testing done on bone marrow aspirate after induction therapy revealed no abnormalities. However, day 42 bone marrow aspirate showed rearrangement of the ABL1 locus, consistent with residual disease. Other consecutive samples showed negative FISH results with BCR/ABL1 probe.FISH analysis at the time of diagnosis revealed a gain of ABL1 signal in 78% of analyzed cells, consistent with the ABL1 rearrangement due to the t . FISH wa IKZF1 gene  noted by karyotype analysis was a completely balanced rearrangement and was thus not detected by CMA.CMA detected an approximately 70 kb copy deletion at 7p12.2 and 90 kb deletion at 13q14.2. The 70 kb deletion at 7p12.2 results in a disruption of theZF1 gene . The 90  ABL1 gene, t. This translocation involves the RCSD1 gene at 1q21 and has previously been described in only four patients and confirmed by cytogenetic or molecular methods only in two patients [ ABL1 rearrangements is that the N-terminal portion of the RCSD1 gene is fused to ABL1 starting from exon 4, while the other ABL1 fusion products also contain ABL1 exons 2 and 3 [ ABL1 gene, the RCSD1/ABL1 protein retains only a part of the ABL SH2 domain, which would predict association of the RCSD1/ABL1 fusion with ALL rather than CML [ RCSD1 gene codes for a protein kinase substrate CapZIP (CapZ-interacting protein), which is highly expressed in skeletal muscle and more weakly expressed in cardiac muscle. It has been shown that stress-induced phosphorylation of CapZIP may regulate the ability of CapZ to remodel actin filament assembly which is an important step in mitosis [ ABL1 protein has a tyrosine kinase activity and its fusion proteins act as constitutively activated tyrosine kinases, the formation of the RCSD1-ABL1 fusion gene could therefore result in CapZIP phosphorylation with loss of interaction between CapZIP and CapZ, leading to an alteration of the cellular function by affecting the cytoskeleton regulation, which could be an important step in leukemogenesis. It is important to note that B-ALL with the RCSD1-ABL1 fusion has recently been included into a specific molecular subtype of high-risk B-ALL, known as Philadelphia- (Ph-) like ALL, which is characterized by an expression signature resembling that of Philadelphia-positive ALL [We describe a patient with B-cell ALL associated with a particularly rare translocation affecting thepatients –9. The u 2 and 3 . As a cothan CML . Indeed, mitosis . As ABL1tive ALL . Similartive ALL –14, but The patient reported by Mustjoki et al.  received IKZF1 gene. IKZF codes for a DNA-binding protein Ikaros which displays crucial tumor suppressor functions in the hematopoietic system. Loss of Ikaros function has been linked to the development of lymphoid leukemia, in particular precursor B-ALL, and has been shown to confer a poor treatment outcome. Somatic aberrations of IKZF1 have been observed with particularly high frequency in B-ALL cases carrying a BCR/ABL1 rearrangement. More recently, it has also been demonstrated that cases of Ph-like ALL frequently carry IKZF1 abnormalities [ IKZF1 deletions still represent an independent predictor of poor prognosis [ IKZF1 deletion in our patient. No clinical correlates exist for this rare association to date. Although long-term follow-up information is not available for this case, we anticipate that the simultaneous presence of two high-risk genetic abnormalities in leukemia cells (RCSD1-ABL1 fusion and IKZF1 deletion) would predict a very high risk of relapse.Other factors that may modify the disease course and outcome in patients with RCSD1-ABL1 fusion are associated secondary genetic changes, including mutations and deletions of themalities , 13. Des IKZF1 abnormalities. Additionally, the effectiveness of combined treatment with chemotherapy and TKIs for patients with the RCSD1-ABL1 fusion and other genetic lesions associated with Ph-like ALL will have to be explored through large multicenter clinical trials.Our findings illustrate the importance of comprehensive genetic evaluation using a variety of assays in order to achieve fully accurate risk stratification for B-ALL patients. Additional cases will have to be identified and studied to ascertain the prognostic significance of the RCSD1-ABL1 fusion with and without cooccurrence of


The development of the Pediatric Rheumatology Program in Toronto began in 1984 with the recruitment of 3 junior but well-trained pediatric rheumatologists. Their goal, to create the finest pediatric rheumatology program in the world, was predicated on the need to both create and disseminate new knowledge. This required that all the elements of a strong academic program would be supported; (1) scholarly clinical care; (2) education at all levels including medical student, resident, fellow and continuing education; and (3) research, both clinical and basic. Early salary support from The Arthritis Society was critical to the recruitment and the hiring of a clinical fellow.Recognition of the expertise of the individual staff provided opportunities to pursue specific interests in a collegial and non-competitive environment. The shared vision was embraced such that individual goals and successes all contributed to the greater good and allowed each member to share in the successes of the others; each achievement not only advanced an individual's program but the entire division benefitted. Early provision of resources by the institution  provided the opportunity to rapidly build the clinical program. The development of sub-specialty interests was encouraged leading to a variety of specific clinics that built clinical experience and expertise, provided large cohorts for clinical research and allowed trainees to see a large number of patients with rare diseases even if they had otherwise limited exposure to rheumatology. The strong Research Institute affiliated with the hospital provided excellent scientific support for both clinical and basic science research endeavours. Presentation and publication of our work and international meetings gave us early credibility and highlighted training opportunities. To date, over 70 trainees from 27 countries have come through the program, and graduates continue to promote the program as a centre of excellence for clinical and research training.The presentation will review the development of the program and highlight the elements that have allowed the Toronto program to remain one of the leading academic programs for pediatric rheumatology in the world.R. Laxer Grant/Research Support from: Novartis.


