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Journal of Pediatric Urology (2013) xx, 1e6
Caregivers of children with a disorder of sex development: Associations between parenting capacities and psychological distress Cortney Wolfe-Christensen a,*, David A. Fedele b, Katherine Kirk c, Larry L. Mullins d, Yegappan Lakshmanan a, Amy B. Wisniewski e a
Department of Pediatric Urology, Children’s Hospital of Michigan, 3901 Beaubien, Detroit, MI 48201, USA b University of Florida, Department of Health and Clinical Psychology, Gainesville, FL, USA c University of Oklahoma College of Nursing, Oklahoma City, OK, USA d Department of Psychology, Oklahoma State University, Stillwater, OK, USA e Department of Pediatrics, University of Oklahoma Health Sciences Center, Oklahoma City, OK, USA Received 30 November 2012; accepted 22 November 2013
KEYWORDS Disorders of Sex Development; Parenting; Psychosocial
Abstract Objective: Caregivers of children with a disorder of sex development (DSD) are at increased risk for maladaptive parenting capacities, such as high levels of parental overprotection and perceived vulnerability of their child, in addition to parenting stress. The current study aims to examine whether there are relationships between these parenting capacities and psychological distress, including depressive and anxious symptoms. Patients and methods: Participants included 134 caregivers of 90 children with a DSD. Caregivers completed measures of parental overprotection, perceived vulnerability, parenting stress, anxiety, and depression. Results: Hierarchical regression analyses revealed that higher levels of parenting stress were related to more anxious and depressive symptoms in caregivers. Higher levels of perceived vulnerability were related to more anxious symptoms. Levels of parental overprotection were unrelated to anxious or depressive symptoms. Conclusions: There is a relationship between parenting capacities and mental health outcomes in caregivers of children with DSD, although the direction of this relationship is not clear. Given
* Corresponding author. Tel.: þ1 313 745 5588; fax: þ1 313 993 8738. E-mail addresses: [email protected], [email protected] (C. Wolfe-Christensen). 1477-5131/$36 ª 2013 Journal of Pediatric Urology Company. Published by Elsevier Ltd. All rights reserved. http://dx.doi.org/10.1016/j.jpurol.2013.11.016
Please cite this article in press as: Wolfe-Christensen C, et al., Caregivers of children with a disorder of sex development: Associations between parenting capacities and psychological distress, Journal of Pediatric Urology (2013), http://dx.doi.org/10.1016/ j.jpurol.2013.11.016
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C. Wolfe-Christensen et al. the strong relationships between parenting stress and anxious and depressive symptoms, targeting parenting stress and/or psychological distress in these caregivers could result in better functioning overall. ª 2013 Journal of Pediatric Urology Company. Published by Elsevier Ltd. All rights reserved.
Introduction The National Institutes of Health (NIH) has called for researchers to investigate specific “parenting capacities” that could both directly and indirectly affect the functioning of children [1]. Parenting capacities refer to specific behaviors, beliefs, or values that parents may hold about their child. Previous research indicates that caregivers of children with chronic medical conditions are at increased risk for higher levels of psychological distress and maladaptive parenting capacities [2e6]. Three frequently studied parenting capacity variables include parental overprotection (OP; i.e., parenting behaviors that are overprotective based on the developmental level of the child), perceived child vulnerability (PCV; i.e., perceiving the child as sick and vulnerable), and parenting stress (PS; i.e., stress related to relationship between the parent and child, as well as to characteristics of both the parent and child). There is evidence that high levels of OP, PCV, and PS can negatively affect the child, resulting in poorer emotional, behavioral, and social functioning [7e10]. However, there is little research on the relationship between these parenting capacities and caregiver psychological distress, such as depression and anxiety. We previously reported that caregivers of children with a disorder of sex development (DSD), a group of congenital conditions in which chromosomal, phenotypic, and/or gonadal sex are discordant [10], are at risk for increased levels of OP, PCV, and PS [11]. These elevated parenting capacity variables may be related to a variety of reasons. Often, parents and caregivers of children with a DSD are faced with difficult decisions, such as choosing a gender of rearing or whether or not the child should undergo genitoplasty at an early age. Additionally, some DSD diagnoses, such as congenital adrenal hyperplasia (CAH), require lifelong pharmacological management. Finally, nearly 50% of patients with 46,XY DSD will never receive a clinical diagnosis, leading to increased uncertainty and potential psychological distress [12]. The current study aimed to examine the role of OP, PCV, and PS in a large sample of caregivers of children with a DSD in addition to investigating the relationship between these parenting capacities and psychological distress, specifically anxious and depressive symptoms. It was hypothesized that higher levels of OP, PCV, and PS would be associated with higher levels of anxious and depressive symptoms.
Methods Participants/procedures Caregivers of children with a DSD were recruited to participate in a larger study that examined how caregivers
are affected, both in terms of their parenting behavior and their psychological distress, when they have a child with a DSD. Participants were recruited from six pediatric hospitals that specialize in treating children with DSDs, in addition to being recruited from two national support groups for parents and caregivers of children with DSDs. The caregivers were invited to participate if their child had been diagnosed with a DSD for at least 6 months prior to recruitment. The 6-month cut-off was chosen to insure that caregivers of children newly diagnosed with DSD were not included, as they are expected to exhibit elevated levels of depressive and anxious symptoms. Inclusion criteria included the caregiver (a) being 18 years of age or older, (b) able to provide informed consent, (c) and able to read English at an eighth-grade reading level. The exclusion criteria included children with significant developmental delay or cognitive impairment. In families that included two caregivers, both were invited to participate. Questionnaire packets were mailed to participants who were recruited either during the child’s outpatient clinic appointment or via a mailed letter from the hospital where the child receives care for DSD. The caregivers completed the forms, which included a written informed consent document, and mailed them back to the researchers. Caregivers from support groups were recruited at national conventions. Each caregiver received $25 as a thank-you for their participation in the study. The study was approved by the Institutional Review Board at each recruitment site.
