The Psychosocial Impact of Parental Multiple Sclerosis on Children and Adolescents: A Systematic Review

It is estimated that approximately 10% of children worldwide have a parent with a chronic medical condition.¹ Multiple studies have found evidence of increased risks of emotional and behavioral problems in children of parents with chronic somatic illnesses.^2–4 It is suspected that chronic illnesses interfere with parenting through changes in parental availability, household routines, and responsibilities, as well as by contributing to parental depression and family conflict.⁵

Affecting approximately 2.3 million people worldwide,⁶ multiple sclerosis (MS) is the most prevalent chronic inflammatory disease of the central nervous system.⁷ The demyelinating disease manifests 70% of the time in women aged 20 to 40 years.⁸ Thus, people with MS are commonly diagnosed during their reproductive years. People with MS can experience various physical and cognitive symptoms ranging from mild paresthesia and impaired vision to fatigue, limited mobility, and severe disability.⁹ Consequently, MS can affect the ability to work and to care for children or the household.^10,11 The impact of life-threatening parental illnesses, such as cancer, on children’s development has been shown in the past.^12,13 Having a parent with a debilitating but not life-threatening illness, such as MS, may also affect a child’s psychosocial status.

There have been 3 systematic reviews on this subject,^14–16 with Razaz et al¹⁴ providing the latest review in 2014 (literature search in 2012). All 3 reviews found evidence of predominantly negative consequences, but a few of the included studies indicated positive psychosocial effects or no significant effects.^14–16 Considering the time frame of the last reviews, an updated, critical, systematic review is needed. By reviewing the current knowledge on this subject, we aimed to highlight further research needs and implications for clinical care. In this review, we do not specifically address the impact of children taking care of an ill parent.

METHODS

Search Strategy

In August 2020, we searched the following databases via Ovid: MEDLINE, PsycINFO, and PSYNDEX. The research question “What is the psychosocial impact of parental multiple sclerosis on children?” meant that the search consisted of 4 elements: multiple sclerosis AND parents/family AND children/adolescents AND psychosocial impact. Based on these main search terms, we built search strings using all associated terms and available synonyms and spellings using the operator “OR.” Subject headings were used if available for the searched term. The detailed search strings can be accessed in TABLE S1, which is accessible online at IJMSC.org.

Eligibility Criteria

Types of Studies

A few restrictions maximized the number of relevant search results. We included all empirical studies published in English or German and excluded data from case reports, comments, and conference abstracts. We also checked the references of previous reviews for eligible publications.

Types of Participants

We did not establish age restrictions for the children included in the study because many studies include children of a wide age range and often do not differentiate between different age groups. We included studies that had children with a parent with MS as participants or studies that referenced them by proxy.

Types of Settings

To be considered for inclusion, families had to have at least 1 parent with a confirmed diagnosis of MS of any clinical course. We excluded studies that had children with an MS diagnosis.

Types of Outcomes

All outcomes that could generally be allocated to psychosocial effects, including quality of life issues and adjustment issues, were considered psychiatric disorders. “Good” adjustment in children and adolescents refers to healthy, normative, and age-appropriate behavior that aims toward positive adult functioning.¹⁷ Psychosocial adjustment encompasses a child’s emotions, behaviors, and social functioning. Behavior difficulties, often categorized into internalizing and externalizing symptoms, were included as well. Internalizing refers to negative behaviors that focus on inner emotions and are experienced as anxiety, depression, and somatization.¹⁸ Children’s externalizing expresses through negative behaviors focusing on the outside, such as hyperactivity, aggression, and disruptive conduct.¹⁹ We excluded studies investigating other outcomes, such as predominantly somatic or genetic effects.

Data Collection and Analysis

Selection of Studies

We exported all search results to a reference management program (Citavi, version 6.3.0.35; Swiss Academic Software GmbH), with which we detected and deleted possible duplicates. The remaining search results were then exported to the screening tool Rayyan (Qatar Computing Research Institute – Data Analytics Group). One of us (M. Haker) reviewed all results; 2 others (J.P. and A.C.R.) each reviewed one-half of the results. The reviewers were screen blinded from each other. Conflicts were resolved by discussion. The potentially eligible studies agreed on were then subjected to blinded full-text screening in which M. Haker screened all papers and J.P. and A.C.R. each screened one-half according to the eligibility criteria using a structured screening sheet; see TABLE S2. Screening conflicts were resolved by discussion.

Data Extraction and Analysis

We created a data extraction sheet to ascertain the most relevant aspects of each included study; for details, see TABLE S3. We then extracted the data of all included studies available in full-text. The heterogeneity of the study types, outcomes, and participants did not allow a meta-analysis of the data. We performed a descriptive analysis, combining studies with similar outcomes and differentiating negative, neutral, and positive findings.

