1. Introduction
A caregiver is defined as a person who provides care to those who have difficulties in completing the tasks of daily living and thus need supervision or assistance due to some form of illness or disability [
1]. They may provide the care in an institution or an organization, which is called formal caregiving. The concept also refers to an informal caregiver who has a social relationship with the care recipient including family member, relative, friend, or neighbor. With the rapid growth of the global economy, institutionalized long-term care may be an option for children with disabilities (CWD), but the informal caregivers are preferable because of children’s high dependency and cognitive deficiency. Therefore, the caregivers of CWD generally refer to informal caregivers especially family caregivers including parents, grandparents, siblings, or other family members. In recent years, there has been a tremendous change in the health care system which exerts a shift toward family-centered services. This shift highlights the primary and important role of the family in children’s caring and thus increases the responsibilities of informal caregivers [
2]. In addition, their contribution on children’s home-based rehabilitation and maintenance of rehabilitation effect can be increasingly enormous [
3].
The World Health Organization has reported that around 15% of the world population (one billion people) live with some form of disability and 95 million are children (aged 0–14) [
4]. The total number of CWD presents the rising trend, which aggravates the informal caregiver’s childcare burden. Compared with the general population, previous studies have found that caregivers of CWD experienced high stress [
5], depression, anxiety, and low satisfaction with life [
6,
7], resulting in a detrimental effect on their health-related quality of life (HRQOL). In China, the investigated number of CWD in 2006 was 3.87 million, roughly 4.66% of the total population with disabilities [
8]. Additionally, caregivers, especially mothers, may feel guilt or remorse for having a child with disability and thus would do their utmost to care for the disabled child alone [
9]. These factors will further worsen HRQOL of this special population and therefore should urgently be concerned. However, the current corresponding attention is far from enough.
To our knowledge, there is a wealth of research on the HRQOL of caregivers of CWD abroad, and the relevant literature has mentioned many influencing factors [
10,
11], including the disability types and behavioral problems of children [
12,
13,
14,
15], caregivers’ employment status [
16], social participation [
17], parental stress [
5], and coping strategies [
18]. In recent years, domestic preliminary explorations have begun in Taiwan [
19,
20] and the underdeveloped central and western areas of the mainland, such as Anhui and Hunan [
9,
21], but neglecting developed areas, especially Shanghai. Comparing the results of existing studies, factors affecting the HRQOL of caregivers of CWD vary with the level of economic development, culture, social support, and other circumstances [
22,
23,
24]. The key and intervening factors also change, suggesting that the results of existing studies do not necessarily apply to caregivers of CWD in Shanghai.
Moreover, with the advances of rehabilitation technology and the support of rehabilitation-related policies, CWD have more rehabilitation and development possibilities, which shift the pressure for rehabilitation from external factors to internal factors for caregivers. This change not only increases the pressure on caregivers to enable their children to receive rehabilitation services, but also reduces opportunities for caregivers’ self-development. Both could lower caregivers’ HRQOL. Shanghai, as an international metropolis, is prosperous in economy and is at the forefront of rehabilitation services for CWD in China. Therefore, it is crucial to explore the HRQOL among caregivers of CWD. Furthermore, the findings could provide references for some developed countries and other similar areas in developing countries.
The International Classification of Functioning, Disability and Health (ICF), which was approved in May 2001 by the World Health Assembly, is a universal framework of functioning and health [
25]. It is organized in two parts, one part includes body functions and structures, activities and participation, the other part encompasses contextual factors [
26]. ICF emphasizes the dynamic interaction between health condition and personal, environmental factors, which means any factors of caregivers, children, and the environment may have impact on caregiver’s HRQOL. In October 2007, the International Classification of Functioning, Disability and Health—Child and Youth (ICF-CY) was published, and was designed specifically for children and youths [
27]. Both ICF and ICF-CY provide a scientific basis for identifying the potential determinants of the caregivers’ HRQOL of CWD comprehensively.
Therefore, the purpose of this study was to measure the HRQOL of caregivers of CWD in Shanghai and to explore the potential influencing factors associated with HRQOL among these caregivers under the guidance of ICF and ICF-CY. Ultimately, the findings could provide references for local health care policymakers and public health researchers to design appropriate health management strategies for the vulnerable population.
2. Materials and Methods
2.1. Participants and Procedure
We recruited the caregivers of CWD from one district of Shanghai. This district has a well-constructed rehabilitation service system and high-level government attention to CWD. Since the exact number of CWD is not yet available, we determined the sample size based on the number of children who received the Sunshine Baby Cards. The Sunshine Baby Card is the most important project of rehabilitation service system provided for CWD by the Shanghai Health Bureau, Shanghai Municipal Education Commission, Shanghai Finance Bureau, and the Shanghai Disabled Person’s Federation. It can be applied for any child who has been certified as disabled by a professional organization. According to the regulation, every child who holds a Sunshine Baby Card can receive rehabilitation training in designated rehabilitation institutions and enjoy the corresponding subsidies. Thus, the number of children with a Sunshine Baby Card approximates the actual number of CWD. In 2019, there were 900 children with the Sunshine Baby Card in the district, 15% of the total number of cardholders were selected for the survey. Then the sample size was expanded by 20% to take into account the dropout rate, which resulted in a sample size of 900 × 15% × (1 + 20%) = 162. Finally, 170 cases were included in the survey.
