Parental stress in families of children with special educational needs: a systematic review

Background This systematic review aimed to identify the risk factors and protective factors for parental stress in families with children with special educational needs. Studies have indicated that the wellbeing of families is related to the physical, psychological, and social conditions of the children, as well as the family functioning, stress coping strategies, and social and professional support of their parents. The parents of children with special educational needs experience high levels of parental stress. Identifying the associated risk factors and protective factors may shed light on the provision of interventions to promote the mental wellbeing of these parents. Methods Boolean operators were used to search multiple online databases, and the Preferred Reporting Items for Systematic Reviews and Meta-Analyses guidelines were applied in this systematic review. Data were extracted into categories of population, age, region, the child's diagnosis, the stress-measuring instrument, and the risk factors and protective factors. Results Twenty-six studies, including 5,169 parents and 3,726 children, were reviewed. The following four major risk factors and protective factors were found to be associated with parental stress: the sex of the parents, diagnosis-related coping issues, socioeconomic characteristics, and social isolation of the parents. Conclusions This systematic review identified four significant risk factors and protective factors related to social support from couples, family members, and social circles. Various agencies may provide financial and manpower assistance and professional support and services to improve the parents' knowledge and coping skills, as well as affectional support, early screening, and continuous assessment of the parents' progress. Social policies and interventions offering continuous and diagnosis-related support to the parents of children with special educational needs are highly recommended.


Introduction
Over 291 million children and adolescents younger than 20 years globally were estimated to have developmental disabilities and special educational needs (1). These children may have disabilities, such as intellectual disability, epilepsy, hearing or vision loss, autism spectrum disorder (ASD), or attention deficit hyperactivity disorder (2). Parenting a child with SEN may be a wonderful journey with a sense of accomplishment and excitement, but it may also be challenging or even impose a caregiving burden (3).
Parents of children with SEN may be prone to psychological distress, such as anxiety, sleep disturbances, and frustration, when facing the behavioral problems of their children (4,5). For example, a study by Caley (6) found that the mother, as the primary caretaker of SEN .
/fpsyt. . experienced higher stress levels. Stress may come from managing the challenging behavior and special needs of children. Aif et al. (7) concluded that parents bringing up children with SEN face overall family life changes, and burdens affect many aspects of life. Prolonged stress from handling children's daily-life problems and diverse and challenging needs may affect family functioning and wellbeing (8,9). Parents may also feel stress and experience additional financial strain when taking care of their children who have SEN in combination with fluctuating health conditions or the requirement for repeated hospitalization (10-12). The impact of taking care of children with SEN is multifaceted. It is not uncommon for such parents to have marital problems, physical and psychological distress, or mental health issues (13)(14)(15). Studies have provided some hints of the possible factors contributing to the mental wellbeing of the parents of children with SEN. For example, families who receive services designed based on person-centered and family-centered approaches have been found to experience less marital stress. These families may develop more appropriate or adaptive techniques to take care of their children and may feel more supported by healthcare service providers (3). Psychoeducation programs designed to address the challenging behavior and health conditions of children with SEN (e.g., ASD) have been shown to effectively reduce the maternal burden (16).
To improve the wellbeing of the parents or informal caregivers of children with SEN, it is essential to identify the risk factors and protective factors for stress in this population. The aim of this study was to identify the risk factors and protective factors affecting the stress level of the parents of children with SEN. A better understanding of these factors may help to provide guidelines for service providers to design effective interventions (17)(18)(19).

Operation definition
This is a systematic review of studies on stress experienced by the parents of children (aged 3-17) with SEN. In this paper, SEN include specific learning difficulties, intellectual disability, ASD, attention deficit hyperactivity disorder, physical disability, visual impairment, hearing impairment, and speech and language impairments (20). Parent-related stress represents the level of dysfunction in the parent-child system related to the parent's functioning (15).

