The Cleft Palate–Craniofacial Journal 52(1) pp. 3–11 January 2015 Ó Copyright 2015 American Cleft Palate–Craniofacial Association
ORIGINAL ARTICLE Longitudinal Analysis of Parenting Stress in Mothers and Fathers of Infants With and Without Single-Suture Craniosynostosis Kristen E. Gray, Ph.D, Mary Michaeleen Cradock, Ph.D., Kathleen A. Kapp-Simon, Ph.D., Brent R. Collett, Ph.D., Lynette D. Pullmann, Ph.D., Matthew L. Speltz, Ph.D. Objective: To examine longitudinal differences in reported stress between parents of children with and without single-suture craniosynostosis and to compare the stress reports of mothers and fathers. Design: Multi-site, nonrandomized prospective study. Setting/Participants: Children with single-suture craniosynostosis (cases) were identified via referral of the treating surgeon or physician at the time of diagnosis, and those without singlesuture craniosynostosis (controls) were recruited from pediatric practices, birthing centers, and announcements in print media. When children were aged 6, 18, and 36 months (on average), mothers and fathers of children with and without single-suture craniosynostosis completed the Parenting Stress Index. For cases, 247 mothers and 211 fathers completed the Parenting Stress Index at the first visit; corresponding numbers for controls were 254 and 220, respectively. Main Outcome Measures: The Parenting Stress Index Parent and Child Domains and subscales scores. Results: We found few differences between parents of infants with and without single-suture craniosynostosis, regardless of parent gender. Irrespective of case status, mothers consistently reported higher stress than fathers on the Parent Domain. Within the Parent Domain, mothers reported more stress than fathers on the Role Restriction and Spousal Support subscales. Conclusions: The parents of children with single-suture craniosynostosis reported levels of stress similar to those reported by parents of same-aged, unaffected children. Mothers reported greater stress than fathers, and these differences remained remarkably stable over time. This may reflect widely held perceptions of gender differences in parenting roles. KEY WORDS: craniosynostosis
craniofacial condition, fathers, mothers, parenting stress, single-suture
Because parenting stress may undermine a child’s psychosocial adjustment, there has been great interest in understanding how various craniofacial conditions, including single-suture craniosynostosis (SSC), are related to parent functioning (e.g., Endriga and Kapp-Simon, 1999; Pope et al., 2005). Parents of infants with SSC experience many presumably stressful events, including the child’s unusual appearance and prolonged cranial surgery soon after the initial diagnosis (Marsh et al., 1991). Many parents also fear the possibility of future medical and developmental problems including headaches (Beck et al., 2002), surgical revisions (Marsh et al., 1991), and developmental delays (Speltz et al., 2004). Consideration of these and other potential stressors led to the recent recommendation by McCarthy et al. (2012) that services to address family coping, parent-child issues, and ‘‘barriers to medical and healthcare’’ (p. S3) be included in practice parameters for craniosynostosis. However, empirical data on observed or reported parental responses to the diagnosis of SCC are scant. Most studies of parenting stress in children with craniofacial anomalies have focused on orofacial clefts,
Ms. Gray is Senior Fellow, Department of Health Services, University of Washington, Seattle, Washington. Dr. Cradock is Director of Behavioral Health, Department of Psychology, St. Louis Children’s Hospital, and Assistant Professor, Department of Pediatrics, Washington University School of Medicine, St. Louis, Missouri. Dr. Kapp-Simon is Associate Professor, Department of Surgery, Northwestern University, and Licensed Clinical Psychologist, Pediatric Psychologist, Shriners Hospital for Children, Chicago, Illinois. Dr. Collett is Attending Psychologist, Psychiatry and Behavioral Medicine, Seattle Children’s Hospital, and Assistant Professor, Department of Psychiatry and Behavioral Sciences, University of Washington, Seattle, Washington. Dr. Pullmann is Clinical Assistant Professor, Department of Psychiatry and Behavioral Sciences, University of Washington, Seattle, Washington. Dr. Speltz is Attending Psychologist, Psychiatry and Behavioral Medicine, Seattle Children’s Hospital, and Professor, Department of Psychiatry and Behavioral Sciences, University of Washington, Seattle, Washington. This work was supported by the National Institute of Dental and Craniofacial Research (grant no. R01DE 13813). Submitted September 2013; Revised February 2014; Accepted March 2014. Address correspondence to: Dr. Matthew L. Speltz, Seattle Children’s Hospital, 4800 Sand Point Way NE, Mailstop CL-08, Seattle, WA 98105. E-mail [email protected]. DOI: 10.1597/13-239 3
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Cleft Palate–Craniofacial Journal, January 2015, Vol. 52 No. 1
with most lacking control groups and multiple assessments (Endriga and Kapp-Simon, 1999). In one of the only previous studies of the psychological impact of SSC on parents (Rosenberg et al., 2011), we assessed stress in mothers and fathers immediately following their infants’ diagnosis of SSC. Parents of same-aged infants without SSC served as a control group. We used the Parenting Stress Index (PSI; Abidin, 1995), a well-validated measure of psychological response to stress as well as events (‘‘stressors’’) that precede or are associated with stress reactions (we will use the term stress to connote both). The PSI distinguishes between parent-related stress (e.g., lack of spousal support) and child-related stress (e.g., the child placing many demands on the parent). We found minimal differences in child-related stress, and parent-related stress was slightly lower for parents of affected children, though statistically nonsignificant. Differences by parent gender, however, were striking: Regardless of a child’s SSC diagnosis, fathers reported higher child-related stress than mothers. Conversely, mothers reported higher parentrelated stress than fathers in such areas as health, role restriction, depression, and spousal support. This lack of presurgery case-control differences suggests that parents of children with SSC may be highly resilient in their reaction to their child’s initial diagnosis and treatment. Alternatively, parents may experience a suppressed or delayed stress response related to the effects of caring for an infant in the midst of an acute medical situation. For example, parents may pull together in response to a crisis, benefiting from temporary support from friends or the child’s medical team. Furthermore, concerns about developmental delays, which would not typically emerge clearly until the preschool years, may be a major source of stress for parents of children with SSC. Indeed, affected children have 1.5 to 2 times the risk of developmental delay compared with their unaffected peers (Starr et al., 2012), and 35% to 50% of school-aged children with SSC have adverse neurodevelopmental outcomes (Kapp-Simon et al., 2007). If this is the case, we would anticipate increasing stress among parents of cases and larger differences between case and control group participants at later assessments. A pattern of increasing stress in parents of children with various disabilities (Down syndrome, motor impairment, developmental delay) has been reported previously (Hauser-Cram et al., 2001), such that very few parents displayed clinically meaningful levels of stress prior to age 3 years but showed increases in the years afterward (.38% in the clinical range by age 10). This finding is consistent with theoretical expectations that parenting stress continually fluctuates in relation to the child’s developmental stages and the environmental demands placed on child and parents (Baker et al., 2003). This study examined the stability of parent stress over time in the same cohort, providing opportunities to address the above hypotheses (e.g., suppressed or delayed stress response). We examined parent stress at two visits subsequent to the affected child’s surgery, when children
were approximately aged 18 and 36 months. We hypothesized that parenting stress among cases would increase over time in relation to control parents, as the buffering effects of social support fade and other potential stressors emerge (e.g., child developmental delays). We also expected to observe the same parent gender pattern reported previously (i.e., with fathers reporting higher child-related stress and mothers reporting higher parent-related stress). Finally we examined the influence of parents’ ‘‘defensive’’ response bias on case-control group differences as measured by the PSI Defensive Responding scale, a measure of respondents’ tendency to present a favorable self-impression (Abidin, 1995). METHODS Participants Participants included parents of infants with SSC (cases) and demographically similar, unaffected infants (controls) enrolled between 2001 and 2006. We followed participants longitudinally and performed assessments at three time points (visit 1, 2, and 3). The study was approved by the institutional review boards at each of five participating centers (see Kapp-Simon et al., 2012). This research was in full compliance with Health Insurance Portability and Accountability Act standards. Cases Infants with SSC were referred to the project at the time of diagnosis by the treating surgeon or pediatrician. Eligibility criteria included (1) diagnosed SSC (sagittal, metopic, unilateral coronal, or unilateral lambdoid synostosis), confirmed on computed tomography scans; (2) participation prior to surgery; and (3) child age 30 months. Exclusions included (1) prematurity (,34 weeks’ gestation); (2) major medical or neurological conditions (e.g., cardiac defects, seizure disorders); (3) presence of three or more extracranial minor malformations (Leppig et al., 1987); or (4) presence of other major malformations. Among the 266 enrolled case patients, 247 biological mothers and 211 biological fathers completed the Parenting Stress Index (PSI) at visit 1. Of these, 220 (89.1%) and 179 (84.8%) of the same mothers and fathers completed the PSI at visit 2, respectively, and 182 mothers (73.7%) and 137 fathers (64.9%) at visit 3. A total of 176 mothers and 131 fathers completed the PSI at all three visits, and 131 participants had PSI protocols from both parents for all three visits. Controls Unaffected infants were eligible if they had no known craniofacial anomaly and met none of the exclusion
Gray et al., STRESS IN PARENTS OF CHILDREN WITH SINGLE-SUTURE CRANIOSYNOSTOSIS
criteria for cases. Control families were recruited through pediatric practices, birthing centers, and announcements in print media. Controls were group matched to cases on the following factors: (1) age at enrollment (63 weeks); (2) sex; (3) family socioeconomic status (SES; Hollingshead, 1975); and (4) race/ ethnicity. Among 259 eligible control patients, 254 biological mothers and 220 biological fathers completed the PSI at visit 1. Of these, 221 (87.0%) and 195 (88.6%) of the same mother and father respondents, respectively, completed the PSI at visit 2, and 201 mothers (79.0%) and 170 fathers (77%) at visit 3. A total of 188 mothers and 159 fathers completed the PSI at all three visits, and 157 controls had PSI data available for both parents across all three time periods.