Measures Demographic form Caregivers completed an investigator-created demographic form, which assessed the child’s diagnosis, age, gender of rearing, and grade in school. Additionally, the caregiver’s age and sex, marital status, education level, self-identified race/ethnicity, and annual family income were also collected. Child Vulnerability Scale [13] The Child Vulnerability Scale (CVS) is an eightitem measure that assesses the caregiver’s level of perceived child vulnerability. The respondent is asked to read the item and to use a 4-point Likert-type scale ranging from “definitely false” to “definitely true.” Higher scores are indicative of more perceived vulnerability, with a cut-off score of 10 indicating a clinically significant level. The CVS shows moderate to high internal reliability (a Z 0.74), high testeretest reliability (r Z 0.84) and adequate internal consistency (a Z 0.78). Cronbach’s alpha for the current sample was 0.80, meaning that the internal reliability for the measure was high.
Please cite this article in press as: Wolfe-Christensen C, et al., Caregivers of children with a disorder of sex development: Associations between parenting capacities and psychological distress, Journal of Pediatric Urology (2013), http://dx.doi.org/10.1016/ j.jpurol.2013.11.016
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Caregiver Distress in DSD
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Parent Protection Scale [14] Caregiver overprotection was measured with the Parent Protection Scale (PPS), a 25-item self-report that assesses dimensions of protective parenting of children aged 2e18 years. Caregivers use a 4-point scale ranging from “never” to “always” to answer each question. Higher scores indicate greater levels of overprotection of their child. The PPS shows moderate to high internal reliability (a Z 0.73), high testeretest reliability (r Z 0.86), and adequate internal consistency (a Z 0.74) [14]. Cronbach’s alpha for the current sample was 0.70, meaning that the internal reliability for the measure was adequate. Parenting Stress Index/Short Form (PSI/SF) [15] Caregiver stress was measured with the Parenting Stress Index/Short Form (PSI/SF), a 36-item self-report that uses a 5-point scale ranging from “strongly agree” to “strongly disagree.” Higher PSI/SF scores indicate greater caregiver stress. The PSI/SF was developed for caregivers of children 1 month to 12 years of age. The PSI/SF shows high internal consistency (a Z 0.92) and its validity has been established in parents of children with chronic medical conditions such as diabetes and asthma. Cronbach’s alpha for the current sample was 0.92, meaning that the internal reliability for the measure was excellent. Beck Anxiety Index [16] Caregiver levels of anxious symptoms were measured with the Beck Anxiety Index (BAI), a 21-item self-report measure developed for 17e80 year olds that uses a 4-point response scale ranging from “not at all” to “severely.” Higher scores indicate greater levels of parental anxiety. Total scores can be categorized as minimal (0e7), mild (8e15), moderate (16e25), and severe (26e63). The BAI has good psychometric properties including high internal reliability (a Z 0.92) and good testeretest reliability (r Z 0.75) after 1 week [17]. Cronbach’s alpha for the current sample was 0.91, meaning that the internal reliability for the measure was high. Beck Depression Inventory-2nd Edition (BDI-2) [18] Caregiver levels of depressive symptoms were measured with the Beck Depression Inventory-2nd Edition (BDI-2), a 21-item self-report measure developed for 13e80 year olds
Table 1
Statistical analyses First, descriptive statistics were conducted to better characterize the nature of the sample. Next, hierarchical regression analyses were conducted to determine whether relationships existed between the parenting capacity variables (i.e., OP, PCV, and PS) and the caregiver distress variables (i.e., anxious and depressive symptoms). Covariates for the regression analyses were statistically chosen based on significant correlations between demographic and study variables (Table 1) and included child age, child’s gender of rearing, caregiver sex, and total family income for all analyses. Variables were entered with covariates on step 1, the parenting capacities on step 2, and the total scores from the BAI and BDI served as the dependent variables in separate equations. All reported p values are two sided, statistical significance was considered at a 0.05, and R2 was used as the measure of effect size because it estimates the factor of interest while controlling for covariates. Statistical analyses were conducted with IBM SPSS Statistics version 20.
Results A total of 134 caregivers (93 F, 41 M) of 90 children (gender of rearing: 65 F, 25 M) with a DSD were included in the study (Table 2). One hundred and thirty-seven caregivers were approached across the six DSD clinics and 119 consented to participate, resulting in a response rate of 86.9%, while the remaining 15 caregivers were recruited from one of the national support groups. Comparisons between these groups (clinic vs. support group) with regard to caregiver age, sex, and marital status, child age and gender of
Zero-order correlations between the study variables. 1
1. 2. 3. 4. 5. 6. 7. 8. 9.
that uses a 4-point response scale ranging from “not at all” to severely.” Higher BDI-2 scores indicate greater levels of depressive symptomology. Total scores can be categorized as minimal (0e13), mild (14e19), moderate (20e28), and severe (29e63) [18]. The BDI-2 possesses excellent internal reliability (a Z 0.90e0.92) and testeretest reliability (r Z 0.93) after 1 week [19]. Cronbach’s alpha for the current sample was 0.91, meaning that the internal reliability for the measure was high.
Child age Sex of rearing Caregiver sex Total income CVS total PPS total PSI total BAI total BDI total
2
3
4
5
6
7
8
9
0.08
0.17 .01
0.11 0.17 0.11
0.11 0.09 0.12 0.06
0.52** 0.04 0.04 e0.09 0.35**
0.15 0.12 0.16 0.26** 0.26** 0.01
0.11 0.02 0.16 0.09 0.35** 0.14 0.39**
0.11 0.22** 0.26** 0.12 0.31** 0.08 0.65** 0.64**
Note: CVS Z Child Vulnerability Scale; PPS Z Parental Protection Scale; PSI Z Parenting Stress Index; BAI Z Beck Anxiety Inventory; BDI Z Beck Depression Inventory. *p < 0.05; **p < 0.01.
