Stress and Family Satisfaction in Parents of ... - Wiley Online Library

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*New York University Medical Center, Rusk Institute of Rehabilitation Medicine, and †Laser and Skin Surgery. Center of New York and New York University ...
CLINICAL AND LABORATORY INVESTIGATIONS Pediatric Dermatology Vol. 16 No. 3 190–197, 1999

Stress and Family Satisfaction in Parents of Children with Facial Port-Wine Stains A. Cate Miller, Ph.D.,* Ineke M. Pit-ten Cate, M.A.,* Heather S. Watson, B.A.,* and Roy G. Geronemus, M.D.† *New York University Medical Center, Rusk Institute of Rehabilitation Medicine, and †Laser and Skin Surgery Center of New York and New York University Medical Center, New York, New York

Abstract: A cross-sectional survey was employed to assess parenting stress, family satisfaction, and parental concerns and to determine predictors of stress in parents of children with port-wine stains (PWSs). The participants were 46 parents of 24 children receiving treatment with pulsed dye laser photocoagulation for facial PWS at an outpatient dermatology clinic based at a university medical center. Outcome measures used were self-report instruments assessing psychosocial adjustment (Parenting Stress Index, Family Satisfaction Scale, and Parental Concerns Questionnaire). As a group, parents scored in the average range on the stress and family satisfaction measures when compared with a normative sample; five parents (11%) scored in the clinical range for stress. Forty-nine percent of the variance in parenting stress was accounted for by four variables: the child’s age (␤ = 0.34; p = 0.031), the parents’ degree of family satisfaction (␤ = −0.27; p = 0.077), the level of parental concern regarding the child’s facial PWS (␤ = 0.45; p = 0.005), and the parents’ satisfaction with staff communication (␤ = −0.51; p = 0.002). The data suggest that while, as a group, parents of children with a facial PWS report to be in the average range for psychological stress, some do not fare as well as others. Factors associated with lower stress include younger children, more family cohesion and adaptation, fewer parental concerns, and greater satisfaction with parent-staff communication. The potential for the development of medical complications and psychological problems over time suggests the need for treatment of the PWS at an early age. Health care providers should be prepared to screen for clinical levels of distress and to refer parents for psychological intervention when needed.

A port-wine stain (PWS) is a benign, disfiguring vascular birthmark consisting of ectatic dermal capillaries. This congenital anomaly is relatively common, occurring in an estimated 3 children per 1000 births, and approxiAddress correspondence to A. Cate Miller, Ph.D., New York University Medical Center, Rusk Institute of Rehabilitation Medicine, 400 East 34th St., RR 506, New York, NY 10016. Ineke M. Pit-ten Cate is now affiliated with the Centre for Research into Physiological Development, University of Southampton, UK.

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mately 80% of port-wine stains are found on the face or neck (1). It presents as a pink-to-red area which typically darkens to purple with age and grows in proportion to the general growth of the child’s body. Proliferative growth

Miller et al: Stress in Parents of Children with PWS

of PWSs has been found in two-thirds of patients by the age of 45. The consequences of this proliferative growth include thickening, nodularity, spontaneous bleeding, and infection arising within the PWSs (2). A PWS is formed by malformed blood vessels close to the skin surface and currently the treatment of choice is pulsed dye laser photocoagulation. The energy from the laser is absorbed by the hemoglobin in the large blood vessels of the PWS, causing photocoagulation and heat damage to the endothelium. The diameter of these cutaneous vessels is lessened and the PWS is significantly diminished, if not completely removed. The PWS is associated with two uncommon syndromes: Sturge–Weber syndrome and Klippel– Trenaunay syndrome. Sturge–Weber syndrome is a neurocutaneous disorder characterized by cutaneous facial angioma, leptomeningeal angioma associated with seizures, and other neurologic complications including mental retardation and glaucoma (3). Klippel–Trenaunay is characterized by capillary malformations, atypical varicosities or venous malformations, and bony or soft tissue hypertrophy of the upper or lower extremities (4). Although the medical aspects of PWSs and their treatment have been well reported, data regarding the psychological implications for children and their parents are limited. Because access to children with disfigurement has usually been through plastic surgery clinics, the vast majority of available research has focused on children with craniofacial anomalies and cleft lips and palates, conditions which have a long history of surgical intervention. Recent advances in the use of lasers to treat children with PWSs have now made this population accessible for study. Sociologic studies on attractiveness conducted in the 1970s and studies of children with facial anomalies were the bases for the initial hypotheses that people with a facial PWS suffer from psychological and social impairments. Some studies have substantiated these claims, stating that children born with facial vascular malformations suffer psychological and emotional difficulties (5). Similarly, Lanigan and Cotterill (6) found that people with PWSs suffer a significant degree of social stigmatization and have lowered levels of self-esteem, which has an adverse effect on social development. In contrast, results of standardized psychological screening tests for psychiatric illness, depression, and anxiety reveal that these patients have similar or less evidence of morbidity than controls (6). The authors of the latter study suggest that a considerable degree of psychological disability does exist but is suppressed. The hypothesis of psychological morbidity has not been substantiated by other studies. For example, in a study of the first patients to be treated in this country by laser, Kalick et al. (7) failed to find noteworthy differences between the adult patient