Quality Assessment

To assess the quality of the included studies, we created a quality rating form based on the quality appraisal in a previous systematic review,¹⁴ an adapted version of the Graphic Appraisal Tool for Epidemiology.²⁰

In assessing the risk of bias for multiple categories, we assigned an overall quality rating of low, medium, or high to each of the included studies. Because 12 of the included studies were already appraised by a systematic review,¹⁴ we assessed the risk of bias for the remaining 16 studies only. One of us (M. Haker) rated all included studies; 2 others (J.P. and A.C.R.) each rated one-half. We then compared, discussed, and combined the 2 separate ratings into a final rating. For the detailed criteria used, see TABLE S4.

RESULTS

Literature Search

The database search yielded 719 references, of which 111 were identifieds as duplicates and subsequently deleted. We screened the titles and abstracts of the remaining 608 references for eligibility. During this process, we excluded 536 references. We analyzed the remaining 72 references and 2 additional studies, identified in reference lists of previous reviews, in full-text. After the full-text screening we excluded 46 studies, leaving 28 studies for inclusion. For an overview of the inclusion process, see FIGURE S1.

Description of Included Studies

We extracted data from 28 references: 19 cross-sectional studies, 5 cohort studies, and 4 case-control studies. In 2 studies, the same participant sample was examined. Seventeen studies used comparison groups with children of healthy parents; 5 studies collected longitudinal data.

Eight studies originated in the United States, 4 in Canada, 3 in Switzerland, 3 in Australia, 3 in Israel, 2 in Greece, 2 in Denmark, 1 in the United Kingdom, and 1 in Norway; 1 study reported data from the trio of Switzerland, Greece, and Germany. Overall, the sample size varied widely and ranged from 30 to 4177 children with a parent with MS. The ages of the children studied ranged from 1.5 to 26 years, with most studies focusing on children aged 4 to 18 years.

The types of outcomes ascertained and instruments used in the studies are quite heterogeneous. The most frequently assessed outcomes were child adjustment, including emotional and behavioral problems, children’s mental health, and coping with the parent’s illness. Accordingly, the most frequently used instruments were the Strengths and Difficulties Questionnaire,²¹ the Child Behavior Checklist,²² and the Youth Self-Report.²³ In 8 studies, the respondent was a third party (a schoolteacher or an external rater); 6 studies had at least 1 parent and the child as respondents; 7 studies had only children as respondents; 3 studies had only 1 parent as the respondent; and 4 studies had findings based on databases or registries. For an overview of the included studies, see TABLE 1.

Summary of Included Studies

Summary of Included Studies

Quality Assessment

In the quality appraisal, we rated 5 studies as high quality, 6 as medium quality, and 5 as low quality. Combined with the 12 studies previously appraised in the quality assessment by Razaz et al,¹⁴ we report data from 5 high-quality studies, 11 medium-quality studies, and 12 low-quality studies in this review.

Regarding the risk of bias, the most common issue was missing data on participating children (exact age, eligibility criteria, and recruiting), as well as the children’s exposure to the parental illness, eg, exposure duration, MS course, and MS severity. Another frequent concern was the small sample size of many of the studies.

Main Findings

To summarize the findings, we grouped the studies based on their outcomes and reported the direction of their results.

Psychosocial Adjustment

Fifteen studies^24–38 investigated the impact of parental MS on children’s psychosocial adjustment. Eight studies^{25,27–30,35,36,38} found that children with a parent with MS (N = 700) experienced negative emotions (eg, sadness, anxiety) and had behavior difficulties. Five studies^{25,28,30,36,38} compared children with a parent with MS with children with parents without MS (N = 668). Six studies,^{26,28,29,34,35,37} found that depression, fatigue, physical disability, and marital discord for parents with MS correlated with negative outcomes for their children (N = 491).

One cross-sectional study³² compared how much the children of people with MS (N = 56) knew about their parent’s disease. Children with partial information internalized more (eg, felt lonely, sad) and had more social problems compared with children with full or no information about their parent’s MS. One caveat: the psychosocial status of children with no information was reported by their parents because these children were not allowed to complete their own questionnaires.

Three studies^24,31,33 found that the children of people with MS (N = 548) had normal psychosocial development and reported no deviation from expected norms. Whereas Rintell³³ did not use a control group, Andersen et al²⁴ compared their findings with data from 42,016 children of mothers without MS

One cross-sectional study²⁹ found that children of people with MS (N = 48), especially girls, expressed prosocial behavior more often than children of parents without MS. One case-control study³¹ reported that children of a parent with MS (N= 126) were less likely to report peer problems than children of parents without MS (N = 126).