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Case-Control and Parent Gender Differences
At visit 1, infants were evaluated following their diagnosis at a mean age of 9 months, on average 46.5 days prior to surgery. Target ages for visits 2 and 3 were 18 and 36 months, respectively. Children were developmentally tested at each visit and their parents completed the PSI during the visit or, when necessary, at home and returned it by mail. At visit 1, mothers were interviewed to obtain medical history and family constellation data.
To evaluate whether parents of children with SSC reported higher levels of stress than parents of unaffected children and whether mothers and fathers differed in their PSI reports, we used a generalized estimating equations approach (GEE; Liang and Zeger, 1986) to linear regression for each outcome at each of the three visits. Outcomes were the PSI Child and Parent Domain scores, as well as selected subscales based on observed case-control or mother-father differences (Rosenberg et al., 2011): Acceptability, Demandingness, and Distractibility subscales from the Child Domain and Role Restriction and Spousal Support from the Parent Domain. Only participants with the same eligible respondents from visit 1 were included in these analyses (e.g., the same biological mother or father). All regression models were adjusted for matching variables, including SES (continuous; Hollingshead, 1975), child age at visit (months), race (white versus nonwhite), patient gender, and study site. We included an interaction term between case status and respondent (mother versus father), which allowed for examination of both case-control and father-mother differences within the same model. The P value of the interaction terms in these models indicated whether case-control differences varied by respondent and whether motherfather differences varied by case status. Regardless of statistical significance, stratified results were reported to provide a more complete picture of the findings. To facilitate comparisons across outcomes with different distributions (e.g., the domain and subscale scores), we calculated standardized differences between cases and controls and mothers and fathers, using control mothers’ mean and standard deviation of each outcome at each visit as the reference for standardization. Results are presented as standardized differences. To evaluate whether defensive responding influenced case-control differences, we repeated the above regressions for the Child and Parent Domain scores, excluding all respondents who met the PSI manual’s definition of excessively biased reporting (i.e., scores 24 on the Defensive Responding scale; Abidin, 1995). To examine whether the odds of defensive responding differed between cases and controls at visit 1, we performed logistic regression adjusting for SES, race, and gender, including an interaction term between case status and respondent.
Data Analysis
Change Over Time
We examined demographic characteristics separately for cases and controls and among cases within each affected suture site. We also compared demographic characteristics and visit 1 PSI scores of participants with PSI scores at visit 3 versus those lost to follow-up.
To determine whether case-control differences changed over time, we performed linear regression separately for mothers and fathers, using GEE with an interaction between case status and visit. To evaluate whether mother-father differences changed over time,
Measures Parenting Stress Index The PSI (Abidin, 1995) assesses the presence or intensity of potential stress/stressors in the family environment. The Parent Domain score summarizes parents’ personal characteristics and social support as they relate to the demands and tasks of parenting. Subscales include Depression, Attachment to Child, Role Restriction, Competence, Isolation, Health, and Spousal Support. The Child Domain score summarizes stress related to child characteristics or temperament, represented by the following subscales: Adaptability, Acceptability, Demandingness, Mood, Distractibility, and Reinforces Parent. Higher scores on all scales reflect higher levels of stress. Test-retest reliability data from several studies provide values ranging from .55 to .82 for Child Domain scores and .69 to .91 for the Parent Domain (Abidin, 1995). Procedure
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Cleft Palate–Craniofacial Journal, January 2015, Vol. 52 No. 1
TABLE 1
Demographic Characteristics of Single-Suture Craniosynostosis Cases and Controls at Visit 1 Cases Controls
Total
Sagittal
Metopic
Unicoronal
Lambdoid
n
%
n
%
n
%
n
%
n
%
n
%
Total
256
100.0
249
100.0
115
100.0
58
100.0
63
100.0
13
100.0
Gender Female Male
93 163
36.3 63.7
90 159
36.1 63.9
26 89
22.6 77.4
16 42
27.6 72.4
42 21
66.7 33.3
6 7
46.2 53.8
Age, mo ,6 6–9 9þ
111 73 72
43.4 28.5 28.1
121 62 66
48.6 24.9 26.5
77 15 23
67.0 13.0 20.0
23 18 17
39.7 31.0 29.3
19 23 21
30.2 36.5 33.3
2 6 5
15.4 46.2 38.5
Race Nonwhite White
75 181
29.3 70.7
63 186
25.3 74.7
27 88
23.5 76.5
17 41
29.3 70.7
16 47
25.4 74.6
3 10
23.1 76.9
Socioeconomic status 1 (high) 2 3 4 5 (low)
70 127 34 16 9
27.3 49.6 13.3 6.3 3.5
58 84 58 33 16
23.3 33.7 23.3 13.3 6.4
27 41 22 20 5
23.5 35.7 19.1 17.4 4.3
12 20 14 8 4
20.7 34.5 24.1 13.8 6.9
14 19 19 5 6
22.2 30.2 30.2 7.9 9.5
5 4 3 0 1
38.5 30.8 23.1 0.0 7.7
93 97 28 38
36.3 37.9 10.9 14.8
89 91 34 35
35.7 36.5 13.7 14.1
42 28 23 22
36.5 24.3 20.0 19.1
21 24 3 10
36.2 41.4 5.2 17.2
21 34 6 2
33.3 54.0 9.5 3.2
5 5 2 1
38.5 38.5 15.4 7.7
Marital status Single Married/cohabitating
22 232
8.6 90.6
21 227
8.4 91.2
12 103
10.4 89.6
2 55
3.4 94.8
6 57
9.5 90.5
1 12
7.7 92.3
Children in household 1 2 3 4þ
116 88 37 13
45.3 34.4 14.5 5.1
86 93 43 26
34.5 37.3 17.3 10.4
43 40 19 12
37.4 34.8 16.5 10.4
22 23 8 5
37.9 39.7 13.8 8.6
18 25 12 8
28.6 39.7 19.0 12.7
3 5 4 1
23.1 38.5 30.8 7.7
Adults in household 1 2 3þ
9 227 18
3.5 88.7 7.0
12 212 24
4.8 85.1 9.6
5 101 8
4.3 87.8 7.0
3 44 11
5.2 75.9 19.0
4 56 3
6.3 88.9 4.8
0 11 2
0.0 84.6 15.4
Site Seattle Chicago St. Louis Atlanta
for each participant at each visit we calculated the difference between mothers’ and fathers’ scores (father minus mother). Using these difference scores as the outcome and visit as the exposure, we then performed linear regression using GEE. We included the visit-bycase status interaction to examine whether change over time differed between cases and controls. All longitudinal analyses were adjusted for SES, age at first visit, race, gender, and study site. Only participants with the same respondents across time were included. For change over time in father-mother differences, only parents maintaining the same marital status were included because any observed changes in differences could otherwise be attributable to changing marital status.
Controls were slightly more likely than cases to be of white race and to report higher SES. For cases, mean age at surgery was 8.8 months (range, 1.8 to 34.1 months). Compared with participants at visit 3, cases and controls lost to follow-up were more likely to be girls (40% of controls, 43% of cases) and of lower SES (22% of controls and 34% of cases in the two lowest SES categories) (data not shown). Cases lost to follow-up were also more likely to be nonwhite (36%). Average PSI scores at visit 1 were similar between participants and nonparticipants for mothers and fathers (i.e., absolute differences 3.5 points). Case-Control Group Differences Child Domain
RESULTS Cases and controls had similar distributions of gender, age at visit 1, study site, and marital status (Table 1).
Case mothers reported higher Child Domain scores than control mothers at visit 1 (standardized difference [std diff] ¼ 0.2; 95% confidence interval [CI], 0.0 to 0.4),
Gray et al., STRESS IN PARENTS OF CHILDREN WITH SINGLE-SUTURE CRANIOSYNOSTOSIS
but differences at visits 2 and 3 were negligible (std diff ¼ 0.0) (Table 2). Case fathers also reported higher Child Domain scores than control fathers at visit 1 (std diff ¼ 0.2; 95% CI, 0.0 to 0.4), but these differences diminished and were negligible at visits 2 and 3 (std diff 0.1). Parent Domain Case mothers consistently reported lower Parent Domain scores than control mothers, though these differences were small and imprecise, with std diff ranging from 0.1 (95% CI, 0.3 to 0.1) to 0.2 points (95% CI, 0.3 to 0.0). Case-control differences in fathers were similar, with std diff ranging from 0.1 (95% CI 0.3, 0.1) to 0.2 (95% CI 0.3, 0.1). Subscales Across all visits, case mothers and fathers reported slightly higher scores than controls on the Acceptability and Demandingness subscales, with std diff ranging from 0.1 to 0.5 for mothers and 0.1 to 0.4 for fathers, but these differences decreased over time (Table 2). Differences on the Distractibility subscale were negligible for both mothers and fathers. Among mothers and fathers, cases reported slightly lower scores on the Role Restriction and Spousal Support subscales than controls across visits (mothers’ std diff ¼0.1 to 0.3; fathers’ std diff ¼ 0.1 to 0.2), although these differences were small and often imprecise. Defensive Responding Across the three visits, 17.6% to 20.2% of case mothers were categorized as ‘‘defensive’’ compared with 9.5% to 12.2% of control mothers. Defensive responding was similar in case and control fathers (20.4% to 24.6% for cases, 20.0% to 23.6% controls). After removing defensive responders, case-control differences among mothers on the Child Domain became slightly larger (std diff ¼ 0.0 [95% CI, 0.2 to 0.2] to 0.3 [95% CI, 0.1 to 0.5]) but aside from visit 1 were mostly small and imprecise; differences on the Parent Domain were negligible (std diff 0.1j; data not shown). When removing defensively responding fathers, all casecontrol differences were small and imprecise (std diff ¼ 0.0 [95% CI, 0.2 to 0.2] to 0.2 [95% CI, 0.0 to 0.4]).
