- Email: [email protected]
Family functioning as a mediating variable affecting psychosocial adjustment of children with cystic fibrosis
B E H A V I O R A L
P E D I A T R I C S
Richard W. Olmsted, Editor
Family functioning as a mediating variable affecting psychosocial adjustment of children with cystic fibrosis Family functioning was examined as a mediating variable affecting adjustment in children with cystic fibrosis. Three groups o f children (cystic fibrosis, asthma, and healthy control subjects) were initially compared on two dimensions o f adjustment: self-concept and frequency o f behavior problems. The families o f these children were compared according to functional level o f "'cohesion" and "'adaptability" (assessed by an as yet unvalidated family functioning measure). No significant difference was found between the groups in family functioning or children's self-concept. The two groups o f chronically ill children did have significantly higher frequencies o f behavior problems than had the healthy children. When there was statistical control for effects related to family functioning, however, this difference was not significant. Family functioning was found to be a better predictor o f child adjustment than was the presence o f illness. These results are consistent with recent research suggesting that children with cystic fibrosis adjust in basically healthy ways and that any deficits result more from mediating factors. In this study family functioning was found to be an important mediating factor in this process.
Brian L. Lewis, Ph.D.,* and Kon-Taik Khaw, M.D., Boston, Mass.
EARLY STUDIES of the psychosocial effects of cystic fibrosis were generally supportive of the commonly held belief that the presence of CF leads to profound deficits in individual adjustment and family functioning. 1-6In reviewing these studies, Gayton and Friedman 7 noted a number of methodologic problems: Most utilized few subjects and inadequate controls and were primarily descriptive in nature, relying on clinical experience and subjective evaluations. Recent evidence indicates that children with C F are not as maladjusted as has been previously reportedY ~ Although these children, as a group, do sometimes score more poorly on specific measures of psychologic adjustment, 8,9 most appear to be coping with the illness in a
From the Cystic Fibrosis Program, Division o f Cell Biology, Children's Hospital Medical Center, Department o f Pediatrics, Harvard Medical School. Supported in part by the Ina S. Pulmutter Cystic Fibrosis Research Fund. *Reprint address: Ethan Allen Medical Center, 1100 Ethan Allen Ave., Winooski, VT 05404.
636
The Journal of P E D I A T R I C S Vol. 10l, No. 4, pp. 636-640
"normal," well-adjusted manner) ~ Based on this finding, it has been suggested that a more fruitful course for future research would be to identify mediating variables that are important in the coping process and Which differentiate healthy from unhealthy adjustment. Abbreviation used FACES: Family Adaptability and Cohesiveness Evaluation Scales Bedell et a111 have identified "life stress" as one such mediating variable. These researchers found that the self-concepts of chronically ill children (including children with CF) were related to the level of stress they experienced. The implication of this result was that the poor adjustment often reported in chronically ill children might result more from life stress than from the disease. Olson et aW have proposed a conceptual system for examining family functioning called the "circumplex model," which is based on family systems theory. Within this model families can be classified according to how they function on two salient dimensions, "cohesion" and "adaptability." Cohesion is defined as "the emotional
0022-3476/82/100636+05500.50/0
9 1982 The C. V. Mosby Co.