Please cite this article in press as: Wolfe-Christensen C, et al., Caregivers of children with a disorder of sex development: Associations between parenting capacities and psychological distress, Journal of Pediatric Urology (2013), http://dx.doi.org/10.1016/ j.jpurol.2013.11.016
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C. Wolfe-Christensen et al. Table 2
some of our previous reports [11,20e22]; however, the relationships between parenting capacities and psychological distress have not yet been investigated in this population. Results of the regression analyses revealed that the models for both anxious and depressive symptoms were significant, (F(7, 123) Z 5.42, p < 0.001; F(7, 123) Z 18.10, p < 0.001), respectively. Specifically, after controlling for child age, child’s gender of rearing, caregiver sex, and total family income, level of perceived vulnerability, t(130) Z 2.40, p Z 0.018, and level of parenting stress, t(130) Z 3.48, p Z 0.001, were both independent, significant predictors of level of anxious symptoms. Notably, the combination of the parenting capacities accounted for 19.1% of the variance in level of anxious symptoms above and beyond the variance accounted for by the covariates. With regard to depressive symptoms, after controlling for child age and gender, caregiver sex, and total family income, level of parenting stress was a significant predictor of level of depressive symptoms (t(130) Z 8.54 p < 0.001). Level of parental overprotection and level of perceived vulnerability were unrelated to the level of depressive symptoms. Additionally, the parenting capacities accounted for 36.5% of the variance in depressive symptoms over and above the variance accounted for by the covariates.
Demographics of caregivers (N Z 134).
Sex Age (years) Marital status Married Single Other Race/ethnicity Caucasian African American Asian Hispanic Native American Other Annual household income $60,000 Missing
41 M, 93 F 19e68 (M Z 35.73 8.71) 71.3% (N Z 97) 22.1% (N Z 30) 6.6% (N Z 9) 77.9% 8.1% 4.4% 3.7 2.9% 2.9%
(N (N (N (N (N (N
Z Z Z Z Z Z
106) 11) 6) 5) 4) 4)
26.4% 30.9% 37.5% 1.5%
(N (N (N (N
Z Z Z Z
41) 42) 51) 2)
rearing, and total family income were not significant (all p > 0.05), providing support for including all 134 caregivers in the analyses. The children were categorized into four groups on the basis of karyotype and gender of rearing: (a) 46,XX DSD reared female, which includes children with a 46,XX karyotype and presence of the SRY gene, and CAH due to deficiencies in either 21-hydroxylase (OH), 18-OH, or 11-OH; (b) 46,XX reared male, which includes one child with CAH due to 21-OH deficiency; (c) 46,XY reared male, which includes diagnoses of mixed and complete gonadal dysgenesis, micropenis, penoscrotal hypospadias, and idiopathic DSD; and (d) 46,XY reared female, which includes diagnoses of partial and complete androgen insensitivity syndrome, mixed and complete gonadal dysgenesis, and micropenis (Table 3). The majority of the caregivers self-identified as Caucasian (77.6%) and reported being married (72.2%). Examination of the caregivers who met the clinical cut-off scores on the parenting measures revealed that 45 caregivers (33.6%; 45/134) met the cut-off for at least one of the parenting capacity measures, with 26.7% of these caregivers (12/45) meeting the cut-off on more than one measure (Table 4). The majority of caregivers reported minimal to mild levels of anxious and depressive symptoms. However, 12.7% (17/134) of them reported moderate to severe levels of anxious symptoms on the BAI, and 13.4% (18/134) of caregivers had BDI-2 scores in the moderate to severe ranges. It should be noted that a subset of these caregivers (N Z 87) have been included in
Table 3
Discussion We have previously reported that parents of children with a DSD are at increased risk for maladaptive parenting capacities; however, the relationships of these variables to psychological distress were not examined. The current study sought to identify relationships between three discrete parenting capacity variables, namely parental overprotection, perceived child vulnerability, and parenting stress, and psychological distress, specifically anxious and depressive symptoms in caregivers of children with a DSD. The findings indicate that higher levels of parenting stress were significantly related to higher levels of both anxiety and depression, while higher levels of perceived vulnerability were significantly related to higher levels of anxiety. Given that causality of these relationships cannot be determined from the current study, the findings suggest that interventions targeting psychological distress and/or parenting stress and perceived vulnerability could result in an improvement in the other domain. Given the robust bidirectional relationship between parent and child adjustment to chronic medical conditions, interventions that improve caregiver functioning (i.e., lowering overprotection, perceived vulnerability, parenting stress,
Demographics of children (N Z 90).
Gender of rearing Age (years) Diagnosis and gender of rearing 46,XX DSD reared female 46,XX DSD reared male 46,XY DSD reared male 46,XY DSD reared female
25 M, 65 F M Z 6.70 5.69 (0.50e21) 56.7% 1.1% 25.6% 16.7%
(N (N (N (N
Z Z Z Z
51) 1) 23) 15)
Age mean SD (range) 6.38 5.14 (0.50e21) 11 5.33 4.96 (0.50e15) 8.81 8.72 (0.05e21)
Please cite this article in press as: Wolfe-Christensen C, et al., Caregivers of children with a disorder of sex development: Associations between parenting capacities and psychological distress, Journal of Pediatric Urology (2013), http://dx.doi.org/10.1016/ j.jpurol.2013.11.016
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Caregiver Distress in DSD Table 4
5
Parenting capacity measures.
