Methods

Participants

Caregivers of children with SCD were recruited from two comprehensive pediatric SCD centers (Buffalo and Pittsburgh). Eligibility criteria were: (1) primary caregiver of a child, from birth through 10 years of age, diagnosed with any type of SCD; (2) English-speaking; and (3) residing within 30 miles of the respective study site.

Procedures

Medical care teams were informed about BI and previous research demonstrating success (acceptability and effectiveness) amongst caregivers of pediatric oncology patients. Medical providers then approached eligible caregivers during the child’s routine outpatient hematology appointments and/or inpatient admissions, to obtain caregivers permission to be contacted regarding their interest in this project (Figure 1). After obtaining consent, caregivers were asked to complete baseline assessments (T1) and a time was scheduled to start BI sessions.
B right IDEAS Problem-Solving Skills Training
BI is an evidence-based manualized intervention, based foundationally on the tenets of Problem-Solving Therapy23. In previously published work, the intervention was titled Problem-Solving Skills Training18,19 (PSST); however, during an ongoing dissemination science grant, professionals trained to deliver the intervention, as well as patient advocates, suggested that relabeling the intervention (i.e., Bright IDEAS rather than PSST) would make it more acceptable to families. In addition, professionals and advocates felt the Bright IDEAS label was a more accurate depiction of the goals of the intervention.
Bright signifies the concept of optimism (i.e., positive problem orientation - You can do it ), which is essential to successful problem-solving24. ‘IDEAS’ is a mnemonic for each of the BI problem-solving steps with each letter signifying a step in the problem-solving process: I (Identify the problem), D (Determine all possible options), E (Evaluate your options - pros and cons of each option), A (Act - create an action plan based on D and E), and S (See if it worked - if the plan does not work go back to steps D and E respectively).
During BI sessions, participants are taught to utilize a five-step approach to problem-solving18,19,20 (Figure 2). Over the past 25 years of developing and assessing the effectiveness of BI, a comprehensive instructor’s manual (Supplement 1: Instructor’s Manual) was developed to serve as a guide for therapists when delivering the intervention to caregivers. It was not changed for this research. For this study, we modified the Cancer Parent Manual previously developed for caregivers of children with cancer, to include relevant information for caregivers of children with SCD (Supplement 2: SCD Parent Manual). We also used BI worksheets (Supplement 3: Worksheets) during sessions, as these materials contain attractive graphics and simplify the problem-solving process for caregivers. The worksheets were not changed for this research.
BI was presented to caregivers as a systematic approach that could be used to overcome any life challenge, including those commonly faced when caring for a child diagnosed with SCD. Following our template for providing BI to caregivers of children with cancer, BI was delivered over the course of 6-8 individual sessions, with each session following a structured format, and lasting approximately 30-60 minutes. During the first session therapists focused on: establishing rapport; learning about the family and the child’s medical history; and explaining BI. Caregivers were also provided a copy of the SCD Parent Manual and Worksheets. During subsequent sessions (i.e., sessions 2-8) therapists focused on: reviewing the BI program and worksheets; assisting caregivers with identifying challenges; systematically guiding caregivers through the problem-solving steps using the ‘IDEAS’ mnemonic; and during the final session the principles of the program were reviewed and strategies for relapse prevention were discussed.
Although the content of BI was identical to previous efforts with PSST, in the current study, delivery times and locations were markedly more flexible and determined based on the caregiver’s preference and needs to reduce the impact of logistical barriers (e.g., lack of transportation, reliable childcare, etc.) on participation. In prior BI studies within pediatric oncology, sessions were primarily conducted during the child’s hospitalizations or clinic visits. In the present study, our flexible community-based approach was modeled after BI work with caregivers of children recently diagnosed with autism25. BI sessions were completed in a multitude of settings including home visits, meeting at the caregiver’s work setting during lunch, Skype, and so on. Not only could caregivers choose the location of each session, but they could also select any time that worked for them, which included evening and weekend sessions. Our ultimate goal was to make BI as accessible as possible. Participants were also encouraged to use the BI Worksheets between sessions (i.e., homework), but this was not required.

