Courtesy stigma among primary caregivers of children with autism spectrum disorder in eastern China

Introduction The experience and perception of stigma is a common problem among primary caregivers of children with autism spectrum disorder (ASD), and has a profound adverse impact on primary caregivers and children with ASD; however, few studies have explored courtesy stigma among primary caregivers of children with ASD in the Chinese context. The aim of this study was to explore the status of courtesy stigma among primary caregivers of children with ASD in Lianyungang, Jiangsu Province, Eastern China, and to conduct in-depth analysis of its predictors from multiple perspectives. Methods An institution-based multi-center cross-sectional survey was conducted in the rehabilitation department of a large specialized hospital and 10 rehabilitation centers for children with special needs in Lianyungang, Jiangsu Province, Eastern China, from October 2022 to February 2023. A structured questionnaire to assess child-related factors, primary caregiver-related factors, courtesy stigma, general self-efficacy, and social support, was used to collect data. Predictors of courtesy stigma among primary caregivers of children with ASD were identified by linear regression. Results A total of 428 primary caregivers of children with ASD were recruited. The mean ± standard deviation (SD) score for courtesy stigma was 7.49 ± 4.13. Multiple linear regression analysis revealed that primary caregivers of children with ASD who were not too satisfied with their current marital status (β = 1.21, 95% CI: 0.34–2.08, p < 0.05) were more likely to have a high courtesy stigma; however, significantly lower courtesy stigma was observed in primary caregivers of children with ASD who were not picky eaters (β = −1.33, 95% CI: −2.08 – –0.58, p < 0.05), and who reported low level challenge in caring for children with ASD (β = −1.16, 95% CI: −2.20 – –0.12, p < 0.05), good general self-efficacy (β = −0.16, 95% CI: −0.25 – –0.06, p < 0.05), and good social support (β = −0.04, 95% CI: −0.08 – –0.01, p < 0.05). Conclusion There is a high level of courtesy stigma among primary caregivers of children with ASD in eastern China, and it is affected by numerous factors. More resources should be directed to groups that are more likely to experience stigma.