Clinical cut-off met
N (%)
PSI þ PPS þ CVS PSI þ PPS PSI þ CVS PPS þ CVS PSI Only PPS Only CVS Only
2 1 4 5 13 11 9
(4.4) (2.2) (8.8) (11.1) (28.9) (24.4) (20.0)
Note: PSI Z Parenting Stress Index, PPS Z Parental Protection Scale, CVS Z Child Vulnerability Scale.
parenting and 19% reported either anxious or depressive symptoms in the moderate-severe range. Additionally, caregivers who reported higher levels of parenting stress, and those who perceive their child as more vulnerable also reported experiencing more depressive and anxious symptoms. Family-based interventions that target the reduction of these maladaptive parenting practices and/or psychological distress have the potential to directly improve the functioning of caregivers. Finally, given the strong bidirectional relationship between parent and child adjustment to illness, improving the functioning of caregivers could also indirectly improve adjustment outcomes in their children.
Conflict of interest anxiety, and depressive symptoms) can potentially indirectly improve child coping and functioning as well. Notably, although relatively small proportions of the caregivers reported clinically significant levels of the measures of overprotection, perceived vulnerability, and parenting stress, there was little overlap in the caregivers that reached the cut-offs on each of the measures. As a whole, almost one-third of the caregivers in the sample met clinical cut-offs on the parenting capacity measures, which is consistent with research in other groups of children with chronic illnesses [23]. Although it is estimated that the majority of parents adjust well to a child’s diagnosis, a consistent subgroup (approximately 25%) have some difficulty, which can be communicated, either directly or indirectly, to the child and may result in poor adjustment of the child [24]. The findings of the current study should be interpreted in light of several limitations. The study is cross-sectional in nature; therefore, we are precluded from identifying the temporal relationships between variables. We also acknowledge that there is some dependency within the data since a subset of caregivers completed measures assessing their interactions with or perceptions of the same child. Additionally, there is a selection bias, as all of the participants were responsible for completing the questionnaires and mailing them back. It is certainly possible that people who made the effort to complete the study differed from the non-completers. Unfortunately, we did not collect data from those who did not complete the study. Finally, the majority of the participants selfidentified as Caucasian and reported to be married. This threatens the external validity of the data by affecting the generalizability of the results, especially with regard to functioning in families of minority status. While the abovementioned limitations exist, the study does have some strengths as well. The sample included a large number of caregivers of children with DSD, which is an understudied population, and validated questionnaires were utilized. Additionally, participants were recruited from several medical centers and national support groups, which increases variability in the sample.
Conclusions Overall, 33% of caregivers in the sample met the cut-off score for clinically significant levels of maladaptive
None.
Funding None.
References [1] National Institutes of Health. PA-06-97: parenting capacities and health outcomes in youths and adolescents. Retrieved 10.05.06, from: http://grants.nih.gov/grants/guide/pa-files/ PA-06-097.html. [2] Kashikar-Zuck S, Lynch AM, Slater S, Graham TB, Swain NF, Noll RB, et al. Family factors, emotional functioning, and functional impairment in juvenile fibromyalgia syndrome. Arthritis Rheum 2008;59:1392e8. [3] Lea ˜o LL, Zhang L, Sousa PL, Medoza-Sassi R, Chadha R, Lovatel R, et al. High prevalence of depression amongst mothers of children with asthma. J Asthma 2009;46:388e91. [4] Majnemer A, Limperopoulos C, Shevell M, Rohlicek C, Rosenblatt B, Tchervenkov C. Health and well-being of children with congenital cardiac malformations, and their families, following open-heart surgery. Cardiol Young 2006;16: 157e64. [5] Rentinck IC, Ketelaar M, Jongmans MJ, Gorter JW. Parents of children with cerebral palsy: a review of factors related to the process of adaptation. Child Care Health Dev 2007;33:161e9. [6] Patin ˜o-Ferna ´ndez AM, Pai AL, Alderfer M, Hwang WT, Reilly A, Kazak AE. Acute stress in parents of children newly diagnosed with cancer. Pediatr Blood Cancer 2008;50:289e92. [7] Holmbeck GN, Johnson SZ, Wills KE, McKernon W, Rose B, Erklin S, et al. Observed and perceived parental overprotection in relation to psychosocial adjustment in preadolescents with a physical disability: the meditational role of behavioral autonomy. J Consult Clin Psychol 2002;70:96e110. [8] Anthony KK, Gil KM, Schanberg LE. Parental perceptions of child vulnerability in children with chronic illness. J Pediatr Psychol 2003;28:185e90. [9] Colletti CJ, Wolfe-Christensen C, Carpentier MY, Page MC, McNall-Knapp RY, Meyer WH, et al. The relationship of parental overprotection, perceived vulnerability, and parenting stress to behavioral, emotional, and social adjustment in children with cancer. Pediatr Blood Cancer 2008;51: 269e74. [10] Hughes IA, Houk C, Ahmed SF, Lee PA, Lawson Wilkins Endocrine Society/European Society for Paediatric Endocrinology Consensus Group. Consensus statement on management of intersex disorders. J Pediatr Urol 2006;2:148e62.