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sample and a normative sample on measures of neuroticism, introversion/extroversion, depression, hostility, self-esteem, and anxiety. In a study of children with PWSs and children with prominent ears, Sheerin et al. (8) found that the children with PWSs scored as well as or better than nondisfigured peers on measurements of psychosocial adjustment, while the children with prominent ears did not fare very well. Hill-Beuf and Porter (9) found that for children with vitiligo, a disfiguring condition that involves depigmentation of the skin, those who developed competencies that build self-esteem appeared to cope well with the disorder. Older age and a change in living circumstances were associated with greater stress (9). A facial disfigurement, then, may be best conceptualized as a risk factor, the significance of which is mediated by factors such as maternal adjustment, parent-child interaction, parental social support, and medical severity of disfigurement (10). There have been no systematic investigations of the parents of children with PWSs, or of the impact of parental factors on the well-being of children with PWSs. Wagner and Wagner (11) used anecdotal interview data to describe ways in which parents and the parent-child relationship may be impacted by the presence of a PWS. They suggested that parents may be acutely aware of the potential social difficulties the child will encounter. Consequently they may develop a style of interaction with the child ranging from overprotection to the encouragement of fierce independence. The parent may also experience some degree of narcissistic vulnerability, and may cope by either emotionally distancing him/herself from the child or minimizing the PWS. More systematic inquiries have been conducted with families of children with craniofacial anomalies. Campis et al. (10) found that the mothers in their study resembled a normal population in terms of their psychological functioning and the quality of the mother-child relationship. The mother’s perception of her relationship with her child better predicted the child’s emotional adjustment than either the medical severity of the anomaly or the mother’s social support network. Pillemer and Cook (12) found that parents who were highly stressed rated their children as more anxious, depressed, and socially inadequate than less-stressed parents. The children also had a lower self-concept than other children. Another study reported higher stress levels, lower self-competence evaluations, and more marital conflict in mothers of children with craniofacial anomalies than in mothers of healthy children (13). However, observational measures revealed no group differences in maternal response to the child or in the behavior and responsiveness of the children themselves. Similarly parents of children with craniofacial anomalies also reported less available social support and less satisfaction with their support than a

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matched sample of parents of children with no significant physical or behavioral problems (14). Of interest, the parents of the children with anomalies in this study underreported the presence of behavioral-psychological problems in their children. Barden et al. (15) found that while mothers of infants with craniofacial anomalies rated their current life and parental satisfaction more positively than mothers in a control group, they exhibited consistent patterns of less nurturing behavior. This suggests that the quality of the parent-child interaction may be affected without the parents’ awareness. However, caution should be taken in interpreting these results since only 10 mother-infant dyads were studied (5 mothers of children with anomalies; 5 controls). In a recent study involving children with craniofacial anomalies, types of parental involvement were found to predict the child’s social competence and satisfaction with facial appearance (16,17). Parents who were highly concerned about the child’s peer interactions had children with lower social competence and greater dissatisfaction with their facial appearance. Similarly parents who provided advice/support had children with greater dissatisfaction with their facial appearance. In contrast, when parents used more active orchestration their children evidenced greater social competence. The current study is a response to the paucity of data regarding parents of children with PWSs. The purpose is to explore the psychological experiences of parents of children undergoing laser treatment for facial PWSs. The aims of this study are twofold: to describe the parents’ psychological and family functioning, and to determine which child and parent characteristics predict parental distress. Predictors to be examined include demographic characteristics, family satisfaction, parental concerns, parents’ knowledge regarding the PWS, and parent-staff communication. METHODS Subjects The parents (24 mothers and 22 fathers) of 24 children receiving laser treatment for facial PWSs in an outpatient dermatology clinic based at a university medical center participated in this study (n ⳱ 46). This represents a participation rate of 65% of the parents who were invited to take part in this study. All parents of children consecutively admitted over an 11-month period and who met the following inclusion criteria were approached: their child’s PWS must be located on the face or neck, and their child must be a new referral for laser treatment, that is, their child must not have begun receiving treatment. Parents were solicited prior to their first consultation with the dermatologist. The instruments (PSI, FSS, CBCL, and PCQ) and a written informed consent form

