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Health related quality of life in parents of six to eight year old children with Down syndrome
Research in Developmental Disabilities 34 (2013) 4239–4247
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Research in Developmental Disabilities
Health related quality of life in parents of six to eight year old children with Down syndrome Jan Pieter Marchal a, Heleen Maurice-Stam a, Janneke Hatzmann a, A.S. Paul van Trotsenburg b, Martha A. Grootenhuis a,* a b
Academic Medical Center/Emma Children’s Hospital, Psychosocial Department, Amsterdam, The Netherlands Academic Medical Center/Emma Children’s Hospital, Department of Endocrinology, Amsterdam, The Netherlands
A R T I C L E I N F O
A B S T R A C T
Article history: Received 13 May 2013 Received in revised form 4 September 2013 Accepted 4 September 2013 Available online
Raising a child with Down syndrome (DS) has been found to be associated with lowered health related quality of life (HRQoL) in the domains cognitive functioning, social functioning, daily activities and vitality. We aimed to explore which socio-demographics, child functioning and psychosocial variables were related to these HRQoL domains in parents of children with DS. Parents of 98 children with DS completed the TNO-AZL adult quality of life questionnaire (TAAQOL) and a questionnaire assessing socio-demographic, child functioning and psychosocial predictors. Using multiple linear regression analyses for each category of predictors, we selected relevant predictors for the final models. The final multiple linear regression models revealed that cognitive functioning was best predicted by the sleep of the child (b = .29, p < .01) and by the parent having given up a hobby (b = .29, p < .01), social functioning by the quality of the partner relation (b = .34, p < .001), daily activities by the parent having to care for an ill friend or family member (b = .31, p < .01), and vitality by the parent having enough personal time (b = .32, p < .01). Overall, psychosocial variables rather than socio-demographics or child functioning showed most consistent and powerful relations to the HRQoL domains of cognitive functioning, social functioning, daily activities and vitality. These psychosocial variables mainly related to social support and time pressure. Systematic screening of parents to detect problems timely, and interventions targeting the supportive network and the demands in time are recommended. ß 2013 Elsevier Ltd. All rights reserved.
Keywords: Down syndrome Parents Health related quality of life Predictors Cross-sectional study Social support Leisure
1. Introduction Down syndrome (DS) is characterized by developmental disability and is associated with various health problems such as congenital heart disease, hearing loss and endocrine disorders (Roizen & Patterson, 2003). The estimated birth prevalence of Down syndrome in the Netherlands is 14–16 per 10,000 live births, resulting in an annual estimated 275–322 live births of children with DS (De Graaf et al., 2011; Weijerman et al., 2008). The moment that parents are first informed about the condition of their child is an emotional and stressful event (Hedov, Wikblad, & Annere´n, 2002). Aside from the initial emotional impact, children with DS also have an influence on family life, since they need extra care throughout their life, which is most often provided by parents (Crowe & Florez, 2006). It is therefore not surprising that raising a child with DS has been found to be associated with higher levels of parental stress and reduced well-being in parents (Dabrowska & Pisula,
* Corresponding author at: Psychosocial Department, Emma Children’s Hospital, Academic Medical Center, A3-241, Post Box 22660, 1100 DD Amsterdam, The Netherlands. Tel.: +31 20 566 5674. E-mail address: [email protected] (M.A. Grootenhuis). 0891-4222/$ – see front matter ß 2013 Elsevier Ltd. All rights reserved. http://dx.doi.org/10.1016/j.ridd.2013.09.011
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2010; Hedov, Annere´n, & Wikblad, 2002; Sloper, Knussen, Turner, & Cunningham, 1991). Yet, some authors suggest that the impact of raising a child with DS is relatively mild compared to raising children with other disabilities (Dumas, Wolf, Fisman, & Culligan, 1991; Hodapp, Ly, Fidler, & Ricci, 2001). In addition, it is important to note that families of children with DS also report positive perceptions of the impact of raising a child with DS (Hastings & Taunt, 2002; Skotko, Levine, & Goldstein, 2011). Several studies identified factors that were associated with higher levels of stress and reduced well-being in parents of children with DS. These include socio-demographic factors like lower socio-economic status, a higher number of children and (higher or lower) age of the child with DS (Dabrowska & Pisula, 2010; Fidler, Hodapp, & Dykens, 2000; Hodapp, Ricci, Ly, & Fidler, 2003; Sloper et al., 1991). Also, child functioning variables such as behavioral problems, communicative impairment, health problems and diminished self-sufficiency of the child were found to be of negative influence (Abbeduto et al., 2004; Bourke et al., 2008; Hodapp et al., 2003; Sloper et al., 1991). Finally, psychosocial factors such as maladaptive coping styles of parents, less dispositional optimism, lower quality of the (marital) relationship, and a lack of social support were associated with negative consequences (Greenberg, Seltzer, Krauss, Chou, & Hong, 2004; Hauser-Cram, Warfield, Shonkoff, & Krauss, 2001; Sloper et al., 1991; van der Veek, Kraaij, & Garnefski, 2009a). All of these findings provide valuable insight into the issues parents face. Yet, the outcome-measures stress and well-being cannot depict the wide spectrum of possible affected and unaffected domains of life. For a more extensive evaluation of parental functioning, health related quality of life (HRQoL) is a useful concept. HRQoL is a widely adopted multidimensional concept that emerged during the 1980s, and comprises a range of possible domains, including physical, emotional and intellectual functioning, the ability to participate in valued activities within the family, in the workspace and in community, and the resulting sense of well-being and comfort (Naughton & Shumaker, 2003; Patrick & Erickson, 1988). Although HRQoL is frequently used to evaluate the functioning of parents with children with various disorders, few reports on the HRQoL of parents of children with DS are available. The HRQoL of parents of children with chronic conditions, both with or without developmental disabilities, has been found to be lower compared to parents of children without chronic conditions, with different disorders being associated with varying profiles of HRQoL in parents (Davis & Gavidia-Payne, 2009; Hatzmann, Heymans, Ferrer-iCarbonell, van Praag, & Grootenhuis, 2008; Hatzmann, Valstar, et al., 2009; Khanna et al., 2011; Lin et al., 2009). Concerning HRQoL in parents of children with DS, a Swedish study showed that mothers of children with DS reported poorer vitality than their spouses, and poorer vitality and mental health than mothers in the control group (Hedov, Annere´n, & Wikblad, 2000). In two small, exploratory studies, Brazilian parents of children with DS scored lower in the psychological and environmental domain of the WHOQOL-BREF questionnaire, the latter domain encompassing financial means, housing and opportunities for recreation (Buzatto & Beresin, 2008; Murphy, Herrman, Hawthorne, Pinzone, & Evert, 2000; Oliveira & Limongi, 2011). The Dutch Care-project, of which we employed the database for our current study, revealed that parents of children with DS scored lower in the domains cognitive and social functioning, daily activities and vitality than parents of children without a chronic condition (Hatzmann et al., 2008). In summary, all these HRQoL-studies showed that parents of children with DS tend to experience lowered HRQoL regarding vitality, leisure (social functioning, daily activities, and recreation), and mental- or psychological health. However, none of the above-mentioned studies analyzed predictors of parental HRQoL thoroughly. Predictors of HRQoL have been studied in the broader group of parents of children with developmental disabilities, which also comprises children with DS. Factors that were found to be associated with lower HRQoL show considerable overlap with those related to parental stress and well-being in parents of children with DS. These factors concern mainly child functioning and psychosocial variables. A lower household income, higher levels of functional impairment and behavioral problems of the child, higher experienced burden, less social support, maladaptive coping style of parents, less participation in health promoting activities, and poorer professional support were all related to lower HRQoL outcomes (Bourke-Taylor, Pallant, Law, & Howie, 2012; Browne & Bramston, 1998; Davis & Gavidia-Payne, 2009; Khanna et al., 2011; Lin et al., 2009). For the current study, aimed at tracing factors that are associated with HRQoL in parents of children with DS, we analyzed the data from the Care-project, described in Hatzmann et al. (2008). As a conceptual framework for variables that could be related to HRQoL of parents of children with DS, we used the model of Hatzmann, Maurice-Stam, Heymans, and Grootenhuis (2009). In their model, variables were clustered as ‘socio-demographics’, ‘disease related variables’, and a range of psychosocial mediating variables (e.g. leisure time and emotional support). Since our sample size was more limited, we could not analyze mediation. We therefore employed these as direct predictors of HRQoL instead of analyzing mediation, and renamed those variables ‘psychosocial variables’. Finally, ‘disease related variables’ were renamed into ‘child functioning’ because this covers the meaning of the concerning variables in our sample. This resulted in a model comprising three clusters of variables that were hypothesized to be directly related to HRQoL: ‘socio-demographic variables’, ‘child functioning’ and ‘psychosocial variables’. The objective of our study was to determine whether socio-demographics, child functioning and psychosocial variables were related to cognitive functioning, social functioning, daily activities and vitality, the HRQoLdomains in which parents of children with DS scored lower than parents of children without a chronic condition. 