Article selection
The initial search was first applied to the Cochrane Database of Systematic Reviews to determine if any previous reviews covered our topic. After confirming that there was no equivalent review, an extended search was conducted.
This systematic review followed the Preferred Reporting Items for Systematic Reviews and Meta-Analyses protocols (21). Boolean searches were used to interrogate PubMed, Embase, Cochrane Library, PsycINFO (via ProQuest), and Web of Science databases. Studies published from 1960 to 2021 were searched. The following search terms were entered into the databases: "parental stress, " "special educational needs, " "risk factor, " and "stress level measurement." The detailed search terms included "parental stress AND special educational needs, " "parental stress AND special educational needs and/or disabilities, " "parental stress AND special educational needs AND measuring risk level, " "parental stress AND special educational needs AND level of risk, " "parental stress AND special educational needs AND risk factor, " "parental stress AND special educational needs AND stress level measurement, " "parental stress level measurement AND special educational needs, " "parenting stress level measurement AND special educational needs, " and "parent stress level AND special educational needs." Articles were included in the analysis if: • they reported primary research investigating stress experienced by the mother, father, or both parents of children with SEN, with at least one quantitative measurement or in a qualitative format; • they were published in a peer-reviewed journal; and • the full-text publication was available in English.
Articles were excluded if: • they reported a study unrelated to parental stress or • they reported secondary research or were a conference presentation or unpublished thesis.
The primary database search identified 3,092 records. Information was imported to EndNote 20 (Clarivate, London, UK) for the deduplication process to eliminate redundant data. After completing this process, 2,524 articles were excluded. In addition, 169 articles were excluded as they were not relevant. Another 169 articles were excluded as they did not evaluate both parental stress and SEN. Another 204 articles were excluded because they reported secondary research, consisted of a manuscript or conference material with an abstract only, or did not report parental stress. No further eligible studies were identified during the manual screening process. Finally, 26 articles were included in this systematic review. The details of the screening process, which followed the Preferred Reporting Items for Systematic Reviews and Meta-Analyses guidelines (22), are described in Figure 1.

Description of the articles reviewed
After screening the articles, relevant data were extracted and summarized in tabular format. The characteristics of the included studies are summarized in Table 1.
This review covered 26 studies, which included seven studies from the USA; five from Europe; and 14 from Asia, including five from China and Hong Kong. Of these 26 studies, 11 involved ASD, six involved ADHD, and 11 involved intellectual disability. For the articles involving intellectual disability, different terms, such as cognitive delay .

Risk factors and protective factors
The following four major categories of risk factors and protective factors were identified: the sex of the parents, diagnosisrelated coping issues, socioeconomic characteristics, and social isolation of the parents.

Sex of the parents
Mothers were reported to perceive more parental stress than fathers in four studies, mainly due to their labor workload and their need to handle unpredictable situations (28- 30,36). Only one study reported the same stress level between mothers and fathers in 15 families (39). Parental stress was found to increase with the increasing age of the parents (29).

Diagnosis-related coping issues
Twelve studies reported that the major risk factors were the challenging behavior or maladaptive behavior of children with SEN. Behavioral problems related to ASD or ADHD or the emotional problems of children with SEN were related to higher levels of parental stress (23- 25,27,28,31,33,35,39,47). Higher levels of emotional problems of SEN children, higher parental stress were recorded. Parents who do not have adequate professional or social support may develop poor parenting practices. Negative coping habits or handling methods may result from the lack of appropriate knowledge and competence to handle stressful situations (24,31,32,43).
Protective factors, such as professional support (23, 25, 27-32, 34, 35,41,44), and positive coping skills and techniques, such as stress management strategies, were related to lower levels of parental stress (31,42,45,47). That is, a better understanding of the child's progress and gaining knowledge and skills to manage the behaviors of children with SEN were also protective factors. Furthermore, a study of parents reported that those who knew the diagnosis of their child since birth were able to adjust their expectations (23). For example, the parents were able to cope better if the cognitive deficits of their children were identified at birth, rather than acquired later.

Socioeconomic characteristics
Having financial problems (31,34,45) and a family's lower socioeconomic level (44,46) were related to higher levels of parental stress in parents of children with SEN compared with parents of typically developing children.
Five studies (31,34,(44)(45)(46) from countries including Israel, Vietnam, Malawi of Africa, and China reported that financial issues were a risk factor, but parents demonstrated hesitation and resistance to seek financial assistance due to social pressure and associated feelings of shame about disclosing the family's situation. Another two studies found that the self-disclosure of the family's information and needs when approaching government services was a source of shame and conflict with other family members.