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6 [11] Kirk KD, Fedele DA, Wolfe-Christensen C, Phillips TM, Mullins LL, et al. Parenting characteristics of female caregivers of children affected by chronic endocrine conditions: a comparison between disorders of sex development and type 1 diabetes mellitus. J Pediatr Nursing 2011; 26:e29e36. [12] Morel Y, Rey R, Teinturier C, Nicolino M, Michel-Calemard L, Mowszowicz I, et al. Aetiological diagnosis of male sex ambiguity: a collaborative study. Eur J Pediatr 2002;16:49e59. [13] Forsyth BWC, Horwitz SM, Leventhal JM, Leventhal JM, Burger J, Leaf PJ. The Child Vulnerability Scale: an instrument to measure parental perceptions of child vulnerability. J Pediatr Psychol 1996;21:89e101. [14] Thomasgard M, Metz WP, Edelbrock C, Shonkoff JP. Parentchild relationship disorders: I. Parental overprotection and the development of the Parent Protection Scale. J Dev Behav Pediatr 1995;16:244e50. [15] Abdin RR. Parenting stress index/short form manual. Los Angeles, CA: Western Psychological Services; 1990. [16] Beck AT, Epstein N, Brown G, Steer RA. An inventory for measuring clinical anxiety: psychometric properties. J Consult Clin Psychol 1988;56:893e7. [17] Wilson KA, Chambless DL, de Beurs E. Beck anxiety inventory. In: Maruish ME, editor. The use of psychological testing for treatment planning and outcomes assessment. 3rd ed. Mahwah, NJ: Lawrence, Erlbaum; 1999. pp. 399e421.
C. Wolfe-Christensen et al. [18] Beck AT, Steer RA, Brown GK. Manual for the Beck Depression Inventory-II. San Antonio, TX: Psychological Corporation; 1996. [19] Dozios DJA, Dobson KS, Ahnberg JL. A psychometric evaluation of the Beck Depression Inventory-II. Psychol Assess 1998;10: 83e90. [20] Fedele DA, Kirk K, Wolfe-Christensen C, Phillips TM, Mazur T, Mullins LL, et al. Primary caregivers of children affected by disorders of sex development: mental health and caregiver characteristics in the context of genital ambiguity and genitoplasty. Int J Pediatr Endocrinol 2010;2010:690674. [21] Wolfe-Christensen C, Fedele DA, Kirk K, Phillips TM, Mazur T, Mullins LL, et al. Degree of external genital malformation at birth in children with a disorder of sex development and subsequent caregiver distress. J Urol 2012;188:1596e600. [22] Hullmann SE, Fedele DA, Wolfe-Christensen C, Mullins LL, Wisniewski AB. Differences in adjustment by child developmental stage among caregivers of children with disorders of sex development. Int J Pediatr Endocrinol 2011;16. [23] Hullmann SE, Wolfe-Christensen C, Ryan JL, Fedele DA, Rambo PL, Chaney JM. Parental overprotection, perceived child vulnerability, and parenting stress: a cross-illness comparison. J Clin Psychol Med Settings 2010;17:357e65. [24] Herzer M, Godiwala N, Hommel KA, Driscoll K, Mitchell M, Crosby LE, et al. Family functioning in the context of pediatric chronic conditions. J Dev Behav Pediatr 2010;31:26e34.
Please cite this article in press as: Wolfe-Christensen C, et al., Caregivers of children with a disorder of sex development: Associations between parenting capacities and psychological distress, Journal of Pediatric Urology (2013), http://dx.doi.org/10.1016/ j.jpurol.2013.11.016
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Journal of Pediatric Urology (2013) xx, 1e6
Caregivers of children with a disorder of sex development: Associations between parenting capacities and psychological distress Cortney Wolfe-Christensen a,*, David A. Fedele b, Katherine Kirk c, Larry L. Mullins d, Yegappan Lakshmanan a, Amy B. Wisniewski e a
Department of Pediatric Urology, Children’s Hospital of Michigan, 3901 Beaubien, Detroit, MI 48201, USA b University of Florida, Department of Health and Clinical Psychology, Gainesville, FL, USA c University of Oklahoma College of Nursing, Oklahoma City, OK, USA d Department of Psychology, Oklahoma State University, Stillwater, OK, USA e Department of Pediatrics, University of Oklahoma Health Sciences Center, Oklahoma City, OK, USA Received 30 November 2012; accepted 22 November 2013
KEYWORDS Disorders of Sex Development; Parenting; Psychosocial
Abstract Objective: Caregivers of children with a disorder of sex development (DSD) are at increased risk for maladaptive parenting capacities, such as high levels of parental overprotection and perceived vulnerability of their child, in addition to parenting stress. The current study aims to examine whether there are relationships between these parenting capacities and psychological distress, including depressive and anxious symptoms. Patients and methods: Participants included 134 caregivers of 90 children with a DSD. Caregivers completed measures of parental overprotection, perceived vulnerability, parenting stress, anxiety, and depression. Results: Hierarchical regression analyses revealed that higher levels of parenting stress were related to more anxious and depressive symptoms in caregivers. Higher levels of perceived vulnerability were related to more anxious symptoms. Levels of parental overprotection were unrelated to anxious or depressive symptoms. Conclusions: There is a relationship between parenting capacities and mental health outcomes in caregivers of children with DSD, although the direction of this relationship is not clear. Given
* Corresponding author. Tel.: þ1 313 745 5588; fax: þ1 313 993 8738. E-mail addresses: [email protected], [email protected] (C. Wolfe-Christensen). 1477-5131/$36 ª 2013 Journal of Pediatric Urology Company. Published by Elsevier Ltd. All rights reserved. http://dx.doi.org/10.1016/j.jpurol.2013.11.016
Please cite this article in press as: Wolfe-Christensen C, et al., Caregivers of children with a disorder of sex development: Associations between parenting capacities and psychological distress, Journal of Pediatric Urology (2013), http://dx.doi.org/10.1016/ j.jpurol.2013.11.016
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C. Wolfe-Christensen et al. the strong relationships between parenting stress and anxious and depressive symptoms, targeting parenting stress and/or psychological distress in these caregivers could result in better functioning overall. ª 2013 Journal of Pediatric Urology Company. Published by Elsevier Ltd. All rights reserved.