2. Materials and methods 2.1. Participants The data regarding HRQoL of parents of children with DS were gathered within the scope of the Care-project: a large, cross-sectional study on the consequences for parents of caring for chronically ill children, including parents of 100 children
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with DS (Hatzmann et al., 2008). The parents of children with DS were recruited in two ways. The majority was recruited from a cohort of 196 children born between 1999 and 2001 who were studied and described within the scope of an RCT by Van Trotsenburg et al. (2005). Parents of 177 children with DS (19 lost to follow up or unknown addresses) were sent an introductory letter explaining the aim of the study and inviting them to complete the enclosed HRQoL questionnaire and informed consent, and to return these using the enclosed return envelope. Out of this cohort, 98 parents responded. In addition, two parents were recruited in the outpatient clinic of Academic Medical Center. Each family received one questionnaire, and it was not specified whether father or mother was requested to respond. Since we aimed to study the HRQoL in parents of children with DS within a relatively narrow age range, the two parents who were recruited from the outpatient clinic were excluded from our analyses. The calendar ages of their children (14.6 and 16.2 years) differed widely from that of the rest of the sample (range 6.0–8.4 years). The Medical Ethical Committee of the Academic Medical Center Amsterdam approved the CARE project. 2.2. Measurement 2.2.1. Outcomes: HRQoL To assess HRQoL, the ‘TNO-AZL Questionnaire for Adult’s Health Related Quality Of Life’ (TAAQOL) was used (Bruil, Fekkes, Vogels, & Verrips, 2004). This questionnaire was developed by the Netherlands Organization for Applied Scientific Research (TNO) and the Leiden University Medical Center. The TAAQOL is a validated, generic HRQoL questionnaire which consists of 45 items that address 12 domains: gross and fine motor functioning, cognitive functioning, sleep, pain, social functioning, daily activities, sexuality, vitality, positive emotion, depressive emotions and aggressiveness. The four domains we used as outcomes (cognitive functioning, social functioning, daily activities, and vitality) consist of four items each. These items consist of two parts: the first part assesses whether a health status problem is present, with answers on a four-point scale ranging from ‘no/never’ to ‘a lot/always’. If the answer on this first part is affirmative, the second part inquires the subjective response to the reported problem (‘how much did that bother you?’), with answers on a four-point scale ranging from ‘not at all’ to ‘very much’. The scores in each domain are to be added up, and transformed to a scale score between 0-100, with higher scores indicating better functioning. The Cronbach’s alphas of the four domains of the TAAQOL we used were ranging from .85 to .88 in the norm sample (Bruil et al., 2004). In our sample, Cronbach’s alphas for the four domains ranged from .85 to .91. 2.2.2. Predictors Predictors of HRQoL were drawn from self-formulated questions in the Care-questionnaire, which was developed for the entire group of parents of children with chronic diseases, most of which were not associated with developmental disability, and not specifically for parents of children with DS. A pediatrician and a psychologist listed all available predictors from the Care-questionnaire, and independently selected predictors they deemed relevant for HRQoL in parents of children with DS, based on literature and their clinical experience. After this, the final selection of predictors was made by consensus. As mentioned earlier, predictors were clustered as ‘socio-demographic variables’, ‘child functioning’ and ‘psychosocial variables’: 2.2.2.1. Socio-demographic variables (10 items). (1) Gender of the parent, (2) age of the parent, (3) gender of the child, (4) age of the child, (5) current relationship status of the respondent: married/living together, separated/divorced/widowed, single or other, (6) low educational level of the parent: low versus intermediate and high educational level (educational level was defined as the highest educational level completed; low: primary education, lower or middle general secondary education; intermediate: middle vocational education, higher secondary education, or pre-university education; high: higher vocational education, or university), (7) chronically ill parent – yes/no, (8) country of birth of the parent: the Netherlands versus elsewhere, (9) total number of children living at home, and (10) current employment status of the respondent: paid employment – yes/no. 2.2.2.2. Child functioning (4 items). (1) Care dependency: the number of domains of life where the child needs assistance, with the number of domains ranging from 0 to 8: physical care, mobility, eating and drinking, medication use, coping with devices, amusing him/herself, contact with other children, and education. Cronbach’s alpha of this scale was .70 (2) sleep: average number of hours of sleep per night of the child with DS, (3) diaper during daytime – yes/no, and (4) difficulties making contact: ‘did your child ever show problems in making contact with you? (e.g. does not seek contact, or does so in a strange way)’ – yes/no. 2.2.2.3. Psychosocial variables (8 items). (1) Emotional support from partner, family, friends and neighbors, each rated on a three-point scale with 0 = ‘no support’ and 2 = ‘very good support’. The sum of these scores resulted in the variable ‘Emotional support’, ranging from 0 to 8. Cronbach’s alpha of this scale was .60. (2) Quality of the partner relation: constructed from five questions: ‘do you and your partner agree on how to raise your child(ren)?’; ‘do you and your partner agree on how to share the care for your child(ren)?’; ‘can you discuss any problem or worry with your partner?’; ‘if you disagree with your partner, can you come to a resolve?’; ‘do you think you and your partner have enough time for each other?’. Answers on these questions were rated on four-point scales, ranging from 0 = ‘(almost) never’ to 3 = ‘(almost) always’. The sum of these scores resulted in the ‘Partner relation’ variable, ranging from 0 to 15. Cronbach’s alpha of this scale was .83. Other psychosocial
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variables were: (3) enough personal time: ‘do you feel you have enough time for yourself? – yes/no, (4) given up hobby since birth of child – yes/no, (5) loss of friendship since diagnosis – yes/no, (6) job adjustment due to condition of child – yes/no, (7) caring for an ill friend or family member (not child or partner) – yes/no, and (8) adverse life events: ‘have you ever experienced an adverse life event, such as losing your partner or a close family member, a car accident or major financial problems?’ – yes/no. 2.3. Statistical analysis Scales of the TAAQOL were constructed and missing data were imputed based on the guidelines provided by the TAAQOL manual. Scales of the predictor variables were constructed as described in Section 2.2.2. Internal consistency of all scales was assessed by the Cronbach’s alpha. Subsequently, descriptive analyses were performed for socio-demographics, child functioning and psychosocial variables. To limit the number of predictors in the final predictive model, we selected variables for each of the four HRQoL-domains using multiple linear regression analyses, separately for each of the three clusters of predictors: socio-demographic variables, child functioning and psychosocial variables. The condition for selection of a variable for the final regression analysis was set at p < .15. The assumptions for multiple linear regression analyses were met. The selected variables were used in the final multiple linear regression analyses to predict the HRQoL outcomes, resulting in four multiple linear regression models: one for each HRQoL-outcome. In all multiple linear regression models, a significance level of .05 was deemed significant. Given the exploratory nature of this study, no correction for multiple testing was applied. Standardized regression coefficients of .1 were considered small, .3 medium and .5 large for continuous variables, after Cohen (1988). For binary coded variables, standardized regression coefficients of .2 can be considered small, .5 as medium and .8 as large. The explained variances (R2) were determined of all multiple linear regression models. All data in this study were analyzed using SPSS version 20. 