Social isolation of parents
Five studies reported that social isolation and a poor social life were sources of parenting burdens and increased the stress level of parents (24,(31)(32)(33)43). This occurred because the lack time for oneself and insufficient social support may increase perceived levels of stress. Another three studies reported that poor family support induced extra stress during the caregiving journey (37, 38,43). Four studies indicated that stigma and judgment from others induced stress in the parents of children with SEN (25,31,40,47).
However, it was found that supportive family and marital relationships and sufficient labor help were protective factors. Support from professionals (25, 27-29, 31, 32, 37, 41, 42, 45, 47) and family members (7,26,27,29,44) were factors contributing to a reduction in the levels of parental stress. Regarding family support, four studies showed that positive marital relationships and support from spouses were essential protective factors (35,38,39,44). Three studies conducted in the Asian region: Jordan, Malaysia, and Israel (30,37,40) indicated that strong family support promoted a positive perception of the parental task as a challenge rather than a burden. Sufficient professional services were found to be useful for improving the mindfulness and attitudes of the parents (25) and their knowledge of disease and behavioral management (29, 32,45,47). Ongoing assessments of the parenting situation (41) and stress management techniques (30,42, 47) may also reduce parental stress. Thirteen studies applied the Parenting Stress Index-Short Form to evaluate parental stress.

Sex of the parents
The results indicated that there were differences in parental stress levels between fathers and mothers, with mothers perceiving higher stress levels than fathers. A higher stress level may be induced by closer interactions with children to handle behavior related to their diagnosis. Role identification within the family may be one of the underlying reasons for this finding; that is, the father may focus on breadwinning rather than caring or daily house activities. A heavy workload for the daily care of children and more frequent interactions with problematic behavior were some of the sources of parental stress. The major caring role was shouldered by the mother, leading to higher stress levels for mothers. Studies (48,49) have shown that parental stress is induced by heavy physical and psychological burdens related to daily caring and continuous worrying about the child's future. This may have negative effects on the entire family system and directly impact the quality of life of the whole family (50)(51)(52). In China, mothers generally take greater responsibility for childcare than fathers (45). Mothers face unique challenges in handling SEN children's emotional and behavioral symptoms. The mothers may perceive being left alone in the caring duties as they play a significant role in raising SEN children. When adequate external support is available, the mothers may gain confidence to adopt a more positive living style. Professional support may include caring techniques, knowledge of SEN, and information on available services. Direct intervention from different professions may reduce parental stress more effectively.
The father was identified as an immeasurable source of support (39), Further studies on how the family role and the father-child relationship affect parental stress are recommended. In the meanwhile, service providers may consider to actively involve father into training as a partner, to fully utilize their strengths to support their spouses. Adequate support to fathers and recognitions afterwards are therefore foundations to development of fathers.

Diagnosis-related coping issues
For diagnosis-related issues, one of the identified risk factors was handling the challenging behavior of the children. The underlying deficits leading to the challenging behavior of children with SEN are varied. The lack of knowledge and skills in behavioral management of children with SEN may induce negative coping behaviors (e.g., reacting with anger), which cause further anxiety and stress to the parents (6,53). Therefore, diagnosisspecific psychoeducation programs (e.g., knowledge of the specific conditions of children with SEN and parenting, behavioral management, problem-solving, and stress management skills) would be helpful to reduce the stress levels of the parents (5,10,54).
Formal support from professionals and government policy may provide structured and sustainable assistance to parents to improve their knowledge and skills to handle problems or crisis in daily care. The informal support system, on the other hand, including        support from family members, relatives and friends, and even neighborhoods, can act as a moderating factor to reduce parental stress and improve the wellbeing of mothers (40). Multiple studies indicated social support is an essential protective factor to reduce perceived parental stress (39,44,47). Some parents may consider their social life is being sacrificed. Muñoz-Silva et al. (24) found that the strongest predictor of mothers' stress is the negative impact on their social life, but not the children's emotional problems nor the mothers' perceived social support. Therefore, the parent's social life should be considered when designing an improvement plan for the family. Because of negative coping habits or handling methods, parents have a feeling of shame when children exhibit uncontrollable challenging behavior in a social environment (40) and they blame themselves (7,38) or have low confidence in managing their children with SEN (46). Long-term follow-up by a professional is recommended. For example, professionals may provide ongoing monitoring of the parents' stress levels and refer them to suitable professional services as required.