Millions of children across North America and Europe live in families with alcohol or drug abusing parents. These children are at risk for a number of negative social, emotional and developmental outcomes, including an increased likelihood of developing a substance use disorder later in life. Family-based intervention programs for children with substance abusing parents can yield positive outcomes. This study is a realist review of evaluations of family-based interventions aimed at improving psychosocial outcomes for children of substance abusing parents (COSAPs). The primary objectives were to uncover patterns of contextual factors and mechanisms that generate program outcomes, and advance program theory in this field.Realist review methodology was chosen as the most appropriate method of systematic review because it is a theory-driven approach that seeks to explore mechanisms underlying program effectiveness (or lack thereof). A systematic and comprehensive search of academic and grey literature uncovered 32 documents spanning 7 different intervention programs. Data was extracted from the included documents using abstraction templates designed to code for contexts, mechanisms and outcomes of each program. Two candidate program theories of family addiction were used to guide data analysis: the family disease model and the family prevention model. Data analysis was undertaken by a research team using an iterative process of comparison and checking with original documents to determine patterns within the data.Programs originating in both the family disease model and the family prevention model were uncovered, along with hybrid programs that successfully included components from each candidate program theory. Four demi-regularities were found to account for the effectiveness of programs included in this review: (1) opportunities for positive parent-child interactions, (2) supportive peer-to-peer relationships, (3) the power of knowledge, and (4) engaging hard to reach families using strategies that are responsive to socio-economic needs and matching services to client lived experience.This review yielded new findings that had not otherwise been explored in COSAP program research and are discussed in order to help expand program theory. Implications for practice and evaluation are further discussed.The online version of this article (doi:10.1186/s13643-015-0158-4) contains supplementary material, which is available to authorized users. Substance abuse is a persistent and longstanding issue in society. It is estimated that 7 to 11 % of adults have an alcohol use disorder in the USA and UK, respectively . The impOver the past few decades, a small number of programs specifically targeting COSAPs have emerged and many take a family-focused approach to intervention. Family-based programming seeks to elicit change by involving multiple family members in an effort to strengthen family functioning and reduce negative childhood outcomes. It is argued that family-based intervention programs can be more effective than working with children or parents alone –12. EvidA systematic review of COSAP programs conducted in 2012 examined effect sizes of nine programs and concluded that there was preliminary evidence for their effectiveness, particularly for proximal outcomes such as family relationships, social behaviours and program-related knowledge . That paThe current study sought to fill this gap by adopting a theory-focused approach to improve understanding family-based interventions for COSAPs. This realist review focused on the following key research questions: What patterns of contextual factors and mechanisms can be identified, and how do they generate outcomes? Can a COSAP program theory be articulated and refined, and if so, what are the implications for program implementation? A theory-building approach was used to guide to this realist review.This review seeks to synthesize existing knowledge of family-based interventions for COSAPs and articulate a theoretical framework for how such programs work. Realist review was chosen as the methodological approach for this study. A realist review, also known as a realist synthesis, is a type of systematic review that examines research evidence on social interventions in an effort to explain how and why they work, or do not work, in particular contexts . The proA realist approach to a systematic review is ideal for examining social interventions, particularly those delivered in community settings, because it is recognized that programs are rarely delivered in precisely the same way, nor will they have the same outcomes, due to contextual variables that can never be fully controlled , 17. As Part of theory refinement process of a realist review is to examine the relationship between contextual factors and outcomes and the underlying mechanisms that connect the two . This isThis realist review followed practice guidelines and current publication standards for document selection, appraisal, data extraction and presentation of results, as outlined by the RAMESES project , 21. ThiA scoping search was conducted in order to identify any existing theories on family-based addiction interventions with a focus on children, as well as identify key programs or authors of note. We began our scoping with an extensive review of the literature on family-based theories of addiction. This process was guided by the collective professional knowledge of the field and lived experience of family addiction of the research team, as well as consultation with expert informants in the field. Based on this scoping search, we selected two candidate program theories of how parental addiction progressively impacts children, upon which COSAP programs are theoretically based: (1) the family disease model and (2) the family prevention model. These theoretical models of how addiction progresses within the family were helpful for the research team in making preliminary classifications of each program according to underlying addiction theory.The origins of family disease model programs are rooted in the abstinence and 12-step facilitation movements. Addiction is viewed as a family disease, whereby the entire