3. Results 3.1. Participants Table 1 shows the socio-demographic characteristics of the parents and children with DS. Of our participants, 86% was mother and 98% was married or living together. In our sample, 41% of the parents received higher education, and 63% had a paid employment. Functioning of the children with DS and psychosocial variables of their parents are shown in Table 2. 3.2. Predictors of parental HRQoL Table 3 shows the results of the linear regression analyses we used to select variables for the final regression models: standardized regression coefficients corresponding to variables that met criteria for selection (p < .15) are presented in bold. Table 4 presents the results of the final linear regression models. All final models were highly significant (p < .001) with explained variances ranging from 37% to 57%. Significant predictors had standardized regression coefficients of small to medium size (.17–.34). The most powerful predictors were psychosocial variables. Some child functioning variables also contributed significantly to the model, but none of the socio-demographic variables appeared as significant predictors in the final model. Cognitive functioning was best predicted by the number of hours of sleep per night of the child (b = .29, p < .01), and by whether the parent had given up a hobby since the birth of the child with DS (b = .29, p < .01). Apart from that, enough personal time (b = .19, p = .03) and more emotional support (b = .17, p = .04) predicted better cognitive functioning, while loss of friendship (b = .19, p = .03) predicted poorer cognitive functioning. Social functioning was best predicted by the quality of the partner relation (b = .34, p < .001). Further, emotional support (b = .22, p = .02) and having enough personal time (b = .19, p = .04) were related to better social functioning. The score for daily activities was best predicted by whether the parent had to care for an ill friend or family member (b = .31, p < .01). Besides that, enough personal time (b = .24, p = .02) predicted higher scores for daily activities. For vitality, the parent having enough personal time (b = 0.32, p < 0.01) was the most powerful predictor for better functioning. The child wearing a diaper during daytime (b = .23, p = .02) and loss of friendship (b = .20, p = .03) were related to poorer vitality. 4. Discussion The objective of this study was to identify variables that are related to HRQoL regarding cognitive functioning, social functioning, daily activities and vitality of parents of children with DS. In conclusion we found that mainly psychosocial variables were associated with these HRQoL outcomes, rather than child functioning or socio-demographic variables. To our knowledge, this is the first study that analyzed factors related to HRQoL in parents of children with DS. The finding that socio-demographics did not show strong relations with HRQoL is in line with most of the literature concerning stress in parents of children with DS, and concerning HRQoL in parents of children with chronic disorders or developmental disabilities (Hatzmann, Maurice-Stam, et al., 2009; Sloper et al., 1991).
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Table 1 Socio-demographic characteristics. Socio-demographic Gender parent (n, %) Male Female Age parent (range) (M, SD) Gender child (n, %)a Male Female Age child (range) (M, SD) Current relationship status (n, %)a Married/living together Separated/divorced/widowed Single Other Educational level of the parent (n, %)a High Intermediate Low Chronically ill parent (n, %)a Yes No Country of birth of the parent (n, %)a Netherlands Elsewhere Total number of children (M, SD)a One (n, %) Two (n, %) Three (n, %) >Three (n, %) Paid employment (n, %) Yes No a b
14 (14%) 84 (86%) 32.4–51.9 years 41.3 years (4.4) 56 (60%) 38 (40%) 6.0–8.4 yearsb 7.1 years (0.6) 96 1 1 0
(98%) (1%) (1%) (0%)
40 (41%) 38 (39%) 19 (20%) 12 (13%) 84 (88%) 91 (94%) 6 (6%) 2.5 (1.0) 9 (9%) 47 (48%) 29 (30%) 12 (12%) 62 (63%) 36 (37%)
Numbers not adding up to the total number of participants (N = 98) caused by missing values. n = 95.
Child functioning was clearly related to the outcome variables in the models we used for selecting variables. Yet, in the final model, which included psychosocial variables as well, the predictive value of child functioning diminished. The level of care dependency, i.e. the number of domains the child needs practical help on, did not predict HRQoL in the final model. This is very similar to what Hatzmann, Maurice-Stam, et al. (2009) found in parents of children with metabolic diseases (some of which comprise developmental disabilities). Yet, Sloper et al. (1991) found that self-sufficiency of the child with DS, assessed with a 317-item scale, was associated with maternal satisfaction with life, but not with the paternal satisfaction with life. Perhaps, these latter findings cannot be generalized to the current situation of children with DS in the Netherlands, with a different standard of care and changed attitudes in society toward children with DS. Other studies showed that, with regard to child functioning, in particular behavioral problems are related to parental stress or well-being in parents of children with DS (Abbeduto et al., 2004; Hauser-Cram et al., 2001; Hodapp et al., 2003). Given the wide spectrum of chronic illnesses in the Care project, most of which were not associated with developmental disability, behavioral problems of the children were not assessed in this project. However, some of our variables, such as sleep and wearing a diaper during daytime may reflect behavioral problems in the children. Sleep disturbance, for instance, has been found to be related to behavioral problems in children with typical development, and to a range of psychiatric problems in children with DS (Gregory, Eley, O’Connor, & Plomin, 2004; Richdale, Francis, Gavidia-Payne, & Cotton, 2000; Stores & Stores, 2013). Also, an association between incontinence and behavioral problems has been described in children without developmental disabilities (von Gontard, Mauer-Mucke, Plu¨ck, Berner, & Lehmkuhl, 1999). Psychosocial variables showed most consistent and most powerful relations with parental HRQoL. This finding is in line with previous findings regarding predictors for stress in parents of children with DS (Dabrowska & Pisula, 2010; Sloper et al., 1991) and with findings in parents of children with other chronic conditions (Ten Hoedt et al., 2011). The beneficial relation between a supportive social network and well-being is well-established in parents of children with DS, and in parents of children with various other conditions (Dunst, Trivette, & Cross, 1986; Hatzmann, Valstar, et al., 2009; Hauser-Cram et al., 2001; Khanna et al., 2011; Sloper et al., 1991; Van der Veek, Kraaij, & Garnefski, 2009b). Our finding that a stronger partner relation is associated with better HRQoL in the social domain is in line with studies that showed the predictive value of the partner relation for HRQoL (Wiedebusch et al., 2010; Wiedebusch, Pollmann, Siegmund, & Muthny, 2008), for well-being in parents of children with DS (Van der Veek et al., 2009b). Van der Veek et al. (2009b) suggested that in parents of children with DS the positive effect of a supportive partner relation could be seen as a ‘‘constant condition that may or may not facilitate a
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Table 2 Functioning of children with Down syndrome and psychosocial variables of their parents. N = 98 Child functioning Care dependency score (M, SD) Physical care (n, %) Movement/mobility (n, %) Eating and drinking (n, %) Medication use (n, %) Coping with devices (n, %) Amusing him/herself (n, %) Contact with other children (n, %) Education (n, %) Sleep (range)a (M, SD) Diaper during daytime (n, %) Yes No Difficulties making contact (n, %)b Yes No Psychosocial variables Emotional support (range)a (M, SD) Quality of partner relation (range) (M, SD) Enough personal time (n, %)b Yes No Given up hobby (n, %)b Yes No Loss of friendship (n, %)b Yes No Job adjustment (n, %) Yes No Caring for ill friend/family member (n, %)b Yes No Adverse life event (n, %) Yes No a b
3.6 (1.8) 71 (72%) 21 (21%) 42 (43%) 26 (27%) 6 (6%) 53 (54%) 50 (50%) 86 (88%) 3–13 h 9.8 h (1.9) 36 (37%) 62 (63%) 18 (18%) 75 (77%)
0–8 5.5 (1.9) 2–15 10.9 (2.9) 27 (29%) 67 (71%) 27 (30%) 64 (70%) 31 (33%) 62 (67%) 70 (71%) 28 (29%) 8 (8%) 87 (92%) 79 (81%) 19 (19%)
n = 94. Numbers not adding up to the total number of participants (N = 98) is caused by missing values.