Introduction
Autism spectrum disorder (ASD) is a neurodevelopmental condition, characterized by social and communication impairments and limited repetitive behavioral features, the prevalence of which is increasing worldwide (1,2).A recent meta-analysis reported that the prevalence of ASD in China is 26 per 10,000 people and rising (3).Due to insufficient comprehension and misunderstanding of ASD in mainstream society, the characteristics of children with ASD often lead to discrimination toward them (4).Notably, some characteristics of children with ASD are often also blamed on their caregivers, who may be accused of being unskilled and inefficient in caring for their children; thus, caregivers are also exposed to stigma (5)(6)(7).Primary caregivers of children with ASD often experience stigma, including courtesy stigma and affiliate stigma, particularly in school and community settings, and more so than primary caregivers of children with intellectual and physical disabilities (6,8).Courtesy or affiliate stigma are types of social stigma experienced due to association with a stigmatized person (9).In courtesy stigma, primary caregivers experience stigma due to sharing interpersonal relationships with an affected child, while affiliate stigma focuses on negative stereotypes internalized by the primary caregiver (4).Although there are high levels of stigma among primary caregivers of children with ASD, there has been insufficient research into stigma, particularly courtesy stigma, in this context.
Stigma can lead to low self-esteem and social withdrawal of primary caregivers of children with ASD (10).Further, it can lead to reduced health-seeking behavior and even impaired caregiving, which can affect the mood and behavior of children (11,12).Longitudinal studies have confirmed that parental experiences of discrimination have a significant impact on internalized and externalized features in children with ASD (13).Primary caregivers of children with ASD who experience stigma also have increased levels of psychological distress, including sadness, helplessness, hopelessness, tension, worry, irritability, and fear, which can all have negative impacts on their relationships with their spouses and children (13,14); however, empirical research on stigma interventions among primary caregivers of children with ASD is inadequate (4).Therefore, study of this phenomenon and factors predictive of stigma among primary caregivers of children with ASD is necessary to, to provide a reference for formulation of intervention measures.
In recent years, there has been an increased research focus on stigma in primary caregivers of children with ASD.Studies conducted in multiple countries have shown that child-related factors, including the sex, age, and severity of symptoms of children, are predictors of stigma among primary caregivers of children with ASD (15)(16)(17)(18).In terms of primary caregiver-related factors, resilience, relationship with children, depression, anxiety, selfcompassion, cognitive fusion, burden, self-esteem, trait mindfulness, and perceived stress, marital satisfaction of the primary caregiver were correlated with stigma (17-24).In addition, unmet ASD care needs, family structure, and the number of children with ASD in the family were also factors influencing stigma (25).Marital satisfaction was found to be associated with higher negative emotions and stigma in mothers of children with ASD (26,27).Despite these findings, research to date has been confined to investigation of factors influencing stigma from limited perspectives.Notably, studies from other populations have found that caregivers of picky eaters face criticism from others and experience stigma because their parenting skills are often considered ineffective (28).Income, selfefficacy, and social support are also important predictors of stigma (29-33).Income can affect the parenting ability of children with ASD.Caregivers with higher income may have better knowledge of behavior management through education and access to ASD services (34), and thus better manage their children and reduce the negative views of others.Evidence already suggests that low income is a predictor of emotional problems in mothers of children with ASD (35).Self-efficacy refers to a relatively stable, confident personality trait and a sense of optimism about one's ability to face life's difficulties (36).Caregivers with higher self-efficacy are more likely to deal with difficulties with confidence and may experience lower levels of stigma.Family support as part of social support has been elucidated to be significantly negatively associated with stigma (37).However, there is a lack of research on the relationship between social support and courtesy stigma.Perceived social support may be key to improving caregiver resilience during the diagnosis and management of children with ASD (38).Lack of social support may feel more social exclusion and isolation, increasing stigma.Further, family caregivers with high caregiving burden experienced more severe stigma (39).In addition, challenging parenting experiences predict anxiety, depression, and stress symptoms in mothers of children with ASD (40).Caregivers with challenging parenting experiences may encounter more difficulties in the process of caregiving, such as managing children's behavioral problems, others' perceptions of children, and parenting confidence.However, whether challenging parenting experiences influence stigma is unclear.Based on these publications, we inferred that the reported variables may impact courtesy stigma among primary caregivers of children with ASD.
It is difficult for primary caregivers of children with ASD to avoid struggling with stigma.Identifying predictors of stigma among primary caregivers of children with ASD could help to identify populations at risk and develop effective interventions; however, stigma may also vary depending on socio-cultural contexts, such as political systems, etiquette customs, and value systems (41).Most research on stigma to date has been limited to western developed countries.Numerous researchers have noted that further research on stigma among primary caregivers of children with ASD in developing countries is needed to understand the experience of the primary caregivers of children with ASD in different contexts (15,42).Stigma may be more prevalent in Asian societies, due to the concept of "face" and stigma socialization (4)."Face" describes the "self-image" that a person experiences because of the evaluation of a particular situation by others.People may have the feeling of losing their "face, " due to negative social evaluation (43).In addition, stigma may be more prominent in China's collectivist culture (41), as individuals who deviate from societal norms in collectivist cultures tend to experience more stigmatization (44).Different cultural practices in China may also contribute to heightened levels of stigma relative to those in other countries (37).Due to the emphasis on social acceptance and validation in Chinese culture, individuals tend to exhibit heightened sensitivity towards negative public perceptions; however, there has been insufficient research on courtesy stigma among primary caregivers of children with ASD in eastern China.

Aims of the current study
The aims of the current study were to explore the present status of courtesy stigma among primary caregivers of children with ASD in Lianyungang, Jiangsu Province, Eastern China, and to conduct deep analysis of the effects of child-related factors (sex, age, duration of rehabilitation, comorbidities, severity of symptoms, single child, and picky eater), primary caregiver-related factors (sex, age, education level, relationship to children with ASD, area of residence, employment status, monthly family income, satisfaction with marital status, levels of challenge in caring for children, and economic burden), general self-efficacy, and social support on courtesy stigma.Our findings will increase the current limited knowledge of courtesy stigma among primary caregivers of children with ASD in China, and will be instructive for the design and development of effective interventions to reduce courtesy stigma among primary caregivers of children with ASD.

Study design
An institution-based multi-center cross-sectional survey was conducted from October 2022 to February 2023 in the rehabilitation department of a large specialized hospital and 10 rehabilitation centers for children with special needs in Lianyungang, Jiangsu Province, Eastern China; these facilities are responsible for providing services for children with ASD.