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Father-Mother Differences Child Domain Among cases, fathers reported higher scores than mothers on the Child Domain at visit 1 (std diff ¼ 0.3; 95% CI, 0.1 to 0.4), but both the magnitude and precision of these differences decreased thereafter (visit 2 std diff ¼ 0.1; 95% CI, 0.0 to 0.2; visit 3 std diff ¼ 0.0; 95% CI, 0.2 to 0.3) (Table 2). Among controls, differences were less consistent, with fathers reporting higher scores than mothers at visit 1 (std diff ¼ 0.3; 95% CI, 0.2 to 0.4) but negligible differences at visits 2 and 3 (std diff ¼ 0.0). Parent Domain Among both cases and controls and at all visits, fathers had lower Parent Domain scores than mothers, with standardized differences ranging from 0.1 (95% CI, 0.3 to 0.0) to 0.3 (95% CI, 0.5 to 0.2). The differences were most pronounced at visit 3 for both cases and controls. Subscales Father-mother differences on Child Domain subscales were negligible for cases and controls (Table 2). However, in both groups, fathers reported lower stress than mothers on the Parent Domain subscales Role Restriction (std diff ¼0.2 [95% CI, 0.3 ¼0.1] to 0.4 [95% CI, 0.6 to 0.2]) and Spousal Support (std diff ¼ 0.3 [95% CI, 0.4 to 0.2] to 0.5 [95% CI, 0.7 to 0.3]). Change Over Time Among cases, father-mother differences in the Child and Parent Domain scores decreased by 1.5 points for each visit (Child Domain 95% CI, 2.9 to 0.1; Parent Domain 95% CI, 3.3 to 0.3) (Table 3). In controls, the differences decreased by 1.9 (95% CI, 3.2 to 0.6) points for each visit on the Child Domain and by 0.5 (95% CI, 2.0 to 1.0) points on the Parent Domain. Thus, father-mother differences became less apparent on the Child Domain and more apparent on the Parent Domain. Interactions between case status and visit were not statistically significant for either the Child or Parent Domain (P ¼ .66 and .40, respectively).
Changes Over Time Interaction Between Case Status and Respondent Case-control differences for mothers decreased by 0.8 points at each subsequent visit for the Child Domain and increased by 0.7 points for the Parent Domain at each visit (Table 3). Case-control difference among fathers remained essentially unchanged over time.
None of the interactions between case status and respondent (mother versus father) were statistically significant when using the domain scores as outcomes in cross-sectional analyses (P values, .32 to .97).
Controls
96.5 (20.1) 96.7 (18.1) 95.3 (17.7) 95.0 (16.7) 114.6 (23.9) 107.2 (20.2) 116.7 (19.4) 109.9 (19.9)
12.0 18.2 23.4 17.6 17.5
Subscales Acceptability Demandingness Distractibility Role Restriction Spousal Support
12.0 17.7 23.7 16.1 15.2
(3.4) (4.6) (4.5) (4.3) (4.1)
(3.9) (4.6) (4.2) (4.2) (4.1)
11.6 17.3 23.3 18.5 18.1
11.5 16.8 25.1 18.1 17.9
(2.9) (4.5) (4.7) (4.1) (4.3)
(3.2) (4.1) (4.3) (4.1) (4.7)
11.5 16.7 23.3 16.9 16.1
11.5 16.2 24.2 16.8 15.8
(2.9) (4.2) (4.2) (4.2) (4.2)
(3.2) (3.9) (4.2) (4.3) (4.3)
0.1 0.2 0.0 0.2 0.1
0.0 0.2
0.1 0.2 0.2 0.1 0.2
0.0 0.1
0.5 0.5 0.1 0.3 0.2
0.2 0.2 to to to to to
0.7 0.6 0.1 0.1 0.0
to to to to to
0.3 0.4 0.0 0.2 0.0
0.1 0.0 0.2 0.5 0.3 to to to to to
0.3 0.4 0.2 0.0 0.1
0.2 to 0.3 0.4 to 0.1
0.1 0.1 0.3 0.3 0.4
0.2 to 0.2 0.3 to 0.1
0.3 0.3 0.2 0.5 0.4
0.0 to 0.4 0.3 to 0.0
.34 .08 .99 .04 .29
.70 .18
.37 .01 .11 .60 .10
.99 .44
,.001 ,.001 .40 .004 .02
.03 .09
0.2 0.2 0.1 0.2 0.2
0.0 0.2
0.1 0.4 0.1 0.2 0.1
0.1 0.1
0.4 0.4 0.1 0.1 0.1
0.2 0.1 to to to to to
0.6 0.6 0.0 0.1 0.1
to to to to to
0.3 0.6 0.3 0.0 0.1
0.1 0.0 0.1 0.4 0.4
to to to to to
0.4 0.4 0.3 0.0 0.0
0.2 to 0.3 0.4 to 0.1
0.1 0.2 0.1 0.4 0.3
0.1 to 0.3 0.3 to 0.1
0.1 0.3 0.3 0.3 0.2
0.0 to 0.4 0.3 to 0.1
95% CI
95% CI‡
P Value Std Diff
Fathers
Mothers
Case-Control Differences
.20 .05 .56 .06 .09
.66 .18
.50 ,.001 .42 .09 .16
.31 .31
.002 ,.001 .10 .19 .38
.07 .49
0.0 0.1 0.1 0.3 0.5
0.0 0.3
0.0 0.0 0.1 0.4 0.4
0.1 0.2
0.2 0.0 0.1 0.2 0.3
0.3 0.1
P Value Std Diff
to to to to to
0.4 0.2 0.2 0.1 0.2
to to to to to
0.2 0.2 0.2 0.2 0.2
0.1 0.2 0.1 0.5 0.7
to to to to to
0.2 0.1 0.3 0.2 0.3
0.1 to 0.2 0.5 to 0.1
0.1 0.2 0.1 0.6 0.5
0.0 to 0.2 0.4 to 0.1
0.0 0.1 0.1 0.3 0.4
0.1 to 0.4 0.3 to 0.0
95% CI
Cases
.59 .33 .19 ,.001 ,.001
.58 ,.001
.77 .93 .32 ,.001 ,.001
.16 .01
.04 .71 .44 .01 ,.001
,.001 .06
0.0 0.1 0.0 0.4 0.4
0.0 0.3
0.0 0.1 0.2 0.3 0.4
0.0 0.2
0.4 0.1 0.1 0.3 0.5
0.3 0.2
P Value Std Diff
to to to to to
0.5 0.2 0.3 0.2 0.3
to to to to to
0.2 0.0 0.0 0.1 0.3
0.2 0.2 0.1 0.5 0.6
to to to to to
0.2 0.0 0.2 0.2 0.3
0.1 to 0.2 0.5 to 0.2
0.1 0.3 0.3 0.5 0.6
0.1 to 0.1 0.4 to 0.0
0.2 0.1 0.0 0.5 0.6
0.2 to 0.4 0.4 to 0.1
95% CI
Controls
Father-Mother Differences
* M ¼ mothers; F ¼ fathers; CI ¼ confidence interval; SD ¼ standard deviation; std diff ¼ standardized difference. † All standardized differences adjusted for socioeconomic status, age at visit, race (white versus nonwhite), gender, and study site; based on distribution of control mothers’ scores at each visit. ‡ All 95% confidence intervals calculated using robust standard error estimates and unstructured correlation matrix. § Case mothers: all n 244; case fathers: all n 210; control mothers: all n 251; control fathers: all n 221. || Case mothers: all n 217; case fathers: all n 179; control mothers: all n 221; control fathers: all n 194. } Case mothers: all n ¼ 182; case fathers: all n¼137; control mothers: all n ¼ 201; control fathers: all n ¼ 170.
(3.9) (5.0) (5.1) (4.9) (4.9)
12.1 18.1 25.0 16.3 15.2
(3.1) (3.7) (3.6) (4.2) (4.0)
Visit 3} Child Domain Parent Domain
(4.1) (4.8) (4.4) (5.0) (5.4)
11.4 15.7 24.8 16.9 15.5
12.0 18.1 24.7 18.0 17.1
(2.8) (4.1) (4.1) (4.3) (5.1)
Subscales Acceptability Demandingness Distractibility Role Restriction Spousal Support
10.4 15.6 24.5 18.5 18.0
96.8 (18.6) 98.3 (17.6) 95.5 (16.6) 94.4 (15.8) 113.7 (24.5) 109.0 (21.4) 114.3 (20.3) 109.3 (21.5)
(3.7) (3.9) (3.9) (4.3) (4.0)
Visit 2|| Child Domain Parent Domain
12.4 17.5 24.5 16.5 15.1
11.9 17.5 24.4 17.4 16.9
Subscales Acceptability Demandingness Distractibility Role Restriction Spousal Support
(3.7) (4.4) (4.4) (4.9) (5.1)
93.9 (18.3) 97.6 (16.7) 90.3 (14.8) 94.1 (15.0) 113.0 (22.8) 109.5 (21.4) 115.6 (20.5) 109.9 (19.4)
M F M F Mean (SD) Mean (SD) Mean (SD) Mean (SD) Std Diff†
Cases
Differences in Parenting Stress Index Scores Between Parents of Infants With and Without Single-Suture Craniosynostosis and Between Mothers and Fathers*
Visit 1§ Child Domain Parent Domain
TABLE 2
.93 .13 .51 ,.001 ,.001
.61 ,.001
.61 .14 .02 .001 ,.001
.96 .02
,.001 .33 .10 ,.001 ,.001
,.001 .001
P Value
8 Cleft Palate–Craniofacial Journal, January 2015, Vol. 52 No. 1
Gray et al., STRESS IN PARENTS OF CHILDREN WITH SINGLE-SUTURE CRANIOSYNOSTOSIS
9
TABLE 3 Changes Over Time in Differences Between Parents of Infants With and Without Single-Suture Craniosynostosis and Between Mothers and Fathers* Change in Difference for Each Visit Case-Control Differences Mothers
Child Domain Parent Domain
Father-Mother Differences Fathers
Cases
Controls
Change†
95% CI‡
P Value
Change
95% CI
P Value
Change
95% CI
P Value
Change
95% CI
P Value
0.8 0.7
2.5 to 0.9 1.0 to 2.4
.33 .42
0.2 0.2
2.0 to 1.6 2.1 to 1.7
.84 .84
1.5 1.5
2.9 to 0.1 3.3 to 0.3
.04 .10
1.9 0.5
3.2 to 0.6 2.0 to 1.0
.004 .50
* CI ¼ confidence interval. † All change values adjusted for socioeconomic status, age at first visit, race (white versus nonwhite), gender, and study site. ‡ All 95% confidence intervals with robust standard error estimates and unstructured correlation matrix.