positive adaptation, as it may be a potential source of relief or stress on its own’’. A strong partner relation relating specifically to the HRQoL domain of social functioning may be explained by the willingness of a partner to make arrangements regarding the care and support the other in maintaining their social network. Also, parents with a strong social network may have the opportunity of sharing the burden of their worries and care with more people than just their partner, and so reduce stress in their marital relationship. The importance of loss of friendships, together with emotional support and the quality of the partner relation underline the relevance of a supportive social network around the parent, be it the partner, friends, or family and neighbors. Our results also highlight the importance of having enough time for leisure. Parents who reported having enough personal time showed better HRQoL, while having given up a hobby was related to poorer cognitive functioning. Also, caring for an ill friend or family member was related to lower scores for daily activities, which can be explained by the burden in terms of time too. Interestingly, a Dutch study showed that providing informal care for more than 11 h a week was associated with lower overall life satisfaction, although providing care for less than 6 h a week, and combining it with paid employment lead to higher life satisfaction (Van Campen, de Boer, & Iedema, 2013). Also, a previous study that assessed HRqoL in parents of children with DS using the WHOQOL-BREF measure, found that parents scored lower on environmental domain, which encompasses opportunities for recreation (Oliveira & Limongi, 2011). These results all relate to the time-pressure parents have to deal with. In our sample, more than two-thirds (71%) reported not having enough personal time. Several other studies showed that parents of children with DS spend more time on child care, and less on recreational and social activities than parents of children without a disorder (Barnett & Boyce, 1995; Crowe & Florez, 2006; Hedov, Annere´n, et al., 2002; O’Mullan Wayne & Krishnagiri, 2005; Sanders & Morgan, 1997). In a study asking parents with DS which important needs
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Table 3 Selection of variables for the final predictive model of HRQoL: standardized regression coefficients (b). Cognitive functioning Socio-demographic variables (b) Gender parent (male) Age parent Gender child (male) Age child Low educational level Chronically ill parent Country of birth (elsewhere) Total number of children Paid employment 2 R F (df) Child functioning (b) Care dependency Sleep Diaper during daytime Difficulties making contact R2 F (df) Psychosocial variables (b) Emotional support Quality of partner relation Enough personal time Given up hobby Loss of friendship Job adjustment Caring for ill friend/family member Adverse life event R2 F (df)
.09 .04 .10 .15 .30** .13 .07 .00 .13 .10 1.07 (9, 77)
Social functioning
Daily activities
Vitality
.14 .01 .10 .14 .09 .23* .04 .03 .02 .10 .94 (9, 77)
.08 .05 .08 .16 .16 .12 .01 .12 .00 .07 .63 (9, 77)
.11 .12 .07 .25** .21* .21* .01 .16 .14 .16 1.59 (9, 77)
.24** .36**** .01 .16* .33 10.46 (4, 84)****
.11 .14 .05 .27** .18 4.46 (4, 84)***
.09 .24** .18* .12 .17 4.15 (4, 84)***
.13 .17* .21** .18* .20 5.11 (4, 84)***
.24** .04 .30*** .35*** .23** .12 .00 .01 .48 7.85 (8, 69)****
.22** .31*** .17* .25** .06 .05 .06 .10 .48 7.83 (8, 69)****
.20* .08 .17* .10 .16* .02 .29*** .15 .38 5.20 (8, 69)****
.23** .16 .28** .13 .22** .09 .00 .05 .37 5.13 (8, 69)****
Note. For selected variables (p < .15), the corresponding standardized regression coefficients are printed in bold. * p < .15. ** p < .05. *** p < .01. **** p < .001.
Table 4 Final multiple linear regression models for parental HRQoL: standardized regression coefficients (b). Cognitive functioning Socio demographic variables (b) Age child Low educational level Chronically ill parent Total number of children Child functioning (b) Care dependency Sleep Diaper during daytime Difficulties making contact Psychosocial variables (b) Emotional support Quality of partner relation Enough personal time Given up hobby Loss of friendship Caring for ill friend/family member R2 F (df)
.16
Social functioning
Daily activities
.13 .11 .04
.04
.04 .29***
.18 .15
.06
.14
.17**
.22** .34**** .19** .19
**
.19 .29*** .19** .57 11.57 (8, 71)****
.46 10.77 (6, 76)****
Vitality
.11 .23** .15
.14
.18 **
.24
.18 .31*** .37 7.19 (6, 74)****
Note: For significant predictors, the variable names and corresponding standardized regression coefficients are presented in bold. ** p < .05. *** p < .01. **** p < .001.
.32*** .20** .46 6.60 (9, 71)****
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were not or only partly met, 75% reported to want a break from their responsibilities, and 72% needed weekend and afterschool activities for their child (Siklos & Kerns, 2006). Recently, Bourke-Taylor et al. (2012) found that in mothers of children with developmental disabilities, participation in health-promoting leisure activities was related to better HRQoL outcomes. So, it seems a quite consistent finding that the parents feel they need more time for themselves, but have difficulty arranging this. Our results showed that although it may be challenging to take time for oneself due to the demands of everyday life, it seems important for the well-being of parents of children with DS. One of the limitations of our study was the limited number of predictors we could include in our regression analyses to obtain reliable results. Further, we did not define whether fathers or mothers were requested to complete the questionnaires, which resulted in a high percentage of mothers participating. Since the impact of raising a child with DS has been found to differ between mothers and fathers, it is recommended to include both parents of each child in future research (Hedov et al., 2000; Hornby, 1995; Roach, Orsmond, & Barratt, 1999). Also, future research aiming to construct a more complete predictive model should incorporate other factors that are expected to predict HRQoL in parents of children with DS. These include in particular behavioral problems of the child, which were not included in the current study, because of the broad spectrum of disorders included in the Care project the present study was part of. Most disorders were not associated with developmental disabilities or behavioral disturbances. Also parental stress, parental coping styles or personality features such as dispositional optimism could be included in a more complete model. Moreover, instead of self-formulated questionnaires and self-constructed scales, standardized and validated questionnaires should be used if available. Finally, since age of the child has been identified as a predictor for parental stress in previous studies, the generalizability of our results to parents of children with DS in age categories other than in the present study is limited. It is therefore recommended to study HRQoL of these parents longitudinally, since this will provide insight in the changes in HRQoL over time, and possibly in the causality and direction of the associations (Haveman, van Berkum, Reijnders, & Heller, 1997). Despite these limitations, our findings showed that HRQoL-outcomes of parents of six to eight years old children with DS were mainly associated with psychosocial variables relating primarily to social support and time pressure. Health care providers should be aware of the risk for lowered HRQoL in parents of children with DS. To pay systematic attention to possible psychosocial problems in order to identify parents most at risk, we recommend the use of valid, short screening instruments. As we found social support and creating enough (leisure) time for oneself to be related to better HRQoL outcomes, interventions should target these social and personal resources. For instance, social workers could pay attention to the informal support network of parents and to the quality of the relationship with their partner, and they should stimulate parents to take time for activities with family, friends, or neighbors, or to connect to groups of other parents of children with DS (Law, King, Stewart, & King, 2001). Also, attention should be paid to the time-pressure parents face, and the amount of time available for leisure. Most likely it is hard for parents to create personal time. For this reason parents could be referred to respite care services or be stimulated to find similar help in their informal network. Respite care comprises interventions aimed at temporarily easing the burden of the caregiving task by, for instance, providing overnight residential care for a weekend (Caples & Sweeney, 2011; Chan & Sigafoos, 2001; Douma, Dekker, & Koot, 2006). In conclusion, we found psychosocial variables rather than socio-demographics and child functioning to be most consistently and powerfully related to HRQoL in parents of children with DS. These psychosocial variables were primarily related to social support and time pressure. Health care providers should be aware that parents of children with DS are at risk for lower HRQoL. Systematic screening of parents to detect problems at an early stage, and interventions that target the supportive network and the demands in time are recommended.