Volume 101 Number 4 bonding members have with one another and the degree of individual autonomy a person experiences in the family" (p. 5). Adaptability is defined as, "the ability of a marital or family system to change its power structure, role relationships, and relationships rules in response to situational and developmental stress" (p. 12). According to the theory, healthy families function in the moderate or balanced ranges on both dimensions. We hypothesized that healthy adjustment in the child with C F is more dependent upon healthy family functioning (balanced levels of cohesion and adaptability) than on the mere presence of the illness. METHOD Subjects. Eighty-four children and their mothers participated as subjects in this study. The children were between the ages of 7 and 12 years and represented one of the following three groups: (1) children with cystic fibrosis (n = 31), randomly selected from the population of children being treated on an outpatient basis in the CF clinic at Children's Hospital Medical Center in Boston; (2) children with chronic asthma (n = 26), randomly selected from the population of asthmatic children being treated on an outpatient basis in the allergy clinic at Children's Hospital in Boston; and (3) healthy children, i.e., children with no previous history of chronic illness (n -- 27), randomly selected from a consenting school system in the suburban Boston area. The three groups of children were matched by age, gender, and race (only white children were selected as C F occurs predominantly in white subjects). An effort was made to control for socioeconomic status by selecting the healthy children from a school, within the consenting suburban school system, that seemed to best reflect the socioeconomic level of the clinic populations. In addition, the chronically ill children (those in groups 1 and 2) had to meet the following selection criteria: They had to have been diagnosed as having their illness for at least two years; they could not have siblings diagnosed with the same illness; and they could not have been hospitalized for exacerbations of the illness over the preceding year. These additional selection criteria were used to ensure that the coping and adjustment variables being assessed would reflect long-term adaptation to the illness rather than short-term responses to recent crisis situations or atypical stress. One further criterion used in selecting the asthmatic children was that their illness had to be severe enough to require medication daily. Prior to participating in the study all parents reviewed and signed consent forms approved by the Hospital Consent Committee. Tests administered. The procedure for administering the
Psychosocial adjustment in cystic fibrosis
637
questionnaires was identical for all groups. Mothers completed the Family Adaptability and Cohesiveness Evaluation Scales 13 and the Behavior Problem Checklist. TM Children completed the Piers-Harris Children's Self-Concept Scale) 5
Family Adaptability and Cohesiveness Evaluation Scales. This is a self-report instrument based on the circumplex model previously discussed, consisting of 111 statements concerning various aspects of family dynamics. The statements are of the type, "Family members are concerned with each other's welfare." The person completing the instrument indicates, on a scale from 1 to 4, the degree to which each statement is felt to be true of his/her family. Two separate scores are obtained from an analysis of the responses, one for cohesion and one for adaptability. Because of the newness of this instrument, validity studies are still underway and have not yet been reported. The circumplex model upon which FACES was developed does appear to have empiric validity in terms of differentiating families under stress and setting treatment goals for family therapy.16. J7
Behavior Problem Checklist. This checklist consists of 55 problem behaviors that often occur in childhood and adolescence; it can be completed by anyone familiar with a child's behavior. The person completing the instrument goes through the list, placing a check next to each item that is felt to be a problem for the targeted child. The total number of items checked provides a rating of the frequency of behavioral problems for that child. The scale is reported to be internally consistent and to have test-retest reliability of about 0.82.18 O'Leary and Johnson's 19 review suggested that the behavior checklist has high concurrent and construct validity, and that it reliably differentiates groups of children with known behavior disorders. Piers-Harris Children's Self-Concept Scale. This is an 80-item self-report questionnaire designed to assess the child's self-evaluation. The items are of the type, "I am smart" and "It is hard for me to make friends," to which the child indicates whether the statement is true or false for him/her by circling either "yes" or "no." The instrument is scored to provide an overall indication of the child's self-concept. Reports of internal consistency for the scale range from 0.78 to 0.93, with test-retest reliability estimates about 0.72.15 A correlation coefficient of about 0.65 was found, with similar measures of self-esteem, and of 0.40 with teacher and peer ratings. 2~ RESULTS Subject match. Analysis of demographic data for the three groups of children indicated that they were similar in age, gender, and percentage who came from single-parent families. Although specific data concerning socioeconomic
638
Lewis and K h a w
The Journal of Pediatrics October 1982
Table. Results of t tests examining differences between groups on mean Behavior Problem Checklist scores Group comparisons
Value
t Value
P
CF vs control Asthma vs control CF vs asthma
2.71 3.63 -0.92
2.12 2.72 -0.71
0.037* 0.004]0.480
*Significantat P = 0.05. ~Sigrfificantat P = 0.01