DISCUSSION Case-Control Group Differences We found no persisting differences in PSI scores between parents of infants with and without SSC, regardless of parent gender. We anticipated higher stress reports among parents of infants with SSC for the reasons listed previously (e.g., unusual child appearance, the growing potential for associated medical complications and/or developmental delays), as well as findings from previous studies of parent stress in diagnostically heterogeneous samples of children with craniofacial disorders (Pope et al., 2005). Why then did we not find differences? First, SSC may have little relative impact on infant and family psychological functioning in the early years of life compared with the generally stressful events that surround the birth and early years of any child. Previous studies comparing the reported stress of parents with and without children with various disabilities or health problems have generated mixed findings. Although many have observed higher stress among parents of affected children (Fuller and Rankin, 1994; Pelchat et al., 1999; Ong et al., 2001; Oelofsen and Richardson, 2006; Gupta, 2007; Ong et al., 2011), several have reported null findings (Pelchat et al., 1999; Singer et al., 1999; Gupta, 2007; Collett et al., 2011; Treyvaud et al., 2011; Cloonan et al., 2012; Gray et al., 2012). Such inconsistency is not surprising, given the wide variation in sample demographics and ascertainment methods as well as the many different child health problems and disabilities under study. Another possibility is that the multidisciplinary, family-centered care typically received by infants and children with SSC mitigated the stressors associated with this diagnosis and surgical treatment. In the family adjustment and adaptation response model (Patterson, 1998), a family crisis occurs when the demands on the family exceed the capabilities of the family (e.g., a diagnosis of SSC), necessitating changes in the resources or coping behaviors of the family, ultimately resulting in adjustment to the crisis. These resources can emerge at
the individual, family, or community level, the latter including medical and other health care providers/ facilities. Recent care guidelines for craniosynostosis emphasize the importance of coordinated, multidisciplinary care that cuts across specialties (e.g., anesthesiology, neurosurgery, craniofacial surgery, psychology, speech-language pathology) and embraces family-centered care (McCarthy et al., 2012). All of the craniofacial teams in this study embraced this model of care, which may have served as a community-level resource that helped families to better cope with the typically stressful events surrounding their child’s medical condition. Indeed, social support has been observed to predict stress in parents of children with disabilities or serious medical conditions (Hauser-Cram et al., 2001; Shin and Nhan, 2009; Singer et al., 2010; Jeong et al., 2013) and without them (Ostberg and Hagekull, 2000). The lack of case-control differences may also be attributable to the study sample characteristics. Both case and control parents were generally affluent, white, and married. Because the study required multiple visits over time, retained families may have been more stable than those from the general population, further contributing to their lower risk. Additionally, parents received feedback on their child’s cognitive and motor development after each visit. If delays were detected, parents were encouraged to consult with their child’s pediatrician to determine whether further assessment or intervention was needed. Ultimately, 26% of cases and 15% of controls received some form of developmental intervention by visit 3, which may have provided additional support, minimizing case-control group differences. Another possibility is that case-control group differences were attenuated by parent response bias, which we assessed with the PSI’s Defensiveness Responding scale. Mothers of children with SSC were about twice as likely as control group mothers to meet the PSI’s definition of defensive responding. However, their removal from analyses had little effect on overall case-control group differences. A final possibility is that elevated parenting stress is potentially observable only among parents of infants
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Cleft Palate–Craniofacial Journal, January 2015, Vol. 52 No. 1
with more severe, multi-suture forms of craniosynostosis and related genetic syndromes (e.g., Apert or Crouzon syndromes; Wilkie et al., 1995).
small proportion of single parents, who tend to report elevated parenting stress regardless of gender (Skari et al., 2002).
Father-Mother Differences
Clinical Implications
We observed remarkably consistent differences between mothers and fathers across time, regardless of whether the child was affected by SSC. A number of studies have reported higher stress in mothers than fathers of children with various health problems; however, few have examined whether this pattern is shown by parents of healthy children. In one study of psychological responses after the birth of a healthy infant, mothers reported higher levels of insomnia, anxiety, and other negative emotions than fathers (Skari et al., 2002). Many of these differences dissipated during infancy, but reemerged when evaluated 5 years later (Skreden et al., 2008). The finding of father-mother differences of similar magnitude in both case and control parents suggests that these differences were unrelated to having a child with a health problem but instead reflected underlying gender differences in parenting experiences. In particular, mothers reported greater stress related to role restriction and spousal support, which may be attributable to traditional parenting roles in which mothers serve as the primary caregiver and fathers as the provider (Pelchat et al., 2007). Similar to mothers of typically developing children, mothers of children with disabilities tend to assume more responsibility for the care of their child and to express greater dissatisfaction with the division of tasks (Pelchat et al., 2007). This likely contributed to the elevated stress reports we observed in both case and control group mothers. Therefore, results suggest that levels of reported stress in one parent cannot be generalized to the other parent. Mother and father reports of stress should be treated separately in future studies of family responses to children’s health problems or other stressors.
Although the PSI did not reveal differences between cases and controls, within-group variation in PSI scores among parents of cases may be predictive of subsequent child outcomes. This question has not yet been addressed in analyses of this cohort, though previous studies of other craniofacial disorders (e.g., oral clefts) have found the PSI in infancy to have good predictive validity to later psychosocial outcomes (Krueckeberg and Kapp-Simon, 1993; Speltz et al., 1997; Pope et al., 2005). Although findings suggest that on average, parents of infants with SSC are doing about as well as parents of typically developing babies, the identification of individual parents within the SSC population with relatively high levels of reported stress could lead to interventions that mitigate later childhood problems, interventions like those recommended by the McCarthy et al. (2012) practice parameters. As a next step, we will examine models predicting school-age outcomes for children with SSC, which will include earlier scores from the PSI as potential predictors.
Strengths and Limitations The strengths of the study include the use of a control group with which parents of children with SSC were compared, rather than using norms from the PSI. In addition, we assessed parental stress at multiple points in time—both before and after corrective surgery— allowing for examination of changes over time. Last, unlike earlier studies that focused only on mothers, we also included fathers, allowing for father-mother as well as case-control comparisons. The limitations of this study include constrained demographic representation (mostly white parents of high SES), limiting generalizability to nonwhite or lower SES populations. An additional factor limiting generalizability is the sample’s
Acknowledgment. We would like to thank Joseph Picciano, Ph.D., for his assistance with this project.