family is affected by one person’s addiction . This thIn contrast, the family prevention model views addiction as one of many risk factors that characterize dysfunctional families. This theory suggests that parental addiction leads to poor parenting skills, poor emotion regulation and poor family cohesion, which in turn leads to childhood psychosocial problems, delinquency and eventually substance use , 26. ImpBoth candidate program theories are similar in that they share short-term goals of improving child psychosocial outcomes, parenting behaviours and family functioning, as well as a longer term goal of eventually reducing probability that COSAPs will develop substance use disorders later in life. Both approaches use the family unit as the vehicle for change. However, differences lie within the pathways to achieving those short- and longer term goals. A distinction is made between knowledge versus skill, as well as parent versus child as the primary target for family change. Programs based in family prevention will emphasize skills over knowledge and primarily target parents, while those taking a family disease perspective typically accentuate knowledge and the child’s experience. In sum, while both models ultimately aspire to similar ends , the origins and pathways inherent to each model is slightly different. Our goal was to refine these program models and articulate a COSAP program theory which could account for their success (or lack thereof). We hypothesized that differing mechanisms would be at play for each model of program.The document search process began with a systematic search of academic databases in the psychology, social services and health fields including PsycINFO, Medline, Scopus, CINAHL, Social Work Abstracts and Social Services Abstracts, in September 2013. Search terms included [program OR intervention OR treatment OR therapy] AND [child OR youth OR adolescent OR teenager OR student OR COA OR COSAP] AND [substance OR addiction OR drug OR alcohol] AND [family based OR family skills OR parent training OR parenting skills]. Other search engines such as Google and grey literature databanks were used in an attempt to identify grey literature such as community evaluations, government reports, conference proceedings and other documents not found in academic databases. This was an iterative process, and snowball searching was also undertaken by combing through article reference lists to identify any relevant documents that may have been missed through the initial search process. Additional documents were also revealed through correspondence with COSAP program researchers. Snowball searching continued until December 2014 to ensure that all relevant literature was identified. Only documents written in English were considered for inclusion.Documents were reviewed in stages at the title, abstract and full-text level to determine whether they met inclusion/exclusion criteria. These criteria were as follows: participants were children between the ages of 6 and 18 who have a parent who is a substance user; the parent with the addiction or another caregiver must attend the program with the child and programs are delivered in a group format. A decision was made by the research team to exclude programs geared towards infants and toddlers, as program format and content would likely be fundamentally distinct from those aimed at school-age children and their parents. Any program that was not explicitly family-based (i.e. did not involve both parents and children as participants) was excluded. Appraisal of selected documents were further screened by consensus for relevance and rigour, defined as ability to make a theoretical contribution to the review and trustworthiness of evidence presented in empirical studies . With reAn iterative process of data extraction was adopted for this study using a team approach. The research team consisted of a senior academic researcher with expertise in realist methodology, a doctoral level graduate student and a volunteer research assistant. The expertise of team members spanned child and clinical psychology, abstinence and harm reduction, marginalized populations, community-based research and personal lived experience with substance abuse. A coding abstraction sheet template, created by the research team, was used with each document in order to extract data relating to program descriptions, contexts, mechanisms and outcomes. Two members of the research team (AU and CD) independently coded all documents. The coding templates were revised and refined in an iterative fashion throughout the data extraction and analysis phase. Completed abstraction sheets were reviewed as a team for consistency, and disagreements were settled through consensus. Documents were then re-coded according to the final agreed upon coding abstraction sheets. Completed abstraction sheets were then reviewed among all research team members and an initial round of discussion took place about potential CMO configurations emerging from the data. CMO configurations were revised based on consensus, and documents were subsequently re-coded to ensure that CMOs had been properly identified. The research team convened on a regular basis to discuss demi-regularities emerging from the data and their degree of fit with candidate program theories. Demi-regularities were identified and labelled based on patterns of CMO configurations. When recurring patterns of program functioning were noted, CMO configurations were sorted and categorized in an attempt to explain the emerging demi-regularities. In cases where CMO configurations did not fully explain the demi-regularities, the research team returned to the data so as to refine the CMOs. Clarification of demi-regularities was then undertaken through a process of modification or generation of new demi-regularities, based on the CMO data. This was done in an iterative process through team discussions in an attempt to fully elicit sound explanatory themes. Document selection, appraisal and extraction for this realist review followed standard guidelines as outlined by the RAMESES project , 21.n = 16), grey literature community evaluation reports (n = 9), qualitative studies (n = 3), book chapters (n = 3) and conference presentations (n = 1). Additional file Figure A wide range of outcomes was reported for these programs. The majority were proximal and measured shortly after program completion. We summarized these to reflect general categories: child behavioural changes , child emotional changes , parenting , relapse prevention  and family cohesion . Very few studies were longitudinal and measured reduction or prevention of child substance use later in life. There were also slight variations in terms of dosage, structural format, content, target population  and eligibility for program participation .The COSAP programs included in this review were classified by the research team according to their potential support for the two candidate program theories, based on the program descriptions found in supporting documents. For example, programs that promoted a disease-based conceptualization of addiction, emphasized the important of abstinence and implemented a primarily knowledge-based curriculum were categorized within the family disease model. Programs that used a skills-based curriculum aimed at reducing risk factors for substance use and enhancing protective factors within the family were categorized within the family prevention model. Programs that appeared to draw elements from both models were classified as hybrids , Moving Parents and Children Together (MPACT), Family Competence Program (FCP)) discussed that the program succeeded in bringing families together for shared time that would not have otherwise been spent. It was frequently noted that providing opportunities for parent-child interactions in an enjoyable and supportive environment (context) led to improvements in family cohesion (outcome).hopeful enjoyment through which this outcome is achieved. Providing multiple opportunities for positive parent-child interactions (context) during the program was found to foster a sense of joy and pleasure (mechanism) among family members and an increased sense of hope that the family unit could be restored (mechanism). A qualitative evaluation of MPACT program provides a useful example of this process:I think the sheer fact that we went every week and we didn’t miss a week and we all did it together, just that alone I think … It made us feel good about ourselves .We identified the mechanism of Our review documented multiple instances of programs that encouraged families to spend time together in a supportive and non-punitive environment. This allowed parents to develop empathy (mechanism) for their children, and in turn children were allowed a safe space (context) to express themselves to their parent during the program . Having hopeful enjoyment did not have sufficient opportunity to fire in this case. Further, older children actually reported negative effects of parental involvement, suggesting that attempts by parents to increase parent-child interaction time were not only lacking in enjoyment but were in fact met with rejection. The authors of that paper hypothesized that older children who were accustomed to lack of supervision (context) perceived increased family time as an unwelcome intrusion [hopeful enjoyment needs to be triggered in order for positive outcomes in family cohesion to occur, in the context of child age and prior experience with parental supervision. Programs that facilitate positive parent-child interactions can help families achieve a restored connection when it is developmentally appropriate for them to do so, as is more likely to be the case for younger children. For older children who are at an individuation developmental stage, attempts at eliciting hopeful enjoyment of family interactions may misfire and fail to yield positive outcomes. The accumulation of CMO evidence found in support of this demi-regularity suggests that positive parent-child interactions occurring within a safe and supportive environment, which can be actively fostered by the COSAP program, are instrumental in yielding increased family cohesion.2. Supportive peer-to-peer relationshipsAnother useful illustration of this demi-regularity is an example where desired program outcomes were not achieved. The Focus on Families (FOF) evaluation indicated that the program did not achieve desired outcomes for family cohesion –33. In tntrusion . This prtrust and safety within the group as well as validation of experience. Improvements in child psychosocial functioning were consistently reported in these cases Environments that fostered supportive peer relationships among child participants and among parents were noted across many programs as being instrumental in achieving positive child psychosocial outcomes and to a lesser extent positive parenting outcomes. Evidence was found to support this process in a couple of ways. First, supportive peer-to-peer relationships between the child participants elicited mechanisms of es e.g. , 34, 35), 35trusttrust and safety mechanism is instrumental in achieving positive child behavioural outcomes. This finding would suggest that attention to participant characteristics such as similar age and lived experience, when forming program peer groups, is warranted as it will facilitate positive peer-bonding and will more likely trigger perceptions of trust and safety.Conversely, negative peer-bonding between child participants was noted in one COSAP program evaluation , furthervalidation of experience. Parents struggling with parenting at the same time as recovering from substance abuse (context) were reported to have found the group dynamics and peer relationships fostered with other parents during the COSAP programs to be beneficial. The process of validation (mechanism) for these parents can be described as the normalization of experience and sharing of mutual struggles among supportive peers. Further, at least one program evaluation discussed the possibility that the strong bonds formed between participants were a motivator to continue attending sessions (outcome) [Both adults and children appeared to benefit greatly from meeting others and making friends, specifically with people who lived in similar circumstances. For many, this seemed to bring mutual understanding as families’ experiences were normalized and they realized that they were not alone with their struggles. Second, we noted that parent participants who were placed in supportive groups with other peers also exhibited positive outcomes through a mechanism of outcome) . It is p3.