were mailed to those who offered verbal consent for participation. Parents completed the questionnaires at home and returned them by mail or in person prior to or at the time of the first laser treatment appointment. The Medical Information Form was administered immediately following the consultation with the dermatologist. Parents completed this questionnaire in the physician’s office and without consulting each other or written materials. Institutional review board approval and written informed consent from each parent participant were obtained. Two of the children were diagnosed with Sturge– Weber syndrome and one was diagnosed with Klippel– Trenaunay syndrome. There were no differences between these three children and the other children on individual or family demographic characteristics. The average size of the children’s PWSs was 15 cm2, with a range of 3 to 300 cm2. All subjects are the biological parents of the children being treated, with the exception of one who is a stepfather. All participants are Caucasian. The mothers’ ages range from 25 to 40 years (mean ⳱ 32.8; SD ⳱ 4.26), and the fathers’ ages range from 30 to 48 years (mean ⳱ 36.0; SD ⳱ 4.72). The ages of the 7 boys and 17 girls range from 1 month to 12.3 years (median ⳱ 1.9 years). Eighty-three percent of the children are 5 years of age or younger. Frequency and percentage distributions for additional demographic variables are found in Table 1. As the table shows, almost all of the parents are married. The number of years married ranges from less than 1 to 14 (mean ⳱ 6.5; SD ⳱ 3.82). Measures Parenting Stress Index-Short Form (PSI-SF) The PSI-SF (18) is a shortened version of the Parenting Stress Index (PSI). A 5-point Likert scale is used to rate 36 items. The PSI-SF contains three subscales: parental distress, parent-child dysfunctional interaction, and difficult child. Summing these three subscales yields the total stress score. The author reports an alpha reliability coefficient of 0.91 for the total scale. Alpha reliability coefficients for the subscales of parental distress, parentchild dysfunctional interaction, and difficult child are 0.87, 0.80, and 0.85, respectively. Test-retest reliability coefficients range from 0.68 to 0.85 for the subscale and total scores. The manual includes research regarding content and concurrent validity. The final descriptive statistics and norms for the PSI-SF were produced by combining the initial and replicative samples of the norming studies for the PSI. The first sample consisted primarily of parents who were bringing their children for the child’s 1-year wellcare visit to a group pediatric practice in a small city in Virginia. The PSI was administered as part of a routine screening of all parents and was an integrated component

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TABLE 1. Frequency and Percentage Distributions for Demographic Characteristics Mothers (n ⳱ 24) Variable Marital status Married Separated Single (never married) Employment status Employed Unemployed Missing Religion Catholic Protestant Jewish Other None Missing Education Some high school High school diploma Some college Bachelors degree Masters degree Doctoral degree Number of children 1 2 3 Family income $20,000–29,999 $30,000–39,999 $40,000–49,999 $50,000–59,999 $60,000–79,000 $80,000–99,999 ⱖ$100,000 Missing

Fathers (n ⳱ 22)

Frequency

Percent*

Frequency

Percent*

22 1 1

92 4 4

20 2 0

91 9 0

14 9 1

58 38 4

19 1 2

86 5 9

13 3 4 1 1 2

54 13 17 4 4 8

13 4 5 0 0 0

59 18 23 0 0 0

0 8 7 4 2 3

0 33 29 17 8 13

1 2 11 4 4 0

5 9 50 18 18 0

10 8 6

42 33 25

1 2 4 3 4 1 6 3

4 8 17 13 17 4 25 13

* Percentages may not total 100 due to rounding.

of the comprehensive health care services provided. The second sample consisted of parents from the same group pediatric practice who completed the PSI during a wellchild check for day care, kindergarten, or first grade. The mean age of the mothers and fathers from the PSI norming studies was 30.9 years (SD ⳱ 5.9 years) and 32.1 years (SD ⳱ 6.0 years), respectively. The mean number of children in the home was 2.1 (SD ⳱ 1.2) and the average age of the target child was 4.9 years (SD ⳱ 3.1 years). Eighty percent of these samples reported an annual income of ⱕ$40,000; 77% were married; 27% of the mothers and 28% of the fathers had earned a college degree (or beyond); 60% of the mothers and 94% of the fathers were employed; and 76% of the sample was Caucasian. The alpha reliability coefficient for the total scale when administered to the sample in the current study was 0.93. Alpha coefficients for the three subscales in this sample ranged from of 0.81 to 0.90.