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Contents lists available at ScienceDirect
Research in Developmental Disabilities
Health related quality of life in parents of six to eight year old children with Down syndrome Jan Pieter Marchal a, Heleen Maurice-Stam a, Janneke Hatzmann a, A.S. Paul van Trotsenburg b, Martha A. Grootenhuis a,* a b
Academic Medical Center/Emma Children’s Hospital, Psychosocial Department, Amsterdam, The Netherlands Academic Medical Center/Emma Children’s Hospital, Department of Endocrinology, Amsterdam, The Netherlands
A R T I C L E I N F O
A B S T R A C T
Article history: Received 13 May 2013 Received in revised form 4 September 2013 Accepted 4 September 2013 Available online
Raising a child with Down syndrome (DS) has been found to be associated with lowered health related quality of life (HRQoL) in the domains cognitive functioning, social functioning, daily activities and vitality. We aimed to explore which socio-demographics, child functioning and psychosocial variables were related to these HRQoL domains in parents of children with DS. Parents of 98 children with DS completed the TNO-AZL adult quality of life questionnaire (TAAQOL) and a questionnaire assessing socio-demographic, child functioning and psychosocial predictors. Using multiple linear regression analyses for each category of predictors, we selected relevant predictors for the final models. The final multiple linear regression models revealed that cognitive functioning was best predicted by the sleep of the child (b = .29, p < .01) and by the parent having given up a hobby (b = .29, p < .01), social functioning by the quality of the partner relation (b = .34, p < .001), daily activities by the parent having to care for an ill friend or family member (b = .31, p < .01), and vitality by the parent having enough personal time (b = .32, p < .01). Overall, psychosocial variables rather than socio-demographics or child functioning showed most consistent and powerful relations to the HRQoL domains of cognitive functioning, social functioning, daily activities and vitality. These psychosocial variables mainly related to social support and time pressure. Systematic screening of parents to detect problems timely, and interventions targeting the supportive network and the demands in time are recommended. ß 2013 Elsevier Ltd. All rights reserved.
Keywords: Down syndrome Parents Health related quality of life Predictors Cross-sectional study Social support Leisure
1. Introduction Down syndrome (DS) is characterized by developmental disability and is associated with various health problems such as congenital heart disease, hearing loss and endocrine disorders (Roizen & Patterson, 2003). The estimated birth prevalence of Down syndrome in the Netherlands is 14–16 per 10,000 live births, resulting in an annual estimated 275–322 live births of children with DS (De Graaf et al., 2011; Weijerman et al., 2008). The moment that parents are first informed about the condition of their child is an emotional and stressful event (Hedov, Wikblad, & Annere´n, 2002). Aside from the initial emotional impact, children with DS also have an influence on family life, since they need extra care throughout their life, which is most often provided by parents (Crowe & Florez, 2006). It is therefore not surprising that raising a child with DS has been found to be associated with higher levels of parental stress and reduced well-being in parents (Dabrowska & Pisula,
* Corresponding author at: Psychosocial Department, Emma Children’s Hospital, Academic Medical Center, A3-241, Post Box 22660, 1100 DD Amsterdam, The Netherlands. Tel.: +31 20 566 5674. E-mail address: [email protected] (M.A. Grootenhuis). 0891-4222/$ – see front matter ß 2013 Elsevier Ltd. All rights reserved. http://dx.doi.org/10.1016/j.ridd.2013.09.011
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2010; Hedov, Annere´n, & Wikblad, 2002; Sloper, Knussen, Turner, & Cunningham, 1991). Yet, some authors suggest that the impact of raising a child with DS is relatively mild compared to raising children with other disabilities (Dumas, Wolf, Fisman, & Culligan, 1991; Hodapp, Ly, Fidler, & Ricci, 2001). In addition, it is important to note that families of children with DS also report positive perceptions of the impact of raising a child with DS (Hastings & Taunt, 2002; Skotko, Levine, & Goldstein, 2011). Several studies identified factors that were associated with higher levels of stress and reduced well-being in parents of children with DS. These include socio-demographic factors like lower socio-economic status, a higher number of children and (higher or lower) age of the child with DS (Dabrowska & Pisula, 2010; Fidler, Hodapp, & Dykens, 2000; Hodapp, Ricci, Ly, & Fidler, 2003; Sloper et al., 1991). Also, child functioning variables such as behavioral problems, communicative impairment, health problems and diminished self-sufficiency of the child were found to be of negative influence (Abbeduto et al., 2004; Bourke et al., 2008; Hodapp et al., 2003; Sloper et al., 1991). Finally, psychosocial factors such as maladaptive coping styles of parents, less dispositional optimism, lower quality of the (marital) relationship, and a lack of social support were associated with negative consequences (Greenberg, Seltzer, Krauss, Chou, & Hong, 2004; Hauser-Cram, Warfield, Shonkoff, & Krauss, 2001; Sloper et al., 1991; van der Veek, Kraaij, & Garnefski, 2009a). All of these findings provide valuable insight into the issues parents face. Yet, the outcome-measures stress and well-being cannot depict the wide spectrum of possible affected and unaffected domains of life. For a more extensive evaluation of parental functioning, health related quality of life (HRQoL) is a useful concept. HRQoL is a widely adopted multidimensional concept that emerged during the 1980s, and comprises a range of possible domains, including physical, emotional and intellectual functioning, the ability to participate in valued activities within the family, in the workspace and in community, and the resulting sense of well-being and comfort (Naughton & Shumaker, 2003; Patrick & Erickson, 1988). Although HRQoL is frequently used to evaluate the functioning of parents with children with various disorders, few reports on the HRQoL of parents of children with DS are available. The HRQoL of parents of children with chronic conditions, both with or without developmental disabilities, has been found to be lower compared to parents of children without chronic conditions, with different disorders being associated with varying profiles of HRQoL in parents (Davis & Gavidia-Payne, 2009; Hatzmann, Heymans, Ferrer-iCarbonell, van Praag, & Grootenhuis, 2008; Hatzmann, Valstar, et al., 2009; Khanna et al., 2011; Lin et al., 2009). Concerning HRQoL in parents of children with DS, a Swedish study showed that mothers of children with DS reported poorer vitality than their spouses, and poorer vitality and mental health than mothers in the control group (Hedov, Annere´n, & Wikblad, 2000). In two small, exploratory studies, Brazilian parents of children with DS scored lower in the psychological and environmental domain of the WHOQOL-BREF questionnaire, the latter domain encompassing financial means, housing and opportunities for recreation (Buzatto & Beresin, 2008; Murphy, Herrman, Hawthorne, Pinzone, & Evert, 2000; Oliveira & Limongi, 2011). The Dutch Care-project, of which we employed the database for our current study, revealed that parents of children with DS scored lower in the domains cognitive and social functioning, daily activities and vitality than parents of children without a chronic condition (Hatzmann et al., 2008). In summary, all these HRQoL-studies showed that parents of children with DS tend to experience lowered HRQoL regarding vitality, leisure (social functioning, daily activities, and recreation), and mental- or psychological health. However, none of the above-mentioned studies analyzed predictors of parental HRQoL thoroughly. Predictors of HRQoL have been studied in the broader group of parents of children with developmental disabilities, which also comprises children with DS. Factors that were found to be associated with lower HRQoL show considerable overlap with those related to parental stress and well-being in parents of children with DS. These factors concern mainly child functioning and psychosocial variables. A lower household income, higher levels of functional impairment and behavioral problems of the child, higher experienced burden, less social support, maladaptive coping style of parents, less participation in health promoting activities, and poorer professional support were all related to lower HRQoL outcomes (Bourke-Taylor, Pallant, Law, & Howie, 2012; Browne & Bramston, 1998; Davis & Gavidia-Payne, 2009; Khanna et al., 2011; Lin et al., 2009). For the current study, aimed at tracing factors that are associated with HRQoL in parents of children with DS, we analyzed the data from the Care-project, described in Hatzmann et al. (2008). As a conceptual framework for variables that could be related to HRQoL of parents of children with DS, we used the model of Hatzmann, Maurice-Stam, Heymans, and Grootenhuis (2009). In their model, variables were clustered as ‘socio-demographics’, ‘disease related variables’, and a range of psychosocial mediating variables (e.g. leisure time and emotional support). Since our sample size was more limited, we could not analyze mediation. We therefore employed these as direct predictors of HRQoL instead of analyzing mediation, and renamed those variables ‘psychosocial variables’. Finally, ‘disease related variables’ were renamed into ‘child functioning’ because this covers the meaning of the concerning variables in our sample. This resulted in a model comprising three clusters of variables that were hypothesized to be directly related to HRQoL: ‘socio-demographic variables’, ‘child functioning’ and ‘psychosocial variables’. The objective of our study was to determine whether socio-demographics, child functioning and psychosocial variables were related to cognitive functioning, social functioning, daily activities and vitality, the HRQoLdomains in which parents of children with DS scored lower than parents of children without a chronic condition. 