(Addiction) Knowledge is powerThus, according to this demi-regularity, programs that address the social isolation common to substance using families through strategies such as appropriate peer grouping are more likely to trigger mechanisms of validation and trust.parental recognition and responsibility and children relinquishing responsibility for parental addiction.Programs that specifically emphasized knowledge about addiction and education around the impact that substance abuse has on children and families were found to yield improvements in parenting and child psychosocial outcomes. The following key mechanisms were identified within this demi-regularity: relinquishing responsibility is triggered when children are provided with information about parental addiction that had been previously withheld or downplayed (context). The process of relinquishing responsibility appears to be a key element of the knowledge provision demi-regularity and may set the stage for subsequent improvements in child emotional and behavioural outcomes to occur, either directly through this demi-regularity or through one of the others identified in this review.In the Betty Ford program, for example, knowledge was described as “opening the door for them”  parental recognition and responsibility mechanism is triggered under these circumstances, whereby parents are able to recognize the impact of their behaviours and take responsibility for how it has affected their children. For example, evaluations of the Celebrating Families! program and MPACT program both documented instances where parents gained new understanding of the impact that alcoholism has on the family (mechanism) [Further, parents who attend these programs are also provided with knowledge about how their alcohol or drug use has impacted their children and the family unit. The chanism) , a realichanism)  and “thechanism) , p.18). chanism) .engagement emerged as an important intermediary factor that was necessary in order for outcomes to be achieved. Interestingly, the process of engagement is not explicit within the family prevention model nor the family disease model, perhaps because engagement is assumed to occur once recruitment is established or that engagement is equated with program attendance. As such, engagement did not initially emerge within either candidate program theory. However, as data abstraction progressed, we noted that only those programs classified in this review as aligning with the family prevention model were attuned to this issue. Information on recruitment best-practices exists within family-based intervention literature (e.g. [trust (mechanism) among families who are typically marginalized (context). For example, SFP and the Safe Haven program both went to extensive lengths to encourage and incentivize participants, such as providing meals, transportation, childcare, basic necessities and vouchers redeemable for family activities. Families participating in these programs were contextually characterized as low income, low education, having unstable housing, child welfare involvement and, unsurprisingly, often mistrustful of service providers. The key mechanism here is the sense of trust and acceptance that is developed on the part of the client in response to these staff/program efforts, as evidenced here: “Basic material supports provide a message to needy families that the staff really care about them” ([Responsiveness to client socio-economic status (SES) needs. Programs that are responsive to the SES realities and needs of their clients (context) will encourage program engagement (outcome) by fostering a sense of t them” The Safe Haven staff began to know and understand the unique circumstances of each of the participating families. This seemed to increase staff empathy for the families. The families, in turn, reported to the process evaluator that they felt the staff “cared about them” .For certain programs where the participating families were recruited from particularly marginalized populations , re (e.g. ); howevere (e.g. ). In theb)trust and personal identification with the program materials (mechanism). This was true of programs such as Safe Haven where extensive efforts to make the program and staff culturally consistent with an African-American worldview led to increases in client acceptance and engagement: “They put it in a way Black people can understand” ([trust (mechanism) in program and led to increased engagement (outcome). We hypothesize that once engagement is established, other program outcomes will be more easily facilitated via the demi-regularities explored above, creating a series of CMO chains.Matching to client lived experience. Programs that took appropriate steps to match staff and client lived experience of family addiction and/or cultural background (context) were more successful in engaging clients (outcome) by fostering rstand” rstand” In the example above, the program’s responsiveness to client SES needs (context) affected both the staff’s ability to engage with the families and vice versa (outcome), through a process of trust building (mechanism).Note that our review revealed that cultural adaptation, such as the one described in the Safe Haven program, did not always lead to better outcomes. For example, a number of SFP formats have been culturally adapted for a variety of different ethnic groups in the USA. However, comparisons between generic SFP and culturally adapted versions yielded no improvements in positive outcomes, beyond an increase in retention . Based oAs part of the analysis process, the four demi-regularities described above were examined with respect to their alignment with the candidate program theories. Programs classified within the family disease model were supported with evidence from the “knowledge” and “supportive peer relationships” demi-regularities. This suggests that the provision of knowledge