Family Satisfaction Scale (FSS) The FSS (19) is a tool that assesses two of the three dimensions of the Circumplex Model of marital and family systems. It consists of 14 items which comprise two subscales: family cohesion and family adaptability. Family cohesion is defined as the emotional bonding that family members have toward one another. Family adaptability is defined as the ability of a marital or family system to change its power structure, role relationships, and relationship rules in response to situational and developmental stress. The Chronbach alpha coefficients are 0.92, 0.85, and 0.84 for the total scale, cohesion subscale, and adaptability subscale, respectively. Test-retest reliability coefficients of 0.75, 0.76, and 0.67 are reported for the total score, the cohesion subscale, and the adaptability subscale, respectively. The manual includes research regarding the construct validity of the instrument. Norms are available for both parents and adolescents.

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The alpha reliability coefficient for the FSS in this sample was 0.90. Alpha coefficients of 0.82 and 0.81 were found for the cohesion and adaptability subscales, respectively. Parental Concerns Questionnaire (PCQ) The PCQ is a nine-item instrument developed for use in this study. Six items measure the parent’s current concerns for their child regarding the psychological and social impact of having a PWS. For example, “How often are situations avoided because people’s reactions to the PWS may have a negative affect on your child?” and “How worried are you that your child’s physical appearance is currently being negatively affected by the PWS?” Three items measure the parent’s concerns regarding the child’s future should the PWS not be removed such as, “If the port-wine stain is not removed, how worried are you that your child’s future peer relationships will be negatively affected by the PWS?” A 7-point Likert scale (0 ⳱ never/not at all; 6 ⳱ always/extremely) is used to rate each item. The parent’s item responses are averaged to create a total score. The items of the PCQ are based on years of clinical experience with this population, on a review of the literature, and on unstructured interviews with parents whose children were already receiving treatment and were therefore not eligible for participation in this study. This instrument was piloted on four parents whose children were already receiving treatment. An alpha reliability coefficient of 0.80 was found for parents in this sample. Medical Information Form (MIF) The MIF is a 10-item instrument developed for use in this study. Parents respond to these questions immediately following their first consultation with the dermatologist. Parents use a Likert scale (0 ⳱ not at all; 6 ⳱ extremely) to respond to two items: how informed they are regarding PWSs, and how satisfied they feel regarding the quality of communication with medical staff. A multiple-choice format for eight items tests the parent’s acquisition of key facts regarding PWSs and laser surgery. A summary score reflects the responses to these eight items. The MIF has high face validity as the questions test the acquisition of facts regarding the etiology and treatment of PWSs. The dermatologist verbally explained this information and distributed a two-page written synopsis to the parents. The actual wording of the MIF items matches that in the written synopsis. Similarly the dermatologist was accustomed to using the same terminology during the parent consultation. This instrument was piloted on two parents whose children were already receiving treatment and were therefore ineligible for this study, and on three hospital staff members who were employed in other departments. The latter were first

given access to the written synopsis and then tested using the MIF. Child Behavior Checklist Two versions of the CBCL (20,21) are used to assess problem behaviors in children ages 2 to 3 years and 4 to 18 years. The former consists of 100 items and the latter consists of 118 items related to behavioral problems which are scored by parents on a 3-point scale ranging from “Not true” to “Often true.” Norms (T scores) are provided for children by gender and age. The instruments yield a total problem behavior score and two broad-band scores (internalizing and externalizing). Eight minorband factor scores can be computed: withdrawn, somatic complaints, anxious/depressed, social problems, thought problems, attention problems, delinquent behavior, and aggressive behavior. The CBCL is well standardized and the psychometric properties have been extensively evaluated and discussed in detail in the manuals. RESULTS T tests were used to compare mothers’ and fathers’ responses on each of the measures. The two groups did not differ on the total scores or on the subscale scores for the PSI-SF or the FSS. Nor did they differ on the total score of the PCQ or the MIF. Therefore the scores were pooled for further analyses. The means, standard deviations, and percentiles for the parents’ scores are presented in Table 2. Twelve of the 24 children in this study fell in the age range required for CBCL use. CBCL scores were obtained for 11 of the 12 eligible children. The children’s total problem behavior T scores fell in the average range (50%) (mean ⳱ 49.0; SD ⳱ 16.95). The T scores for only one child reached the “clinical” range. The parents in this study were quite similar to the normative group of the PSI in mother’s age, father’s age, child’s age, marital status, employment status, and number of children. There were moderate differences in educational level, however, the greatest difference was noted in family income, with the parents in this study reporting higher levels than parents in the normative group. All of the parents in this sample passed the protocol validity indicator, suggesting that they were not responding in a defensive manner. The parents in this study placed in the low-average to average range (30%– 40%) for parenting stress (Table 2), however, five parents met the criteria for clinically significant levels of stress, that is, they scored in the 91% or above (18) on one or more of the subscales. T tests and chi-square analyses were used to test for differences between these five parents and the other parents on the FSS, PCQ, CBCL, MIF, and demographic characteristics, including the family’s income, and the child’s age, gender, and