2. Materials and methods 2.1. Participants The data regarding HRQoL of parents of children with DS were gathered within the scope of the Care-project: a large, cross-sectional study on the consequences for parents of caring for chronically ill children, including parents of 100 children
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with DS (Hatzmann et al., 2008). The parents of children with DS were recruited in two ways. The majority was recruited from a cohort of 196 children born between 1999 and 2001 who were studied and described within the scope of an RCT by Van Trotsenburg et al. (2005). Parents of 177 children with DS (19 lost to follow up or unknown addresses) were sent an introductory letter explaining the aim of the study and inviting them to complete the enclosed HRQoL questionnaire and informed consent, and to return these using the enclosed return envelope. Out of this cohort, 98 parents responded. In addition, two parents were recruited in the outpatient clinic of Academic Medical Center. Each family received one questionnaire, and it was not specified whether father or mother was requested to respond. Since we aimed to study the HRQoL in parents of children with DS within a relatively narrow age range, the two parents who were recruited from the outpatient clinic were excluded from our analyses. The calendar ages of their children (14.6 and 16.2 years) differed widely from that of the rest of the sample (range 6.0–8.4 years). The Medical Ethical Committee of the Academic Medical Center Amsterdam approved the CARE project. 2.2. Measurement 2.2.1. Outcomes: HRQoL To assess HRQoL, the ‘TNO-AZL Questionnaire for Adult’s Health Related Quality Of Life’ (TAAQOL) was used (Bruil, Fekkes, Vogels, & Verrips, 2004). This questionnaire was developed by the Netherlands Organization for Applied Scientific Research (TNO) and the Leiden University Medical Center. The TAAQOL is a validated, generic HRQoL questionnaire which consists of 45 items that address 12 domains: gross and fine motor functioning, cognitive functioning, sleep, pain, social functioning, daily activities, sexuality, vitality, positive emotion, depressive emotions and aggressiveness. The four domains we used as outcomes (cognitive functioning, social functioning, daily activities, and vitality) consist of four items each. These items consist of two parts: the first part assesses whether a health status problem is present, with answers on a four-point scale ranging from ‘no/never’ to ‘a lot/always’. If the answer on this first part is affirmative, the second part inquires the subjective response to the reported problem (‘how much did that bother you?’), with answers on a four-point scale ranging from ‘not at all’ to ‘very much’. The scores in each domain are to be added up, and transformed to a scale score between 0-100, with higher scores indicating better functioning. The Cronbach’s alphas of the four domains of the TAAQOL we used were ranging from .85 to .88 in the norm sample (Bruil et al., 2004). In our sample, Cronbach’s alphas for the four domains ranged from .85 to .91. 2.2.2. Predictors Predictors of HRQoL were drawn from self-formulated questions in the Care-questionnaire, which was developed for the entire group of parents of children with chronic diseases, most of which were not associated with developmental disability, and not specifically for parents of children with DS. A pediatrician and a psychologist listed all available predictors from the Care-questionnaire, and independently selected predictors they deemed relevant for HRQoL in parents of children with DS, based on literature and their clinical experience. After this, the final selection of predictors was made by consensus. As mentioned earlier, predictors were clustered as ‘socio-demographic variables’, ‘child functioning’ and ‘psychosocial variables’: 2.2.2.1. Socio-demographic variables (10 items). (1) Gender of the parent, (2) age of the parent, (3) gender of the child, (4) age of the child, (5) current relationship status of the respondent: married/living together, separated/divorced/widowed, single or other, (6) low educational level of the parent: low versus intermediate and high educational level (educational level was defined as the highest educational level completed; low: primary education, lower or middle general secondary education; intermediate: middle vocational education, higher secondary education, or pre-university education; high: higher vocational education, or university), (7) chronically ill parent – yes/no, (8) country of birth of the parent: the Netherlands versus elsewhere, (9) total number of children living at home, and (10) current employment status of the respondent: paid employment – yes/no. 2.2.2.2. Child functioning (4 items). (1) Care dependency: the number of domains of life where the child needs assistance, with the number of domains ranging from 0 to 8: physical care, mobility, eating and drinking, medication use, coping with devices, amusing him/herself, contact with other children, and education. Cronbach’s alpha of this scale was .70 (2) sleep: average number of hours of sleep per night of the child with DS, (3) diaper during daytime – yes/no, and (4) difficulties making contact: ‘did your child ever show problems in making contact with you? (e.g. does not seek contact, or does so in a strange way)’ – yes/no. 2.2.2.3. Psychosocial variables (8 items). (1) Emotional support from partner, family, friends and neighbors, each rated on a three-point scale with 0 = ‘no support’ and 2 = ‘very good support’. The sum of these scores resulted in the variable ‘Emotional support’, ranging from 0 to 8. Cronbach’s alpha of this scale was .60. (2) Quality of the partner relation: constructed from five questions: ‘do you and your partner agree on how to raise your child(ren)?’; ‘do you and your partner agree on how to share the care for your child(ren)?’; ‘can you discuss any problem or worry with your partner?’; ‘if you disagree with your partner, can you come to a resolve?’; ‘do you think you and your partner have enough time for each other?’. Answers on these questions were rated on four-point scales, ranging from 0 = ‘(almost) never’ to 3 = ‘(almost) always’. The sum of these scores resulted in the ‘Partner relation’ variable, ranging from 0 to 15. Cronbach’s alpha of this scale was .83. Other psychosocial
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variables were: (3) enough personal time: ‘do you feel you have enough time for yourself? – yes/no, (4) given up hobby since birth of child – yes/no, (5) loss of friendship since diagnosis – yes/no, (6) job adjustment due to condition of child – yes/no, (7) caring for an ill friend or family member (not child or partner) – yes/no, and (8) adverse life events: ‘have you ever experienced an adverse life event, such as losing your partner or a close family member, a car accident or major financial problems?’ – yes/no. 2.3. Statistical analysis Scales of the TAAQOL were constructed and missing data were imputed based on the guidelines provided by the TAAQOL manual. Scales of the predictor variables were constructed as described in Section 2.2.2. Internal consistency of all scales was assessed by the Cronbach’s alpha. Subsequently, descriptive analyses were performed for socio-demographics, child functioning and psychosocial variables. To limit the number of predictors in the final predictive model, we selected variables for each of the four HRQoL-domains using multiple linear regression analyses, separately for each of the three clusters of predictors: socio-demographic variables, child functioning and psychosocial variables. The condition for selection of a variable for the final regression analysis was set at p < .15. The assumptions for multiple linear regression analyses were met. The selected variables were used in the final multiple linear regression analyses to predict the HRQoL outcomes, resulting in four multiple linear regression models: one for each HRQoL-outcome. In all multiple linear regression models, a significance level of .05 was deemed significant. Given the exploratory nature of this study, no correction for multiple testing was applied. Standardized regression coefficients of .1 were considered small, .3 medium and .5 large for continuous variables, after Cohen (1988). For binary coded variables, standardized regression coefficients of .2 can be considered small, .5 as medium and .8 as large. The explained variances (R2) were determined of all multiple linear regression models. All data in this study were analyzed using SPSS version 20. 