DISCUSSION

The purpose of the present study was to modify the delivery of BI to caregivers of children with SCD in ways that would increase accessibility and reduce stigma associated with receiving behavioral health services. We anticipated that delivery modifications could improve rates of recruitment and retention from previous attempts to recruit caregivers of children with SCD for participation in psychosocial research. With recruitment and delivery modifications (described above), 94% of caregivers of children with SCD consented to participate and 48.6 % completed 6+ sessions of face-to-face BI. These rates of recruitment and retention were higher than those reported in other studies of children with SCD suggesting that our strategies to reduce barriers to participation were moderately successful for this group of caregivers17.
We anticipated that caregivers of children with SCD who successfully completed the BI program (6+ sessions), would report improvements in problem-solving skills and lower levels of distress between T1 and T2 and also between T1 and T3. Based on our clinical experience and the behavioral health literature regarding families of children with SCD (i.e., minority, single parents, low SES), we initially believed these caregivers would report below average levels of problem-solving skills and considerable levels of distress at T1 due to the complexity of caring for a child with SCD and challenges associated with being a minority parent. Our data showed that caregivers of children with SCD reported average levels of problem-solving skills and non-clinical levels of distress at T1, T2, and T3.
While the majority of our sample of caregivers were low SES, single, minority parents who were caring for a child with a severe, chronic illness, our data suggested that these caregivers of children with SCD experience characteristics of psychological hardiness that we did not anticipate. We suspect our belief that caregivers of children with SCD would report below average levels of problem-solving skills and higher levels of distress, in comparison to other populations of caregivers, may have been attributable to existing research demonstrating significant levels of parenting stress and other psychosocial challenges, as well as our own intrapersonal professional biases, as we did not initially consider the possibility that caregivers of children with SCD may have solid problem-solving skills and little evidence of distress at baseline12,16,17,39,. Thus, T1 data from the present study suggest that as a group, caregivers of children with SCD who participated in this study have broadly average problem-solving skills and they are experiencing non-clinical levels of distress.
Future research with caregivers of children with SCD might focus on families who score within the targeted or clinical level for psychosocial risk, which could be accomplished through comprehensive screening efforts utilizing the Psychosocial Assessment Tool (PAT)40. Prior investigations of the PAT with families of children with SCD, demonstrated increased risk for parental stress when the child with SCD did not have government health benefits, lower caregiver education, more children residing within the home, and greater financial difficulties40. Thus, identifying and targeting caregivers for participation in BI, who report higher levels of overall psychosocial risk on the PAT, may produce more robust results with regards to the effectiveness of the intervention with this population of caregivers.
Although we demonstrated successful recruitment of caregivers of children with SCD, levels of retention in this study were modest. In light of the rapidly evolving use of telehealth and iPhone/Android application-based psychosocial intervention programs, researchers may consider utilizing electronic mediums for disseminating BI-content. However, recent work comparing face-to-face BI to BI provided entirely via a website showed that face-to-face delivery was more effective41. With consideration of these findings41, future researchers may consider using the website-BI program as an adjunct to face-to-face sessions in order to further improve accessibility of the BI intervention for caregivers.
Our study had several limitations. First, our sample size of caregivers of children with SCD was small and underpowered to detect small or medium differences. Second, the study design did not include a control group of caregivers of children with SCD who did not receive BI. Third, although we recruited caregivers from two sites, it is possible that our sample of caregivers is not representative of the larger population of caregivers of children with SCD. Thus, future studies designed to implement a psychosocial intervention with caregivers of children with SCD, may benefit from collaborating with multiple sites (i.e., 3+), similar to prior large-scale randomized controlled trials of BI in pediatric oncology, to increase the sample size and thus statistical power18,19,20. We also strongly recommend screening caregivers, who report low levels of problem-solving skills and/or high levels of distress, prior to delivery to ascertain those in highest need.
In summary, data obtained from the present study provides limited evidence to support further efforts in designing and conducting a randomized control trial (RCT) of BI for this population of caregivers. However, focusing future efforts on identifying caregivers with the highest risk (i.e., low levels of problem-solving skills and high levels of distress) based on screening and subsequently delivering BI may be of clinical benefit.
Conflict of Interest Statement
Melissa A. Young, PsyD: No conflicts of interest to declare.
Megan Voll, LPC: No conflicts of interest to declare.
Robert B. Noll, PhD: No conflicts of interest to declare.
Diane L. Fairclough DrPH: No conflicts of interest to declare.
Cate Flanagan-Priore, PhD: No conflicts of interest to declare.
This work was supported by a grant to RBN from the National Center for Advanced Translational Sciences Grant Number UL1 TR001857-02.