Introduction The National Institutes of Health (NIH) has called for researchers to investigate specific “parenting capacities” that could both directly and indirectly affect the functioning of children [1]. Parenting capacities refer to specific behaviors, beliefs, or values that parents may hold about their child. Previous research indicates that caregivers of children with chronic medical conditions are at increased risk for higher levels of psychological distress and maladaptive parenting capacities [2e6]. Three frequently studied parenting capacity variables include parental overprotection (OP; i.e., parenting behaviors that are overprotective based on the developmental level of the child), perceived child vulnerability (PCV; i.e., perceiving the child as sick and vulnerable), and parenting stress (PS; i.e., stress related to relationship between the parent and child, as well as to characteristics of both the parent and child). There is evidence that high levels of OP, PCV, and PS can negatively affect the child, resulting in poorer emotional, behavioral, and social functioning [7e10]. However, there is little research on the relationship between these parenting capacities and caregiver psychological distress, such as depression and anxiety. We previously reported that caregivers of children with a disorder of sex development (DSD), a group of congenital conditions in which chromosomal, phenotypic, and/or gonadal sex are discordant [10], are at risk for increased levels of OP, PCV, and PS [11]. These elevated parenting capacity variables may be related to a variety of reasons. Often, parents and caregivers of children with a DSD are faced with difficult decisions, such as choosing a gender of rearing or whether or not the child should undergo genitoplasty at an early age. Additionally, some DSD diagnoses, such as congenital adrenal hyperplasia (CAH), require lifelong pharmacological management. Finally, nearly 50% of patients with 46,XY DSD will never receive a clinical diagnosis, leading to increased uncertainty and potential psychological distress [12]. The current study aimed to examine the role of OP, PCV, and PS in a large sample of caregivers of children with a DSD in addition to investigating the relationship between these parenting capacities and psychological distress, specifically anxious and depressive symptoms. It was hypothesized that higher levels of OP, PCV, and PS would be associated with higher levels of anxious and depressive symptoms.
Methods Participants/procedures Caregivers of children with a DSD were recruited to participate in a larger study that examined how caregivers
are affected, both in terms of their parenting behavior and their psychological distress, when they have a child with a DSD. Participants were recruited from six pediatric hospitals that specialize in treating children with DSDs, in addition to being recruited from two national support groups for parents and caregivers of children with DSDs. The caregivers were invited to participate if their child had been diagnosed with a DSD for at least 6 months prior to recruitment. The 6-month cut-off was chosen to insure that caregivers of children newly diagnosed with DSD were not included, as they are expected to exhibit elevated levels of depressive and anxious symptoms. Inclusion criteria included the caregiver (a) being 18 years of age or older, (b) able to provide informed consent, (c) and able to read English at an eighth-grade reading level. The exclusion criteria included children with significant developmental delay or cognitive impairment. In families that included two caregivers, both were invited to participate. Questionnaire packets were mailed to participants who were recruited either during the child’s outpatient clinic appointment or via a mailed letter from the hospital where the child receives care for DSD. The caregivers completed the forms, which included a written informed consent document, and mailed them back to the researchers. Caregivers from support groups were recruited at national conventions. Each caregiver received $25 as a thank-you for their participation in the study. The study was approved by the Institutional Review Board at each recruitment site.
Measures Demographic form Caregivers completed an investigator-created demographic form, which assessed the child’s diagnosis, age, gender of rearing, and grade in school. Additionally, the caregiver’s age and sex, marital status, education level, self-identified race/ethnicity, and annual family income were also collected. Child Vulnerability Scale [13] The Child Vulnerability Scale (CVS) is an eightitem measure that assesses the caregiver’s level of perceived child vulnerability. The respondent is asked to read the item and to use a 4-point Likert-type scale ranging from “definitely false” to “definitely true.” Higher scores are indicative of more perceived vulnerability, with a cut-off score of 10 indicating a clinically significant level. The CVS shows moderate to high internal reliability (a Z 0.74), high testeretest reliability (r Z 0.84) and adequate internal consistency (a Z 0.78). Cronbach’s alpha for the current sample was 0.80, meaning that the internal reliability for the measure was high.
Please cite this article in press as: Wolfe-Christensen C, et al., Caregivers of children with a disorder of sex development: Associations between parenting capacities and psychological distress, Journal of Pediatric Urology (2013), http://dx.doi.org/10.1016/ j.jpurol.2013.11.016
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Caregiver Distress in DSD
3
Parent Protection Scale [14] Caregiver overprotection was measured with the Parent Protection Scale (PPS), a 25-item self-report that assesses dimensions of protective parenting of children aged 2e18 years. Caregivers use a 4-point scale ranging from “never” to “always” to answer each question. Higher scores indicate greater levels of overprotection of their child. The PPS shows moderate to high internal reliability (a Z 0.73), high testeretest reliability (r Z 0.86), and adequate internal consistency (a Z 0.74) [14]. Cronbach’s alpha for the current sample was 0.70, meaning that the internal reliability for the measure was adequate. Parenting Stress Index/Short Form (PSI/SF) [15] Caregiver stress was measured with the Parenting Stress Index/Short Form (PSI/SF), a 36-item self-report that uses a 5-point scale ranging from “strongly agree” to “strongly disagree.” Higher PSI/SF scores indicate greater caregiver stress. The PSI/SF was developed for caregivers of children 1 month to 12 years of age. The PSI/SF shows high internal consistency (a Z 0.92) and its validity has been established in parents of children with chronic medical conditions such as diabetes and asthma. Cronbach’s alpha for the current sample was 0.92, meaning that the internal reliability for the measure was excellent. Beck Anxiety Index [16] Caregiver levels of anxious symptoms were measured with the Beck Anxiety Index (BAI), a 21-item self-report measure developed for 17e80 year olds that uses a 4-point response scale ranging from “not at all” to “severely.” Higher scores indicate greater levels of parental anxiety. Total scores can be categorized as minimal (0e7), mild (8e15), moderate (16e25), and severe (26e63). The BAI has good psychometric properties including high internal reliability (a Z 0.92) and good testeretest reliability (r Z 0.75) after 1 week [17]. Cronbach’s alpha for the current sample was 0.91, meaning that the internal reliability for the measure was high. Beck Depression Inventory-2nd Edition (BDI-2) [18] Caregiver levels of depressive symptoms were measured with the Beck Depression Inventory-2nd Edition (BDI-2), a 21-item self-report measure developed for 13e80 year olds
Table 1
Statistical analyses First, descriptive statistics were conducted to better characterize the nature of the sample. Next, hierarchical regression analyses were conducted to determine whether relationships existed between the parenting capacity variables (i.e., OP, PCV, and PS) and the caregiver distress variables (i.e., anxious and depressive symptoms). Covariates for the regression analyses were statistically chosen based on significant correlations between demographic and study variables (Table 1) and included child age, child’s gender of rearing, caregiver sex, and total family income for all analyses. Variables were entered with covariates on step 1, the parenting capacities on step 2, and the total scores from the BAI and BDI served as the dependent variables in separate equations. All reported p values are two sided, statistical significance was considered at a 0.05, and R2 was used as the measure of effect size because it estimates the factor of interest while controlling for covariates. Statistical analyses were conducted with IBM SPSS Statistics version 20.
