Research paperDevelopmental evaluation of family functioning deficits in youths and young adults with childhood-onset bipolar disorder
Introduction
Childhood-onset bipolar disorder (BD) is a complex condition affecting 1–2% of youths (Van Meter et al., 2011). Compared to individuals with late adolescent- and adult-onset BD, youths with childhood-onset BD spend more time symptomatic with mixed depressive and manic presentations, rapid mood fluctuations, and subthreshold symptoms (Birmaher et al., 2009, Birmaher et al., 2014, Geller et al., 2008). These youths also have greater functional impairment (Perlis et al., 2009), poorer quality of life (Perlis et al., 2009), and higher risk for suicidality (Perlis et al., 2004). In addition, childhood-onset BD often persists into adulthood, leading to further impairment and negative outcomes (Axelson et al., 2011, Birmaher et al., 2009, Birmaher et al., 2014, Geller et al., 2008, Leverich et al., 2007). Given the enduring nature of this disorder, there is a critical need for studies to directly evaluate developmental effects by aggregating data from children, adolescents, and adults in order to examine the phenomenology and mechanisms of BD across the lifespan, and thereby enhance diagnosis and treatment efforts.
Family functioning is one such process relevant to BD and important to understand from a developmental perspective, as findings could indicate optimal family involvement in treatment and age-specific intervention targets. In addition to the patient, families of individuals with childhood-onset BD are quite impaired. Compared to healthy controls (HCs) and youths with other psychiatric conditions, families of youths with BD display high levels of conflict, control, aggression, quarreling, forceful punishment, tension, stress, and negative expressed emotion; and low levels of warmth, affection, intimacy, cohesion, expressiveness, organization, and positive expressed emotion (Belardinelli et al., 2008, Keenan-Miller et al., 2012, Nader et al., 2013, Perez Algorta et al., 2017, Schenkel et al., 2008). Family dysfunction also predicts worse course of BD in youths, including: 1) low maternal warmth (Geller et al., 2008); 2) chronic stress in family, romantic, and peer relationships (Kim et al., 2007, Siegel et al., 2015); 3) frequency and severity of stressful life events (Kim et al., 2007); 4) low levels of cohesion and adaptability (Sullivan et al., 2012); and 5) high levels of conflict (Sullivan et al., 2012). This relationship is also bidirectional, with patients’ symptoms/behaviors reciprocally influencing caregivers’ burden/distress (Reinares et al., 2016b). Thus, psychosocial evidence-based treatments (EBTs) for childhood-onset BD incorporate family-based strategies including psychoeducation, communication, problem solving, and affect regulation to address these impairments (Fristad and MacPherson, 2014).
Familial caregivers (e.g., parents, spouses, close relatives) of adults with BD display comparable dysfunction, including low levels of cohesion, expressiveness, and organization; and high levels of conflict (Miklowitz, 2011, Miklowitz and Johnson, 2009, Reinares et al., 2016a, Solomon et al., 2008, Weinstock et al., 2006). In addition, high expressed emotion (Kim and Miklowitz, 2004, Yan et al., 2004) and familial negative affective style (O'Connell et al., 1991) predict recurrence in adults with BD. However, no research has examined the persistence of family dysfunction into adulthood among individuals with childhood-onset BD. One study demonstrated that adults with retrospectively obtained childhood-onset BD experienced sustained psychosocial/functional impairment during prospective observation on a measure that assessed work, relationships (including family), recreation, and life satisfaction (Perlis et al., 2009). Though, family functioning in particular was not assessed in this study, and determination of childhood-onset BD diagnoses may have been influenced by retrospective recall bias (Leboyer et al., 2005). Importantly, no studies have directly compared family functioning in youths with BD vs. adults with prospectively verified childhood-onset BD (youth participants with BD followed into adulthood).
