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Family Functioning in Children with Chronic Illness Compared with Healthy Controls: A Critical Review

2007; Elsevier BV; Volume: 150; Issue: 3 Linguagem: Inglês

10.1016/j.jpeds.2006.11.063

1097-6833

Catherine B. McClellan, Lindsey L. Cohen,

Adolescent and Pediatric Healthcare

Advances in medical care and technology have increased the lifespan and decreased the disease-related suffering of children with chronic illnesses. In addition to physical health outcomes, health care professionals are increasingly evaluating other parameters, such as child and family adjustment. Among these psychosocial factors, family functioning is a key variable that has been shown to play an essential role in children's adjustment to chronic illness.1Wallander J.L. Thompson R.J. Psychosocial adjustment of children with chronic physical conditions.in: Roberts M.C. Handbook of pediatric psychology. 2nd ed. Guilford, New York1995: 124-141Google Scholar Family functioning is a broad concept and is often used as an umbrella term encompassing numerous constructs, including parents' satisfaction with their parenting role, positive parent–child interactions, family communication, family adaptability, and family cohesion.2Rolland J. Mastering family challenges in serious illness and disability.in: Walsh F. Normal family processes. 2nd ed. Guilford, New York1993: 444-473Google Scholar Research has shown that families of a child with a chronic illness may have deficits in family cohesion, family adaptability, parent-child interactions, family conflict, and family problem-solving skills.3Brandt P.A. Childhood diabetes: Behavioral research.Annu Rev Nurs Res. 1998; 16: 63-82PubMed Google Scholar, 4Sawyer E.H. Family functioning when children have cystic fibrosis.J Pediatr Nurs. 1992; 7: 304-311PubMed Google Scholar Understanding the nature and development of these problems in family functioning is critical to formulating effective interventions. Although we know that family functioning is related to the well-being of children with chronic illness, we lack a clear understanding of whether families of chronically ill children are significantly more likely to have difficulties in functioning compared with families of healthy children. In this article we review the research into the functioning of families with children with a range of chronic illnesses compared with healthy controls, critique these studies, highlight parallels across the literature, and provide directions for future study. The inclusion of demographically matched healthy control groups allows researchers to eliminate a number of methodological limitations and draw strong conclusions regarding the impact of childhood illness on family functioning. This is particularly important with measures of family functioning that were not recently normed, or were normed with a group that is significantly different from the sample being studied. For example, researchers examining family functioning in the predominately African-American sickle cell disease (SCD) population have stressed the importance of including a demographically matched healthy comparison sample rather than relying on the norm-group statistics for comparisons.5Barakat L.P. Lutz M. Smith-Whitley K. Ohene-Frempong K. Is treatment adherence associated with better quality of life in children with sickle cell disease?.Qual Life Res. 2005; 14: 407-414Crossref PubMed Scopus (30) Google Scholar, 6Noll R.B. McKellop J.M. Vannatta K. Kalinyak K. Child-rearing practices of primary caregivers of children with sickle cell disease: the perspective of professionals and caregivers.J Pediatr Psychol. 1998; 23: 131-140Crossref PubMed Scopus (17) Google Scholar Including this group allows researchers to better distinguish the disease-specific effects on family functioning independent from the sociocultural influences, such as ethnic minority socioeconomic status.5Barakat L.P. Lutz M. Smith-Whitley K. Ohene-Frempong K. Is treatment adherence associated with better quality of life in children with sickle cell disease?.Qual Life Res. 2005; 14: 407-414Crossref PubMed Scopus (30) Google Scholar, 6Noll R.B. McKellop J.M. Vannatta K. Kalinyak K. Child-rearing practices of primary caregivers of children with sickle cell disease: the perspective of professionals and caregivers.J Pediatr Psychol. 