Background
The prevalence of Pervasive Developmental Disorders (PDD) in children has increased from 0.4 in 1.000 during the 1970s to current estimates of up to 6 per 1.000. This increase is presumably a consequence of improved ascertainment and considerable broadening of the diagnostic concept [
1]. While PDDs were previously only diagnosed in children with mental retardation, recent studies suggest that approximately 50% of individuals diagnosed with PDDs have normal intelligence [
2], and a minimum prevalence of 2 out of every 1.000 for PDDs in mainstream school children was reported in a recent study [
3]. Asperger syndrome (AS) and high-functioning autism (HFA) are PDD diagnoses in individuals of normal intelligence [
4] characterized by pervasive impairment in several areas of development: reciprocal social interaction skills, communication skills, and the presence of stereotyped behaviour, interests, or activities. AS is distinguished from HFA primarily by a lack of clinically significant language delay [
5]. The majority of children with AS or HFA live in families along with their parents. Caregiving of a child with a PDD may be associated with high levels of distress and burden [
2] which potentially undermine the mental and physical health of the parents of these children. While there is much data available about parenting children with PDDs and associated intellectual impairment, only a few studies have explored the health-related quality of life (HRQL) in parents of children with AS or HFA [
6].
Parenting children with developmental disabilities, among them PDDs with intellectual disability, is associated with impaired mental health [
7,
8], higher levels of stress [
8‐
11], sense of devaluation and blame [
9], and also impaired physical functioning, tiredness or exhaustion in mothers and fathers [
12,
13]. For example, Weiss [
7] reported that many parents of children with PDDs experienced feelings of intense anger, guilt, depression or anxiety most of the time. Moreover, these feelings were frequently expressed in psychosomatic problems.
Due to the scarcity of data about the HRQL of parents of children of normal intelligence with PDDs, we deem it relevant to also take into account research on the well-being of parents of children with other types of disorders, such as developmental disabilities or severe mental health problems. Using data from the Wisconsin Longitudinal Study, Seltzer et al. [
14] explored parental attainment and well-being at mid-life in parents of children with developmental disabilities and parents of children with severe mental health problems. The parents of children with developmental disabilities accommodated to their child's needs early on, for example, by restricting their social life and making changes in family routines. However, parents of children with severe mental health problem were not as accommodating. At a follow-up, the physical and mental health of parents of children with developmental disabilities did not differ from that of a normative group, while parents of children with severe mental health problems displayed poorer physical health and elevated levels of depressive symptoms. Similarly, Magana et al. [
15] also found higher rates of physical health problems in mothers caring for their adult children with mental illness. Notably, neither Seltzer et al. nor Magana et al. stated that individuals with PDDs were included in their studies.
Comparisons between mothers and fathers of a child with a developmental disability have displayed different [
12,
16,
17] as well as similar [
11] levels of perceived stress and impaired health. A Swedish report about parents of children with Down's syndrome [
12] indicated that mothers had lower scores of self-perceived vitality, and also that they spent more time caring for their child than the fathers. Moreover, a recent family study by Little [
6], including children with AS, reported that mothers experienced more stress and pessimism about the child's future, and used antidepressants or other therapy more frequently than the fathers. In the same report, mothers of children with AS found coping strategies such as communication and consultation with family, friends, and professionals more helpful than the fathers did.
Parental stress and health outcome is related to child characteristics such as the severity of the core disability or main diagnosis, the age of the child, and the extent of coexisting behaviour problems [
18,
19]. It has been suggested that such coexisting behaviour problems in the child predict parental stress to a higher extent than the severity of the intellectual or adaptive functioning [
19]. Notably, coexisting behaviour or psychiatric problems are common in individuals with AS or HFA [
20‐
22].
The present study which is a part of a longitudinal investigation of school-age children with AS/HFA and their families [
23] focused on the HRQL in parents of children with AS/HFA. More specifically, the aims were to explore: 1) whether the raising of a child with AS/HFA is associated with impaired parental HRQL; 2) if there are differences in the HRQL between mothers and fathers in families with a child diagnosed with AS/HFA; and 3) whether parents' health within the AS/HFA group is related to child behaviour characteristics.
Discussion
Results indicate that mothers, but not fathers, who are caregivers of school-age children with AS/HFA are at increased risk of impaired physical well-being. We also found that the impaired maternal HRQL in the AS/HFA group is related to the extent of symptoms of hyperactivity and conduct problems in the child.
Since there is sparse data about the HRQL of parents who are caregivers of children with AS or HFA, we need to attempt to compare our results with the results of studies dealing with the well-being of parents of children with other types of disorders. Hence, our findings that the mothers of children with AS/HFA report impaired physical well-being resembles previous findings on caregivers of children with intellectual disability [
13] cerebral palsy [
40] and mental disorders [
14,
15]. For example, Emerson [
13] reported that 20 percent of mothers of children with intellectual disability versus three percent of mothers of children without intellectual disability considered themselves to be "physically ill" due to the child's difficulties. Seltzer et al. [
14], and Magana et al. [
15] also found more physical symptoms or increased rates of physical health problems among mothers of adult children with severe mental disorders. Notably, there are also studies which suggest a genetically-linked increased rate of autoimmune disorders in parents of individuals with PDDs [
41].