Psychiatric Disorders

Four studies^39–42 assessed the incidence of psychiatric disorders, including mood and anxiety disorders as well as attention-deficit/hyperactivity disorder (ADHD). All 4 of these studies indicated a possible negative impact for children with a parent with MS.

In 2 studies,^39,40 children with a parent with MS (N = 1388) had higher rates of psychiatric symptoms, indicating a higher risk of psychiatric disorders, compared with children with parents without MS (N = 5217). One study⁴¹ reported that children of parents with peripartum depression were more likely to have higher rates of psychiatric disorders and that parents with MS were more likely to be affected by peripartum depression than parents without MS. One cross-sectional study⁴¹ assessed children of people with MS (N = 41) regarding their indication for psychotherapy. According to this study, 22 children of people with MS (54%) needed psychotherapy. This study did not draw a comparison with children of parents without MS.

One case-control study⁴² investigated the influence of maternal chronic illnesses on the prevalence of ADHD in their children, comparing 47,944 children with ADHD with 2,274,713 children without ADHD. Maternal MS was related to an 80% higher chance of the children having ADHD (31 children of mothers with MS out of 47,944 children with ADHD [0.06%] vs 880 children of mothers with MS out of 2,274,713 children without ADHD [0.04%]).

Coping

Four studies^30,41,43,44 (N = 274) reported on the coping skills of children with a parent with MS. Three studies^41,43,44 found that these children (N = 342) likely coped better as they got older and that their coping correlated with their parent’s coping ability, especially that of the parent without MS. One of the studies⁴⁴ compared data with those from children of a parent with other severe illnesses (n = 116). One cross-sectional study³⁰ reported that children of a parent with MS (N = 48) were less likely to use coping strategies that were considered beneficial than children of parents without MS (n = 145) and tended to use coping mechanisms that correlated with negative outcomes.

Feelings Toward Parents

Two studies^45,46 with the same study population (N = 56) ivestigated children’s feelings toward their parents. Yahav et al⁴⁶ found that children of a parent with MS (expressed more feelings of obligation, concern, fear, and anger toward their parent than children of parents without MS (N = 156). Duvdevany et al⁴⁵ found a high prevalence of ambivalence in children of people with MS.

Early Childhood Development

The vulnerability in the early childhood development of children with a parent with MS was examined in 2 studies^47,48 (N = 936). In 2015, Razaz et al⁴⁷ found no general difference in the early childhood development of children of parents with MS (n = 153) and children of parents without MS (n = 876), but parental mental comorbidities, as well as longer exposure to the parental illness, were associated with increased developmental vulnerability in children. A 2016 study⁴⁸ indicated only 1 difference from children of healthy parents (n = 2988): children of people with MS (n = 783) appeared less vulnerable to problems with social competence. Mental and physical comorbidities in mothers with MS seemed to be associated with increased developmental vulnerability in their children.

Body Image

Two cross-sectional studies^33,49 examined the body image of children with a parent with MS (N = 154) and found no significant impact. One of the studies⁴⁹ compared the findings with those from children of parents without MS (n = 60).

Mother-Daughter Interaction

One cross-sectional study⁵⁰ found that in the social interactions of mother-daughter dyads, daughters and their mothers with MS (n = 31) were similar to daughters and mothers without MS (n = 34).

Education

Moberg et al⁵¹ found that children of people with MS (n = 4,177) earned significantly higher school grades, but did not differ in highest degree attained from children of parents without chronic illness (n = 33,416). Furthermore, children of a parent with MS, especially daughters, tended to attain a health-related degree more frequently than children of parents without MS.

DISCUSSION

We conducted a systematic literature review of the psychosocial impact of having a parent with MS on children and adolescents. Most of the 28 studies reported a negative impact, but there is also evidence for positive associations as well as studies claiming no association between having a parent with MS and children’s psychosocial status. The results are largely in accordance with previous systematic reviews on the subject.^14–16,52 Six studies^{24,34,35,37, 42,45} in the present review were not described in previous work. These studies were of mixed quality, and most of them propose negative effects on children of a parent with MS, especially if the parent experiences depression. In contrast to these findings, 1 high-quality study²⁴ reported no impact of maternal MS on children’s mental health.

In summary, 8 studies^{25,27–30,35,36,38} described associations between parental MS and negative outcomes in children’s psychosocial adjustment, mainly difficulties in children’s moods, behavior, and social interactions. This is also corroborated in the broader literature. A parent’s physical illness is assumed to be a risk factor for psychosocial maladjustment in children, mainly in their internalizing and externalizing problems.^2,53,54 Four studies^39–42 reported increased risks for children with a parent with MS of developing mental health conditions, including mood and anxiety disorders and ADHD. Nonetheless, the described effects were usually small, and study limitations have to be considered.