1998; 23: 131-140Crossref PubMed Scopus (17) Google Scholar A review of studies of chronic illness adjustment and family functioning indicates that previously established differences are not significant when healthy controls are included.7Ievers C.E. Drotar D. Family and parental functioning in cystic fibrosis.J Dev Behav Pediatr. 1996; 17: 48-55Crossref PubMed Scopus (56) Google Scholar We selected cystic fibrosis (CF), juvenile rheumatoid arthritis (JRA), type 1 diabetes/juvenile-onset diabetes, asthma, hemophilia, and SCD for this review because these are some of the most common chronic childhood illnesses and represent a broad spectrum of disease. Although there are differences across these 6 groups, there are similarities in chronicity, creation of disability, and need to adhere to demanding regimens. Articles included in the review were identified through searches in MEDLINE or PsycINFO databases. The following search terms were used: "cystic fibrosis," "juvenile rheumatoid arthritis," "diabetes," "asthma," "hemophilia," and "sickle cell disease." These terms were combined with the search terms "family functioning," "family coping," and "family adjustment." Additional inclusion criteria were that the study focused on pediatric rather than adult chronic illness, and a healthy control group was included. A total of 15 articles were identified (Table; available at www.jpeds.com). Given the small number of studies and range of measures, evaluating this body of work by meta-analysis was not feasible.TableSummary of reviewed studiesAuthorIllnessSampleMeasure of family functioningPrimary findingsGoldberg et al (1990)CF•15 families with CF•45 families with congenital heart disease•50 healthy control familiesAge range: 1 to 9 monthsParenting Stress Index (parent report)•CF and CHD families reported significantly greater stress resulting from overall parenting stress than healthy control families.•Parents of CF children reported the highest levels of stress resulting from the child's demands.Cowen et al (1985)CF•41 families with with CF•31 parents of healthy childrenAge range: 2 to 5 yearsFamily Assessment Measure (FAM) (parent report)•Fathers of CF children rated their family functioning as significantly better than did the fathers of healthy controls.•FAM social desirability subscale was elevated for both mother and father reports.•Denial subscale was elevated for the mothers of CF children.Sawyer (1992)CF•32 families with CF•32 healthy control familiesAge range: 4 months to 3 yearsFeetham Family Functioning Survey (mother report)Mothers in the CF group reported significantly less time available to spend with their spouses.Spieth et al (2001)CF•29 families with CF•29 healthy control familiesAge range: 2 to 5 yearsMcMaster Mealtime Family Interaction Coding System (MICS) (observation of family mealtime)Functioning of CF families was significantly lower than that of healthy control families.Blair et al (1995)CF•27 families with CF•27 families of anorexic adolescents•31 healthy control familiesFAM (parent and adolescent self-report) (observational problem-solving task to assess family functioning)•Significantly more CF families were categorized as good problem solvers compared with the healthy control families and families of anorexic adolescents.•No differences on the FAM were seen in the 3 groups.Lewis and Khaw (1982)CF, asthma•31 families with CF•26 families with asthma•27 healthy control familiesAge range: 7 to 12 yearsFamily Adaptability and Cohesiveness Evaluation Scales (FACES) (mother report)No significant differences in family functioning were seen in the 3 groups.Hamlett et al (1992)Diabetes, asthma•13 families with diabetes•17 families with asthma•30 healthy control familiesAge range: 6 to 14 yearsFamily Environment Scale (FES) (mother report)No significant group differences were seen on any of the family functioning scales.Standen et al (1985)Diabetes•20 families with diabetes•20 healthy control familiesAge range: 4 to 12 yearsFES (parent report)Parents of children with type 1 diabetes described their families as being less achievement oriented (ie, concerned about achievements at school, work) than did the parents of healthy children.Frank et al (1998)Diabetes, JRA•40 families with diabetes•27 families with JRA•62 healthy control familiesAge range: 0 to 16 yearsFACES III and FACES III-K for children under age 12 (parent and child report); longitudinal study (4 assessments in an 18-month period)No significant differences were seen between the illness groups.Harris et al (1991)JRA•12 families with JRA•12 matched healthy control familiesAge range: 6 to 11 yearsFES (parent and child report)No significant differences were seen between the 2 groups on the FES.Huygen et al (2000)JRA•47 families with JRA•52 healthy control familiesAge range: two age categories: 7 to 11 years (child group) and 12 to 16 years (adolescent group)FACES (parent and child report)•Families with JRA children reported higher ratings of cohesion and lower adaptation compared with families with healthy children.