3. Results 3.1. Participants Table 1 shows the socio-demographic characteristics of the parents and children with DS. Of our participants, 86% was mother and 98% was married or living together. In our sample, 41% of the parents received higher education, and 63% had a paid employment. Functioning of the children with DS and psychosocial variables of their parents are shown in Table 2. 3.2. Predictors of parental HRQoL Table 3 shows the results of the linear regression analyses we used to select variables for the final regression models: standardized regression coefficients corresponding to variables that met criteria for selection (p < .15) are presented in bold. Table 4 presents the results of the final linear regression models. All final models were highly significant (p < .001) with explained variances ranging from 37% to 57%. Significant predictors had standardized regression coefficients of small to medium size (.17–.34). The most powerful predictors were psychosocial variables. Some child functioning variables also contributed significantly to the model, but none of the socio-demographic variables appeared as significant predictors in the final model. Cognitive functioning was best predicted by the number of hours of sleep per night of the child (b = .29, p < .01), and by whether the parent had given up a hobby since the birth of the child with DS (b = .29, p < .01). Apart from that, enough personal time (b = .19, p = .03) and more emotional support (b = .17, p = .04) predicted better cognitive functioning, while loss of friendship (b = .19, p = .03) predicted poorer cognitive functioning. Social functioning was best predicted by the quality of the partner relation (b = .34, p < .001). Further, emotional support (b = .22, p = .02) and having enough personal time (b = .19, p = .04) were related to better social functioning. The score for daily activities was best predicted by whether the parent had to care for an ill friend or family member (b = .31, p < .01). Besides that, enough personal time (b = .24, p = .02) predicted higher scores for daily activities. For vitality, the parent having enough personal time (b = 0.32, p < 0.01) was the most powerful predictor for better functioning. The child wearing a diaper during daytime (b = .23, p = .02) and loss of friendship (b = .20, p = .03) were related to poorer vitality. 4. Discussion The objective of this study was to identify variables that are related to HRQoL regarding cognitive functioning, social functioning, daily activities and vitality of parents of children with DS. In conclusion we found that mainly psychosocial variables were associated with these HRQoL outcomes, rather than child functioning or socio-demographic variables. To our knowledge, this is the first study that analyzed factors related to HRQoL in parents of children with DS. The finding that socio-demographics did not show strong relations with HRQoL is in line with most of the literature concerning stress in parents of children with DS, and concerning HRQoL in parents of children with chronic disorders or developmental disabilities (Hatzmann, Maurice-Stam, et al., 2009; Sloper et al., 1991).
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Table 1 Socio-demographic characteristics. Socio-demographic Gender parent (n, %) Male Female Age parent (range) (M, SD) Gender child (n, %)a Male Female Age child (range) (M, SD) Current relationship status (n, %)a Married/living together Separated/divorced/widowed Single Other Educational level of the parent (n, %)a High Intermediate Low Chronically ill parent (n, %)a Yes No Country of birth of the parent (n, %)a Netherlands Elsewhere Total number of children (M, SD)a One (n, %) Two (n, %) Three (n, %) >Three (n, %) Paid employment (n, %) Yes No a b
14 (14%) 84 (86%) 32.4–51.9 years 41.3 years (4.4) 56 (60%) 38 (40%) 6.0–8.4 yearsb 7.1 years (0.6) 96 1 1 0
(98%) (1%) (1%) (0%)
40 (41%) 38 (39%) 19 (20%) 12 (13%) 84 (88%) 91 (94%) 6 (6%) 2.5 (1.0) 9 (9%) 47 (48%) 29 (30%) 12 (12%) 62 (63%) 36 (37%)
Numbers not adding up to the total number of participants (N = 98) caused by missing values. n = 95.
Child functioning was clearly related to the outcome variables in the models we used for selecting variables. Yet, in the final model, which included psychosocial variables as well, the predictive value of child functioning diminished. The level of care dependency, i.e. the number of domains the child needs practical help on, did not predict HRQoL in the final model. This is very similar to what Hatzmann, Maurice-Stam, et al. (2009) found in parents of children with metabolic diseases (some of which comprise developmental disabilities). Yet, Sloper et al. (1991) found that self-sufficiency of the child with DS, assessed with a 317-item scale, was associated with maternal satisfaction with life, but not with the paternal satisfaction with life. Perhaps, these latter findings cannot be generalized to the current situation of children with DS in the Netherlands, with a different standard of care and changed attitudes in society toward children with DS. Other studies showed that, with regard to child functioning, in particular behavioral problems are related to parental stress or well-being in parents of children with DS (Abbeduto et al., 2004; Hauser-Cram et al., 2001; Hodapp et al., 2003). Given the wide spectrum of chronic illnesses in the Care project, most of which were not associated with developmental disability, behavioral problems of the children were not assessed in this project. However, some of our variables, such as sleep and wearing a diaper during daytime may reflect behavioral problems in the children. Sleep disturbance, for instance, has been found to be related to behavioral problems in children with typical development, and to a range of psychiatric problems in children with DS (Gregory, Eley, O’Connor, & Plomin, 2004; Richdale, Francis, Gavidia-Payne, & Cotton, 2000; Stores & Stores, 2013). Also, an association between incontinence and behavioral problems has been described in children without developmental disabilities (von Gontard, Mauer-Mucke, Plu¨ck, Berner, & Lehmkuhl, 1999). Psychosocial variables showed most consistent and most powerful relations with parental HRQoL. This finding is in line with previous findings regarding predictors for stress in parents of children with DS (Dabrowska & Pisula, 2010; Sloper et al., 1991) and with findings in parents of children with other chronic conditions (Ten Hoedt et al., 2011). The beneficial relation between a supportive social network and well-being is well-established in parents of children with DS, and in parents of children with various other conditions (Dunst, Trivette, & Cross, 1986; Hatzmann, Valstar, et al., 2009; Hauser-Cram et al., 2001; Khanna et al., 2011; Sloper et al., 1991; Van der Veek, Kraaij, & Garnefski, 2009b). Our finding that a stronger partner relation is associated with better HRQoL in the social domain is in line with studies that showed the predictive value of the partner relation for HRQoL (Wiedebusch et al., 2010; Wiedebusch, Pollmann, Siegmund, & Muthny, 2008), for well-being in parents of children with DS (Van der Veek et al., 2009b). Van der Veek et al. (2009b) suggested that in parents of children with DS the positive effect of a supportive partner relation could be seen as a ‘‘constant condition that may or may not facilitate a
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Table 2 Functioning of children with Down syndrome and psychosocial variables of their parents. N = 98 Child functioning Care dependency score (M, SD) Physical care (n, %) Movement/mobility (n, %) Eating and drinking (n, %) Medication use (n, %) Coping with devices (n, %) Amusing him/herself (n, %) Contact with other children (n, %) Education (n, %) Sleep (range)a (M, SD) Diaper during daytime (n, %) Yes No Difficulties making contact (n, %)b Yes No Psychosocial variables Emotional support (range)a (M, SD) Quality of partner relation (range) (M, SD) Enough personal time (n, %)b Yes No Given up hobby (n, %)b Yes No Loss of friendship (n, %)b Yes No Job adjustment (n, %) Yes No Caring for ill friend/family member (n, %)b Yes No Adverse life event (n, %) Yes No a b
3.6 (1.8) 71 (72%) 21 (21%) 42 (43%) 26 (27%) 6 (6%) 53 (54%) 50 (50%) 86 (88%) 3–13 h 9.8 h (1.9) 36 (37%) 62 (63%) 18 (18%) 75 (77%)
0–8 5.5 (1.9) 2–15 10.9 (2.9) 27 (29%) 67 (71%) 27 (30%) 64 (70%) 31 (33%) 62 (67%) 70 (71%) 28 (29%) 8 (8%) 87 (92%) 79 (81%) 19 (19%)
n = 94. Numbers not adding up to the total number of participants (N = 98) is caused by missing values.
