Children and adolescents adjustment to parental multiple sclerosis: a systematic review

In Western societies, 4 to 12% of children and adolescents aged 18 and under live in households where a parent has a chronic illness [1‐3]. Increasing evidence suggests that having a parent with a chronic condition can put children at a higher risk of developing emotional and behavioural difficulties due to changes in parent–child interactions [3].

Multiple sclerosis (MS) is a chronic degenerative disease of the central nervous system and is the most common non-traumatic cause of neurological disability among young adults in the Western world [4]. MS typically manifests between the ages of 20 and 40 years, at a life stage when parenting is an important issue for many [5]. Interestingly, reproductive decision-making by people with MS seems to follow the same pattern as the general population [6]. Consequently, many children are exposed to a parent trying to cope with a potentially disabling chronic condition. MS is a particularly challenging disease, and the unpredictable and variable clinical course can cause considerable stress and anxiety on patients and their families [7, 8].

While much research and resources now focus on the child or adolescent who has MS, less is known about the child living with a parent who has MS [9, 10]. We aimed to systematically review the literature to address the question - what are the possible effects on children and adolescents’ psychosocial adjustment of having a parent with MS? In doing so, we aimed to illuminate the impact of parental health issues on children’s development, and open avenues for early identification and potential preventive interventions.

In this systematic review we evaluated the association between parental MS and adjustment in children and adolescents. Overall, while most studies tended to report that children of MS patients exhibited negative psychosocial behaviour compared with children of “healthy” parents, some positive aspects in caring for a parents with MS were also highlighted. However, overall the strength of the evidence was rather weak, with only 2 of 18 studies rated as ‘high quality’ , which makes it difficult to draw evidence-based conclusions [10, 14].

Nonetheless, our findings are broadly consistent with other systematic reviews, which also report negative psychosocial effects in children living with a parent with a physical disability or a chronic illness [3, 38, 39]. A meta-analysis looking at children who have a parent with a chronic illness found that overall these children displayed significantly more internalizing related behaviour (i.e. anxiety, depression, withdrawal) than children with healthy parents [3]. Furthermore, in a population-based sample of children with a parent dealing with a serious physical illness, there was an elevated risk of psychosocial maladjustment, with internalizing problems being more prevalent than externalizing problems, such as aggression and delinquent behaviour [1].

Within our systematic review we found suggestion that children had higher rates of body concern, depression, anxiety, somatization, difficulty in relating to others and greater emotional and behavioural problems [5, 7, 12, 15, 19, 20, 23, 25]. Children also perceived their families as being less cohesive with greater tension and isolation, as compared with the general population [14, 22]. Furthermore, uncertainty regarding the future, as well as illness exacerbation, posed a degree of fear and anxiety in children [25, 40]. Caregiving roles and the stigma attached to a parent’s MS were also sources of stress for children [20]. Specifically for adolescents, greater family responsibilities were linked to fewer social relationships and higher distress [19, 20, 24]. However, several studies found no measurable effect (negative or positive) of having a parent with MS [5, 8, 16, 18], and a few found some positive effects, such as higher personal competence [13, 14].

This pattern of positive and negative outcomes could reflect the costs and benefits associated with caregiving that is also evident in adult caregivers [41]. Some children in our review described pride in their caregiving abilities, as these children completed tasks above those of their peers [10]. However, there are circumstances, when children may feel they have no choice but to become caregivers, and that can intensify the overall family stress [19, 38]. Children are more prone to becoming caregivers in single-parent families, low-income families, families who do not have access to home care support, and families with little social support [5, 42].

Our systematic review highlighted that one of the factors associated with poor adjustment in children was their limited knowledge and understanding of MS [17, 21]. This concurs with other studies which indicate that it is important to provide children information about MS, tailored to their developmental level, as this lack of information has caused some children to believe that their own or other people’s behaviour affected their parents’ illness [43, 44]. Young children, in particular, appear to have a need for information that is currently not being met [43]. Children may need better information on the etiology of MS and be reassured that the risk for them to contract MS is minimal [43]. Children who are unaware of their parent’s illness may display high levels of anxiety and distress as they witness family tension without being aware of its source [21]. Interestingly, educated mothers are less likely to provide information regarding their illness to their children [21]. This observation is worthy of further investigation, especially as several studies show that MS patients who participate in research have a higher socioeconomic status compared with other chronic illness patients, which could potentially lead to selection and reporting bias [5, 45].