The present study did not detect statistically significant differences between mothers in the AS/HFA group and mothers of the control group regarding their self-perceived mental well-being. This is in contrast with many previous studies that have shown that mothers' mental health is related to the child's disability [
8,
12‐
14,
40,
42]. Of course it is possible that our failure to match such findings is due to the low power of the current study, given that the relatively small differences in mental health between parents in the AS/HFA and control groups did not reach statistical significance. Nevertheless, could there be any way to explain our findings of relatively good mental, but poor physical well-being among the mothers? Drawing on previous studies, we note that Weiss [
7] reported that psychosomatic problems were common manifestations of stress related to caregiving in parents of children with PDDs, and based on their findings, Magana et al. [
15] discussed whether mothers of adult children with mental illness were particularly vulnerable to physical health problems. From another standpoint, one might speculate whether the poorer self-rated maternal physical health in the AS/HFA group could be associated with particular personality traits. From a strictly theoretical perspective, a discrepancy between mental and physical health in these mothers could be related to the presence of alexithymic traits, meaning a reduced ability to engage in explicit emotional processing. A relationship between alexithymic personality and somatization has been reported [
43,
44], and research on adults with AS has also found high rates of alexithymia in these individuals [
45]. However, the current study did not determine the presence of alexithymic traits in parents of children with AS/HFA.
Our finding that maternal physical health was poorer than paternal physical health in the AS/HFA group resembles results in a previous report on parenting a child with Down's syndrome, where mothers were more exhausted than fathers [
12]. However, our finding, that self-rated mental health did not differ between mothers and fathers of children with AS/HFA, is in contrast with previous studies. To illustrate, other researchers have reported more anxiety [
17] exhaustion [
12] child-care related stress, pessimism about the child's future, and use of antidepressants in mothers of these children [
6].
In similarity with the results by Hastings [
16,
17], we found that maternal, but not paternal health in the AS/HFA group was related to particular behaviour characteristics of the child. Maternal mental health was related to the extent of symptoms of hyperactivity and conduct problems in the child, and maternal physical and mental health were related to the prosocial behaviour of the child. Previous research has suggested that coexisting behaviour problems in a child could be more stressful for parents than the severity of the child's core disability [
16,
19]. Thus, our finding that maternal health was related to the extent of general behaviour problems of the child, and not to the degree of autistic symptoms reflected in the ASSQ-score, may be in similarity with findings in other studies. However, regarding the social competence of the child, which is a primary aspect of PDDs, we do note that our SDQ data indicates a relationship between maternal health and the prosocial behaviour of the child (ability to be considerate, to share, to be helpful and to be kind to younger children). Notably, the items and wordings of the ASSQ and of the prosocial behaviour scale of the SDQ cover somewhat different aspects of social competence in children. In consistency with other authors [
2], we believe that the prosocial behaviour scale of the SDQ may yield additional useful information about the behaviour characteristics of children with PDDs.
The main strength of the present study is the use of a well-defined sample of 32 school-age children with ICD-10 diagnosed AS or HFA and the control group of typically developing children. Likewise, the use of the SF-12, a well-validated measure of HRQL, and parent as well as teacher-ratings of the children's behaviour, strengthen our report.
However, there are also limitations of the present study, which must be acknowledged. The sample of individuals with AS/HFA was rather small. During the sampling procedure, children with comorbid medical disorders or ongoing medication were excluded from our sample. Whether the sampling method biased parental results in a positive direction is unknown. Thus, considering the issue of low power, it is quite possible that small differences in HRQL between parents in the AS/HFA and control groups were not detected in the current report. More, there were no statistically significant differences with regard to sociodemographic data between parents in the AS/HFA and control groups in this material. However, more mothers in the AS/HFA group were not employed and were lone parents. These important sociodemographic factors need further investigation in larger studies. Finally, the fact that parental health was only measured by the SF-12, and that no physical examination or review of the parents' medical records was performed, are also limiting factors.
To summarize, we found that parenting a child with AS/HFA was associated with impaired HRQL in mothers, but not in fathers, and that impaired maternal HRQL was associated with higher levels of behaviour problems in the child. We conclude that parental HRQL in children with AS/HFA needs further exploration in larger studies. Moreover, studies exploring the issues related to HRQL and sociodemographic circumstances in these parents would be of great interest.
Authors' contributions
HA was the main principal investigator collecting the data and preparing the manuscript together with J-OL and HS.
J-OL supervised and participated with great impact at all stages of preparation of this manuscript, and advised on the statistical analysis.
HS was co-conceiver of the idea of this study and made substantial contribution to the analysis and interpretation of data and preparation of the manuscript.