Socioeconomic characteristics
Financial and human resource constraints may be some of the major risk factors for parental stress, as parents from lower socioeconomic levels need extra support for expenses and manpower to care for their children with SEN (55). For example, the parents of children with SEN may perceive extra physical, time, and financial concerns when bringing their children to receive treatment. To minimize the traveling costs, telehealth or telerehabilitation may be alternative options to support the parents. However, further enhancement of telehealth or tele-rehabilitation systems is needed. Government policies on financial support for the parents of children with SEN are also recommended.

Social isolation of parents
Emotional support was one of the main protective factors identified. Professional staff may provide opportunities for parents to express their concerns, unmet needs, and worries. Social groups were able to provide opportunities for parents to share their unique stories with others with similar experiences. Studies have also suggested the importance of social relationships in reducing parental stress (3,6).
Chu et al. (37) conducted a study which recruited 110 parents of children with ASD, with 80 mothers and 30 fathers, with ages mainly around 21-50, from an online support group. It was found that although stressful situations and parenting difficulties occurred due to time-demanding tasks required by the children, parents expressed that affiliate stigma does not affect their stress levels and even quality of life. It is worth to note that the participants were active support group members who shared knowledge and offered social support in caring for ASD children. The members of this support group were also exposed to massive amounts of information and experience sharing and also receiving therapy from different professionals. Sharing positive coping strategies can tackle stress associated with feelings of helplessness and isolation (56). This indicates some essential features of peer support group that may help reducing parental stress. Cultural factors should be considered as 93.6% of the participants in that study are Malay. Malay perceives the child as a form of test from God, and parents will be judged in the afterlife (57). This may have an impact on the view of caring duties of caring for their children with SEN.

Limitations
This systematic review has several limitations. First, most of the research participants were mothers (3,585 mothers and 699 fathers). The participants joined the studies voluntarily, but actively participating attendants may have a more positive attitude, which may have biased the results. Second, not all studies included a control group or applied a randomized control trial in the research design. Third, the screening, selection, and data extraction processes were performed by a single researcher. Therefore, further studies on parental stress experienced by fathers, performed with the involvement of a second researcher are recommended.

Conclusions
This systematic review synthesized recent findings on the risk factors and protective factors for parental stress in the families of children with SEN. Regarding risk factors, handling the challenging behavior of children is the major component of diagnosis-related coping issues. Financial problems and lower socioeconomic levels restricted the time and resources to care for SEN children. Social isolation of parents is considered as a risk factor caused by poor social life, insufficient family support, and even stigma and judgment from others. On the other hand, protective factors such as professional support can improve positive coping skills and gain a better understanding of a child's progress. Supporting family systems and positive marital relationships can lead to better resilience and coping techniques for parents, leading to better wellbeing.
Healthcare professionals have a unique role in remediating parents' burdens. Parents expected a gracious explanation or time from professionals. Parents treasured being taken by the professionals seriously. Considering the parental stress induced by taking caring of SEN children, healthcare professionals can act as a source of information and direct support as well as assisting parents in developing positive coping techniques. A valid parental stress assessment is essential. It is noticed that the most commonly applied parental measurement tool by healthcare professionals was the Parenting Stress Index-Short Form. Moreover, measurement tool identifying the risk factor and protector factor would be needed to promote the resilience of the parents.
Parenting a child with SEN may be a complicated and demanding task, which may lead to additional financial, physical, psychological, and social burdens. This review identified the possible risk factors and protective factors for parental stress, with the aim of identifying ways to relieve this stress.
The findings from the reviews suggest when identified parents of SEN with high stress, service provider should provide choices .
/fpsyt. . of stress-relieving programs, direct assistance in manpower or financial issue, and also provide professional opinion to develop positive handling technique. When existing services cannot fulfill particular needs, referral to health care professionals should be considered, in order to design custom made program. It may guide the practice of service providers. The development of diagnosisspecific parental support programs and future studies on their impact on parents with different family roles are recommended.

Data availability statement
The original contributions presented in the study are included in the article/supplementary material, further inquiries can be directed to the corresponding author.