status were unavailable, the three groups were similar in range of educational and occupational levels of the parents. Childhood illness and family functioning. In order to test the hypothesis that families caring for children with CF are more likely to be functioning at the extremes within the circumplex model of adaptability and cohesion, FACES scores were utilized in the following manner: First, grand means were calculated for adaptability and for cohesion based on the pooled data from all 84 mothers. Next, a deviation score was calculated for each of the mothers on each of the two dimensions. These scores were obtained by taking the absolute value of the difference between a mother's score on one dimension and the grand mean for that dimension. If caring for a child with CF is stressful enough to cause deficits in family functioning, one would expect FACES deviation scores to be greater in this group than in the healthy control subjects. It was also hypothesized that there would be a difference between the deviation scores for CF and asthma families, given the life-threatening nature of the former illness. Differences between the group means were examined by multivariate analysis of variance. The results indicated no significant difference between any of the groups for deviation adaptability or cohesion scores. Apparently, caring for a child with a chronic illness was not related to extreme family functioning within the circumplex model. Childhood illness and child adjustment. To test the hypothesis that CF affects the psychological adjustment of afflicted children, mean behavioral and self-concept scores were calculated for each of the three groups of children, and t tests were used to examine differences between the group means. No significant difference was found between any of the three groups when self-concept was used as the dependent variable. Based on this result it appears that the child's self-concept is not affected by the presence of CF. When frequency of behavior problems was used as the dependent variable, however, the presence of a chronic illness was related to the child's adjustment (Table). Both the CF and the asthmatic children had significantly more
reported behavior problems than had the control children (P < 0.05 for CF children; P < 0.01 for asthmatic children). There was no significant difference in number of reported behavior problems between the two groups of chronically ill children. Family functioning and child adjustment. To test the hypothesis that extreme family functioning is negatively related to child adjustment, regardless of the presence of illness, two multiple regression analyses were performed with the pooled data (n = 84), with deviation adaptability and cohesion scores used as the independent variables. Piers-Harris score was the dependent variable in the first analysis and Behavior Problem Checklist score in the second. When the relationship between FACES deviation scores and the child's self-concept was evaluated, an F value of 2.97 was obtained (P = 0.059). With the reported number of behavior problems of the child as the dependent variable, the multiple regression analysis yielded highly significant results (F = 7.76, P = 0.001): There was a significant relationship between extreme family functioning and the number of behavior problems reported in the children. In order to determine if family functioning is an important mediating variable affecting adjustment in the child with CF, a post hoe statistical analysis was performed to see if there was a significant difference in frequency of behavior problems between the CF and healthy children when controlling for the effects of family functioning. An analysis of covariance was employed, with deviation adaptability and cohesion scores as the covariates. With this procedure, the difference previously noted between the two groups was no longer significant (P > 0.10). DISCUSSION Before the implications of the major findings of this study are discussed, a word should be said about limitations related to the design. The procedures used to obtain subjects from the three populations were somewhat different. The control group of healthy subjects were all selected from the same elementary school system and therefore probably represent a more homogeneous population than either of the chronically ill groups. Moreover, socioeconomic status was not formally assessed and therefore cannot be ruled out as a possible contaminating factor. On the positive side, the three groups of children were similar on a number of important dimensions, i.e., age, race, gender, and were all from roughly the same geographic area. Few studies have used healthy control groups for purposes of comparison. We believe that the control groups used in this study, although not perfect, represent a methodologic improvement over the use of standardized norms. The first major finding in this study was that the
Volume 10l Number 4
presence of a chronic childhood illness, like CF, was not significantly related to extreme family functioning as assessed by mothers' adaptability and cohesion scores on FACES. The circumplex model of family systems proposes that families under stress have a tendency to move toward the extremes on adaptability and cohesion, If this model is correct and if FACES and the method used to interpret FACES scores are valid, there are two possible explanations for the above result: Either families of chronically ill children are not experiencing stress associated with the illness or they are successfully coping with any stress. This second explanation seems more plausible. Caring for seriously ill children must be inherently stressful for affected families; it does not have to lead to deficits in family functioning, however. Circumplex theory suggests that healthy families may move toward the extremes of adaptability and cohesion when initially confronted with a stressful event, but eventually gravitate back toward the moderate ranges as they successfully cope with these events. It is possible that this is what the families of CF children have done; i.e., they have adapted to the stress in a normal and healthy manner. (It should be remembered that all of the chronically ill children were in stable condition when tested.) An interesting test of the circumplex model would be to study changes in family functioning relative to CF over the course of the disease. If the theory is correct, one would expect healthy families to move toward the extremes on adaptability and cohesion at the time of diagnosis and during periods of acute exacerbations of the illness, but to gravitate back toward the middle as the illness is stabilized. Dysfunctional families could be identified by their inability to move back and forth in this fluid manner. The results concerning the relationship between chronic illness and the psychological adjustment of the affected children were mixed. As expected, chronieal!y ill children had significantly more behavior problems than healthy children. Contrary to expectation, they did not differ in self-concept. These two variables were examined separately because they were thought to be measuring very different aspects of the child's adjustment. Self-concept relates to an internal feeling or perception of self-worth, whereas behavior problems reflect a learned style of interaction with the environment. A possible explanation for this discrepancy involves the relationship established between sick child and parent (especially mother). There is a tendency for sick children to be overprotected by their mothers] 1who compensate for their feelings of guilt and helplessness by being more tolerant and less likely to use consistent discipline. This could have the effect of making these children more prone to developing behavior problems without necessarily affecting the way they feel about themselves.