We collected data in both rehabilitation institutions and community-based settings, considering that children aged 0–6 years are more accessible in rehabilitation institutions, while children aged ≥7 years need to go to school. Since there are private rehabilitation institutions, public hospitals, and community health centers in Shanghai, we selected all types of rehabilitation institutions for investigation. For community-based settings, we covered all communities of this district.
Caregivers were included if (1) they were caring for a child diagnosed with disabilities by the responsible departments; (2) their children were aged 0–16 years; and (3) they were the primary caregivers. Caregivers who did not identify themselves as primary caregivers were excluded from the study. In each rehabilitation institution, the children’s basic information and rehabilitation schedule was first obtained from their health records to determine whether they met the criteria or not. Then, we created the investigation list. As for the communities, we first collected the basic information of CWD from the staff of the communities. If children met the sample requirements, their name would be fed back to the staff. The purpose and procedure of the research project were explained to caregivers. If they agreed to participate in the study, the investigators would conduct a one-to-one investigation. Ultimately, we recruited 97 caregivers from the rehabilitation institutions and 73 caregivers from the communities.
Ethics approval was obtained from the Ethics Committees of School of Public Health of Fudan University (Grant No. IRB#2019-10-0782).
2.2. Measures
2.2.1. Measure of Health-Related Quality of Life
HRQOL was measured using the 12-item Short Form Health Survey (SF-12), an abbreviated form of MOS 36-item Short Form Health Survey (SF-36). SF-12 was designed to take less time to complete and was widely used due to its high degree of acceptability and data quality. It contains 8 domains: Physical Functioning (PF), Role-Physical (RP), Bodily Pain (BP), General Health (GH), Energy/Fatigue (VT), Social Functioning (SF), Role-Emotional (RE), and Mental Health (MH) [
28]. Two summary scores are reported: Physical Component Summary (PCS) score and Mental Component Summary (MCS) score which can be used to measure physical HRQOL and mental HRQOL, respectively. For comparison, scores were calculated using the US norm-based scoring algorithm. The test–retest reliability (0.89) and reliability coefficients (0.76) showed the scale was reliable and valid [
28]. In 2005, a study conducted in Hong Kong using SF-12 showed that this instrument was valid and equivalent for the Chinese population as well as the items and scoring algorithm. Therefore, SF-12 can be applied to the Chinese population for further health-related research [
29].
2.2.2. Measure of Determinants of HRQOL
Child characteristics: Child characteristics were designed under the framework of ICF-CY. For body functions and structures, sleep time and emotional stability were taken into consideration. For activities and participation, we measured the frequency of physical activities and group games during the last four weeks. For personal factors, we collected age, gender, cause of disability, disability type, disability severity, and whether they had the Sunshine Baby Card or disability certificate.
According to the People with Disabilities Act of the Peoples Republic of China, disability is divided into visual disability, hearing disability, speech disability, physical disability, intellectual disability, mental disability, multiple disability, and other disabilities. Since our inclusion criteria did not strictly limit the type of disability, all types above were involved in our objects. Additionally, we specially listed cerebral palsy as one specific type of disability because of its high prevalence among children. Disability severity was graded with China’s legal criteria of disability which divided all kinds of disabilities into four levels, ranging from level I (extremely severe) to IV (mild severe).
Caregiver characteristics: Caregiver characteristics were collected under the guidance of ICF. The dimension of body function and structure was measured with the question of “whether you have the following disease or not (e.g., hypertension, diabetes, Parkinson’s disease)”. The feature of activity and participation was identified by caregivers’ employment status and caring time. Furthermore, we collected the personal factors including relationship with the child, gender, household register, marital status, and education. Previous studies mostly focused on the caregiving of parents or merely mothers, rarely involving other families. In this study, we took fathers, mothers, paternal grandfathers, paternal grandmothers, maternal grandfathers, and maternal grandmothers into consideration.
Environmental factors: Environmental factors included number of children, family size, household income, policy satisfaction, and social friendliness. Among these factors, policy satisfaction and social friendliness were reported by subjective perception of caregivers and were evaluated in the form of the Likert scale, ranging from 1 (not friendly at all or dissatisfaction) to 5 (extremely friendly or satisfaction).
2.3. Statistical Analysis
Descriptive statistical methods were applied in presenting basic characteristics for child, caregiver, and environment, and the measures (mean, standard deviation [SD], frequencies, and percentages) were respectively reported. Mean scores of the SF-12 of caregivers of CWD were compared with norm scores of the general population using the one-sample t-test. In order to preliminarily examine the associations between participants’ characteristics and HRQOL, univariate analyses including t-test and one-way analysis of variance (ANOVA) were conducted. To further investigate the potential predictors, two multiple linear regressions were performed with PCS and MCS scores as dependent variables. Any covariates that achieved p < 0.10 for the bivariate analyses were included in the multivariate linear regression models. We examined the multicollinearity among predictors in each model using Variance Inflation Factor (VIF). We removed the predictors which lead to multicollinearity. All statistical analyses were performed using SPSS Statistics version 20.0 (IBM Corp., Armonk, New York, NY, USA).