Unfortunately, research is often artificially bifurcated by regulatory requirements or investigator expertise/training in pediatrics or adults, and few datasets have prospectively established childhood-onset BD (Birmaher et al., 2009, Geller et al., 2008). These limitations make it challenging to evaluate developmental differences in mechanisms and processes implicated in childhood-onset BD. In addition, no studies have specifically examined the developmental progression of familial dysfunction in this condition, despite its relevance to onset and course of the disorder (Geller et al., 2008, Kim et al., 2007, Reinares et al., 2016b, Siegel et al., 2015). Importantly, parent and family variables also influence psychosocial treatment outcomes in childhood-onset BD, serving as both moderators (Miklowitz et al., 2009, Sullivan et al., 2012, Weinstein et al., 2015) and mediators (MacPherson et al., 2016, Mendenhall et al., 2009). Thus, enhanced understanding of family processes in childhood-onset BD is crucial from both a phenomenological and intervention perspective.
To address gaps in the literature and better conceptualize familial dysfunction across development, the current study examined family functioning in youths with BD, adults with prospectively verified childhood-onset BD, and youth and adult HCs. Adults with BD were followed since childhood via their participation in the Brown University site of the Course and Outcome of Bipolar Youth (COBY) study to ensure that retrospective recall bias did not impact BD diagnoses (Birmaher et al., 2009, Leboyer et al., 2005). Hypotheses were based on research documenting a more severe course of illness and functional impairment in youths vs. adults with BD (Birmaher et al., 2009, Geller et al., 2008, Perlis et al., 2009, Perlis et al., 2004). In addition, youths likely had less time to seek treatment and develop strategies for managing symptoms/stressors than adults with childhood-onset BD, given longer duration of illness in the latter, potentially contributing to exacerbated family dysfunction at younger ages. Thus, it was hypothesized that: 1) youths and young adults with childhood-onset BD would demonstrate impaired family functioning compared to HCs; and 2) youths with BD would display worse family functioning compared to adults with childhood-onset BD.
Section snippets
Participants and procedures
Participants were enrolled in one of two studies approved by the Institutional Review Boards of Bradley Hospital and Brown University. Written informed parental consent and child assent were obtained for youths; written informed consent was obtained for adults. Subsequently, parents, youths, and adults completed assessments and measures cross-sectionally. The sample included 116 individuals with childhood-onset BD (70 youths, 46 adults) and 108 HCs (46 youths, 62 adults).
Inclusion criteria for
Demographics
There were no differences between participants with BD vs. HCs for sex and race (Tables 1 and 2). Collapsing across ages, participants with BD were younger and had lower FSIQ than HCs; there was no difference in Hollingshead-categorized SES (Table 1). When examining demographics across diagnostic groups by age groups, youths with BD had significantly lower FSIQ and SES than youth HCs; there was no difference in age (Table 2). Adults with BD were significantly younger and had significantly lower
Discussion
To our knowledge, this is the first study to take a developmental approach to investigate family functioning in youths and young adults with prospectively verified childhood-onset BD. Results indicated that participants with BD had significantly worse family functioning in all domains (problem solving, communication, roles, affective responsiveness, affective involvement, behavior control, general functioning) compared to HCs, even when controlling for SES and FSIQ. The hypothesized
Author's contributions
Heather A. MacPherson and Daniel P. Dickstein conceptualized the current study. Heather A. MacPherson conducted all statistical analyses, led the literature search, and wrote the first draft of the manuscript, as well as reformulated each following draft.
Amanda L. Ruggieri and Rachel E. Christensen constructed and organized the database file for these analyses and assisted with data cleaning.
Amanda L. Ruggieri, Rachel E. Christensen, Elana Schettini, Kerri L. Kim, and Sarah A. Thomas assisted
Funding
This study was supported by Emma Pendleton Bradley Hospital and the National Institute of Mental Health grants K22MH074945 and R01MH087513 (Principal Investigator Daniel P. Dickstein).Young adult participants with bipolar disorder were recruited from Brown University’s site of the Course and Outcome in Bipolar Youth (COBY) study (R01MH059929). The funders had no role in the study design; in the collection, analysis, and interpretation of data; in the writing of the report; or in the decision to
Acknowledgments
We gratefully acknowledge and thank the youths, young adults, and their families for their time and effort participating in these studies, without which this research would not be possible.
Conflicts of Interest
None.
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