Psychosocial adjustment in cystic fibrosis
639
The relationship between extreme family functioning and behavior problems in the children was much more significant than the relationship between chronic illness and behavior problems. It can be concluded from these results that adjustment difficulties in children (defined in terms of behavior problems and self-concept) are more related to extreme family functioning than to the presence of chronic childhood illness. This conclusion suggests that family functioning is an important mediating variable to consider in childhood adjustment to CF.
REFERENCES
1. Kulczyeki LL, Robinson ME, and Berg CM: Somatic and psychosocial factors relative to management of patients with cystic fibrosis, Clin Proc Child Hosp DC 25:320, 1969. 2. LaMer R, Nakielny W, and Wright N: Psychologicalimplications of cystic fibrosis, Can Med Assoc J 94:1043, 1966. 3. Cytrn L, Vanmoore PVP, and Robinson HE: Psychological adjustment of children with cystic fibrosis, in Anthony EJ, and Koupernik C, editors: The child and his family, vol II, New York, 1973, John Wiley & Sons, Inc. 4. Spock A, and Stedman D: Psychological characteristics of children with cystic fibrosis, NC Med J 27426, 1966. 5. Tropauer A, Franz M, and Dilgard V: Psychological aspects of the care of children with cystic fibrosis, Am J Dis Child 119:424, 1970. 6. Turk J: Impact of cystic fibrosis on family functioning, Pediatrics 34:67, 1964. 7. Gayton WF, and Friedman SB: Psychosocial aspects of cystic fibrosis: A review of the literature, Am J Dis Child 126:856, 1973. 8. Drotar D, Doershuk CF, Stern RC: et al: Psyehosoeial functioning of children with cystic fibrosis. Pediatrics 67:338, 1981. ' 9. Gayton WF, Friedman SB, Tavormina JF, et al: Children with cystic fibrosis: Psychological test findings of patients, siblings and parents, Pediatrics 59:888, I977. 10. Tavorimina JB, Kastner LS, Slater PM, et al: Chronically ill children: A psychologicallyand emotionatly deviant population? J Abnorm Child Psycho1 4:99, 1976. 11. Bedell JR, Giordani B, Amour JL, et al: Life stress and the psychological and medical adjustment of chronically ill children, J Psychosom Res 21:237, 1977. 12. OlsonDH, Sprenkle DH, and Russell CS: Circumplex model of marital and family systems. 1. Cohesion and adaptability dimensions, family types, and clinical application, Family Process 18:3, 1979. 13. OlsonDH, Bell R, and Portner J: FACES: Family Adaptability and Cohesion Evaluation Scales, St. Paul, 1978, Family Social Science. 14. Quay HC, and Peterson DR: Manual for the Behavior Problem Checklist, Authors, 1979. 15. Piers E, and Harris D: Manual for Piers-Harris Children's Self-Concept Scale, Nashville, 1969, Counselor Recordings and Tests. 16. Olson DH, and McCubbin HI: Circumplex model of marital and family systems: Application to family stress and crisis intervention, in MuCubbin HI, editor: Family stress, coping and social support, Minneapolis, 1980, Burgress Publishing Co.
640
Lewis and K h a w
17. Olson DH, Russell CS, and Sprenkle DH: Circumplex model of marital and family systems. II. Empirical studies and clinical intervention, in Vincend JP, editor: Advances in family intervention, assessment and theory, Greenwich, Conn, 1979, JAI Press. 18. Quay G: Measuring dimensions of deviant behavior: The Behavior Problem Checklist, J Abnorm Child Psychol 5:277, 1977.
The Journal o f Pediatrics October 1982
19. O'Leary KD, and Johnson SB: Psychological assessment, in Quay H, and Werry J editors: Psychopathological disorders of childhood, ed 2, New York, 1979, John Wiley & Sons, Inc. 20. Piers EV: The Piers-Harris Children's Self-Concept Scale: Research monograph no. 1, Nashville, 1977, Counselor Recordings and Tests. 21. Travis G: Chronic illness in children: Its impact on child and family, Stanford, 1976, Stanford University Press.