•Adolescents with JRA reported lower levels of cohesion than did their parents.•In the adolescent group, there were no significant differences between parents' report of cohesion in JRA and healthy control families.Gerhart et al (2003)JRA•64 families with JRA•64 healthy control familiesAge range: 8 to 14 yearsFES (parent report)No significant differences were seen in parent-reported levels of family functioning.Noll et al (1994)SCD•32 families with SCD•40 healthy control familiesAge range: 8 to 15 yearsFES (caregiver report); Family Conflict Construct (caregiver report)No significant differences were seen in parent-reported levels of family functioning.Midence et al (1996)SCD•39 families with SCD•24 healthy control familiesAge range: 6 to 16 yearsFES (parent report)Families of children with SCD reported more cohesion and less conflict.Evans et al (2000)Hemophilia•24 families with hemophilia•12 healthy control familiesAge range: 4 to 15 years (all male)FAM (parent report)No significant differences were seen in parent-reported levels of family functioning. Open table in a new tab The literature on families with a child with CF reports varying findings regarding the impact of chronic illness on family functioning. Research shows that parents of children with CF report higher parenting stress compared with parents of healthy children. In addition, mothers of children with CF report decreased time available to spend with their spouses. Spieth et al8Spieth L.E. Stark L.J. Mitchell M.J. Schiller M. Cohen L.L. Mulvihill M. et al.Observational assessment of family functioning at mealtime in preschool children with cystic fibrosis.J Pediatr Psychol. 2001; 26: 215-224Crossref PubMed Scopus (47) Google Scholar found similar results and concluded that families of a child with CF score significantly lower than healthy control families in domains of communication, interpersonal involvement, affect management, behavior control, and role allocation. Some research indicates that having a child with CF might not impair family functioning, however. For example, 2 studies have shown no differences in family functioning between families with children with CF and families of physically healthy children.9Blair C. Freeman C. Cull A. The families of anorexia nervosa and cystic fibrosis patients.Psychol Med. 1995; 25: 985-993Crossref PubMed Scopus (59) Google Scholar, 10Lewis B.L. Khaw K. Family functioning as a mediating variable affecting psychosocial adjustment of children with cystic fibrosis.J Pediatr. 1982; 101: 636-640Abstract Full Text PDF PubMed Scopus (70) Google Scholar Further, on an in vivo problem-solving task, families of adolescents with CF were more likely to be categorized as good problem solvers than were families of healthy adolescents.9Blair C. Freeman C. Cull A. The families of anorexia nervosa and cystic fibrosis patients.Psychol Med. 1995; 25: 985-993Crossref PubMed Scopus (59) Google Scholar Cowen et al11Cowen L. Corey M. Keenan N. Simmons R. Arndt E. Levison H. Family adaptation and psychosocial adjustment to cystic fibrosis in the preschool child.Soc Sci Med. 1985; 20: 553-560Crossref PubMed Scopus (35) Google Scholar found that fathers of young children with CF are more likely to report positive family functioning than are fathers of healthy children. For the most part, the research has found few differences in family functioning comparing families of children with type 1 diabetes mellitus with families with healthy children.12Hamlett K.W. Pellegrini D.S. Katz K.S. Childhood chronic illness as a family stressor.J Pediatr Psychol. 1992; 17: 33-47Crossref PubMed Scopus (155) Google Scholar, 13Standen P.J. Hinde F.R.J. Lee P.J. Family involvement and metabolic control of childhood diabetes.Diabetes Med. 1985; 2: 137-140Crossref PubMed Scopus (13) Google Scholar, 14Frank R.G. Thayer J.F. Hagglund K.J. Vieth A.Z. Schopp L.H. Beck N.C. et al.Trajectories of adaptation in pediatric chronic illness: the importance of the individual.J Consult Clin Psychol. 