References
  1. Edwards CL, Scales MT, Loughlin C, Bennett GG, Harris-Peterson S, DeCastro LM, Whitworth E, Abrams M, Feliu M, Johnson S, Wood M, Harrison O, Killough A. A brief review of the pathophysiology, associated pain, and psychosocial issues in sickle cell disease. Int J Behav Med 2005; 12: 171–179.
  2. Hankins J, Wang W. The painful face of poverty. Pediatr Blood Cancer 2009; 52:157-158.
  3. Ware RE, De Montalembert M, Tshilolo L, Abboud MR. Sickle cell disease. Lancet 2017; 390: 311–323.
  4. Centers for Disease Control and Prevention Website. https://www.cdc.gov/ncbddd/sicklecell/data.html Accessed August 9, 2017.
  5. Chaturvedi S, Debaun MR. Evolution of sickle cell disease from a life-threatening disease of children to a chronic disease of adults: The last 40 years. Am J Hematol 2015; 91: 5–14.
  6. Esezobor CI, Akintan P, Nwaogazie U, Akinwunmi E, Temiye E, Akinsulie A, Gbadegesin R. Enuresis in children and adolescents with sickle cell anaemia if more frequent and substantially different from the general population. PLoS One 2018; 8: 1-12.
  7. Iampietro M, Giovannetti T, Tarazi R. Hypoxia and inflammation in children with sickle cell disease: Implications for hippocampal functioning and episodic memory. Neuropsychol Rev 2014; 24: 252–265.
  8. Lawrence C, Webb J. Sickle cell disease and stroke: Diagnosis and management. Curr Neurol Neurosci Rep 2016; 16: 1–10.
  9. Platt OS, Rosse WF, Milner PF, Castro O, Steinberg MH, Mortality in sickle cell disease. Life expectancy and risk factors for early death. N Engl J Med 1994; 330: 1639-1644.
  10. Amid A, Odame I. Improving outcomes in children with sickle cell disease: Treatment considerations and strategies. Paediatr Drugs 2014; 16: 255–266.
  11. Barakat LP, Patterson CA, Tarazi RA, Ely E. Disease-related parenting stress in two sickle cell disease caregiver samples: Preschool and adolescent. Fam Syst Health 2007; 25: 147–161.
  12. Hildenbrand AK, Barakat LP, Alderfer MA, Marsac ML. Coping and coping assistance among children with sickle cell disease and their parents. J Pediatr Hem Onc 2015; 37: 25-34.
  13. Hofmann M, Montalembert M, Beauquier-Maccotta B, Villartay P, Golse B. Posttraumatic stress disorder in children affected by sickle-cell disease and their parents. Am J Hematol 2007; 82: 171-172.
  14. Northington L. Chronic sorrow in caregivers of school age children with sickle cell disease: A grounded theory approach. Issues Compr Pediatr Nurs 2000; 23: 141-154.
  15. Noll R, Swiecki E, Garstein M, Vannatta K, Kalinyak KA, Davies W, Bukowski W. Parental distress, family conflict, and role of social support for caregivers with or without a child with sickle cell disease. Fam Syst Med 1994; 12: 281-294.
  16. Thompson RJ, Gil KM, Gustafson KE, George LK, Keith BR, Spock A, Kinney TR. Stability and change in the psychological adjustment of mothers of children and adolescents with cystic fibrosis and sickle cell disease. J Pediatr Psychol 1994; 19: 171–188.
  17. Barakat LP, Daniel LC, Smith K, Robinson MR, Patterson CA. Parental problem-solving abilities and the association of sickle cell disease complications with health-related quality of life for school-age children. J Clin Psychol Med Settings 2014; 21: 56-65.
  18. Sahler OJZ, Dolgin MJ, Phipps S, Fairclough DL, Askins MA, Katz ER, Noll RB, Butler RW. Specificity of problem-solving skills training in mothers of children newly diagnosed with cancer: Results of a multisite randomized clinical trial. J Clin Oncol 2013; 31: 1329–1335.
  19. Sahler OJZ, Fairclough DL, Phipps S, Mulhern RK, Dolgin MJ, Noll RB, Katz ER, Varni JW, Copeland DR, Butler RW. Using problem solving skills training to reduce negative affectivity in mothers of children with newly diagnosed cancer: Report of a multisite randomized trial. J Clin Coun Psychol 2005; 73: 272–283.
  20. Sahler OJZ, Varni JW, Fairclough DL, Butler RW, Noll RB, Dolgin MJ, Phipps D, Copeland DR, Katz ER, Mulhern RK. Problem-solving skills training for mothers of children with newly diagnosed cancer: A randomized trial. J Dev Behav Pediatr 2002; 23: 77–86.
  21. Gamble VN. Under the shadow of Tuskegee: African Americans and healthcare. Am J Hematol 1977; 87: 1773-1778.
  22. Stevens EM, Patterson CA, Li YB, Smith-Whitley K, Barakat LM. Mistrust of pediatric sickle cell disease clinical trials research. Am J Prev Med 2016; 51: S78-S86.