REFERENCES Abidin RR. The Parenting Stress Index Professional Manual. Odessa: Psychological Assessment Resources; 1995. Baker L, McIntyre LL, Blacher J, Crnic K, Edelbrock C, Low C. Preschool children with and without developmental delay: behaviour problems and parenting stress over time. J Intellect Disabil Res. 2003;47:217–230. Beck J, Parent A, Angel MF. Chronic headache as a sequela of rigid fixation for craniosynostosis. J Craniofac Surg. 2002;13:327–330. Cloonan YK, Collett BR, Speltz ML, Anderka M, Werler MM. Psychosocial outcomes in children with and without non-syndromic craniosynostosis: findings from two studies. Cleft Palate Craniofac J. 2013;50:406–413. Collett BR, Cloonan YK, Speltz ML, Anderka M. Psychosocial functioning in children with and without orofacial clefts and their parents. Cleft Palate Craniofac J. 2011;49:397–405. Endriga MC, Kapp-Simon KA. Psychological issues in craniofacial care: state of the art. Cleft Palate Craniofac J. 1999;36:3–11. Fuller GB, Rankin RE. Differences in levels of parental stress among mothers of learning disabled, emotionally impaired, and regular school children. Percep Mot Skills. 1994;78:583–592. Gray PH, Edwards DM, O’Callaghan MJ, Cuskelly M. Parenting stress in mothers of preterm infants during early infancy. Early Hum Dev. 2012;88:45–49. Gupta V. Comparison of parenting stress in different developmental disabilities. J Dev Phys Disabil. 2007;19:417–425. Hauser-Cram P, Warfied ME, Shonkoff JP, Krauss MW, Sayer A, Upshur CC. Children with disabilities: a longitudinal study of child
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Pelchat D, Ricard N, Bouchard JM, Perreault M, Saucier JF, Berthiaume M, Bisson J. Adaptation of parents in relation to their 6-month-old infant’s type of disability. Child Care Health Dev. 1999;25:377–397. Pope AW, Tillman K, Snyder HT. Parenting stress in infancy and psychosocial adjustment in toddlerhood: a longitudinal study of children with craniofacial anomalies. Cleft Palate Craniofac J. 2005;42:556–559. Rosenberg JM, Kapp-Simon KA, Starr JR, Cradock M, Speltz ML. Mothers’ and fathers’ reports of stress in families of infants with and without single-suture craniosynostosis. Cleft Palate Craniofac J. 2011;8:509–518. Shin JY, Nhan NV. Predictors of parenting stress among Vietnamese mothers of young children with and without cognitive delay. J Intellect Dev Disabil. 2009;34:17–26. Singer LT, Salvator A, Guo S, Collin M, Lilien L, Baley J. Maternal psychological distress and parenting stress after the birth of a very low-birth-weight infant. J Am Med Assoc. 1999;281:799–805. Singer LT, Fulton S, Kirchner HL, Eisengart S, Lewis B, Short E, Min MO, Satayathum S, Kercsmar C, Baley JE. Longitudinal predictors of maternal stress and coping after very low-birth-weight birth. Arch Pediatr Adolesc Med. 2010;164:518–524. Skari H, Skreden M, Malt UF, Dalholt M, Ostensen AB, Egeland T, Emblem R. Comparative levels of psychological distress, stress symptoms, depression and anxiety after childbirth—a prospective population-based study of mothers and fathers. Br J Obstet Gynaecol. 2002;109:1154–1163. Skreden M, Skari H, Bjork M, Malt U, Veenstra M, Faugli A, ¨ Emblem R. Psychological distress in mothers and fathers of preschool children: a 5-year follow-up study after birth. Br J Obstet Gynaecol. 2008;115:462–471. Speltz ML, Endriga MC, Fisher PA, Mason CA. Early predictors of attachment in infants with cleft lip and/or palate. Child Dev. 1997;68:12–25. Speltz ML, Kapp-Simon K, Cunningham M, Marsh J, Dawson G. Single-suture craniosynostosis: a review of neurobehavioral research and theory. J Pediatr Psychol. 2004;29:651–668. Starr JR, Collett BR, Gaither R, Kapp-Simon KA, Cradock MM, Cunningham ML, Speltz ML. Multicenter study of neurodevelopment in 3-year-old children with and without single-suture craniosynostosis. Arch Pediatr Adolesc Med. 2012;166:536–542. Treyvaud K, Doyle LW, Lee KJ, Roberts G, Cheong JL, Inder TE, Anderson PJ. Family functioning, burden and parenting stress 2 years after very preterm birth. Early Hum Dev. 2011;87:427–431. Wilkie AO, Slaney SF, Oldridge M, Poole MD, Ashworth GJ, Hockley AD, Hayward RD, David DJ, Pulleyn LJ, Rutland P. Apert syndrome results from localized mutations of FGFR2 and is allelic with Crouzon syndrome. Nat Genet. 1995;9:165–172.
ORIGINAL ARTICLE Longitudinal Analysis of Parenting Stress in Mothers and Fathers of Infants With and Without Single-Suture Craniosynostosis Kristen E. Gray, Ph.D, Mary Michaeleen Cradock, Ph.D., Kathleen A. Kapp-Simon, Ph.D., Brent R. Collett, Ph.D., Lynette D. Pullmann, Ph.D., Matthew L. Speltz, Ph.D. Objective: To examine longitudinal differences in reported stress between parents of children with and without single-suture craniosynostosis and to compare the stress reports of mothers and fathers. Design: Multi-site, nonrandomized prospective study. Setting/Participants: Children with single-suture craniosynostosis (cases) were identified via referral of the treating surgeon or physician at the time of diagnosis, and those without singlesuture craniosynostosis (controls) were recruited from pediatric practices, birthing centers, and announcements in print media. When children were aged 6, 18, and 36 months (on average), mothers and fathers of children with and without single-suture craniosynostosis completed the Parenting Stress Index. For cases, 247 mothers and 211 fathers completed the Parenting Stress Index at the first visit; corresponding numbers for controls were 254 and 220, respectively. Main Outcome Measures: The Parenting Stress Index Parent and Child Domains and subscales scores. Results: We found few differences between parents of infants with and without single-suture craniosynostosis, regardless of parent gender. Irrespective of case status, mothers consistently reported higher stress than fathers on the Parent Domain. Within the Parent Domain, mothers reported more stress than fathers on the Role Restriction and Spousal Support subscales. Conclusions: The parents of children with single-suture craniosynostosis reported levels of stress similar to those reported by parents of same-aged, unaffected children. Mothers reported greater stress than fathers, and these differences remained remarkably stable over time. This may reflect widely held perceptions of gender differences in parenting roles. KEY WORDS: craniosynostosis
craniofacial condition, fathers, mothers, parenting stress, single-suture
Because parenting stress may undermine a child’s psychosocial adjustment, there has been great interest in understanding how various craniofacial conditions, including single-suture craniosynostosis (SSC), are related to parent functioning (e.g., Endriga and Kapp-Simon, 1999; Pope et al., 2005). Parents of infants with SSC experience many presumably stressful events, including the child’s unusual appearance and prolonged cranial surgery soon after the initial diagnosis (Marsh et al., 1991). Many parents also fear the possibility of future medical and developmental problems including headaches (Beck et al., 2002), surgical revisions (Marsh et al., 1991), and developmental delays (Speltz et al., 2004). Consideration of these and other potential stressors led to the recent recommendation by McCarthy et al. (2012) that services to address family coping, parent-child issues, and ‘‘barriers to medical and healthcare’’ (p. S3) be included in practice parameters for craniosynostosis. However, empirical data on observed or reported parental responses to the diagnosis of SCC are scant. Most studies of parenting stress in children with craniofacial anomalies have focused on orofacial clefts,
Ms. Gray is Senior Fellow, Department of Health Services, University of Washington, Seattle, Washington. Dr. Cradock is Director of Behavioral Health, Department of Psychology, St. Louis Children’s Hospital, and Assistant Professor, Department of Pediatrics, Washington University School of Medicine, St. Louis, Missouri. Dr. Kapp-Simon is Associate Professor, Department of Surgery, Northwestern University, and Licensed Clinical Psychologist, Pediatric Psychologist, Shriners Hospital for Children, Chicago, Illinois. Dr. Collett is Attending Psychologist, Psychiatry and Behavioral Medicine, Seattle Children’s Hospital, and Assistant Professor, Department of Psychiatry and Behavioral Sciences, University of Washington, Seattle, Washington. Dr. Pullmann is Clinical Assistant Professor, Department of Psychiatry and Behavioral Sciences, University of Washington, Seattle, Washington. Dr. Speltz is Attending Psychologist, Psychiatry and Behavioral Medicine, Seattle Children’s Hospital, and Professor, Department of Psychiatry and Behavioral Sciences, University of Washington, Seattle, Washington. This work was supported by the National Institute of Dental and Craniofacial Research (grant no. R01DE 13813). Submitted September 2013; Revised February 2014; Accepted March 2014. Address correspondence to: Dr. Matthew L. Speltz, Seattle Children’s Hospital, 4800 Sand Point Way NE, Mailstop CL-08, Seattle, WA 98105. E-mail [email protected]. DOI: 10.1597/13-239 3
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with most lacking control groups and multiple assessments (Endriga and Kapp-Simon, 1999). In one of the only previous studies of the psychological impact of SSC on parents (Rosenberg et al., 2011), we assessed stress in mothers and fathers immediately following their infants’ diagnosis of SSC. Parents of same-aged infants without SSC served as a control group. We used the Parenting Stress Index (PSI; Abidin, 1995), a well-validated measure of psychological response to stress as well as events (‘‘stressors’’) that precede or are associated with stress reactions (we will use the term stress to connote both). The PSI distinguishes between parent-related stress (e.g., lack of spousal support) and child-related stress (e.g., the child placing many demands on the parent). We found minimal differences in child-related stress, and parent-related stress was slightly lower for parents of affected children, though statistically nonsignificant. Differences by parent gender, however, were striking: Regardless of a child’s SSC diagnosis, fathers reported higher child-related stress than mothers. Conversely, mothers reported higher parentrelated stress than fathers in such areas as health, role restriction, depression, and spousal support. This lack of presurgery case-control differences suggests that parents of children with SSC may be highly resilient in their reaction to their child’s initial diagnosis and treatment. Alternatively, parents may experience a suppressed or delayed stress response related to the effects of caring for an infant in the midst of an acute medical situation. For example, parents may pull together in response to a crisis, benefiting from temporary support from friends or the child’s medical team. Furthermore, concerns about developmental delays, which would not typically emerge clearly until the preschool years, may be a major source of stress for parents of children with SSC. Indeed, affected children have 1.5 to 2 times the risk of developmental delay compared with their unaffected peers (Starr et al., 2012), and 35% to 50% of school-aged children with SSC have adverse neurodevelopmental outcomes (Kapp-Simon et al., 2007). If this is the case, we would anticipate increasing stress among parents of cases and larger differences between case and control group participants at later assessments. A pattern of increasing stress in parents of children with various disabilities (Down syndrome, motor impairment, developmental delay) has been reported previously (Hauser-Cram et al., 2001), such that very few parents displayed clinically meaningful levels of stress prior to age 3 years but showed increases in the years afterward (.38% in the clinical range by age 10). This finding is consistent with theoretical expectations that parenting stress continually fluctuates in relation to the child’s developmental stages and the environmental demands placed on child and parents (Baker et al., 2003). This study examined the stability of parent stress over time in the same cohort, providing opportunities to address the above hypotheses (e.g., suppressed or delayed stress response). We examined parent stress at two visits subsequent to the affected child’s surgery, when children
were approximately aged 18 and 36 months. We hypothesized that parenting stress among cases would increase over time in relation to control parents, as the buffering effects of social support fade and other potential stressors emerge (e.g., child developmental delays). We also expected to observe the same parent gender pattern reported previously (i.e., with fathers reporting higher child-related stress and mothers reporting higher parent-related stress). Finally we examined the influence of parents’ ‘‘defensive’’ response bias on case-control group differences as measured by the PSI Defensive Responding scale, a measure of respondents’ tendency to present a favorable self-impression (Abidin, 1995). METHODS Participants Participants included parents of infants with SSC (cases) and demographically similar, unaffected infants (controls) enrolled between 2001 and 2006. We followed participants longitudinally and performed assessments at three time points (visit 1, 2, and 3). The study was approved by the institutional review boards at each of five participating centers (see Kapp-Simon et al., 2012). This research was in full compliance with Health Insurance Portability and Accountability Act standards. Cases Infants with SSC were referred to the project at the time of diagnosis by the treating surgeon or pediatrician. Eligibility criteria included (1) diagnosed SSC (sagittal, metopic, unilateral coronal, or unilateral lambdoid synostosis), confirmed on computed tomography scans; (2) participation prior to surgery; and (3) child age 30 months. Exclusions included (1) prematurity (,34 weeks’ gestation); (2) major medical or neurological conditions (e.g., cardiac defects, seizure disorders); (3) presence of three or more extracranial minor malformations (Leppig et al., 1987); or (4) presence of other major malformations. Among the 266 enrolled case patients, 247 biological mothers and 211 biological fathers completed the Parenting Stress Index (PSI) at visit 1. Of these, 220 (89.1%) and 179 (84.8%) of the same mothers and fathers completed the PSI at visit 2, respectively, and 182 mothers (73.7%) and 137 fathers (64.9%) at visit 3. A total of 176 mothers and 131 fathers completed the PSI at all three visits, and 131 participants had PSI protocols from both parents for all three visits. Controls Unaffected infants were eligible if they had no known craniofacial anomaly and met none of the exclusion
Gray et al., STRESS IN PARENTS OF CHILDREN WITH SINGLE-SUTURE CRANIOSYNOSTOSIS
criteria for cases. Control families were recruited through pediatric practices, birthing centers, and announcements in print media. Controls were group matched to cases on the following factors: (1) age at enrollment (63 weeks); (2) sex; (3) family socioeconomic status (SES; Hollingshead, 1975); and (4) race/ ethnicity. Among 259 eligible control patients, 254 biological mothers and 220 biological fathers completed the PSI at visit 1. Of these, 221 (87.0%) and 195 (88.6%) of the same mother and father respondents, respectively, completed the PSI at visit 2, and 201 mothers (79.0%) and 170 fathers (77%) at visit 3. A total of 188 mothers and 159 fathers completed the PSI at all three visits, and 157 controls had PSI data available for both parents across all three time periods.
