The purpose of this study was to explore the cross-sectional and prospective relationships between cognitive coping strategies and parental stress in parents of children with Down syndrome. A total of 621 participants filled out questionnaires, including the Cognitive Emotion Regulation Questionnaire to measure cognitive coping and the Nijmeegse Ouderlijke Stress Index—Korte Versie (A. J. L. L. De Brock, A. A. Vermulst, J. R. M. Gerris, & R. R. Abidin, 1992) to measure parental stress. After 8 months, stress was measured again. Cross-sectionally, using acceptance, rumination, positive refocusing, refocusing on planning, and catastrophizing to a greater extent was related to more stress, whereas using positive reappraisal more often was related to less stress. Prospectively, acceptance and catastrophizing were related to more stress, whereas positive reappraisal was related to less stress. Implications for future research and prevention and intervention activities are discussed.
In The Netherlands, the birth of a child with Down syndrome is estimated to occur in 14 per 10,000 live births annually (De Graaf, 2007). Children and adults with Down syndrome have a higher risk of several physical complications like heart disease and hearing loss (Van Wouwe & Weijerman, 2003). In addition, a large percentage of these people function below average mentally and develop behavioral and communicative problems (Roberts, Price, & Malkin, 2007; Van Wouwe & Weijerman, 2003). Because of these issues, researchers have investigated repeatedly whether parents of a child with Down syndrome experience more stress and a diminished psychological well being compared with parents of a typically developing child. Although having a child with Down syndrome might not be as distressing as once thought, there is evidence that these parents experience more stress (Baker, Blacher, Crnic, & Edelbrock, 2002; Hedov, Annerén, & Wikblad, 2000; Roach, Orsmond, & Barrat, 1999; Scott, Atkinson, Minton, & Bowman, 1997; Singer, 2006; Spangenberg & Theron, 2001) and may be at greater risk of developing a depressive disorder (Scott et al., 1997). This additional stress might be specific to the first few months after diagnosis (Glidden & Schoolcraft, 2003). It is likely, however, that parents also experience stress when the child grows older and encounters difficult transitional periods like learning to speak or finding an appropriate school (Kraemer & Blacher, 2001; Most, Fidler, Laforce-Booth, & Kelly, 2006). Parents who experience more distress may be more likely to engage in maladaptive parenting behavior (Hastings & Beck, 2004). Thus, it seems very important to gain understanding on how these levels of stress can be reduced.
When people experience stress, they use certain strategies to cope with this stress to regulate their emotions (Lazarus & Folkman, 1984). Coping strategies can be adaptive (i.e., leading to less distress) or maladaptive (i.e., leading to more distress) and can be executed behaviorally, such as seeking emotional support, or cognitively, such as attaching positive thoughts to a stressful situation. Evidence for the relationship between coping strategies in general and various measures of psychological well being in parents of children with Down syndrome and other developmental disabilities has been well established in the last years. For example, Abbeduto and colleagues (2004) found that problem-focused coping was related to less depressive symptoms in mothers of children with Down syndrome, autism, or fragile X syndrome, whereas emotion-focused coping was related to more depressive symptoms. In parents of children with developmental disabilities, positive reappraisal and accepting responsibility was related to higher levels of subjective well being, whereas for escape–avoidance, different results for mothers and fathers were found (i.e., adaptive for fathers but not adaptive for mothers; Glidden, Billings, & Jobe, 2006). Avoidant coping strategies are also related to more depression and anxiety in parents of children with Down syndrome (Spangenberg & Theron, 2001).