  23. Nezu AM. Efficacy of a social problem-solving therapy approach for unipolar depression. J Clin Coun Psychol 1986; 54: 196-202.
  24. Nezu AM, Nezu CM, D’Zurilla TJ. Problem-solving therapy: A treatment manual. New York, NY: Springer Publishing Company, LLC: 2013.
  25. Nguyen CT, Fairclough DL, Noll RB. Problem-solving skills training for mothers of children recently diagnosed with autism spectrum disorder: A pilot feasibility study. Autism 2016; 20: 55–64.
  26. Kelly AD, Egan AM, Reiter-Purtill J, Gerhardt CA, Vannatta K, Noll RB. A controlled longitudinal study of internalizing symptoms in older adolescents with sickle cell disease. Pediatr Blood Cancer 2015; 62: 637-642.
  27. Noll RB, Kiska R, Reiter-Purtill J, Gerhardt CA, Vannatta K. A controlled longitudinal study of the social functioning of adolescents with sickle cell disease. Pediatrics 2010; 125: e1453-e1459.
  28. Entwisle DR, Astone NM. Some practical guidelines for measuring youth’s race/ethnicity and socioeconomic status. Child Dev 1994; 65: 1521-1540.
  29. Shavers VL. Measurement of socioeconomic status in health disparities research. J Natl Med Assoc 2007; 99: 1013-1023.
  30. D’Zurilla TJ, Nezu AM, Maydeu-Olivares A. Social problem-solving inventory-Revised. North Tonawanda, NY: MHS; 2002.
  31. Bian C, Li C, Duan Q, Wu H. Reliability and validity of patient health questionnaire: Depressive syndrome module for outpatients. Sci Res Ess 2011; 6: 278–282.
  32. Gitlin LN, Chernett NL, Dennis MP, Hauck WW. Identification of and beliefs about depressive symptoms and preferred approaches among community-living older African Americans. Am J Geriatr Psychiatry 2012; 20: 973-984.
  33. Heuchert JP, McNair DM. Profile of mood states - Second edition. Canada, Multi-Health Systems; 2012.
  34. McNair DM, Lorr M, Doppleman LF. Manual for the profile of mood states. San Diego, CA: Educational and Industrial Testing Services; 1971.
  35. Steele RG, Long A, Reddy KA, Luhr M, Phipps S. Change in maternal distress and child-rearing strategies across treatment for childhood cancer. J Pediatr Psychol 2002; 28: 447-462.
  36. Weiss DS. The impact of event scale – revised. Assessing Psychological Trauma and PTSD: A Practitioner’s Handbook – 2nd Edition. New York, NY: Guilford Press; 2007.
  37. Von DM, Russell KM, Carpenter J, Monahan PO, Zhao Q, Tallman E, Ziner KW, Storniolo AM, Miller KD, Giesler RB, Haase J, Otte J, Champion VL. Health-related quality of life of African American breast cancer survivors compared to healthy African American women. Cancer Nurs 2012; 35: 337-346.
  38. Kroenke K, Spitzer R, Williams W. The patient health questionnaire PHQ-9: Validity of a brief depression severity measure. J Gen Int Med 2001; 16: 2–3.
  39. Thompson R, Gil K, Burbach D, Keith, B, Kinney T. Psychological adjustment of mothers of children and adolescents with sickle cell disease: The role of stress, coping methods, and family functioning. J Pediatr Psychol 1993; 18: 549-559.
  40. Karlson CW, Leist-Haynes S, Smith M, Faith MA, Elkin TD, Megason G. Examination of risk and resiliency in a pediatric sickle cell disease population using the psychosocial assessment tool 2.0. J Pediatr Psychol 2012; 37: 1031–1040.
  41. Phipps S, Fairclough DL, Noll RB, Devine KA, Dolgin MJ, Schepers SA, Askins MA, Schneider N, Ingman K, Voll M, Katz ER, McLaughlin J, Sahler OJZ. In-person vs. web-based administration of a problem-solving skills intervention for parents of children with cancer: Report of a randomized non-inferiority trial. E Clinical Medicine, Published by THE LANCET, in press.
Legends
TABLE 1 Demographic Characteristics of Participants (n = 60) and their Spouses (n = 38)
TABLE 2 Comparison of Problem-Solving Skills Standard Score Changes at Baseline (T1) to T2 and T1 to T3
TABLE 3 Comparison of Mood Disturbance T-score Changes at Baseline (T1) to T2 and T1 to T3
TABLE 4 Comparison of Depression and Post-Traumatic Stress Symptoms Mean and Standard Deviation Changes at Baseline (T1) to T2 and T1 to T3
Figure 1 Consort Diagram
Figure 2 Bright IDEAS Model