Results A total of 134 caregivers (93 F, 41 M) of 90 children (gender of rearing: 65 F, 25 M) with a DSD were included in the study (Table 2). One hundred and thirty-seven caregivers were approached across the six DSD clinics and 119 consented to participate, resulting in a response rate of 86.9%, while the remaining 15 caregivers were recruited from one of the national support groups. Comparisons between these groups (clinic vs. support group) with regard to caregiver age, sex, and marital status, child age and gender of
Zero-order correlations between the study variables. 1
1. 2. 3. 4. 5. 6. 7. 8. 9.
that uses a 4-point response scale ranging from “not at all” to severely.” Higher BDI-2 scores indicate greater levels of depressive symptomology. Total scores can be categorized as minimal (0e13), mild (14e19), moderate (20e28), and severe (29e63) [18]. The BDI-2 possesses excellent internal reliability (a Z 0.90e0.92) and testeretest reliability (r Z 0.93) after 1 week [19]. Cronbach’s alpha for the current sample was 0.91, meaning that the internal reliability for the measure was high.
Child age Sex of rearing Caregiver sex Total income CVS total PPS total PSI total BAI total BDI total
2
3
4
5
6
7
8
9
0.08
0.17 .01
0.11 0.17 0.11
0.11 0.09 0.12 0.06
0.52** 0.04 0.04 e0.09 0.35**
0.15 0.12 0.16 0.26** 0.26** 0.01
0.11 0.02 0.16 0.09 0.35** 0.14 0.39**
0.11 0.22** 0.26** 0.12 0.31** 0.08 0.65** 0.64**
Note: CVS Z Child Vulnerability Scale; PPS Z Parental Protection Scale; PSI Z Parenting Stress Index; BAI Z Beck Anxiety Inventory; BDI Z Beck Depression Inventory. *p < 0.05; **p < 0.01.
Please cite this article in press as: Wolfe-Christensen C, et al., Caregivers of children with a disorder of sex development: Associations between parenting capacities and psychological distress, Journal of Pediatric Urology (2013), http://dx.doi.org/10.1016/ j.jpurol.2013.11.016
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4
C. Wolfe-Christensen et al. Table 2
some of our previous reports [11,20e22]; however, the relationships between parenting capacities and psychological distress have not yet been investigated in this population. Results of the regression analyses revealed that the models for both anxious and depressive symptoms were significant, (F(7, 123) Z 5.42, p < 0.001; F(7, 123) Z 18.10, p < 0.001), respectively. Specifically, after controlling for child age, child’s gender of rearing, caregiver sex, and total family income, level of perceived vulnerability, t(130) Z 2.40, p Z 0.018, and level of parenting stress, t(130) Z 3.48, p Z 0.001, were both independent, significant predictors of level of anxious symptoms. Notably, the combination of the parenting capacities accounted for 19.1% of the variance in level of anxious symptoms above and beyond the variance accounted for by the covariates. With regard to depressive symptoms, after controlling for child age and gender, caregiver sex, and total family income, level of parenting stress was a significant predictor of level of depressive symptoms (t(130) Z 8.54 p < 0.001). Level of parental overprotection and level of perceived vulnerability were unrelated to the level of depressive symptoms. Additionally, the parenting capacities accounted for 36.5% of the variance in depressive symptoms over and above the variance accounted for by the covariates.
Demographics of caregivers (N Z 134).