Participants
At the study sites, primary caregivers (including fathers, mothers, and grandparents) who reported that they lived with, and provided the most care services for, a child with ASD were invited to participate in the study.The inclusion criteria were as follows: (1) primary caregivers of children diagnosed with ASD by an occupational clinician, according to the diagnostic criteria of the American Psychiatric Association's Diagnostic and Statistical Manual of Mental Disorders, Fifth Edition (DSM-5), which are widely used in Chinese hospitals; (2) children with ASD were ≤ 12 years old; (3) the primary caregiver was ≥18 years old; (4) able to understand the research process and the content of the questionnaire; (5) consent to participate in the study.In order to exclude stigma caused by other serious physical illness and neurological diseases, to obtain the stigma of primary caregivers caused by children with ASD, and to consider the quality of questionnaire completion, we set exclusion criteria.The exclusion criteria were: (1) children with severe physical illness or other neurological diseases; (2) the primary caregiver had a history of any psychiatric disorder diagnosed by an occupational clinician.The minimum sample size required was calculated using the singlepopulation proportion formula.Due to the lack of relevant previous studies at the study site, to obtain the maximum sample size, we used the prevalence of stigma as 50% (p = 50%), 95% confidence interval (CI), margin error of 5%, and non-response rate of 10%.The required sample size was calculated as 423.A systematic random sampling technique was employed to select participants.To obtain more reliable conclusions, a total of 450 primary caregivers were invited to participate in the survey, and 450 questionnaires were collected, with a response rate of 100%.Among them, 22 questionnaires were excluded as invalid, because they were not fully completed due to time constraints.A total of 428 valid questionnaires were included in the study (effective response rate, 95.1%).The characteristics of the sample are presented in Table 1.

Procedure
One primary caregiver for each child was asked to complete a questionnaire.The investigators were five people with a master's degree or above from a large specialized hospital who had received standardized training.The investigators informed the primary caregivers of children with ASD about the purpose of the current study, the process, and the benefits of participation, etc.They also provided explanations to the primary caregivers when questions in the questionnaire required clarification.In appreciation for participation, we presented a small gift to each primary caregiver who completed the questionnaire.

Measures
A structured questionnaire, designed based on an extensive review of the relevant literature and consultation with experts in the field, was used for data collection.In addition, we conducted a pre-survey at the study site.According to the findings of the pre-survey and additional expert evaluation, the questionnaire was revised and improved.The questionnaire included five parts: child-related factors, primary caregiver-related factors, courtesy stigma, general selfefficacy, and social support.Both child-related factors and primary caregiver-related factors were measured using a single item.Childrelated factors included sex, age, duration of rehabilitation, comorbidities, severity of symptoms, single child, and picky eater (referring to the rejection of numerous of familiar or unfamiliar foods and intake of an inadequate variety of foods).The severity of symptoms was classified as mild, moderate, or severe and was completed by the primary caregivers based on a clinician diagnosis.Picky eater was assessed by asking "Does your child refuse to eat certain foods or eat very little (e.g., vegetables, fruits)?. " Primary caregiver-related factors included sex, age, education level, relationship to children with ASD, area of residence, employment status, monthly family income, satisfaction with marital status, levels of challenge in caring for children, and economic burden.Satisfaction with marital status was measured by asking "Are you satisfied with your current marital status?. " Levels of challenge in caring for children were assessed by asking "How challenging do you think it is to take care of your child?. " Economic burden was determined by asking "How do you feel about the economic burden of your child's treatment on you?" Courtesy stigma was measured using the Perceived Courtesy Stigma Scale (PCSS), modified from the Devaluation of Consumer Families Scale (DCFS) (45).The scale contains 7 items, and each item is scored on a 4-point Likert scale, ranging from "strongly disagree" to "strongly agree, " with scores of 0 to 3, respectively.The total score is the sum of the scores for each item, with higher scores indicating greater courtesy stigma.The effectiveness of the PCSS has been demonstrated among primary caregivers of children with ASD (41).In the current study, the Cronbach's α coefficient value for the PCSS was 0.895.
General self-efficacy was assessed using the General Self-Efficacy Scale (GSES).Self-efficacy refers to an individual's perception or belief about whether they can take appropriate actions in the environment (46).The GSES consists of 10 items, and each item is scored on a 4-point Likert scale ranging from "not at all true" to "exactly true" with scores of 1 to 4, respectively.The scores of each item were summed to obtain the total score, which ranged from 10 to 40.A higher total score indicates greater general self-efficacy.The scale has been shown to have good reliability and validity (47), and its Cronbach's α coefficient value in the current study was 0.911.
Social support was assessed using the Multidimensional Scale of Perceived Social Support (MSPSS), which comprises three dimensions: family support, friend support, and other support (48).The scale consists of 12 items, with four items included in each dimension.Each item is scored on a 7-point Likert scale ranging from "very strongly disagree" to "very strongly agree" with scores of 1 to 7, respectively.The total score is the sum of the scores for each item, with higher scores indicating greater levels of social support.The scale has been widely used in a variety of populations in China and other countries, including primary caregivers of children with ASD, and has good reliability and validity (34,49).In the current study, the Cronbach's α coefficient value was 0.956.