Measures

Measures were administered at three time points: (a) at baseline prior to receiving BI (T1); (b) immediately following completion of 6-8 BI sessions (T2); and three months following completion of BI sessions (T3). Demographic information was only obtained at T1. Of note, attempts were made via phone and/or email to obtain T2 and T3 data from caregivers who did not complete the BI program.
Demographics
The demographic questionnaire included basic information about the caregiver, the child with SCD, and the family system. Questions focused on caregiver education, occupation, marital status, etc. Caregiver socioeconomic status was determined using occupation and education28,29.
Social Problem-Solving Inventory-Revised (SPSI-R)
The SPSI-R is a 52-item self-report measure designed to assess an individual’s ability to solve problems30. SPSI-R statements are endorsed by respondents on a 5-point likert scale ranging from 1 (i.e., not true at all) to 5 (i.e., extremely true). The SPSI-R assesses positive problem orientation (PPO) and rational problem solving (RPS), which are considered constructive/adaptive problem-solving dimensions. Negative problem orientation (NPO), impulsivity/carelessness style (ICS), and avoidance style (AS) are also measured and categorized as dysfunctional problem-solving dimensions. A total score (0-20) is calculated using the five sub-scores with consideration of whether the sub-scores are constructive/adaptive or dysfunctional dimensions. Higher total scores indicate that the individuals utilize constructive problem-solving orientation and rational problem-solving styles more frequently. The SPSI-R was used as a primary measure of intervention efficacy. In previous research, the internal consistency of the SPSI-R was found to be adequate when used with parents of children with SCD (Cronbach’s α=0.86).17 In the current study, the Cronbach’s α for the SPSI-R total score was .87. Cronbach’sα for the SPSI-R subscales were as follows; PPO .80, NPO .87, RPS .95, ICS .79, and AS .78.
Patient Health Questionnaire-9 (PHQ-9)
The PHQ-9 is a 9-item self-report measure of depressive symptoms31. Each of the 9 items are rated on a 4-point likert scale ranging from 0 (i.e., not at all) to 3 (i.e., nearly every day) with respondents required to rate the frequency of the depressive symptoms experienced within the last two weeks. Total scores range from 0 (no symptoms of depression) to 27 (i.e., severe depression). A score of 15 and above is suggestive of clinical depression. The PHQ-9 was used as a secondary measure of intervention efficacy. When used with an older African American population the PHQ-9 Cronbach’s α was found to be 0.7532. In the current study, the PHQ-9 Cronbach’sα was .86.
Profile of Mood States (POMS)
The POMS is a 65-item self-report measure consisting of seven self-report rating scales about feelings experienced over the previous week33,34. In the current study, we used a 15-item short-form of the POMS, as in prior BI studies35. The POMS items were rated from 0 (not at all) to 4 (extremely). The POMS produces seven subscales: (1) tension-anxiety, (2) depression-dejection, (3) anger-hostility, (4) fatigue-inertia, (5) confusion-bewilderment, (6) vigor-activity, and (7) friendliness. The subscale scores were combined to create a total mood disturbance (TMD) score. The POMS was used as a secondary measure of intervention efficacy. In the current study, the POMS TMD Cronbach’s α in the current sample was .85.
Impact of Events Scale-Revised (IES-R)
The IES-R is a 22-item self-report questionnaire that includes three subscales (i.e., hyperarousal, intrusion, and avoidance) that are associated with post-traumatic stress disorder (PTSD)36. Items are rated on a 4-point scale, based on frequency of occurrence, ranging from “not at all” to “often.” Higher scores indicate more symptoms of post-traumatic stress disorder. The IES-R was used as a secondary measure of intervention efficacy. The IES-R IES-R test-retest reliability estimates obtained from African American breast cancer survivors ranged from 0.89 to 0.94 over a 6-month study period37. In the current study, the Cronbach’sα for this sample was .93.
Data Analysis
Descriptive statistics are provided for the demographic data. We anticipated that caregivers would report higher levels of problem-solving skills and lower levels of distress at T2 and T3 compared to T1. To analyze the changes in problem-solving skills and distress over time, the SPSI-R, PHQ-9, POMS, and IES-R raw scores were analyzed using Maximum Likelihood Estimation for unbalanced (i.e., incomplete) repeated measures. As in prior BI studies18,19,20, this approach was chosen to avoid making the assumption that data are missing-completely-at-random, which occurs with case deletion when using traditional multivariate analysis of variance. Thus, all caregivers, regardless of whether they completed BI, were included in the “intent-to-treat” analyses. In addition, because we had clear directional expectations, one-tailed t-tests are reported. All data analyses were completed using IBM SPSS Statistics Premium Grad Pack 25 for Mac.