However, these studies have focused mainly on general measures of psychological well being like depression, instead of focusing on parental stress experiences related to the child, which might be more sensitive to the influence of having a child with Down syndrome. Also, there are a number of specific cognitive coping strategies proved relevant in other samples, which these studies have not investigated. Two cognitive coping strategies have been studied before in parents of children with Down syndrome: positive reappraisal, which was related to higher levels of subjective well being (King, Scollen, Ramsey, & Williams, 2000) and blaming others, which was related to more depressive and angry feelings (Hall, Bobrow, & Marteau, 1997). Nevertheless, strategies like rumination or focus on thought and catastrophizing, which have consistently been found to be related to depressive symptoms in various populations (Garnefski & Kraaij, 2006; Garnefski, Kraaij, & Spinhoven, 2001; Kraaij, Pruymboom, & Garnefski, 2002; Martin & Dahlen, 2005; Nolen-Hoeksema, Parker, & Larson, 1994; Sullivan, Bishop, & Pivik, 2007), have not yet been studied in these parents. In the current study, therefore, we focused on the effects of a multitude of previously investigated and noninvestigated cognitive coping strategies on the level of stress experienced by parents of children with Down syndrome.
A shortcoming of previous studies in this area has been the lack of a prospective study design. Investigating prospective effects of coping on parental stress is important, as it provides insight in processes over time, which is especially relevant for intervention purposes. In the current study, therefore, we investigated both the cross-sectional and prospective effects of cognitive coping strategies on the stress experienced by parents of children with Down syndrome. In accordance with the literature presented above, we expected that using the strategies of blaming others for having a child with Down syndrome, catastrophizing, and rumination to a greater extent would be related to more parental stress, whereas positive reappraisal would be negatively related to parental stress.
Sample and Procedure
Participants were contacted through the Stichting Downsyndroom (SDS), an institution in The Netherlands that gives information to and organizes activities for parents of children with Down syndrome. The SDS has assembled addresses of over 3,000 couples or single parents rearing a child with Down syndrome, aged 0–18 years old. In May 2005 (Time 1 [T1]), 1,000 addresses of families were randomly selected to create the sample of this study. Each family was sent a package containing a letter to introduce the study, two questionnaires, and two prepaid envelopes (one questionnaire and prepaid envelope for each parent), enabling the parents to respond immediately if they wanted to participate. In the letter and introduction of the questionnaire, the objectives and procedure of the study were explained. Furthermore, participants were assured that they were free to stop at any time or to skip questions they explicitly did not want to answer. They were asked to sign a declaration of informed consent, which was added to the questionnaire. As an incentive, participants were informed that 10 gift tokens valued at €10 each would be raffled. They were also asked to give their permission to be contacted again for future research. If they wanted to participate in this lottery and/or give permission for future contact, they were asked to write their address on a paper that would be separated from the questionnaire, to ensure their anonymity. A telephone number and an e-mail address were provided to answer possible questions. Reminders were sent approximately 3 weeks after people received the questionnaires. Families who had already returned one questionnaire were not contacted again. In 221 cases (34.2%), only the mother returned the questionnaire, and in 38 cases (5.4%), only the father did. One hundred and eighty-one families returned both questionnaires (362 questionnaires, 58.3%). The final response rate for T1 was 31.9% (621 returned questionnaires). However, out of the 973 families who correctly received questionnaires, 440 families returned at least one questionnaire. From this viewpoint, the response rate was 45.2%.
In January 2006 (T2), the participants who had given permission to be contacted for future research (N = 381) received another questionnaire. The same procedure was followed as at T1. Reminders were sent after approximately 3 weeks, and 260 questionnaires were returned. Therefore, the response rate of T2 was 68.2%.
In Table 1, the demographic characteristics of the T1 and T2 samples are shown. Those who participated at T2 did not differ significantly on these characteristics from those who only participated at T1 except for gender (more women participated at T2), t(588) = −3.823, p = .000, and work (more participants worked part time and less worked full time at T2), χ2(4, N = 614) = 21.062, p = .000. The age of the children at T1 followed a somewhat skewed distribution to the right; 3.1% of the children were under 6 months of age at T1.