In our systematic review, studies reported that family dysfunction and lack of social support were associated with a child’s externalizing problems, while the severity of impairment of the ill mother predicted children’s internalizing problems [7, 14]. This is consistent with other studies which showed that lower level of depression in the affected parent has been related to positive coping ability of the children [46]. Likewise, the coping ability of the healthy parents appeared to be a strong predictor of whether children successfully cope with the disease [46, 47]. Nonetheless, no study in our systematic review looked at fatigue as a risk factor, whilst fatigue has been shown to be one of the most common, yet “hidden”, symptoms of MS [48]. Parents with MS identified fatigue as one of the primary problem that interferes with important parenting functioning, ranging from difficulties in being involved with day-to-day activities, to lack of patience [20]. Furthermore, in a group of patients with different chronic illnesses, including MS, one study demonstrated that maternal fatigue potentially mediates some of the relationship between maternal depression and maladaptive child outcomes [45]. Other factors which emerged as potentially influencing a child’s adjustment to parental illness from the included studies were: gender of the parent and the child [20, 46, 47], children’s age and developmental stage [10, 19], level of social support [10], physical condition or disability caused by the disease [19], single parenthood and family environment [10, 15, 19, 23].

Interpretation of our systematic review is constrained by limitations in the original studies, particularly exposure assessment and potential sources of bias. First, none of the studies included in the present systematic review used objective measures of child psychosocial adjustment. Second, studies failed to account for important confounding or to provide baseline characteristics of the participants that limit the generalizability of the findings. Third, many studies did not include a comparison group, which is a critical piece in shedding light on whether the patterns of findings in the studies are specific to children who have a parent with an illness. Last, relying on cross-sectional design fails to disentangle the interactions between normal child developmental variations and the variations produced by the progressive nature of MS, such as difficulties in the transition to adulthood. This is particularly relevant given that disability in MS can often be minimal in the early stages of the disease, and the overall lifespan may not be affected as with other chronic diseases. Furthermore, to explore if there is an MS specific characteristics that influence children’s development, it would be of interest to investigate the psychosocial wellbeing of children with MS parents as compared to other chronic diseases.

To overcome many of the deficiencies raised above, we recommend a population-based approach with inclusion of a representative comparison group to avoid selection biases that may have limited many studies. In addition, rigorous, objective, and well-validated measurement tools are needed, of which several are available, namely, the Early Child Development Instrument [49], or the Child Behaviour Checklist [27]. Along with this, measurement of appropriate confounders or explanatory variables, such as socioeconomic status, gender of the child or the marital status of the parents should be considered. Ideally these would be combined with clinical characteristics of the affected parents, such as disease duration, level of disability or presence of comorbidity, to populate a large study-specific dataset of patients and their children. Findings of these studies would help to inform policy making for healthier communities and assist us in developing and evaluating family centered interventions to improve child and family outcomes [50].

Exploring the relationship between parental MS and a child’s psychosocial adjustment is challenging. Due to the relatively few studies of high methodological quality, it is difficult to draw strong evidenced-based conclusions from the present literature and thus more extensive and higher quality research in this area is greatly needed. From the limited available evidence, it appears that exposure to parental MS may put children at a higher risk of psychosocial problems compared with children with parents not living with a chronic disease. Although the few studies examining the impact of parental chronic disease on children’s development and health represent important first steps, many have serious methodological limitations, particularly with respect to ascertaining individuals with definite MS, and potential sources of bias, such as failure to adjust for important demographic variables, i.e. socio-economic status, lack of a suitable comparison group and sub-optimal data collection. To gain further insight and to assess this relationship accurately we need more population-based studies using objective measures of developmental health, using reliable and valid measurement instruments. Further research is needed before appropriate evidenced-based recommendations can be made, however, it appears pragmatic to advise healthcare professionals, and community partners, such as educators, patient group and policy makers, to be cognizant of the broad impact of chronic parental illness on the developing child.