P E D I A T R I C S
Richard W. Olmsted, Editor
Family functioning as a mediating variable affecting psychosocial adjustment of children with cystic fibrosis Family functioning was examined as a mediating variable affecting adjustment in children with cystic fibrosis. Three groups o f children (cystic fibrosis, asthma, and healthy control subjects) were initially compared on two dimensions o f adjustment: self-concept and frequency o f behavior problems. The families o f these children were compared according to functional level o f "'cohesion" and "'adaptability" (assessed by an as yet unvalidated family functioning measure). No significant difference was found between the groups in family functioning or children's self-concept. The two groups o f chronically ill children did have significantly higher frequencies o f behavior problems than had the healthy children. When there was statistical control for effects related to family functioning, however, this difference was not significant. Family functioning was found to be a better predictor o f child adjustment than was the presence o f illness. These results are consistent with recent research suggesting that children with cystic fibrosis adjust in basically healthy ways and that any deficits result more from mediating factors. In this study family functioning was found to be an important mediating factor in this process.
Brian L. Lewis, Ph.D.,* and Kon-Taik Khaw, M.D., Boston, Mass.
EARLY STUDIES of the psychosocial effects of cystic fibrosis were generally supportive of the commonly held belief that the presence of CF leads to profound deficits in individual adjustment and family functioning. 1-6In reviewing these studies, Gayton and Friedman 7 noted a number of methodologic problems: Most utilized few subjects and inadequate controls and were primarily descriptive in nature, relying on clinical experience and subjective evaluations. Recent evidence indicates that children with C F are not as maladjusted as has been previously reportedY ~ Although these children, as a group, do sometimes score more poorly on specific measures of psychologic adjustment, 8,9 most appear to be coping with the illness in a
From the Cystic Fibrosis Program, Division o f Cell Biology, Children's Hospital Medical Center, Department o f Pediatrics, Harvard Medical School. Supported in part by the Ina S. Pulmutter Cystic Fibrosis Research Fund. *Reprint address: Ethan Allen Medical Center, 1100 Ethan Allen Ave., Winooski, VT 05404.
636
The Journal of P E D I A T R I C S Vol. 10l, No. 4, pp. 636-640
"normal," well-adjusted manner) ~ Based on this finding, it has been suggested that a more fruitful course for future research would be to identify mediating variables that are important in the coping process and Which differentiate healthy from unhealthy adjustment. Abbreviation used FACES: Family Adaptability and Cohesiveness Evaluation Scales Bedell et a111 have identified "life stress" as one such mediating variable. These researchers found that the self-concepts of chronically ill children (including children with CF) were related to the level of stress they experienced. The implication of this result was that the poor adjustment often reported in chronically ill children might result more from life stress than from the disease. Olson et aW have proposed a conceptual system for examining family functioning called the "circumplex model," which is based on family systems theory. Within this model families can be classified according to how they function on two salient dimensions, "cohesion" and "adaptability." Cohesion is defined as "the emotional
0022-3476/82/100636+05500.50/0
9 1982 The C. V. Mosby Co.