that is specific to family addiction (context) facilitates children in relinquishing the responsibility for their parent’s addiction (mechanism) and enables parents to recognize and take responsibility for the impact of their addiction on their family (mechanism), leading to improved coping and reduced family stress (outcome). Further, social support provided to families within the context of a supportive peer relationship (context) serves to validate the experiences of families living with addiction (mechanism), leading to improved coping and parenting behaviours (outcomes). The family disease model asserts that defining addiction as a disease is fundamental to the process of relieving oneself from the guilt and responsibility for a family member’s addiction . The “knPrograms developed from the family prevention model were evidenced with the “positive parent-child interactions”, “supportive peer relationships” and the “engagement” demi-regularities. This suggests that opportunities for positive parent-child interactions within the context of a safe and supportive environment encourage families to seek joy in spending time together (mechanism) and find hope in the restoration of the family unit (mechanism), ultimately leading to improved family cohesion (outcome). Family prevention theory argues that involving parents in the promotion of healthy child functioning will reduce risk factors and enhance strength and protective factors . The impEngagement was found to be present within programs originating in family prevention only. Despite the fact that engagement has not previously been noted within COSAP program literature, it has been validated elsewhere. Findings from Jackson and colleagues’  realist It is notable that two programs included in this review were classified as hybrids, as they drew upon elements common to both candidate program theories. Hybrid programs were evidenced from a combination of all demi-regularities to varying degrees, with the exception of engagement. This is an interesting finding in and of itself but also supports the case for using realist methodology in evaluation inquiry. It would suggest that in practice, program implementation is complex. The MPACT program, for example, was reportedly influenced by the SFP model  and was trust, validation, children relinquishing responsibility for parental addiction and parental recognition and responsibility. Family prevention-based programs appear to be effective when they encourage positive parent-child interactions, facilitate supportive peer relationships and are attuned to client engagement. The key mechanisms triggered in those situations are hopeful enjoyment, validation and trust. The interesting case of the hybrid programs highlights that elements from both theoretical orientations can successfully be implemented within a single program. These findings suggest that in practice the differentiation between these two models may be less clear, as some common demi-regularities were noted across candidate program theories (e.g. supportive peer relationships). Given the overlap between theories, the evidence indicates that the CMO configurations uncovered in this review can be situated within a broader and more encompassing COSAP program theory. Previous research has established that COSAP programs can be effective in supporting children and parents living with family addiction . This rA number of limitations are worth noting. Only 7 COSAP programs were found and included in this realist review. While many programs had rich data embedded within the supporting documents, it is entirely possible that other CMOs not reported here might also explain program outcomes. As such we cannot claim that our findings are exhaustive. Second, there were some contextual factors not consistently reported in the evaluations that may have otherwise been relevant. For example, some parents were concurrently enrolled in addiction treatment programs yet no consistent pattern of outcomes could be found. Other parents were described as being “in recovery”, although this was not clearly defined nor were outcomes compared at this level. Level of substance use severity was also inconsistently reported. Child-specific contextual factors were also rarely reported and no discernible patterns of outcomes by age or gender were found across programs. It is possible that had more data been available these contextual factors would have yielded different demi-regularities; however, for the purpose of this review, we concluded that were not enough evidence to confidently make assertions about the impact of those contextual factors. Therefore, while we are confident in the strength of the findings reported, adherence to the CMOs and the demi-regularities identified in this review do not guarantee future program success. Finally, as in any systematic review, the quality and rigour of the evaluations varied considerably. While the realist review methodology is broad in scope and allows for supporting documents to be included to mitigate these factors, there may have been outcomes not sufficiently explored due to methodological constraints of the COSAP program research currently available. This speaks to the need for enhanced evaluation and research of COSAP programs in applied settings.This review sheds much needed light on the mechanisms and processes that contribute to COSAP programs’ effectiveness. Yet, it also highlights some significant gaps in the level of evaluation currently being conducted in the family addiction treatment field. Specific attention to intervention fidelity, contextual factors and the increased use of mixed-methods approaches would significantly enhance the rigour and quality of reporting of program evaluations. The inclusion of qualitative studies in the present realist review was highly valuable to our exploration of CMO configurations and demi-regularities. We suspect that had more mixed-methods approaches been reported in the literature, the richness of data uncovered would have been even greater. Moreover, the collecting and reporting of contextual data in evaluation research in this field needs to be more nuanced. Client and facilitator characteristics that are the easiest to measure  are arguably the least relevant to outcomes . Increas