Ethical approval
This study was approved by the Ethics Committee of Lianyungang Maternal and Child Health Hospital.This study was conducted in line with the principles of the Declaration of Helsinki.All participants were informed of the purpose of the study, the confidentiality of the data, and the right to withdraw from the study at any time before participating in the study.All participants provided written informed consent.

Data analysis
Eligible questionnaires with no missing items were coded and entered into a database created using EpiData 3.1 software (EpiData Association, Odense, Denmark).All data were exported to SPSS21.0 software (IBM Corporation, Armonk, State of New York) for statistical analysis.Continuous data were tested for normality by calculating kurtosis and skewness coefficients (50), and were found to be normally distributed.Continuous data are described as mean and standard deviation (SD), and categorical data are described as frequency and percentage.The significance of differences in courtesy stigma scores among primary caregivers of children with ASD were evaluated using t-tests and one-way ANOVA.Pearson correlation analysis was used to assess correlations between general self-efficacy, social support, and courtesy stigma among primary caregivers of children with ASD.Variables with statistical significance (p < 0.05) in univariate analysis were included in a multiple linear regression model, to exclude the influence of confounding factors and identify independent factors influencing courtesy stigma among primary caregivers of children with

Results
Current status of courtesy stigma among primary caregivers of children with ASD Among the 428 primary caregivers of children with ASD, the mean ± SD courtesy stigma score was 7.49 ± 4.13, and the mean ± SD score for each item was 1.07 ± 0.59.

Child-related factors
Of the participants' children with ASD, a large proportion were male (69.63%) and < 6 years old (66.82%).More than half of children (52.34%) had undergone rehabilitation treatment for >1 year, and a small proportion (6.54%) had co-morbid diseases.Almost one-fifth of the children (18.93%) had severe symptoms, almost half (47.20%) were only children, and approximately threefifths (59.81%) were picky eaters (Table 1).Univariate analyses showed that primary caregiver courtesy stigma scores were significantly associated with severity of symptoms and picky eaters (p < 0.05) (Table 2).

Primary caregiver-related factors
The 428 primary caregivers who participated in this study ranged in age from 21 to 71 years, with a mean age of 34.36 ± 7.22 years.Almost two-thirds of the primary caregivers (64.02%) were female, and nearly three-fifths (58.88%) had below college degree education.Approximately one-third of the primary caregivers (34.58%) were fathers, 60.98% were mothers, and only 4.44% were grandparents of the children with ASD.More than half of the primary caregivers (54.44%) lived in urban areas and almost half (49.77%) were currently unemployed.More than two-fifths of primary caregivers (42.52%) had a monthly family income of ≤5,000 Yuan (Table 1).Univariate analyses indicated that there were significant differences in courtesy stigma scores among primary caregivers of children with ASD according to monthly family income, satisfaction with marital status, levels of challenge in caring for children, and economic burden (p < 0.05) (Table 3).

Correlations of general self-efficacy and social support with courtesy stigma
Continuous data were tested for normality by analysis of kurtosis and skewness (50).The absolute kurtosis and skewness values of the studied variables were within 1.7 and 0.6, respectively, indicating that the data met the criteria for normal distribution.Among the respondents, mean ± SD general self-efficacy and social support scores were 26.36 ± 4.49 and 57.20 ± 13.71, respectively.Correlation analyses showed that both general self-efficacy and social support were negatively correlated with courtesy stigma of primary caregivers of children with ASD (r = −0.297and r = −0.271,respectively, p < 0.001) (Table 4).