Cognitive coping strategies
To measure the specific cognitive strategies parents use when they are coping, the Cognitive Emotion Regulation Questionnaire (CERQ; Garnefski et al., 2001; Garnefski, Kraaij, & Spinhoven, 2002) was administered at T1. For this study, the CERQ was adapted to accommodate the present population, following the guidelines of the CERQ (Garnefski et al., 2002). The following instructions were provided to parents:
You have a child with Down syndrome. Every parent who has a child with Down syndrome copes with this in his or her own way. By means of the following questions, you are asked what you think about having a child with Down syndrome. Please read the sentences below and indicate how often you have the following thoughts by circling the most suitable answer.
The CERQ is a 36-item questionnaire, consisting of the following nine conceptually distinct subscales, each comprised of four items and referring to what someone thinks about a stressful life event (in this case, having a child with Down syndrome): Self-Blame, referring to thoughts of blaming yourself for having a child with Down syndrome (e.g., “I feel that I am the one to blame for having a child with Down syndrome”); Acceptance, referring to thoughts of accepting having a child with Down syndrome and resigning yourself to what has happened (e.g., “I think that I have to accept that I have a child with Down syndrome”); Rumination or Focus on Thought, referring to thinking about the feelings and thoughts associated with having a child with Down syndrome (e.g., “I often think about how I feel about having a child with Down syndrome”); Positive Refocusing, referring to thinking about joyful and pleasant issues instead of thinking about having a child with Down syndrome (e.g., “I think about pleasant experiences”); Refocus on Planning, referring to thinking about what steps to take and how to handle having a child with Down syndrome (it is the cognitive part of action-focused coping, which does not automatically imply that actual behavior will follow; e.g., “I think about a plan of what I can do best”); Positive Reappraisal, referring to thoughts of attaching a positive meaning to having a child with Down syndrome in terms of personal growth (e.g., “I think that I can become a stronger person as a result of having a child with Down syndrome”); Putting Into Perspective, referring to thoughts of playing down the seriousness of having a child with Down syndrome or emphasising its relativity compared with other things that could happen to you (e.g., “I think that having a child with Down syndrome hasn't been too bad compared with other things”); Catastrophizing, referring to thoughts of explicitly emphasizing the terror of having a child with Down syndrome (e.g., “I often think that having a child with Down syndrome is much worse than what others have experienced); and Blaming Others, referring to thoughts of putting the blame of having a child with Down syndrome on others (e.g., “I feel that others are responsible for me having a child with Down syndrome”). The strategies were measured on a 5-point Likert scale, ranging from almost never (1) to almost always (5). Individual scores were obtained by summing the scores belonging to the particular subscale. High scores on the subscales mean often using these strategies. The CERQ has been well validated (Garnefski et al., 2001, 2002) and has been found to have good internal consistencies in numerous populations, with alphas ranging from .62 to .90 (e.g., Garnefski, Baan, & Kraaij, 2005; Garnefski & Kraaij, 2007; Garnefski, Teerds, Kraaij, Legerstee, & van den Kommer, 2004; Kraaij & Garnefski, 2006). In the present study, Cronbach alphas varied from .58 to .83 (Table 2).