5
Case-Control and Parent Gender Differences
At visit 1, infants were evaluated following their diagnosis at a mean age of 9 months, on average 46.5 days prior to surgery. Target ages for visits 2 and 3 were 18 and 36 months, respectively. Children were developmentally tested at each visit and their parents completed the PSI during the visit or, when necessary, at home and returned it by mail. At visit 1, mothers were interviewed to obtain medical history and family constellation data.
To evaluate whether parents of children with SSC reported higher levels of stress than parents of unaffected children and whether mothers and fathers differed in their PSI reports, we used a generalized estimating equations approach (GEE; Liang and Zeger, 1986) to linear regression for each outcome at each of the three visits. Outcomes were the PSI Child and Parent Domain scores, as well as selected subscales based on observed case-control or mother-father differences (Rosenberg et al., 2011): Acceptability, Demandingness, and Distractibility subscales from the Child Domain and Role Restriction and Spousal Support from the Parent Domain. Only participants with the same eligible respondents from visit 1 were included in these analyses (e.g., the same biological mother or father). All regression models were adjusted for matching variables, including SES (continuous; Hollingshead, 1975), child age at visit (months), race (white versus nonwhite), patient gender, and study site. We included an interaction term between case status and respondent (mother versus father), which allowed for examination of both case-control and father-mother differences within the same model. The P value of the interaction terms in these models indicated whether case-control differences varied by respondent and whether motherfather differences varied by case status. Regardless of statistical significance, stratified results were reported to provide a more complete picture of the findings. To facilitate comparisons across outcomes with different distributions (e.g., the domain and subscale scores), we calculated standardized differences between cases and controls and mothers and fathers, using control mothers’ mean and standard deviation of each outcome at each visit as the reference for standardization. Results are presented as standardized differences. To evaluate whether defensive responding influenced case-control differences, we repeated the above regressions for the Child and Parent Domain scores, excluding all respondents who met the PSI manual’s definition of excessively biased reporting (i.e., scores 24 on the Defensive Responding scale; Abidin, 1995). To examine whether the odds of defensive responding differed between cases and controls at visit 1, we performed logistic regression adjusting for SES, race, and gender, including an interaction term between case status and respondent.
Data Analysis
Change Over Time
We examined demographic characteristics separately for cases and controls and among cases within each affected suture site. We also compared demographic characteristics and visit 1 PSI scores of participants with PSI scores at visit 3 versus those lost to follow-up.
To determine whether case-control differences changed over time, we performed linear regression separately for mothers and fathers, using GEE with an interaction between case status and visit. To evaluate whether mother-father differences changed over time,
Measures Parenting Stress Index The PSI (Abidin, 1995) assesses the presence or intensity of potential stress/stressors in the family environment. The Parent Domain score summarizes parents’ personal characteristics and social support as they relate to the demands and tasks of parenting. Subscales include Depression, Attachment to Child, Role Restriction, Competence, Isolation, Health, and Spousal Support. The Child Domain score summarizes stress related to child characteristics or temperament, represented by the following subscales: Adaptability, Acceptability, Demandingness, Mood, Distractibility, and Reinforces Parent. Higher scores on all scales reflect higher levels of stress. Test-retest reliability data from several studies provide values ranging from .55 to .82 for Child Domain scores and .69 to .91 for the Parent Domain (Abidin, 1995). Procedure
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TABLE 1
Demographic Characteristics of Single-Suture Craniosynostosis Cases and Controls at Visit 1 Cases Controls
Total
Sagittal
Metopic
Unicoronal
Lambdoid
n
%
n
%
n
%
n
%
n
%
n
%
Total
256
100.0
249
100.0
115
100.0
58
100.0
63
100.0
13
100.0
Gender Female Male
93 163
36.3 63.7
90 159
36.1 63.9
26 89
22.6 77.4
16 42
27.6 72.4
42 21
66.7 33.3
6 7
46.2 53.8
Age, mo ,6 6–9 9þ
111 73 72
43.4 28.5 28.1
121 62 66
48.6 24.9 26.5
77 15 23
67.0 13.0 20.0
23 18 17
39.7 31.0 29.3
19 23 21
30.2 36.5 33.3
2 6 5
15.4 46.2 38.5
Race Nonwhite White
75 181
29.3 70.7
63 186
25.3 74.7
27 88
23.5 76.5
17 41
29.3 70.7
16 47
25.4 74.6
3 10
23.1 76.9
Socioeconomic status 1 (high) 2 3 4 5 (low)
70 127 34 16 9
27.3 49.6 13.3 6.3 3.5
58 84 58 33 16
23.3 33.7 23.3 13.3 6.4
27 41 22 20 5
23.5 35.7 19.1 17.4 4.3
12 20 14 8 4
20.7 34.5 24.1 13.8 6.9
14 19 19 5 6
22.2 30.2 30.2 7.9 9.5
5 4 3 0 1
38.5 30.8 23.1 0.0 7.7
93 97 28 38
36.3 37.9 10.9 14.8
89 91 34 35
35.7 36.5 13.7 14.1
42 28 23 22
36.5 24.3 20.0 19.1
21 24 3 10
36.2 41.4 5.2 17.2
21 34 6 2
33.3 54.0 9.5 3.2
5 5 2 1
38.5 38.5 15.4 7.7
Marital status Single Married/cohabitating
22 232
8.6 90.6
21 227
8.4 91.2
12 103
10.4 89.6
2 55
3.4 94.8
6 57
9.5 90.5
1 12
7.7 92.3
Children in household 1 2 3 4þ
116 88 37 13
45.3 34.4 14.5 5.1
86 93 43 26
34.5 37.3 17.3 10.4
43 40 19 12
37.4 34.8 16.5 10.4
22 23 8 5
37.9 39.7 13.8 8.6
18 25 12 8
28.6 39.7 19.0 12.7
3 5 4 1
23.1 38.5 30.8 7.7
Adults in household 1 2 3þ
9 227 18
3.5 88.7 7.0
12 212 24
4.8 85.1 9.6
5 101 8
4.3 87.8 7.0
3 44 11
5.2 75.9 19.0
4 56 3
6.3 88.9 4.8
0 11 2
0.0 84.6 15.4
Site Seattle Chicago St. Louis Atlanta
for each participant at each visit we calculated the difference between mothers’ and fathers’ scores (father minus mother). Using these difference scores as the outcome and visit as the exposure, we then performed linear regression using GEE. We included the visit-bycase status interaction to examine whether change over time differed between cases and controls. All longitudinal analyses were adjusted for SES, age at first visit, race, gender, and study site. Only participants with the same respondents across time were included. For change over time in father-mother differences, only parents maintaining the same marital status were included because any observed changes in differences could otherwise be attributable to changing marital status.
Controls were slightly more likely than cases to be of white race and to report higher SES. For cases, mean age at surgery was 8.8 months (range, 1.8 to 34.1 months). Compared with participants at visit 3, cases and controls lost to follow-up were more likely to be girls (40% of controls, 43% of cases) and of lower SES (22% of controls and 34% of cases in the two lowest SES categories) (data not shown). Cases lost to follow-up were also more likely to be nonwhite (36%). Average PSI scores at visit 1 were similar between participants and nonparticipants for mothers and fathers (i.e., absolute differences 3.5 points). Case-Control Group Differences Child Domain
RESULTS Cases and controls had similar distributions of gender, age at visit 1, study site, and marital status (Table 1).