Volume 101 Number 4 bonding members have with one another and the degree of individual autonomy a person experiences in the family" (p. 5). Adaptability is defined as, "the ability of a marital or family system to change its power structure, role relationships, and relationships rules in response to situational and developmental stress" (p. 12). According to the theory, healthy families function in the moderate or balanced ranges on both dimensions. We hypothesized that healthy adjustment in the child with C F is more dependent upon healthy family functioning (balanced levels of cohesion and adaptability) than on the mere presence of the illness. METHOD Subjects. Eighty-four children and their mothers participated as subjects in this study. The children were between the ages of 7 and 12 years and represented one of the following three groups: (1) children with cystic fibrosis (n = 31), randomly selected from the population of children being treated on an outpatient basis in the CF clinic at Children's Hospital Medical Center in Boston; (2) children with chronic asthma (n = 26), randomly selected from the population of asthmatic children being treated on an outpatient basis in the allergy clinic at Children's Hospital in Boston; and (3) healthy children, i.e., children with no previous history of chronic illness (n -- 27), randomly selected from a consenting school system in the suburban Boston area. The three groups of children were matched by age, gender, and race (only white children were selected as C F occurs predominantly in white subjects). An effort was made to control for socioeconomic status by selecting the healthy children from a school, within the consenting suburban school system, that seemed to best reflect the socioeconomic level of the clinic populations. In addition, the chronically ill children (those in groups 1 and 2) had to meet the following selection criteria: They had to have been diagnosed as having their illness for at least two years; they could not have siblings diagnosed with the same illness; and they could not have been hospitalized for exacerbations of the illness over the preceding year. These additional selection criteria were used to ensure that the coping and adjustment variables being assessed would reflect long-term adaptation to the illness rather than short-term responses to recent crisis situations or atypical stress. One further criterion used in selecting the asthmatic children was that their illness had to be severe enough to require medication daily. Prior to participating in the study all parents reviewed and signed consent forms approved by the Hospital Consent Committee. Tests administered. The procedure for administering the
Psychosocial adjustment in cystic fibrosis
637
questionnaires was identical for all groups. Mothers completed the Family Adaptability and Cohesiveness Evaluation Scales 13 and the Behavior Problem Checklist. TM Children completed the Piers-Harris Children's Self-Concept Scale) 5
Family Adaptability and Cohesiveness Evaluation Scales. This is a self-report instrument based on the circumplex model previously discussed, consisting of 111 statements concerning various aspects of family dynamics. The statements are of the type, "Family members are concerned with each other's welfare." The person completing the instrument indicates, on a scale from 1 to 4, the degree to which each statement is felt to be true of his/her family. Two separate scores are obtained from an analysis of the responses, one for cohesion and one for adaptability. Because of the newness of this instrument, validity studies are still underway and have not yet been reported. The circumplex model upon which FACES was developed does appear to have empiric validity in terms of differentiating families under stress and setting treatment goals for family therapy.16. J7
Behavior Problem Checklist. This checklist consists of 55 problem behaviors that often occur in childhood and adolescence; it can be completed by anyone familiar with a child's behavior. The person completing the instrument goes through the list, placing a check next to each item that is felt to be a problem for the targeted child. The total number of items checked provides a rating of the frequency of behavioral problems for that child. The scale is reported to be internally consistent and to have test-retest reliability of about 0.82.18 O'Leary and Johnson's 19 review suggested that the behavior checklist has high concurrent and construct validity, and that it reliably differentiates groups of children with known behavior disorders. Piers-Harris Children's Self-Concept Scale. This is an 80-item self-report questionnaire designed to assess the child's self-evaluation. The items are of the type, "I am smart" and "It is hard for me to make friends," to which the child indicates whether the statement is true or false for him/her by circling either "yes" or "no." The instrument is scored to provide an overall indication of the child's self-concept. Reports of internal consistency for the scale range from 0.78 to 0.93, with test-retest reliability estimates about 0.72.15 A correlation coefficient of about 0.65 was found, with similar measures of self-esteem, and of 0.40 with teacher and peer ratings. 2~ RESULTS Subject match. Analysis of demographic data for the three groups of children indicated that they were similar in age, gender, and percentage who came from single-parent families. Although specific data concerning socioeconomic
638
Lewis and K h a w
The Journal of Pediatrics October 1982
Table. Results of t tests examining differences between groups on mean Behavior Problem Checklist scores Group comparisons
Value
t Value
P
CF vs control Asthma vs control CF vs asthma
2.71 3.63 -0.92
2.12 2.72 -0.71
0.037* 0.004]0.480
*Significantat P = 0.05. ~Sigrfificantat P = 0.01