To measure parental stress at T1 and T2, the Nijmeegse Ouderlijke Stress Index (NOSI-K; De Brock, Vermulst, Gerris, & Abidin, 1992), a shortened version of the adapted Dutch version of the Parenting Stress Index (PSI; Abidin, 1983), was used. The NOSI is a self-report inventory that measures parental experiences of stress in the parent–child relationship. The items are divided into two major domains, a parent domain and a child domain, each containing a number of subscales that describe specific areas thought to promote parental stress. The child domain describes child characteristics that may cause stress and consists of the subscales Adaptability, Acceptability, Demandingness, Mood, Distractibility–Hyperactivity, and Reinforcement to Parent. The parent domain describes characteristics of the parent that might cause stress and consists of the subscales Sense of Competence, Restricted Role, Attachment, Depression, Parent Health, Social Isolation, and Relationship With Spouse. The shorter version was constructed by factor analysis on the original items. A selection of 25 items was made, which all loaded high on one common factor, parental stress. Within these 25 items, 10 of the original 13 scales of the NOSI are represented, and these subscales stem from both the parent and the child domains. The subscales that are not represented in the NOSI-K are Restricted Role, Social Isolation, and Relationship With Spouse (De Brock et al., 1992). Examples of items are, “I notice I am not as capable in taking care of my child as I thought I would be,” “Parenting this child is harder than I thought it would be,” and “If my child is upset, it is usually difficult to calm him/her down.” Participants are asked to rate on a 6-point Likert scale ranging from totally disagree (1) to totally agree (6) whether they agree with the 25 statements. The 25 items are totaled, with high scores reflecting a high level of parental stress. The NOSI-K has been found to discriminate well between a clinical and nonclinical sample, and the scales from which the NOSI-K items are derived have been shown to have good concurrent and discriminative validities (De Brock et al., 1992). The NOSI-K has been found to have high internal consistencies in other studies (e.g., Assher, Hermanns, Deković, & Reitz, 2007; Van der Pal et al., 2008). In the present study, Cronbach's alpha was .94 for both T1 and T2.
Family changes and changes concerning the child with Down syndrome
To control for the effect of any stressors that might have occurred during the 8 months between T1 and T2, participants were asked if any changes had occurred during these months that (a) concerned the family (i.e., birth of a child, parent has new relationship, separation–break-up, death in family, or other) or (b) the child with Down syndrome (i.e., changes in school–day-care [more or less day-care, change in schools, finished school and is now at home–has a job, or other], changes in physical complications, or other, mainly social, new problems [problems within or outside the family, psychological problems of the child, or other]). Occurrence of any of these four categories of changes was scored as absent (0) or present (1). Scores on the categories were summed to make a final score, with higher scores reflecting more changes.
To study the cross-sectional relationships between cognitive coping strategies and parental stress, a multiple regression analysis was used. To study the prospective relationships between these constructs, a hierarchical multiple regression analysis was used. To control for the effects of any relevant stressors that might have occurred between T1 and T2, this variable was entered in the first step of the hierarchical multiple regression analysis. In addition, we controlled for the level of parental stress at T1 in the first step by adding a difference score of parental stress between T1 and T2.
Prior to the main analysis, preliminary analyses were conducted. First, to examine if ages of the parent and the child had any influence on parental stress, Pearson correlations were calculated. Between parental stress at T1 and age of the parent and the child, small significant correlations of .09 were found. Because a scatter plot revealed no consistent patterns throughout the age ranges, we decided it was justified to treat all participants as one group. Next, Pearson correlations were calculated between all variables that would be entered into the regression analyses (Table 2). Although the cognitive coping strategies were significantly related, there was no evidence of logical or statistical problems due to multicollinearity, as the correlations were not higher than .7 or .9, respectively (Tabachnick & Fidell, 2001). Descriptive statistics were calculated for each of the measures (Table 3). Last, to make sure gender did not influence the analyses, an hierarchical multiple regression analysis was performed for the cross-sectional and prospective analysis, with gender as a control variable in the first step and the interaction terms between gender and coping in the third step. No main or interaction effects were found for gender on parental stress at either T1 or T2.
To investigate the cross-sectional relationships between cognitive coping strategies and parental stress, a multiple regression analysis was performed. Two participants were excluded because of a multiple standardized residual higher than 3. A total of six cognitive coping strategies proved to be significant in predicting parental stress cross-sectionally. The total model explained 22.8% of the variance. When parents used more acceptance, rumination, positive refocusing, refocus on planning, and catastrophizing, they reported more stress. When parents reported to use more positive reappraisal, this was associated with less stress (Table 4).