Case mothers reported higher Child Domain scores than control mothers at visit 1 (standardized difference [std diff] ¼ 0.2; 95% confidence interval [CI], 0.0 to 0.4),
Gray et al., STRESS IN PARENTS OF CHILDREN WITH SINGLE-SUTURE CRANIOSYNOSTOSIS
but differences at visits 2 and 3 were negligible (std diff ¼ 0.0) (Table 2). Case fathers also reported higher Child Domain scores than control fathers at visit 1 (std diff ¼ 0.2; 95% CI, 0.0 to 0.4), but these differences diminished and were negligible at visits 2 and 3 (std diff 0.1). Parent Domain Case mothers consistently reported lower Parent Domain scores than control mothers, though these differences were small and imprecise, with std diff ranging from 0.1 (95% CI, 0.3 to 0.1) to 0.2 points (95% CI, 0.3 to 0.0). Case-control differences in fathers were similar, with std diff ranging from 0.1 (95% CI 0.3, 0.1) to 0.2 (95% CI 0.3, 0.1). Subscales Across all visits, case mothers and fathers reported slightly higher scores than controls on the Acceptability and Demandingness subscales, with std diff ranging from 0.1 to 0.5 for mothers and 0.1 to 0.4 for fathers, but these differences decreased over time (Table 2). Differences on the Distractibility subscale were negligible for both mothers and fathers. Among mothers and fathers, cases reported slightly lower scores on the Role Restriction and Spousal Support subscales than controls across visits (mothers’ std diff ¼0.1 to 0.3; fathers’ std diff ¼ 0.1 to 0.2), although these differences were small and often imprecise. Defensive Responding Across the three visits, 17.6% to 20.2% of case mothers were categorized as ‘‘defensive’’ compared with 9.5% to 12.2% of control mothers. Defensive responding was similar in case and control fathers (20.4% to 24.6% for cases, 20.0% to 23.6% controls). After removing defensive responders, case-control differences among mothers on the Child Domain became slightly larger (std diff ¼ 0.0 [95% CI, 0.2 to 0.2] to 0.3 [95% CI, 0.1 to 0.5]) but aside from visit 1 were mostly small and imprecise; differences on the Parent Domain were negligible (std diff 0.1j; data not shown). When removing defensively responding fathers, all casecontrol differences were small and imprecise (std diff ¼ 0.0 [95% CI, 0.2 to 0.2] to 0.2 [95% CI, 0.0 to 0.4]).
7
Father-Mother Differences Child Domain Among cases, fathers reported higher scores than mothers on the Child Domain at visit 1 (std diff ¼ 0.3; 95% CI, 0.1 to 0.4), but both the magnitude and precision of these differences decreased thereafter (visit 2 std diff ¼ 0.1; 95% CI, 0.0 to 0.2; visit 3 std diff ¼ 0.0; 95% CI, 0.2 to 0.3) (Table 2). Among controls, differences were less consistent, with fathers reporting higher scores than mothers at visit 1 (std diff ¼ 0.3; 95% CI, 0.2 to 0.4) but negligible differences at visits 2 and 3 (std diff ¼ 0.0). Parent Domain Among both cases and controls and at all visits, fathers had lower Parent Domain scores than mothers, with standardized differences ranging from 0.1 (95% CI, 0.3 to 0.0) to 0.3 (95% CI, 0.5 to 0.2). The differences were most pronounced at visit 3 for both cases and controls. Subscales Father-mother differences on Child Domain subscales were negligible for cases and controls (Table 2). However, in both groups, fathers reported lower stress than mothers on the Parent Domain subscales Role Restriction (std diff ¼0.2 [95% CI, 0.3 ¼0.1] to 0.4 [95% CI, 0.6 to 0.2]) and Spousal Support (std diff ¼ 0.3 [95% CI, 0.4 to 0.2] to 0.5 [95% CI, 0.7 to 0.3]). Change Over Time Among cases, father-mother differences in the Child and Parent Domain scores decreased by 1.5 points for each visit (Child Domain 95% CI, 2.9 to 0.1; Parent Domain 95% CI, 3.3 to 0.3) (Table 3). In controls, the differences decreased by 1.9 (95% CI, 3.2 to 0.6) points for each visit on the Child Domain and by 0.5 (95% CI, 2.0 to 1.0) points on the Parent Domain. Thus, father-mother differences became less apparent on the Child Domain and more apparent on the Parent Domain. Interactions between case status and visit were not statistically significant for either the Child or Parent Domain (P ¼ .66 and .40, respectively).
Changes Over Time Interaction Between Case Status and Respondent Case-control differences for mothers decreased by 0.8 points at each subsequent visit for the Child Domain and increased by 0.7 points for the Parent Domain at each visit (Table 3). Case-control difference among fathers remained essentially unchanged over time.
None of the interactions between case status and respondent (mother versus father) were statistically significant when using the domain scores as outcomes in cross-sectional analyses (P values, .32 to .97).
Controls
96.5 (20.1) 96.7 (18.1) 95.3 (17.7) 95.0 (16.7) 114.6 (23.9) 107.2 (20.2) 116.7 (19.4) 109.9 (19.9)
12.0 18.2 23.4 17.6 17.5
Subscales Acceptability Demandingness Distractibility Role Restriction Spousal Support
12.0 17.7 23.7 16.1 15.2
(3.4) (4.6) (4.5) (4.3) (4.1)
(3.9) (4.6) (4.2) (4.2) (4.1)
11.6 17.3 23.3 18.5 18.1
11.5 16.8 25.1 18.1 17.9
(2.9) (4.5) (4.7) (4.1) (4.3)
(3.2) (4.1) (4.3) (4.1) (4.7)
11.5 16.7 23.3 16.9 16.1
11.5 16.2 24.2 16.8 15.8
(2.9) (4.2) (4.2) (4.2) (4.2)
(3.2) (3.9) (4.2) (4.3) (4.3)
0.1 0.2 0.0 0.2 0.1
0.0 0.2
0.1 0.2 0.2 0.1 0.2
0.0 0.1
0.5 0.5 0.1 0.3 0.2
0.2 0.2 to to to to to
0.7 0.6 0.1 0.1 0.0
to to to to to
0.3 0.4 0.0 0.2 0.0
0.1 0.0 0.2 0.5 0.3 to to to to to
0.3 0.4 0.2 0.0 0.1
0.2 to 0.3 0.4 to 0.1
0.1 0.1 0.3 0.3 0.4
0.2 to 0.2 0.3 to 0.1
0.3 0.3 0.2 0.5 0.4
0.0 to 0.4 0.3 to 0.0
.34 .08 .99 .04 .29
.70 .18
.37 .01 .11 .60 .10
.99 .44
,.001 ,.001 .40 .004 .02
.03 .09
0.2 0.2 0.1 0.2 0.2
0.0 0.2
0.1 0.4 0.1 0.2 0.1
0.1 0.1
0.4 0.4 0.1 0.1 0.1
0.2 0.1 to to to to to
0.6 0.6 0.0 0.1 0.1
to to to to to
0.3 0.6 0.3 0.0 0.1
0.1 0.0 0.1 0.4 0.4
to to to to to
0.4 0.4 0.3 0.0 0.0
0.2 to 0.3 0.4 to 0.1
0.1 0.2 0.1 0.4 0.3
0.1 to 0.3 0.3 to 0.1
0.1 0.3 0.3 0.3 0.2
0.0 to 0.4 0.3 to 0.1
95% CI
95% CI‡
P Value Std Diff
Fathers
Mothers
Case-Control Differences
.20 .05 .56 .06 .09
.66 .18
.50 ,.001 .42 .09 .16
.31 .31
.002 ,.001 .10 .19 .38
.07 .49
0.0 0.1 0.1 0.3 0.5
0.0 0.3
0.0 0.0 0.1 0.4 0.4
0.1 0.2
0.2 0.0 0.1 0.2 0.3
0.3 0.1
P Value Std Diff
to to to to to
0.4 0.2 0.2 0.1 0.2
to to to to to
0.2 0.2 0.2 0.2 0.2
0.1 0.2 0.1 0.5 0.7
to to to to to
0.2 0.1 0.3 0.2 0.3
0.1 to 0.2 0.5 to 0.1
0.1 0.2 0.1 0.6 0.5
0.0 to 0.2 0.4 to 0.1
0.0 0.1 0.1 0.3 0.4
0.1 to 0.4 0.3 to 0.0
95% CI
Cases
.59 .33 .19 ,.001 ,.001
.58 ,.001
.77 .93 .32 ,.001 ,.001
.16 .01
.04 .71 .44 .01 ,.001
,.001 .06
0.0 0.1 0.0 0.4 0.4
0.0 0.3
0.0 0.1 0.2 0.3 0.4
0.0 0.2
0.4 0.1 0.1 0.3 0.5
0.3 0.2
P Value Std Diff
to to to to to
0.5 0.2 0.3 0.2 0.3
to to to to to
0.2 0.0 0.0 0.1 0.3
0.2 0.2 0.1 0.5 0.6
to to to to to
0.2 0.0 0.2 0.2 0.3
0.1 to 0.2 0.5 to 0.2
0.1 0.3 0.3 0.5 0.6
0.1 to 0.1 0.4 to 0.0
0.2 0.1 0.0 0.5 0.6
0.2 to 0.4 0.4 to 0.1
95% CI
Controls
Father-Mother Differences
* M ¼ mothers; F ¼ fathers; CI ¼ confidence interval; SD ¼ standard deviation; std diff ¼ standardized difference. † All standardized differences adjusted for socioeconomic status, age at visit, race (white versus nonwhite), gender, and study site; based on distribution of control mothers’ scores at each visit. ‡ All 95% confidence intervals calculated using robust standard error estimates and unstructured correlation matrix. § Case mothers: all n 244; case fathers: all n 210; control mothers: all n 251; control fathers: all n 221. || Case mothers: all n 217; case fathers: all n 179; control mothers: all n 221; control fathers: all n 194. } Case mothers: all n ¼ 182; case fathers: all n¼137; control mothers: all n ¼ 201; control fathers: all n ¼ 170.