1998; 66: 521-532Crossref PubMed Scopus (34) Google Scholar However, mothers of children with diabetes report having less time to engage in activities with their children compared with mothers of healthy children.13Standen P.J. Hinde F.R.J. Lee P.J. Family involvement and metabolic control of childhood diabetes.Diabetes Med. 1985; 2: 137-140Crossref PubMed Scopus (13) Google Scholar Parents of children with type 1 diabetes also report different family values than families of healthy children; specifically, they are more likely to describe their families as less achievement-oriented compared with families of physically healthy children.13Standen P.J. Hinde F.R.J. Lee P.J. Family involvement and metabolic control of childhood diabetes.Diabetes Med. 1985; 2: 137-140Crossref PubMed Scopus (13) Google Scholar Research comparing families with a child with JRA and healthy families indicates a general lack of differences.15Harris J.A. Newcomb A.F. Gewanter H.L. Psychosocial effects of juvenile rheumatic disease The family and peer system as a context for coping.Arthritis Care Res. 1991; 4: 123-130Crossref PubMed Scopus (2) Google Scholar, 16Huygen A.C. Kuis W. Sinnema G. Psychological, behavioral, and social adjustment in children and adolescents with juvenile chronic arthritis.Ann Rheum Dis. 2000; 59: 276-282Crossref PubMed Scopus (75) Google Scholar, 17Gerhart C.A. Vannatta K. McKellop J.M. Zeller M. Taylor J. Passo M. et al.Comparing parental distress, family functioning, and the role of social support caregivers with and without a child with juvenile rheumatoid arthritis.J Pediatr Psychol. 2003; 28: 5-15Crossref PubMed Scopus (65) Google Scholar It is notable that Huygen et al16Huygen A.C. Kuis W. Sinnema G. Psychological, behavioral, and social adjustment in children and adolescents with juvenile chronic arthritis.Ann Rheum Dis. 2000; 59: 276-282Crossref PubMed Scopus (75) Google Scholar separately examined families of children (age 6 to 11 years) and families of adolescents (age 12 to 16 years) and found results suggesting that families with children with JRA, but not those with adolescents with JRA, had greater family cohesion and less family adaptability than families without children with JRA.16Huygen A.C. Kuis W. Sinnema G. Psychological, behavioral, and social adjustment in children and adolescents with juvenile chronic arthritis.Ann Rheum Dis. 2000; 59: 276-282Crossref PubMed Scopus (75) Google Scholar There are few studies comparing family functioning in SCD, hemophilia, or asthma with healthy controls. Two studies comparing functioning in families of children with SCD and healthy families have provided divergent conclusions, possibly due to the geographical differences between the samples and the incorporation of extensive cultural sensitivity training and monitoring of the data collectors in one study but not the other. The better functioning found by Midence et al18Midence K. McManus C. Fuggle P. Davies S. Psychological adjustment and family functioning in a group of British children with sickle cell disease: preliminary empirical findings and a meta-analysis.Br J Clin Psychol. 1996; 35: 439-450Crossref PubMed Scopus (32) Google Scholar could reflect a pattern whereby parents demonstrate increases in family protectiveness and a resultant decrease in reporting of family conflict, whereas the emphasis that Noll et al19Noll R.B. Swiecki E. Garstein M. Vannatta K. Kalinyak K. Davies W.H. et al.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: 281-294Crossref Scopus (24) Google Scholar placed on cultural sensitivity training might have resulted in families of children with SCD feeling less threatened and more open. Because hemophilia occurs predominately in males, research in this area highlights the importance of considering the role of the child's sex in family functioning issues. A study found that the parents of boys with hemophilia reported a greater total number of family functioning difficulties, but the small sample size kept these differences from being statistically significant.20Evans M. Cottrell D. Shiach C. Emotional and behavioural problems and family functioning in children with haemophilia: a cross-sectional survey.Haemophilia. 2000; 6: 682-687Crossref PubMed Scopus (20) Google Scholar Research investigating family functioning in the pediatric asthma population found heightened mother-reported problems with social support, child behavior, and stressful events, but no significant differences on measures of family functioning.10Lewis B.L. Khaw K. Family functioning as a mediating variable affecting psychosocial adjustment of children with cystic fibrosis.J Pediatr. 