It remains an open question which aspects of parental illness act as the main stressors for children. It is thought that secondary illness-related factors regarding family life and parental functioning contribute to children’s psychosocial risk.^5,53 Further work needs to clarify to what extent an evolving disease such as MS triggers an outbreak of premorbid conflicts within a couple and affects the family structure. Communication is key in managing chronic disease in families. Several researchers emphasize the value of communication and education for children affected by parental illness, thereby promoting integrated psychosocial support in the medical care of people with MS and their families,^2,54–57 yet few studies have addressed the communication of parents affected by MS with their children and how and when to address the parental disease. Paliokosta et al³² highlighted the importance of communication about parental illness, possible changes to the family structure, and that parents may need support to decide on the “how, what, and how much.”

The present findings imply that communication can be an important factor in a family’s well-being and adjustment, and families may be helped by seeking professional support when they approach the difficult task of disclosure. Parents may need help to consider “how, what, and how much”³² they might disclose to their children about a parental illness and forthcoming changes in family life.

Interestingly, 6 studies in this review found that the effects on children were associated with or mediated at least in part by depression in parents with MS, especially if the mother is affected.^{26,28,37,39,44,48} This is especially relevant considering that the prevalence of depression is high in people with MS,⁵⁸ but even without an accompanying MS diagnosis, maternal depression has a substantial effect on the psychological well-being of children.⁵⁹

Various studies^{24,31,33,49,50,51,60} reached different conclusions about mental health, body image, childhood development, social interaction, and/or school performance, presenting evidence suggesting no difference between children with parents with MS and children with parents without MS. Positive effects of having a parent with MS were also described, such as more prosocial behavior, fewer peer problems, less vulnerability regarding social development, and higher school grades.^29,31,48,51 Razaz et al⁴⁸ mentioned the seemingly higher social competence of children of people with MS. They assumed that the greater involvement in household duties due to the parent’s illness might be one of the reasons for the positive effect on children’s social interaction.⁴⁸ It is also possible that the children develop a better reflective ability to regulate their emotions. Further research could test the role of processing emotions in families of people with MS.

Although these findings indicate that a parent’s MS has at least some psychosocial impact, results must be considered with reservation. This systematic review found several methodological issues that limit the validity of the evidence from these studies.

Approximately one-third of the studies did not investigate a comparison group of children with parents without MS, which reduces the validity of the findings. A minority of the studies collected longitudinal data that would allow for some conclusions to be drawn on the continuity of the identified effects, especially considering the psychosocial dynamics of developing children and of MS as an illness. Furthermore, numerous studies failed to describe eligibility criteria, recruitment processes, outcome definitions, adjusted analyses for confounders, or the children’s exposure to their parent’s MS. For future research, children’s data should be analyzed separately for different age groups, and only 1 child per family should be included to avoid clustering effects. The use of instruments that assess data on children by proxy (eg, parents, teachers) should be evaluated carefully. Evidence indicates discrepancies between children’s self-reported outcomes and parental proxy responses.^61,62 Therefore, the by-proxy approach should be applied only in study settings where children are too young to respond themselves or should be used only in addition to speaking with the children themselves.

This systematic review is also not without limitations. Although we searched 3 medical databases and hand searched the references of previous reviews, some relevent data may have been missed. We also included only quantitative and mixed-method studies published in English or German.

Considering the heterogeneous and partially contradictory evidence found in these 28 studies, we emphasize the need for further research. However, the findings in the present review imply that health care providers should ask people with MS about their children’s response to their illness and that parents with MS should talk to their children about it as well. In addition, neurologists need to consider a family perspective when counseling parents with MS. Because this is not a part of medical education, physicians need support and training in how to address these issues. Furthermore, health systems need to develop integrated structures to organize and finance family counseling for people with chronic illnesses. Not every child with a parent with MS will need psychosocial support, but it could be beneficial for parents and physicians to keep in mind that adjustment problems may occur. As a first step, a decision aid for people with MS considering parenthood could raise awareness and thereby help people with MS talk about this topic with their physicians and family members.

CONCLUSION

Overall, the psychosocial impact of parental MS on children is not yet clearly understood. Evidence suggests negative consequences on children’s psychosocial well-being, but a few studies also indicated positive effects or no difference from children with parents without MS. The variable study quality and contradictory findings emphasize the need for further research to allow definite conclusions and recommendations for clinical practice.

PRACTICE POINTS

Evidence suggests that having a parent with multiple sclerosis (MS) has mixed effects on the psychosocial well-being of children, but further research is needed.

Physicians caring for people with MS need to be aware of the potential effect on the patient’s family and should address this effect when counseling parents with MS. Children affected by their parent’s illness can be directed to support options.