To investigate the prospective effects of cognitive coping on stress, a hierarchical multiple regression analysis was used (Table 4). Changes in parental stress between T1 and T2 and changes concerning the child with Down syndrome and the family were entered first, to control for their possible effects on stress. This first step proved to be significant (R2 = .065), F(2, 247) = 8.604, p < .001. In the second step, all nine cognitive coping strategies were entered. Doing so added 18.4% of explained variance to the model, which was significant. The total model now explained 24.9% of variance. Changes concerning the child with Down syndrome and the family remained significant in the final model, as did change between parental stress at T1 and T2. The cognitive coping strategies of acceptance, positive reappraisal, and catastrophizing were significant predictors of parental stress over the 8 months. Using more acceptance and catastrophizing was related to more stress; using more positive reappraisal was related to less stress.
In the current study, we investigated the cross-sectional and prospective effects of cognitive coping strategies on parental stress in a sample of 621 parents of children with Down syndrome. First, the cross-sectional relationships were studied. The analyses showed that using acceptance, rumination, positive refocusing, refocus on planning, and catastrophizing more often was related to more parental stress. Alternatively, using positive reappraisal to a higher extent was associated with less parental stress. Next, the prospective relationships were investigated. When controlling for stressful situations that occurred in the 8 months between the two measurement points and the changes in stress between T1 and T2, three coping strategies remained relevant in predicting parental stress: acceptance, positive reappraisal, and catastrophizing. Using more acceptance and catastrophizing was related to more stress, whereas using more positive reappraisal was related to less stress.
These results agree partially with previous studies. Although catastrophizing and rumination have been reported to be maladaptive strategies, positive refocusing, acceptance, and refocusing on planning are usually considered adaptive (Garnefski et al., 2001, 2002, 2004, 2006; Kraaij & Garnefski, 2006; Martin et al., 2005; Nolen-Hoeksema et al., 1994). The unexpected finding on positive refocusing might be explained by the fact that this strategy may reflect an avoidant coping style, which is usually considered maladaptive in response to a stressor that remains present (Taylor & Aspinwall, 1996). Indeed, positive refocusing is related to the more behaviorally oriented subscale Escape–Avoidance of the Ways of Coping Questionnaire (Garnefski et al., 2001), which proved to be related to more depressive symptoms and less subjective well being in mothers of children with developmental disabilities (Glidden et al., 2006).
A similar explanation can be provided for acceptance. Although it is usually considered functional to accept a situation you cannot change, as was found by Glidden et al. (2006) in fathers of children with developmental disabilities, acceptance has previously been found to be related to more depressive symptoms and might also reflect a passive “giving up” (Kraaij et al., 2002). Moreover, the measure of acceptance used by Glidden and colleagues, stemming again from the Ways of Coping Questionnaire, differs somewhat in definition from the measure used in the present study, as it has the concomitant theme of trying to “put things right.” This might also explain the different findings.
Last, refocus on planning might be maladaptive because of the uncontrollable nature of having a child with Down syndrome, as it also proved to be maladaptive in another sample that was exposed to an uncontrollable stressor (Garnefski et al., 2005). Moreover, although refocus on planning is related to problem-focused coping, a strategy that was found to be adaptive in several studies on parents of children with developmental disabilities (Abbeduto et al., 2004; Stoneman & Gavidia-Payne, 2006), it is only the cognitive counterpart of action or problem-focused coping (Garnefski et al., 2001, 2002). This means that it is not certain if actual behavior will follow. Thus, refocus on planning might actually reflect a kind of worrying or rumination instead of actively trying to do something to improve the situation.
Note that there seemed to be a suppressor effect at work for positive reappraisal: Although in the multiple regression analyses, this coping strategy was related to parental stress, there was no significant correlation between the two. This suggests that the findings for positive reappraisal should be interpreted with some caution (Tu, Gunnell, & Gilthorpe, 2008). However, that other studies have related positive reappraisal to better well being in parents of children with Down syndrome (King et al., 2000), parents of children with developmental disabilities (Glidden et al., 2006), and other populations (e.g., Garnefski & Kraaij, 2006) shows its relevance.