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TABLE 2. Means, Standard Deviations, and Percentiles for Parent Functioning Variable Parenting Stress Index-SF (n ⳱ 41) Parental distress Parent-child dysfunction Difficult child Total Family Satisfaction Scale (n ⳱ 40) Family cohesion Family adaptability Total Parental Concerns Questionnaire (n ⳱ 44) Current concerns Future concerns Total Medical Information Form (n ⳱ 31) Perceived level of knowledge Satisfaction with staff communication Acquisition of key facts

Mean

SD

Percentile

23.8 17.2 22.1 63.1

7.14 6.77 7.97 18.08

40 40 30 30

29.0 21.9 50.9

5.09 3.72 8.40

67 73 67

1.4 3.7 5.1

1.02 1.41 2.08

3.5 4.0 6.0

1.18 1.07 1.29

following the medical consultation (r ⳱ –0.33; p ⳱ 0.044); and parent’s satisfaction with staff communication (r ⳱ −0.47; p ⳱ 0.006). Results of preliminary stepwise regression analyses then identified four of these variables as the most salient predictors. They were entered into the final hierarchical regression model as follows: the demographic characteristic, child’s age, was entered first because it is a preexisting variable. Family satisfaction, parental concerns, and satisfaction with staff communication were entered next in the same step. The four variables accounted for a total of 49% of the variance in parenting stress (Table 3). The child’s age explained 5% of the variance, and the second set of variables accounted for an additional 44% of the variance. Increases in the children’s ages and increases in parental concerns were associated with increases in parenting stress, while higher levels of family satisfaction and greater satisfaction with parent-staff communication were associated with lower levels of stress.

diagnosis. Parents whose scores fell in the clinical range had significantly older children (mean ⳱ 4.1 years; SD ⳱ 0.83 years) than the other parents (mean ⳱ 2.1; SD ⳱ 2.17). There were proportionally more sons in the clinically stressed group (3, N ⳱ 41; ␹2 ⳱ 7.08; Fisher’s exact test p ⳱ 0.020) and more children diagnosed with Sturge–Weber/Klippel–Trenaunay syndromes (3, N ⳱ 41; ␹2 ⳱ 9.38; Fisher’s exact test p ⳱ 0.017). The levels of family satisfaction for all parent participants fell in the average to high-average range (67%–73%). A hierarchical regression analysis was used to investigate the impact of demographic characteristics, family satisfaction, parental concerns, and medical information variables on all of the parents’ level of stress. The independent variables were examined in order to create the most parsimonious regression equation. Only those variables with zero-order Pearson or point-biserial correlations which were significant at or below the 0.05 level were marked for inclusion in the regression model. These included child’s age (r ⳱ 0.40; p ⳱ 0.005); child’s diagnosis, that is, diagnosis of Sturge–Weber/Klippel– Trenaunay syndromes or not (rpb ⳱ 0.29; p ⳱ 0.035); family satisfaction (r ⳱ –0.41; p ⳱ 0.006); parental concerns (r ⳱ 0.27; p ⳱ 0.049); parent’s knowledge

DISCUSSION This study examines the levels of stress, family satisfaction, and concerns of parents of children undergoing

TABLE 3. Hierarchical Multiple Regression Analysis of Parenting Stress (n = 26) Predictors Demographics Child’s age Parent variables Family satisfaction(FSS) Parent concerns (PCQ) Staff Communication

Adj R2

F

p

R2 Change

0.05

2.43

0.132

0.05

0.49

7.29

0.001

0.44



p

p

0.132

0.34

0.031