Sex Age (years) Marital status Married Single Other Race/ethnicity Caucasian African American Asian Hispanic Native American Other Annual household income $60,000 Missing
41 M, 93 F 19e68 (M Z 35.73 8.71) 71.3% (N Z 97) 22.1% (N Z 30) 6.6% (N Z 9) 77.9% 8.1% 4.4% 3.7 2.9% 2.9%
(N (N (N (N (N (N
Z Z Z Z Z Z
106) 11) 6) 5) 4) 4)
26.4% 30.9% 37.5% 1.5%
(N (N (N (N
Z Z Z Z
41) 42) 51) 2)
rearing, and total family income were not significant (all p > 0.05), providing support for including all 134 caregivers in the analyses. The children were categorized into four groups on the basis of karyotype and gender of rearing: (a) 46,XX DSD reared female, which includes children with a 46,XX karyotype and presence of the SRY gene, and CAH due to deficiencies in either 21-hydroxylase (OH), 18-OH, or 11-OH; (b) 46,XX reared male, which includes one child with CAH due to 21-OH deficiency; (c) 46,XY reared male, which includes diagnoses of mixed and complete gonadal dysgenesis, micropenis, penoscrotal hypospadias, and idiopathic DSD; and (d) 46,XY reared female, which includes diagnoses of partial and complete androgen insensitivity syndrome, mixed and complete gonadal dysgenesis, and micropenis (Table 3). The majority of the caregivers self-identified as Caucasian (77.6%) and reported being married (72.2%). Examination of the caregivers who met the clinical cut-off scores on the parenting measures revealed that 45 caregivers (33.6%; 45/134) met the cut-off for at least one of the parenting capacity measures, with 26.7% of these caregivers (12/45) meeting the cut-off on more than one measure (Table 4). The majority of caregivers reported minimal to mild levels of anxious and depressive symptoms. However, 12.7% (17/134) of them reported moderate to severe levels of anxious symptoms on the BAI, and 13.4% (18/134) of caregivers had BDI-2 scores in the moderate to severe ranges. It should be noted that a subset of these caregivers (N Z 87) have been included in
Table 3
Discussion We have previously reported that parents of children with a DSD are at increased risk for maladaptive parenting capacities; however, the relationships of these variables to psychological distress were not examined. The current study sought to identify relationships between three discrete parenting capacity variables, namely parental overprotection, perceived child vulnerability, and parenting stress, and psychological distress, specifically anxious and depressive symptoms in caregivers of children with a DSD. The findings indicate that higher levels of parenting stress were significantly related to higher levels of both anxiety and depression, while higher levels of perceived vulnerability were significantly related to higher levels of anxiety. Given that causality of these relationships cannot be determined from the current study, the findings suggest that interventions targeting psychological distress and/or parenting stress and perceived vulnerability could result in an improvement in the other domain. Given the robust bidirectional relationship between parent and child adjustment to chronic medical conditions, interventions that improve caregiver functioning (i.e., lowering overprotection, perceived vulnerability, parenting stress,
Demographics of children (N Z 90).
Gender of rearing Age (years) Diagnosis and gender of rearing 46,XX DSD reared female 46,XX DSD reared male 46,XY DSD reared male 46,XY DSD reared female
25 M, 65 F M Z 6.70 5.69 (0.50e21) 56.7% 1.1% 25.6% 16.7%
(N (N (N (N
Z Z Z Z
51) 1) 23) 15)
Age mean SD (range) 6.38 5.14 (0.50e21) 11 5.33 4.96 (0.50e15) 8.81 8.72 (0.05e21)
Please cite this article in press as: Wolfe-Christensen C, et al., Caregivers of children with a disorder of sex development: Associations between parenting capacities and psychological distress, Journal of Pediatric Urology (2013), http://dx.doi.org/10.1016/ j.jpurol.2013.11.016
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Caregiver Distress in DSD Table 4
5
Parenting capacity measures.
Clinical cut-off met
N (%)
PSI þ PPS þ CVS PSI þ PPS PSI þ CVS PPS þ CVS PSI Only PPS Only CVS Only
2 1 4 5 13 11 9
(4.4) (2.2) (8.8) (11.1) (28.9) (24.4) (20.0)
Note: PSI Z Parenting Stress Index, PPS Z Parental Protection Scale, CVS Z Child Vulnerability Scale.
parenting and 19% reported either anxious or depressive symptoms in the moderate-severe range. Additionally, caregivers who reported higher levels of parenting stress, and those who perceive their child as more vulnerable also reported experiencing more depressive and anxious symptoms. Family-based interventions that target the reduction of these maladaptive parenting practices and/or psychological distress have the potential to directly improve the functioning of caregivers. Finally, given the strong bidirectional relationship between parent and child adjustment to illness, improving the functioning of caregivers could also indirectly improve adjustment outcomes in their children.
Conflict of interest anxiety, and depressive symptoms) can potentially indirectly improve child coping and functioning as well. Notably, although relatively small proportions of the caregivers reported clinically significant levels of the measures of overprotection, perceived vulnerability, and parenting stress, there was little overlap in the caregivers that reached the cut-offs on each of the measures. As a whole, almost one-third of the caregivers in the sample met clinical cut-offs on the parenting capacity measures, which is consistent with research in other groups of children with chronic illnesses [23]. Although it is estimated that the majority of parents adjust well to a child’s diagnosis, a consistent subgroup (approximately 25%) have some difficulty, which can be communicated, either directly or indirectly, to the child and may result in poor adjustment of the child [24]. The findings of the current study should be interpreted in light of several limitations. The study is cross-sectional in nature; therefore, we are precluded from identifying the temporal relationships between variables. We also acknowledge that there is some dependency within the data since a subset of caregivers completed measures assessing their interactions with or perceptions of the same child. Additionally, there is a selection bias, as all of the participants were responsible for completing the questionnaires and mailing them back. It is certainly possible that people who made the effort to complete the study differed from the non-completers. Unfortunately, we did not collect data from those who did not complete the study. Finally, the majority of the participants selfidentified as Caucasian and reported to be married. This threatens the external validity of the data by affecting the generalizability of the results, especially with regard to functioning in families of minority status. While the abovementioned limitations exist, the study does have some strengths as well. The sample included a large number of caregivers of children with DSD, which is an understudied population, and validated questionnaires were utilized. Additionally, participants were recruited from several medical centers and national support groups, which increases variability in the sample.
Conclusions Overall, 33% of caregivers in the sample met the cut-off score for clinically significant levels of maladaptive
None.
Funding None.
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