Multiple linear regression analysis of factors associated with courtesy stigma
Multiple linear regression analysis indicated that the primary caregivers of children who were not picky eaters had lower courtesy stigma scores than those with picky eaters (β = −1.33,95% CI: −2.08 --0.58, p < 0.05).Primary caregivers who were not too satisfied with their current marital status had higher courtesy stigma scores than those who were satisfied (β = 1.21, 95% CI: 0.34-2.08,p < 0.05).Primary caregivers who reported a low level of challenge in caring for children with ASD had lower courtesy stigma scores than those who reported a high level of challenge (β = −1.16,95% CI: −2.20 --0.12, p < 0.05).In addition, primary caregivers with good general self-efficacy (β = −0.16,95% CI: −0.25 --0.06, p < 0.05) and social support (β = −0.04,95% CI: −0.08 --0.01, p < 0.05) had low courtesy stigma scores.Therefore, child picky eaters, satisfaction with marital status, challenges of caring for children, general self-efficacy, and social support may be useful for predicting courtesy stigma among primary caregivers of children with ASD (Table 5).

Discussion
Stigma has been identified as a major problem experienced by the primary caregivers of children with ASD (42).This study addressed the gap in research on courtesy stigma among primary caregivers of children with ASD in eastern China, by determining the status of courtesy stigma among primary caregivers of children with ASD in Lianyungang, Jiangsu Province, Eastern China and identifying possible predictors.Our findings show that the mean ± SD score of the courtesy stigma scale was 7.49 ± 4.13, while the mean ± SD score for each item was 1.07 ± 0.59.Previous qualitative studies conducted in China have suggested that parents of children with ASD often experience courtesy stigma, due to their close relationship with the child (6).Therefore, there is an urgent need to develop effective interventions to help reduce courtesy stigma for primary caregivers of children with ASD.We found that the child-related factor, picky eating, was associated with courtesy stigma among primary caregivers of children with ASD.Primary caregiver-related factors, including satisfaction with marital status and level of challenge in caring for children were also associated with courtesy stigma, as were general self-efficacy and social support.In terms of child-related factors, our study showed that sex, age, duration of rehabilitation, comorbidities, severity of symptoms, and single child were not significantly associated with courtesy stigma.In terms of primary caregiver-related factors, sex, age, education level, relationship to children with ASD, area of residence, employment status, monthly family income, and economic burden were not significantly associated with courtesy stigma.

Relationship of child-related factors with courtesy stigma
In this study, 69.63% of the children with ASD were boys, which is consistent with the sex distribution of ASD in epidemiological studies (51); however, we did not find any association between the sex of children with ASD and courtesy stigma of primary caregivers, consistent with a previous study conducted in Guangdong, China (37), but in contrast with a study conducted in eastern India, where parents of girls with ASD reported higher stigma scores (18).This may be due to differences in measurement instruments, sample size, and study subjects.This may also be due to variation in social and cultural background, such as political system and etiquette customs (41) and more extensive future studies in different regions are warranted.Children with ASD are often described as picky or selective eaters (52, 53).Most children with ASD show aversion to specific food colors, textures, odors, or other food characteristics (54).The results of this study suggest that primary caregivers of children with ASD who are picky eaters are more likely to experience courtesy stigma, possibly because they are more likely to perceive their children as unhealthy eaters or eating too few food types, and to have negative views of the eating behavior of their children (55).This may cause caregivers to feel that they will be perceived differently, thus increasing courtesy stigma.In addition, people may also perceive their children as "difficult" and/ or "problematic, " and be critical of their ability to function as caregivers, making it possible for them to experience courtesy stigma in environments where support is expected (28).Children with ASD who are picky eaters can suffer from nutritional deficiencies, and