status were unavailable, the three groups were similar in range of educational and occupational levels of the parents. Childhood illness and family functioning. In order to test the hypothesis that families caring for children with CF are more likely to be functioning at the extremes within the circumplex model of adaptability and cohesion, FACES scores were utilized in the following manner: First, grand means were calculated for adaptability and for cohesion based on the pooled data from all 84 mothers. Next, a deviation score was calculated for each of the mothers on each of the two dimensions. These scores were obtained by taking the absolute value of the difference between a mother's score on one dimension and the grand mean for that dimension. If caring for a child with CF is stressful enough to cause deficits in family functioning, one would expect FACES deviation scores to be greater in this group than in the healthy control subjects. It was also hypothesized that there would be a difference between the deviation scores for CF and asthma families, given the life-threatening nature of the former illness. Differences between the group means were examined by multivariate analysis of variance. The results indicated no significant difference between any of the groups for deviation adaptability or cohesion scores. Apparently, caring for a child with a chronic illness was not related to extreme family functioning within the circumplex model. Childhood illness and child adjustment. To test the hypothesis that CF affects the psychological adjustment of afflicted children, mean behavioral and self-concept scores were calculated for each of the three groups of children, and t tests were used to examine differences between the group means. No significant difference was found between any of the three groups when self-concept was used as the dependent variable. Based on this result it appears that the child's self-concept is not affected by the presence of CF. When frequency of behavior problems was used as the dependent variable, however, the presence of a chronic illness was related to the child's adjustment (Table). Both the CF and the asthmatic children had significantly more
reported behavior problems than had the control children (P < 0.05 for CF children; P < 0.01 for asthmatic children). There was no significant difference in number of reported behavior problems between the two groups of chronically ill children. Family functioning and child adjustment. To test the hypothesis that extreme family functioning is negatively related to child adjustment, regardless of the presence of illness, two multiple regression analyses were performed with the pooled data (n = 84), with deviation adaptability and cohesion scores used as the independent variables. Piers-Harris score was the dependent variable in the first analysis and Behavior Problem Checklist score in the second. When the relationship between FACES deviation scores and the child's self-concept was evaluated, an F value of 2.97 was obtained (P = 0.059). With the reported number of behavior problems of the child as the dependent variable, the multiple regression analysis yielded highly significant results (F = 7.76, P = 0.001): There was a significant relationship between extreme family functioning and the number of behavior problems reported in the children. In order to determine if family functioning is an important mediating variable affecting adjustment in the child with CF, a post hoe statistical analysis was performed to see if there was a significant difference in frequency of behavior problems between the CF and healthy children when controlling for the effects of family functioning. An analysis of covariance was employed, with deviation adaptability and cohesion scores as the covariates. With this procedure, the difference previously noted between the two groups was no longer significant (P > 0.10). DISCUSSION Before the implications of the major findings of this study are discussed, a word should be said about limitations related to the design. The procedures used to obtain subjects from the three populations were somewhat different. The control group of healthy subjects were all selected from the same elementary school system and therefore probably represent a more homogeneous population than either of the chronically ill groups. Moreover, socioeconomic status was not formally assessed and therefore cannot be ruled out as a possible contaminating factor. On the positive side, the three groups of children were similar on a number of important dimensions, i.e., age, race, gender, and were all from roughly the same geographic area. Few studies have used healthy control groups for purposes of comparison. We believe that the control groups used in this study, although not perfect, represent a methodologic improvement over the use of standardized norms. The first major finding in this study was that the
Volume 10l Number 4
presence of a chronic childhood illness, like CF, was not significantly related to extreme family functioning as assessed by mothers' adaptability and cohesion scores on FACES. The circumplex model of family systems proposes that families under stress have a tendency to move toward the extremes on adaptability and cohesion, If this model is correct and if FACES and the method used to interpret FACES scores are valid, there are two possible explanations for the above result: Either families of chronically ill children are not experiencing stress associated with the illness or they are successfully coping with any stress. This second explanation seems more plausible. Caring for seriously ill children must be inherently stressful for affected families; it does not have to lead to deficits in family functioning, however. Circumplex theory suggests that healthy families may move toward the extremes of adaptability and cohesion when initially confronted with a stressful event, but eventually gravitate back toward the moderate ranges as they successfully cope with these events. It is possible that this is what the families of CF children have done; i.e., they have adapted to the stress in a normal and healthy manner. (It should be remembered that all of the chronically ill children were in stable condition when tested.) An interesting test of the circumplex model would be to study changes in family functioning relative to CF over the course of the disease. If the theory is correct, one would expect healthy families to move toward the extremes on adaptability and cohesion at the time of diagnosis and during periods of acute exacerbations of the illness, but to gravitate back toward the middle as the illness is stabilized. Dysfunctional families could be identified by their inability to move back and forth in this fluid manner. The results concerning the relationship between chronic illness and the psychological adjustment of the affected children were mixed. As expected, chronieal!y ill children had significantly more behavior problems than healthy children. Contrary to expectation, they did not differ in self-concept. These two variables were examined separately because they were thought to be measuring very different aspects of the child's adjustment. Self-concept relates to an internal feeling or perception of self-worth, whereas behavior problems reflect a learned style of interaction with the environment. A possible explanation for this discrepancy involves the relationship established between sick child and parent (especially mother). There is a tendency for sick children to be overprotected by their mothers] 1who compensate for their feelings of guilt and helplessness by being more tolerant and less likely to use consistent discipline. This could have the effect of making these children more prone to developing behavior problems without necessarily affecting the way they feel about themselves.