(3.9) (5.0) (5.1) (4.9) (4.9)
12.1 18.1 25.0 16.3 15.2
(3.1) (3.7) (3.6) (4.2) (4.0)
Visit 3} Child Domain Parent Domain
(4.1) (4.8) (4.4) (5.0) (5.4)
11.4 15.7 24.8 16.9 15.5
12.0 18.1 24.7 18.0 17.1
(2.8) (4.1) (4.1) (4.3) (5.1)
Subscales Acceptability Demandingness Distractibility Role Restriction Spousal Support
10.4 15.6 24.5 18.5 18.0
96.8 (18.6) 98.3 (17.6) 95.5 (16.6) 94.4 (15.8) 113.7 (24.5) 109.0 (21.4) 114.3 (20.3) 109.3 (21.5)
(3.7) (3.9) (3.9) (4.3) (4.0)
Visit 2|| Child Domain Parent Domain
12.4 17.5 24.5 16.5 15.1
11.9 17.5 24.4 17.4 16.9
Subscales Acceptability Demandingness Distractibility Role Restriction Spousal Support
(3.7) (4.4) (4.4) (4.9) (5.1)
93.9 (18.3) 97.6 (16.7) 90.3 (14.8) 94.1 (15.0) 113.0 (22.8) 109.5 (21.4) 115.6 (20.5) 109.9 (19.4)
M F M F Mean (SD) Mean (SD) Mean (SD) Mean (SD) Std Diff†
Cases
Differences in Parenting Stress Index Scores Between Parents of Infants With and Without Single-Suture Craniosynostosis and Between Mothers and Fathers*
Visit 1§ Child Domain Parent Domain
TABLE 2
.93 .13 .51 ,.001 ,.001
.61 ,.001
.61 .14 .02 .001 ,.001
.96 .02
,.001 .33 .10 ,.001 ,.001
,.001 .001
P Value
8 Cleft Palate–Craniofacial Journal, January 2015, Vol. 52 No. 1
Gray et al., STRESS IN PARENTS OF CHILDREN WITH SINGLE-SUTURE CRANIOSYNOSTOSIS
9
TABLE 3 Changes Over Time in Differences Between Parents of Infants With and Without Single-Suture Craniosynostosis and Between Mothers and Fathers* Change in Difference for Each Visit Case-Control Differences Mothers
Child Domain Parent Domain
Father-Mother Differences Fathers
Cases
Controls
Change†
95% CI‡
P Value
Change
95% CI
P Value
Change
95% CI
P Value
Change
95% CI
P Value
0.8 0.7
2.5 to 0.9 1.0 to 2.4
.33 .42
0.2 0.2
2.0 to 1.6 2.1 to 1.7
.84 .84
1.5 1.5
2.9 to 0.1 3.3 to 0.3
.04 .10
1.9 0.5
3.2 to 0.6 2.0 to 1.0
.004 .50
* CI ¼ confidence interval. † All change values adjusted for socioeconomic status, age at first visit, race (white versus nonwhite), gender, and study site. ‡ All 95% confidence intervals with robust standard error estimates and unstructured correlation matrix.
DISCUSSION Case-Control Group Differences We found no persisting differences in PSI scores between parents of infants with and without SSC, regardless of parent gender. We anticipated higher stress reports among parents of infants with SSC for the reasons listed previously (e.g., unusual child appearance, the growing potential for associated medical complications and/or developmental delays), as well as findings from previous studies of parent stress in diagnostically heterogeneous samples of children with craniofacial disorders (Pope et al., 2005). Why then did we not find differences? First, SSC may have little relative impact on infant and family psychological functioning in the early years of life compared with the generally stressful events that surround the birth and early years of any child. Previous studies comparing the reported stress of parents with and without children with various disabilities or health problems have generated mixed findings. Although many have observed higher stress among parents of affected children (Fuller and Rankin, 1994; Pelchat et al., 1999; Ong et al., 2001; Oelofsen and Richardson, 2006; Gupta, 2007; Ong et al., 2011), several have reported null findings (Pelchat et al., 1999; Singer et al., 1999; Gupta, 2007; Collett et al., 2011; Treyvaud et al., 2011; Cloonan et al., 2012; Gray et al., 2012). Such inconsistency is not surprising, given the wide variation in sample demographics and ascertainment methods as well as the many different child health problems and disabilities under study. Another possibility is that the multidisciplinary, family-centered care typically received by infants and children with SSC mitigated the stressors associated with this diagnosis and surgical treatment. In the family adjustment and adaptation response model (Patterson, 1998), a family crisis occurs when the demands on the family exceed the capabilities of the family (e.g., a diagnosis of SSC), necessitating changes in the resources or coping behaviors of the family, ultimately resulting in adjustment to the crisis. These resources can emerge at
the individual, family, or community level, the latter including medical and other health care providers/ facilities. Recent care guidelines for craniosynostosis emphasize the importance of coordinated, multidisciplinary care that cuts across specialties (e.g., anesthesiology, neurosurgery, craniofacial surgery, psychology, speech-language pathology) and embraces family-centered care (McCarthy et al., 2012). All of the craniofacial teams in this study embraced this model of care, which may have served as a community-level resource that helped families to better cope with the typically stressful events surrounding their child’s medical condition. Indeed, social support has been observed to predict stress in parents of children with disabilities or serious medical conditions (Hauser-Cram et al., 2001; Shin and Nhan, 2009; Singer et al., 2010; Jeong et al., 2013) and without them (Ostberg and Hagekull, 2000). The lack of case-control differences may also be attributable to the study sample characteristics. Both case and control parents were generally affluent, white, and married. Because the study required multiple visits over time, retained families may have been more stable than those from the general population, further contributing to their lower risk. Additionally, parents received feedback on their child’s cognitive and motor development after each visit. If delays were detected, parents were encouraged to consult with their child’s pediatrician to determine whether further assessment or intervention was needed. Ultimately, 26% of cases and 15% of controls received some form of developmental intervention by visit 3, which may have provided additional support, minimizing case-control group differences. Another possibility is that case-control group differences were attenuated by parent response bias, which we assessed with the PSI’s Defensiveness Responding scale. Mothers of children with SSC were about twice as likely as control group mothers to meet the PSI’s definition of defensive responding. However, their removal from analyses had little effect on overall case-control group differences. A final possibility is that elevated parenting stress is potentially observable only among parents of infants
10
Cleft Palate–Craniofacial Journal, January 2015, Vol. 52 No. 1
with more severe, multi-suture forms of craniosynostosis and related genetic syndromes (e.g., Apert or Crouzon syndromes; Wilkie et al., 1995).
small proportion of single parents, who tend to report elevated parenting stress regardless of gender (Skari et al., 2002).
Father-Mother Differences
Clinical Implications
We observed remarkably consistent differences between mothers and fathers across time, regardless of whether the child was affected by SSC. A number of studies have reported higher stress in mothers than fathers of children with various health problems; however, few have examined whether this pattern is shown by parents of healthy children. In one study of psychological responses after the birth of a healthy infant, mothers reported higher levels of insomnia, anxiety, and other negative emotions than fathers (Skari et al., 2002). Many of these differences dissipated during infancy, but reemerged when evaluated 5 years later (Skreden et al., 2008). The finding of father-mother differences of similar magnitude in both case and control parents suggests that these differences were unrelated to having a child with a health problem but instead reflected underlying gender differences in parenting experiences. In particular, mothers reported greater stress related to role restriction and spousal support, which may be attributable to traditional parenting roles in which mothers serve as the primary caregiver and fathers as the provider (Pelchat et al., 2007). Similar to mothers of typically developing children, mothers of children with disabilities tend to assume more responsibility for the care of their child and to express greater dissatisfaction with the division of tasks (Pelchat et al., 2007). This likely contributed to the elevated stress reports we observed in both case and control group mothers. Therefore, results suggest that levels of reported stress in one parent cannot be generalized to the other parent. Mother and father reports of stress should be treated separately in future studies of family responses to children’s health problems or other stressors.
Although the PSI did not reveal differences between cases and controls, within-group variation in PSI scores among parents of cases may be predictive of subsequent child outcomes. This question has not yet been addressed in analyses of this cohort, though previous studies of other craniofacial disorders (e.g., oral clefts) have found the PSI in infancy to have good predictive validity to later psychosocial outcomes (Krueckeberg and Kapp-Simon, 1993; Speltz et al., 1997; Pope et al., 2005). Although findings suggest that on average, parents of infants with SSC are doing about as well as parents of typically developing babies, the identification of individual parents within the SSC population with relatively high levels of reported stress could lead to interventions that mitigate later childhood problems, interventions like those recommended by the McCarthy et al. (2012) practice parameters. As a next step, we will examine models predicting school-age outcomes for children with SSC, which will include earlier scores from the PSI as potential predictors.
Strengths and Limitations The strengths of the study include the use of a control group with which parents of children with SSC were compared, rather than using norms from the PSI. In addition, we assessed parental stress at multiple points in time—both before and after corrective surgery— allowing for examination of changes over time. Last, unlike earlier studies that focused only on mothers, we also included fathers, allowing for father-mother as well as case-control comparisons. The limitations of this study include constrained demographic representation (mostly white parents of high SES), limiting generalizability to nonwhite or lower SES populations. An additional factor limiting generalizability is the sample’s
Acknowledgment. We would like to thank Joseph Picciano, Ph.D., for his assistance with this project.
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