Relationship of primary caregiver-related factors to courtesy stigma
Children with ASD can impact the healthy family structure and function, particularly the relationship between husband and wife (37).In this study, we found that marital status satisfaction was associated with courtesy stigma among primary caregivers of children with ASD, consistent with a previous report of an adverse association between maternal stigma and marital satisfaction (26).Studies in other populations have also shown that reduced marital satisfaction is associated with stronger self-reported stigma (57).Marital status satisfaction is one of the key factors in determining whether the family will be susceptible to disorganization when the whole family undertakes the task of child care (26).Higher marital status satisfaction may make primary caregivers more comfortable with difficulties associated with the presence of children with ASD, including stigma.Primary caregivers who were satisfied with their current marital status were more likely to have good communication, helpful interaction, emotional support, and accurate and useful information from their partners, which reduced their courtesy stigma to some extent; however, primary caregivers who were less satisfied with their current marital status were likely to experience more verbal aggression, intimidation, non-verbal anger, and disruptive interactions from their partners.Therefore, family-based interventions that promote effective communication, problem solving, and mutual support between couples are of some importance in reducing stigma.Raising a child with ASD requires a considerable amount of care, and many primary caregivers are forced to quit their jobs to care for their ASD child (42).This study demonstrated that primary caregivers who reported a higher level of challenge in caring for children with ASD were more likely to experience courtesy stigma.The high-level challenges experienced by primary caregivers caring for children with ASD may involve dealing with specific behaviors, such as screaming, aggression, and tantrums, which could lead to isolation and exclusion of primary caregivers, as well as difficulties in interacting with others in public places, thereby increasing courtesy stigma.A previous study demonstrated that parents experienced more stigma when their children exhibited higher level ASD-related behaviors (15).Therefore, clinical professionals should train primary caregivers of children with ASD in behavioral management and coping skills, to reduce the challenges of caring for children with ASD, thereby reducing courtesy stigma.

Relationship of general self-efficacy and social support with courtesy stigma
In this study, we found that primary caregivers of children with ASD who had low general self-efficacy had higher courtesy stigma scores, similar to previous findings of a significant negative association between self-esteem and affiliate stigma (37); however, self-efficacy is a dynamic and modifiable characteristic.Therefore, measures should be implemented to improve the general self-efficacy of primary caregivers of children with ASD.It is necessary to help the main caregivers of children with ASD to establish self-understanding and evaluation, so that they realize that their own value, which could reduce their courtesy stigma.More support and counseling services for parents of children with ASD could reduce their stigma (6).Our findings show that social support was negatively correlated with courtesy stigma among primary caregivers of children with ASD.This finding is similar to previous literature showing that social support, particularly from family and friends, is associated with the mental health of mothers of children with ASD (40, 58).Social support can directly affect an individual's well-being and can alleviate distress; therefore, it is important to implement specific interventions to increase social support, to reduce courtesy stigma among primary caregivers of children with ASD.In addition, it is necessary to educate the public about ASD and improve their understanding of ASD (6).

Limitations
This study has important theoretical and practical implications, but also some limitations.First, the cross-sectional design of this study limits the inference of causality between variables, and future longitudinal studies are needed.Second, in this study, we recruited only primary caregivers of children with ASD who were undergoing rehabilitation in a rehabilitation facility, where primary caregivers would receive professional and peer support, which may have resulted in underestimation of courtesy stigma.Future studies are needed to expand the population to include primary caregivers of children with ASD outside rehabilitation facilities.Third, this study was restricted to Lianyungang, Jiangsu Province, Eastern China; hence, the results may only be representative of areas with similar backgrounds, and future studies in other regions are needed.Fourth, some variables in this study were measured by one item, and in the future multiple items or other assessment methods are needed.Finally, only quantitative analysis was conducted in this study, and we did not include qualitative analysis to provide a more comprehensive assessment of courtesy stigma.Future qualitative research is warranted.

Conclusion
There is a high level of courtesy stigma among primary caregivers of children with ASD in eastern China.Children who are picky eaters, marital satisfaction of primary caregivers, the level of challenge in caring for children, general self-efficacy, and social support are factors associated with courtesy stigma.There is an urgent need to establish effective interventions to reduce the courtesy stigma among primary caregivers of children with ASD.Actions taken to address the associated factors discussed above may help to reduce courtesy stigma and its adverse consequences among primary caregivers of children with ASD.

TABLE 2
Relationship between child-related factors and courtesy stigma.

TABLE 3
Relationship between primary caregiver-related factors and courtesy stigma.

TABLE 5
Multiple linear regression model to determine the factors associated with courtesy stigma.