1982; 101: 636-640Abstract Full Text PDF PubMed Scopus (70) Google Scholar, 12Hamlett K.W. Pellegrini D.S. Katz K.S. Childhood chronic illness as a family stressor.J Pediatr Psychol. 1992; 17: 33-47Crossref PubMed Scopus (155) Google Scholar (For more details, see the Table, available at www.jpeds.com.) In summarizing the results that can be drawn from this body of literature, it is essential to consider the findings in light of methodological limitations. Most of the investigations reviewed herein used a single measure of family functioning, and 2 of the studies obtained only mothers' reports.8Spieth L.E. Stark L.J. Mitchell M.J. Schiller M. Cohen L.L. Mulvihill M. et al.Observational assessment of family functioning at mealtime in preschool children with cystic fibrosis.J Pediatr Psychol. 2001; 26: 215-224Crossref PubMed Scopus (47) Google Scholar, 10Lewis B.L. Khaw K. Family functioning as a mediating variable affecting psychosocial adjustment of children with cystic fibrosis.J Pediatr. 1982; 101: 636-640Abstract Full Text PDF PubMed Scopus (70) Google Scholar, 11Cowen L. Corey M. Keenan N. Simmons R. Arndt E. Levison H. Family adaptation and psychosocial adjustment to cystic fibrosis in the preschool child.Soc Sci Med. 1985; 20: 553-560Crossref PubMed Scopus (35) Google Scholar, 12Hamlett K.W. Pellegrini D.S. Katz K.S. Childhood chronic illness as a family stressor.J Pediatr Psychol. 1992; 17: 33-47Crossref PubMed Scopus (155) Google Scholar, 13Standen P.J. Hinde F.R.J. Lee P.J. Family involvement and metabolic control of childhood diabetes.Diabetes Med. 1985; 2: 137-140Crossref PubMed Scopus (13) Google Scholar, 18Midence K. McManus C. Fuggle P. Davies S. Psychological adjustment and family functioning in a group of British children with sickle cell disease: preliminary empirical findings and a meta-analysis.Br J Clin Psychol. 1996; 35: 439-450Crossref PubMed Scopus (32) Google Scholar, 20Evans M. Cottrell D. Shiach C. Emotional and behavioural problems and family functioning in children with haemophilia: a cross-sectional survey.Haemophilia. 2000; 6: 682-687Crossref PubMed Scopus (20) Google Scholar, 21Sawyer M.G. Spurrier N. Kennedy D. Martin J. The relationship between the quality of life of children with asthma and family functioning.J Asthma. 2001; 38: 279-284Crossref PubMed Scopus (35) Google Scholar, 22Goldberg S. Morris P. Simmons R.J. Fowler R.S. Levison H. Chronic illness in infancy and parenting stress: a comparison of three groups of parents.J Pediatr Psychol. 1990; 15: 347-358Crossref PubMed Scopus (163) Google Scholar Family functioning is a multifaceted concept, and multiple measures are needed to capture its important dimensions. Measuring other domains, such as children's medication adherence and parental depression, might be important because they can influence family functioning and child behavior.23Lavigne J.V. Faier-Routman J. Psychological adjustment to pediatric physical disorders: a meta-analytic review.J Pediatr Psychol. 1992; 17: 133-157Crossref PubMed Scopus (454) Google Scholar, 24Quittner A.L. Opipari L.C. Regoli M.J. Jacobsen J. Eigen H. The impact of caregiving and role strain on family life: comparisons between mothers of children with cystic fibrosis and matched controls.Rehabil Psychol. 1992; 37: 289-304Crossref Scopus (84) Google Scholar Other research has shown that parents' reports of activities may not accurately reflect true behaviors. For example, Quittner et al24Quittner A.L. Opipari L.C. Regoli M.J. Jacobsen J. Eigen H. The impact of caregiving and role strain on family life: comparisons between mothers of children with cystic fibrosis and matched controls.Rehabil Psychol. 1992; 37: 289-304Crossref Scopus (84) Google Scholar found that mothers of children with CF do not perceive their parenting role differently than mothers of healthy children; however, behavioral assessment revealed significant differences in the activities of the 2 groups. These findings support the need for multiple informants and multiple methods of assessment when investigating family functioning. Another limitation of this research is the extreme variation in the age of the participants. Illness-related issues that impact family functioning might differ across age groups and developmental periods. For example, whereas families of children with CF and those with healthy children may face many of the same