The Accreditation Council for Graduate Medical Education requires residency programs to provide curricula for residents to engage in scholarly activities but does not specify particular guidelines for instruction. We propose a Resident Scholarship Program that is framed by the self-determination theory (SDT) and emphasize the process of scholarly activity versus a scholarly product.The authors report on their longitudinal Resident Scholarship Program, which aimed to support psychological needs central to SDT: autonomy, competence, and relatedness. By addressing those needs in program aims and program components, the program may foster residents’ intrinsic motivation to learn and to engage in scholarly activity. To this end, residents’ engagement in scholarly processes, and changes in perceived autonomy, competence, and relatedness were assessed.Residents engaged in a range of scholarly projects and expressed positive regard for the program. Compared to before residency, residents felt more confident in the process of scholarly activity, as determined by changes in increased perceived autonomy, competence, and relatedness. Scholarly products were accomplished in return for a focus on scholarly process.Based on our experience, and in line with the SDT, supporting residents’ autonomy, competence, and relatedness through a process-oriented scholarship program may foster the curiosity, inquisitiveness, and internal motivation to learn that drives scholarly activity and ultimately the production of scholarly products. Residency programs accredited by the Accreditation Council for Graduate Medical Education (ACGME) must provide curricula designed to advance residents’ knowledge of the basic principles of research and opportunities for residents to participate in scholarly activity (defined as taking a systematic approach to a question or to a project) , 2. To tThis trend article describes the process-oriented Resident Scholarship Program, developed in the Department of Pediatrics at Baylor College of Medicine in Houston, Texas, USA. It begins with a review of the theoretical framework that guides the Resident Scholarship Program, chronicles program development, and recounts preliminary program evaluation findings. It ends with a brief discussion of how, based on its theoretical framework, the program has had positive effects on residents.In 2012, residency program leadership conducted a needs assessment to ascertain residents’ understanding of, and interest in, scholarly activity and scholarship. The needs assessment revealed that 1) residents valued pursuing scholarly activities that aligned with their personal interests, 2) many residents had participated in research before entering residency but were not currently engaged in any scholarly activities, and 3) residents tended to perceive scholarship as research and discount other types of scholarship .The following year, the program directors collaborated with the principal investigator to create a scholarship program that would address issues identified by the needs assessment and fulfill the ACGME mandate. The principal investigator formed an oversight committee consisting of an education researcher, a quality improvement specialist, and a faculty member interested in education. The oversight committee members acted under the auspices of the residency program to develop, implement, and evaluate a scholarship program for pediatric residents that spanned their three years of training.The oversight committee members (hereafter referred to as we) drafted initial program aims after reviewing the literature and speaking to colleagues who administered scholarship programs in other residencies. As much as possible, we capitalized on existing educational activities; for example, we included lectures on research design and scientific writing in the noon conference curriculum. With the support of residency program directors, the Vice-Chair for Education, and the Chair of Pediatrics, we secured a 4-week required rotation for residents’ scholarly activity in the second year.autonomy entails choices and respecting alternative perspectives. Competence entails a reasonable level of challenge, and assistance in developing knowledge and skills to meet that challenge. Relatedness entails acknowledging human emotions and creating human connections.In the process of developing the Resident Scholarship Program, we familiarized ourselves with the self-determination theory (SDT). We embraced SDT as a theoretical framework, given its alignment with the aims and process-orientation of our program. Briefly, SDT purports that supporting three basic psychological needs – autonomy, competence, and relatedness – fosters intrinsic motivation to learn, that is, actions or behaviors driven by interest or enjoyment, not external pressures or rewards –14. Consrelatedness led us to think about ways to engage the larger pediatric faculty in residents scholarship. As also displayed in With these psychological needs in mind, we revised our program aims to better align with the SDT framework. For example, and as displayed in We set forth intended learning outcomes for program components. As displayed in We designed the first year of the program to be an introduction to scholarship. We secured time early in residents' first year of training to introduce them to Boyer's  broad deWe designed the Resident Scholarship Program to be largely experiential in the second year of residency. All second-year residents must complete a 4-week, scholarly activity rotation. In this rotation, residents have protected time to work on their scholarly project under the supervision of their project mentor. Residents also have opportunity to explore project-related learning opportunities .We encourage residents to use their scholarly activity rotation to delve into the scholarly process rather than concentrate on having a product by the end of the rotation. We use Glassick's criteria , outlineWe designed the third year of the program to be driven by third-year residents’ desire to bring their scholarly project to completion, or