In addition, note that although it was hypothesized that blaming others for having a child with Down syndrome would increase perceptions of stress, this strategy was found to be of no influence. Only a few parents reported using this strategy, as reflected in the low mean score in Table 3, and this might be the reason why we did not find any effects for blaming others in the current study. To use the strategy of blaming others, it seems reasonable to assume that parents need to be able to picture someone to blame, which is not so easy in the case of having a child with Down syndrome. Indeed, in the study (Hall et al., 1997) that found an effect for blaming others, a much smaller and more specific sample was used of parents who had received a false-negative diagnosis on prenatal screening for Down syndrome.
It is interesting that the amount of explained variance for cognitive coping was nearly as high prospectively as it was cross-sectionally (18% vs. 23%). Therefore, cognitive coping strategies seem to keep having a relevant influence on the stress experienced by parents of children with Down syndrome over time, even when 8 months and relevant life changes have passed. Another interesting finding was that no gender effects were found. We believe this might be best explained by the way in which we investigated these gender differences. Whereas other investigators have performed separate regression analyses for mothers and fathers to investigate the effect of gender (e.g., Essex, Seltzer, & Krauz, 1999; Glidden et al., 2006; Hall et al., 1997), we investigated the effect of gender more directly by testing the interaction effects between gender and the several coping strategies. This has the advantage of being able to test if the effect of the coping strategies differs for men and women. When using the other strategy of analysis, it is possible to detect different relationships between coping and parental stress for men and women, but it is not possible to test if these differences are significant.
This study suffers from a number of limitations that should be addressed. First, all data were gathered using self-report, which may have caused some bias. Second, the parents that decided to participate at T1 might have differed significantly from the parents that did not or from those who were not invited to join in on this study because they were not a member of the Dutch Down Syndrome Foundation. This may have caused a selection bias, which means that the results should be generalized with care. Third, the parental stress scores at T1 and T2 were highly correlated, which might be due to the relatively short time interval of 8 months. Future researchers are encouraged to perform prospective studies with larger time intervals. In addition, it should be noted that the Cronbach's alphas of two of the coping strategies—self-blame and blaming others—were relatively low. The reliabilities for self-blame and blaming others in other studies using the CERQ have usually been better (Garnefski et al., 2002). A possible explanation might be that using these strategies might not be as straightforward or common as it would be when one is coping with other types of life events. This might be interesting food for thought for future studies. Last, other factors that may explain levels of stress in parents of children with Down syndrome (e.g., personality characteristics, social support) were not taken into account. Future studies should investigate the influence of these factors as well.
Despite these limitations, this study benefits from its prospective design and shows that catastrophizing, positive reappraisal and acceptance can influence how much stress parents experience over time. Intervention and prevention programs for parents of children with Down syndrome may succeed by trying to discourage parents from catastrophizing the fact that they have a child with Down syndrome. Other studies are needed to sort out whether teaching parents to positively reappraise their having a child with Down syndrome actually reduces parental stress. Similarly, the surprising result concerning acceptance, a strategy that intuitively seems adaptive but here proved to be maladaptive both cross-sectionally and prospectively, should be further investigated before any recommendations for interventions can be made. Apart from the fact that acceptance may reflect a passive giving-up, it might be useful to investigate differential effects and personality characteristics that might influence this strategy. If future research can replicate the current findings for measures of well being, such as anxiety, depression, and/or positive emotions, the present results could contribute to intervention and prevention programs for parents who are struggling to cope with having a child with Down syndrome.
Editor-in-Charge: Steven J. Taylor
Shelley M. C. van der Veek, MSc, PhD Candidate (SVeek@fsw.leidenuniv.nl or email@example.com), Academic Medical Center Amsterdam, Leiden University, Department of Clinical and Health Psychology, P.O. Box 9555, 2300 RB Leiden, The Netherlands. Vivian Kraaij, PhD, Assistant Professor, Medical Psychology Department, Leiden University Medical Center, Leiden, The Netherlands. Nadia Garnefski, PhD, Associate Professor, Clinical and Health Psychology Department, Leiden University, Leiden, The Netherlands.