stressors, the declines in health associated with increased age in individuals with CF may be an important variable when investigating family functioning. Similarly, parents of adolescents with type 1 diabetes and JRA may experience greater conflict associated with treatment adherence issues than they had faced when their children were younger. By collapsing across large age ranges, researchers may have diluted possible effects. Most of the studies selected healthy controls similar in age, sex, and ethnicity to the chronically ill children. Unfortunately, few studies considered additional variables when matching. For example, only 2 studies considered the number of other children in the home and whether any of these other children also had significant medical diagnoses.8Spieth L.E. Stark L.J. Mitchell M.J. Schiller M. Cohen L.L. Mulvihill M. et al.Observational assessment of family functioning at mealtime in preschool children with cystic fibrosis.J Pediatr Psychol. 2001; 26: 215-224Crossref PubMed Scopus (47) Google Scholar, 13Standen P.J. Hinde F.R.J. Lee P.J. Family involvement and metabolic control of childhood diabetes.Diabetes Med. 1985; 2: 137-140Crossref PubMed Scopus (13) Google Scholar By matching not only basic demographics, but also other important variables, researchers are better able to attribute any differences between the families to disease status. Most of the studies neglected to include how illness-related factors, such as disease severity and time since diagnosis, relate to family functioning. Families might have increased difficulties when a child is first diagnosed with a chronic illness.25Jacobsen A.M. Hauser S.T. Lavori P. Willett J.B. Cole C.F. Wolfsdorf J.I. et al.Family environment and glycemic control: a four-year prospective study of children and adolescents with insulin-dependent diabetes mellitus.Psychosom Med. 1994; 56: 401-409Crossref PubMed Scopus (165) Google Scholar The relationship between childhood chronic illness and family functioning may have been less apparent because time since diagnosis, disease severity, and extent of disease activity were not considered. Future examinations of family functioning and childhood chronic illness should include multimethod assessments of family functioning. Ideally, research examining family functioning should include reports of family functioning from all members of the family, in addition to observational measures to directly assess family interactions. Observational measures of family functioning can identify objective indices of family functioning, which cannot reliably be obtained through ratings. Although observational data can be time-consuming and expensive to collect, it can provide researchers and clinicians with objective and quantifiable data to use in evaluating changes over time and responses to intervention. Multisite research, rather than sampling from a single clinic group, will allow researchers to increase the size of their samples and the generalizability of their findings. Along with the site from which participants are drawn, other participant characteristics also deserve consideration in future research, including the impact of previous parenting experience on family functioning, the child's sex and ethnicity, and whether additional children with chronic physical or mental illnesses reside in the home. Although difficult to conduct, longitudinal investigations of family functioning are needed to explore the processes by which chronic childhood illnesses influence family functioning. Evaluating families as they progress from initial diagnosis to key developmental phases (eg, school entry and adolescence) may help identify the periods of greatest challenges and aid in developing protocols to mediate these threats to optimal family functioning. In addition, longitudinal research aids identification of the roles of other variables, such as disease severity, in predicting family functioning. Conducting methodologically sound, high-quality research in this area might make it possible to determine whether families with chronically ill children are indeed at increased risk for problematic functioning. Such research also would bolster the current understanding of the process by which families confront stressful events and how these events relate to their functioning. In addition, assessment work in this vein should highlight directions for intervention and provide recommendations to other families about how to navigate stressful life events.