to engage in another scholarly project. We allow third-year residents to devote an elective rotation to their scholarly project and/or participate in the annual, department-wide Resident Scholarship Day.As stated previously, we used the STD to guide program evaluation . In thisWe conducted formative evaluation in the spring of 2014. We interviewed four project mentors and asked residents who completed the scholarly activity rotation for feedback about the scholarship program. Based on that feedback, we added the end-of-rotation reflective sessions described above. The reflective sessions continue to be a means of collecting ongoing formative feedback about the program from residents, project mentors, and residency program directors. To better appreciate the perspective of project mentors, we asked one of them (AH) to join our oversight committee in 2014.We catalog and categorize residents’ scholarly projects by Boyer's types of scholarship. We get a general sense of residents’ engagement by reviewing their reflective summaries. We administer an online program evaluation survey every 6 months; retrospective pre-post items ask residents directly about gains in confidence in the processes of scholarly activity and indirectly about their sense of autonomy, competence, and relatedness. We also include several items asking specifically about how residents forged relationships with their project mentor.For this report, we applied descriptive statistics to evaluation data. We used McNemar's test to compare (pre- vs. post-rotation) proportions of residents responding to survey items related to autonomy, competence, and relatedness. Our program evaluation has been approved by the IRB at Baylor College of Medicine.In the following section, we share evaluation data that we planned to collect as well as evaluation data that we did not plan to collect but view as evidence of the Resident Scholarship Program achieving its aims.Based on our catalog of scholarly projects for 2013–2014 and 2104–2015 academic years, and our review of reflective summaries, residents engaged in a variety of scholarship: discovery ; application ; teaching ; and integration .“It was a very valuable learning experience to pursue a scholarly activity without competing clinical duties for a full block. And being able to complete a (scholarly) project during residency is intrinsically rewarding.”Data from our program evaluation survey for 2013–2014 and 2014–2015 indicated that residents gained a broader understanding of scholarship as a result of the Resident Scholarship Program, with 96% (48/50), 98% (49/50), and 93% (46/50) seeing application, teaching, and integration, respectively, as scholarship. Nearly all residents reported positive regard for the program, with 68% rating the overall quality as ‘excellent’ or ‘outstanding’ and 30% as ‘good’. Open-ended feedback supported numeric ratings. For example, one resident commented, ‘The scholarship program is a phenomenal opportunity during residency. Please continue with this program as it is a unique opportunity for us work in an area of interest’. Another remarked,p<0.05, McNemar's test) in selecting a project that aligns with their career interests , directing their own learning as it related to their scholarly project , disseminating scholarship , collaborating around scholarly pursuits , and establishing a mentoring relationship . Open-ended feedback also supported the findings. For example, one resident commented, ‘I liked the independence this rotation gives and the protected time to work on a project of our choosing [autonomy]’. Another reported,“I never participated in these things (scholarship) during college or medical school because I thought they were boring. But this month really gave me the confidence that I can write a paper that other people might care about [relatedness]. At this point I would be far more likely to write up something in the future than I have ever been in the past [competence].”Significantly more residents felt confident , 58 (80%) reported either completing their project with recent dissemination  or planning dissemination during the coming year.Residents’ enthusiastic regard for the Resident Scholarship Program caught the attention of the Vice-Chair for Education and the Chair of Pediatrics, resulting in funding for resident travel to present their scholarly projects, for Resident Scholarship Day, and for part-time administrative assistance. In addition, we have disseminated work about the Resident Scholarship Program at several national venues. Locally, our program received an Educational Scholarship Award, funded by Texas Children's Hospital, and was recognized with the Educational Innovation Award in the Department of Pediatrics at Baylor College of Medicine.Based on our experience, and in line with the SDT, we believe that supporting residents’ autonomy, competence, and relatedness through a process-oriented scholarship program may foster the curiosity, inquisitiveness, and internal motivation to learn that drives scholarly activity and ultimately the production of scholarly products. Our use of the SDT as a program theory enables us to go beyond claims that our program worked and speculate about how it worked . First, On a practical level, SDT was a useful theoretical framework and aid for decision making as the Resident Scholarship Program evolved. For example, when deliberating about whether residents should be required to participate in Resident Scholarship Day, we chose to make it optional, thus supporting residents’ autonomy. When deliberating about whether residents could partner on a scholarly project, we decided in favor of partnership, thus supporting relatedness.We acknowledge the limitations of our work, notably the lack of a comparison group and self-reported measures of autonomy, competence, and relatedness. The speculations we offer about our program outcomes are preliminary; in the future, we hope to determine if our appeal to residents’ curiosity, inquiry, and motivation to learn actually results in publications, presentations, and enduring materials.Although others have used SDT to promote autonomy among pediatric residents , ours is