positive adaptation, as it may be a potential source of relief or stress on its own’’. A strong partner relation relating specifically to the HRQoL domain of social functioning may be explained by the willingness of a partner to make arrangements regarding the care and support the other in maintaining their social network. Also, parents with a strong social network may have the opportunity of sharing the burden of their worries and care with more people than just their partner, and so reduce stress in their marital relationship. The importance of loss of friendships, together with emotional support and the quality of the partner relation underline the relevance of a supportive social network around the parent, be it the partner, friends, or family and neighbors. Our results also highlight the importance of having enough time for leisure. Parents who reported having enough personal time showed better HRQoL, while having given up a hobby was related to poorer cognitive functioning. Also, caring for an ill friend or family member was related to lower scores for daily activities, which can be explained by the burden in terms of time too. Interestingly, a Dutch study showed that providing informal care for more than 11 h a week was associated with lower overall life satisfaction, although providing care for less than 6 h a week, and combining it with paid employment lead to higher life satisfaction (Van Campen, de Boer, & Iedema, 2013). Also, a previous study that assessed HRqoL in parents of children with DS using the WHOQOL-BREF measure, found that parents scored lower on environmental domain, which encompasses opportunities for recreation (Oliveira & Limongi, 2011). These results all relate to the time-pressure parents have to deal with. In our sample, more than two-thirds (71%) reported not having enough personal time. Several other studies showed that parents of children with DS spend more time on child care, and less on recreational and social activities than parents of children without a disorder (Barnett & Boyce, 1995; Crowe & Florez, 2006; Hedov, Annere´n, et al., 2002; O’Mullan Wayne & Krishnagiri, 2005; Sanders & Morgan, 1997). In a study asking parents with DS which important needs
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Table 3 Selection of variables for the final predictive model of HRQoL: standardized regression coefficients (b). Cognitive functioning Socio-demographic variables (b) Gender parent (male) Age parent Gender child (male) Age child Low educational level Chronically ill parent Country of birth (elsewhere) Total number of children Paid employment 2 R F (df) Child functioning (b) Care dependency Sleep Diaper during daytime Difficulties making contact R2 F (df) Psychosocial variables (b) Emotional support Quality of partner relation Enough personal time Given up hobby Loss of friendship Job adjustment Caring for ill friend/family member Adverse life event R2 F (df)
.09 .04 .10 .15 .30** .13 .07 .00 .13 .10 1.07 (9, 77)
Social functioning
Daily activities
Vitality
.14 .01 .10 .14 .09 .23* .04 .03 .02 .10 .94 (9, 77)
.08 .05 .08 .16 .16 .12 .01 .12 .00 .07 .63 (9, 77)
.11 .12 .07 .25** .21* .21* .01 .16 .14 .16 1.59 (9, 77)
.24** .36**** .01 .16* .33 10.46 (4, 84)****
.11 .14 .05 .27** .18 4.46 (4, 84)***
.09 .24** .18* .12 .17 4.15 (4, 84)***
.13 .17* .21** .18* .20 5.11 (4, 84)***
.24** .04 .30*** .35*** .23** .12 .00 .01 .48 7.85 (8, 69)****
.22** .31*** .17* .25** .06 .05 .06 .10 .48 7.83 (8, 69)****
.20* .08 .17* .10 .16* .02 .29*** .15 .38 5.20 (8, 69)****
.23** .16 .28** .13 .22** .09 .00 .05 .37 5.13 (8, 69)****
Note. For selected variables (p < .15), the corresponding standardized regression coefficients are printed in bold. * p < .15. ** p < .05. *** p < .01. **** p < .001.
Table 4 Final multiple linear regression models for parental HRQoL: standardized regression coefficients (b). Cognitive functioning Socio demographic variables (b) Age child Low educational level Chronically ill parent Total number of children Child functioning (b) Care dependency Sleep Diaper during daytime Difficulties making contact Psychosocial variables (b) Emotional support Quality of partner relation Enough personal time Given up hobby Loss of friendship Caring for ill friend/family member R2 F (df)
.16
Social functioning
Daily activities
.13 .11 .04
.04
.04 .29***
.18 .15
.06
.14
.17**
.22** .34**** .19** .19
**
.19 .29*** .19** .57 11.57 (8, 71)****
.46 10.77 (6, 76)****
Vitality
.11 .23** .15
.14
.18 **
.24
.18 .31*** .37 7.19 (6, 74)****
Note: For significant predictors, the variable names and corresponding standardized regression coefficients are presented in bold. ** p < .05. *** p < .01. **** p < .001.
.32*** .20** .46 6.60 (9, 71)****
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were not or only partly met, 75% reported to want a break from their responsibilities, and 72% needed weekend and afterschool activities for their child (Siklos & Kerns, 2006). Recently, Bourke-Taylor et al. (2012) found that in mothers of children with developmental disabilities, participation in health-promoting leisure activities was related to better HRQoL outcomes. So, it seems a quite consistent finding that the parents feel they need more time for themselves, but have difficulty arranging this. Our results showed that although it may be challenging to take time for oneself due to the demands of everyday life, it seems important for the well-being of parents of children with DS. One of the limitations of our study was the limited number of predictors we could include in our regression analyses to obtain reliable results. Further, we did not define whether fathers or mothers were requested to complete the questionnaires, which resulted in a high percentage of mothers participating. Since the impact of raising a child with DS has been found to differ between mothers and fathers, it is recommended to include both parents of each child in future research (Hedov et al., 2000; Hornby, 1995; Roach, Orsmond, & Barratt, 1999). Also, future research aiming to construct a more complete predictive model should incorporate other factors that are expected to predict HRQoL in parents of children with DS. These include in particular behavioral problems of the child, which were not included in the current study, because of the broad spectrum of disorders included in the Care project the present study was part of. Most disorders were not associated with developmental disabilities or behavioral disturbances. Also parental stress, parental coping styles or personality features such as dispositional optimism could be included in a more complete model. Moreover, instead of self-formulated questionnaires and self-constructed scales, standardized and validated questionnaires should be used if available. Finally, since age of the child has been identified as a predictor for parental stress in previous studies, the generalizability of our results to parents of children with DS in age categories other than in the present study is limited. It is therefore recommended to study HRQoL of these parents longitudinally, since this will provide insight in the changes in HRQoL over time, and possibly in the causality and direction of the associations (Haveman, van Berkum, Reijnders, & Heller, 1997). Despite these limitations, our findings showed that HRQoL-outcomes of parents of six to eight years old children with DS were mainly associated with psychosocial variables relating primarily to social support and time pressure. Health care providers should be aware of the risk for lowered HRQoL in parents of children with DS. To pay systematic attention to possible psychosocial problems in order to identify parents most at risk, we recommend the use of valid, short screening instruments. As we found social support and creating enough (leisure) time for oneself to be related to better HRQoL outcomes, interventions should target these social and personal resources. For instance, social workers could pay attention to the informal support network of parents and to the quality of the relationship with their partner, and they should stimulate parents to take time for activities with family, friends, or neighbors, or to connect to groups of other parents of children with DS (Law, King, Stewart, & King, 2001). Also, attention should be paid to the time-pressure parents face, and the amount of time available for leisure. Most likely it is hard for parents to create personal time. For this reason parents could be referred to respite care services or be stimulated to find similar help in their informal network. Respite care comprises interventions aimed at temporarily easing the burden of the caregiving task by, for instance, providing overnight residential care for a weekend (Caples & Sweeney, 2011; Chan & Sigafoos, 2001; Douma, Dekker, & Koot, 2006). In conclusion, we found psychosocial variables rather than socio-demographics and child functioning to be most consistently and powerfully related to HRQoL in parents of children with DS. These psychosocial variables were primarily related to social support and time pressure. Health care providers should be aware that parents of children with DS are at risk for lower HRQoL. Systematic screening of parents to detect problems at an early stage, and interventions that target the supportive network and the demands in time are recommended.
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