Psychosocial adjustment in cystic fibrosis
639
The relationship between extreme family functioning and behavior problems in the children was much more significant than the relationship between chronic illness and behavior problems. It can be concluded from these results that adjustment difficulties in children (defined in terms of behavior problems and self-concept) are more related to extreme family functioning than to the presence of chronic childhood illness. This conclusion suggests that family functioning is an important mediating variable to consider in childhood adjustment to CF.
REFERENCES
1. Kulczyeki LL, Robinson ME, and Berg CM: Somatic and psychosocial factors relative to management of patients with cystic fibrosis, Clin Proc Child Hosp DC 25:320, 1969. 2. LaMer R, Nakielny W, and Wright N: Psychologicalimplications of cystic fibrosis, Can Med Assoc J 94:1043, 1966. 3. Cytrn L, Vanmoore PVP, and Robinson HE: Psychological adjustment of children with cystic fibrosis, in Anthony EJ, and Koupernik C, editors: The child and his family, vol II, New York, 1973, John Wiley & Sons, Inc. 4. Spock A, and Stedman D: Psychological characteristics of children with cystic fibrosis, NC Med J 27426, 1966. 5. Tropauer A, Franz M, and Dilgard V: Psychological aspects of the care of children with cystic fibrosis, Am J Dis Child 119:424, 1970. 6. Turk J: Impact of cystic fibrosis on family functioning, Pediatrics 34:67, 1964. 7. Gayton WF, and Friedman SB: Psychosocial aspects of cystic fibrosis: A review of the literature, Am J Dis Child 126:856, 1973. 8. Drotar D, Doershuk CF, Stern RC: et al: Psyehosoeial functioning of children with cystic fibrosis. Pediatrics 67:338, 1981. ' 9. Gayton WF, Friedman SB, Tavormina JF, et al: Children with cystic fibrosis: Psychological test findings of patients, siblings and parents, Pediatrics 59:888, I977. 10. Tavorimina JB, Kastner LS, Slater PM, et al: Chronically ill children: A psychologicallyand emotionatly deviant population? J Abnorm Child Psycho1 4:99, 1976. 11. Bedell JR, Giordani B, Amour JL, et al: Life stress and the psychological and medical adjustment of chronically ill children, J Psychosom Res 21:237, 1977. 12. OlsonDH, Sprenkle DH, and Russell CS: Circumplex model of marital and family systems. 1. Cohesion and adaptability dimensions, family types, and clinical application, Family Process 18:3, 1979. 13. OlsonDH, Bell R, and Portner J: FACES: Family Adaptability and Cohesion Evaluation Scales, St. Paul, 1978, Family Social Science. 14. Quay HC, and Peterson DR: Manual for the Behavior Problem Checklist, Authors, 1979. 15. Piers E, and Harris D: Manual for Piers-Harris Children's Self-Concept Scale, Nashville, 1969, Counselor Recordings and Tests. 16. Olson DH, and McCubbin HI: Circumplex model of marital and family systems: Application to family stress and crisis intervention, in MuCubbin HI, editor: Family stress, coping and social support, Minneapolis, 1980, Burgress Publishing Co.
640
Lewis and K h a w
17. Olson DH, Russell CS, and Sprenkle DH: Circumplex model of marital and family systems. II. Empirical studies and clinical intervention, in Vincend JP, editor: Advances in family intervention, assessment and theory, Greenwich, Conn, 1979, JAI Press. 18. Quay G: Measuring dimensions of deviant behavior: The Behavior Problem Checklist, J Abnorm Child Psychol 5:277, 1977.
The Journal o f Pediatrics October 1982
19. O'Leary KD, and Johnson SB: Psychological assessment, in Quay H, and Werry J editors: Psychopathological disorders of childhood, ed 2, New York, 1979, John Wiley & Sons, Inc. 20. Piers EV: The Piers-Harris Children's Self-Concept Scale: Research monograph no. 1, Nashville, 1977, Counselor Recordings and Tests. 21. Travis G: Chronic illness in children: Its impact on child and family, Stanford, 1976, Stanford University Press.