2.1 STUDY CHARACTERIZATION

This is a case-control study of a quantitative nature. The research was approved by the Ethics Research Committee of the local University and it is in compliance with principles of the Declaration of Helsinki. The Free and Informed Consent Form (TCLE) was applied to all volunteer participants of this study.

2.2 STUDY LOCATIONS

This study was carried out in four small cities in Brazil, located in the region of Piemonte da Diamantina, in the State of Bahia. The total population of the first city is 12,344 people, the second city has a population of 79,247 inhabitants. The third city has 10.376 inhabitants, and the fourth city has a population of 11,847 inhabitants (^{Instituto Brasileiro de Geografia e Estatística [IBGE], 2010}). The main economic source of this region is agriculture and trade of livestock. In agriculture, subsistence activities are predominant. In addition, mining is also one of the main sources of economic activity in the region (^{IBGE, 2010}).

The choice for such regions was due to the fact that it is a context still little explored in researches with outcomes such as those of this study. Moreover, the four cities have units of Specialized Educational Care (SEC) and health units in effective working, being a reference in this region, which facilitated the access of researchers to the target public. Due to the specific cultural and economic characteristics of the region, these municipalities add peculiarities that are not very explored in field studies. Specifically with populations of children and adolescents with disabilities and their caregivers, there are no epidemiological survey data that can describe sociodemographic factors as potential risk or protection factors in these subjects.

Data collection took place in the Specialized Educational Care Unit (SEC), in the regular educational network or directly in the medical/physiotherapeutic centers in health units of these municipalities between July 2016 and September 2016.

2.3 SAMPLING

The selection of cases in this study was based on the location of mothers or primary caregivers of children and adolescents with disabilities who attended the SEC rooms, or who attended health units to perform medication or rehabilitation follow-up, and all of which are residents in the municipalities described above. Control subjects were recruited by observing data such as kinship, age, gender and socioeconomic status that could have the greatest possible similarity in the matching of pairs. Children and adolescents with typical development should be properly enrolled in the regular education network of their municipalities.

The sample calculation was performed considering 95% confidence interval, an unknown prevalence, power of 80% and alpha of 5%. Thus, 100 caregivers, 50 cases and 50 controls would be needed as the minimum sample size of this study. The number of participants in this study was 112 pairs of caregiver-children with and without disabilities, 56 (cases) and 56 (controls), who fulfilled the study inclusion criteria.

2.4 INCLUSION/EXCLUSION CRITERIA

Primary caregivers of children and adolescents with disabilities attending the SEC Unit regular educational institution, or those at follow-up health units of their municipalities and residents in the respective municipalities participated in the study. The caregivers should be 18 years of age or older. Caregivers who were not able to understand the issues present in the data collection instruments were excluded from the study; however, caregivers were not excluded by this criterion.

2.5 VARIABLES AND INSTRUMENTS

In this study, the following variables were investigated: Screening of Common Mental Disorder (CMD) of caregivers and sociodemographic variables of pairs of caregiver - child/adolescent with disabilities.

The Self-Reporting Questionnaire (SRQ-20) was used to identify characteristic signs of CMD, which is a standardized questionnaire for the screening of non-psychotic mental disorders in adults (≥ 18 years). This questionnaire has 20 questions with dichotomic responses (YES or NO) about anxious, depressive, and somatoform symptoms in the last 30 days, and classifies the subject into positive CMD when there are ≥ 8 YES responses and negative CMD when there are < 8 YES responses.

A standardized sociodemographic questionnaire elaborated specifically for the study, based on the main risk and/or protection factors pointed out in epidemiological studies, was used to evaluate sociodemographic factors. This questionnaire contains 55 items that aimed to investigate information about income, schooling, health profile and physical activity.

2.6 PROCEDURES

Participants of the research were initially oriented on the aim of the project and its evaluation instruments. Subsequently, caregivers signed the informed consent form, formally making their participation in this investigation possible. Mothers or primary caregivers of typical children/adolescents and those with disabilities received the information about the day, time and place to respond to the scientific work instruments by means of a letter previously delivered by the researchers to the coordination of the Multifunctional Resource Rooms (MRR), in schools and in the basic health units of the municipalities, in case those included in the research were carrying out procedures in these places. Finally, Self-Reporting Questionnaire (SRQ-20) and the Sociodemographic Questionnaire were answered in the form of an interview, being performed individually with each caregiver.

It is important to emphasize that, due to the case-control design of this study, pairs of caregiver-child/adolescent with disabilities were recruited first and then pairs of caregiver-child/adolescent without disabilities were recruited for adequate matching. Thus, for each child with a disability another child without a disability of the same age and sex was recruited for the study.

2.7 STATISTICAL ANALYSIS

Subjects were characterized by means of absolute frequencies, means and standard deviations. The t-test was applied for independent measurements in order to compare the average age between cases and controls. Bivariate analysis was performed for caregiver’s characteristics between cases and controls, and child’s characteristics between cases and controls using the Chi-square test at significance level of 5% and, where possible, the Odds Ratio analysis to calculate the Odds Ratio (OR) with 95% confidence interval, considering the group factor as independent variable and sociodemographic issues as dependent variables of the study.

Variables with significance level ≤20.0 were included in the Logistic Regression Analysis of the study. The Binary Logistic Regression model was used to verify the effect of the remaining sociodemographic variables between cases and controls. In order to facilitate the understanding of variables remaining in the Logistic Regression model, the discussion was carried out by topics for each of the variables.

4.1 INFORMANT’S HEALTH PERCEPTION

In the social perception, sometimes the person with a disability is perceived as weak and fragile, sometimes as someone with a lot of courage and willpower. Even when socially, culturally and temporally contextualized, disability brings with it a strong emotional burden to people involved in the process - parents, children and other relatives (Sá & Rabinovich, 2006).

When analyzing the results of the caregiver’s health perception, it was evidenced that caregivers of children with disabilities perceive their health in a negative way. Behind this observation, there are some important implications for a better understanding of the reality exposed by informants. The unfavorable sociodemographic conditions of pairs of caregiver-children with disabilities when compared to pairs of caregiver-children without disabilities end up predisposing the main caregivers to have a negative view of their health.

This negative predisposition that caregivers feel about their health is intrinsically related to the lack of support that they receive in the task of caring. This is due to little involvement of family members, overburdening those who directly take care of the child with the disability, not only for having some disease, food restriction or use of controlled medications by the child/adolescent and/or the caregiver, but also because the family is often exposed to complicated financial conditions, which is certainly due to the lack of information to family members about rights guaranteed by law. Given this context, it should be pointed out that the use of the terminology ‘person with disability’ is full of precipitated meanings, since this subject is often loaded with prejudices, stigmas and stereotypes, in a way that negatively instigates relatives own perceptions about their health (^{Sassaki, 2005}).

^{Garip et al (2017)}, in a study on the fatigue of mothers of children with cerebral palsy, showed that care delivery is a natural role for mothers. However, the care for a child with functional limitations and long-term dependence is totally different and negatively influences mothers or primary caregivers in their quality of life in terms of physical and emotional functioning. In this perspective, when fatigue is constant with a continuous overload in the task of caring, the consequence to the caregiver is to visualize or perceive his or her health in a negative way.

Brehaut et al. (2011) reported that caregivers of children with complex medical conditions were more likely to report poorer physical health than caregivers of healthy children. Parents reported feeling emotionally frustrated, anxious, angry, helpless, and hopeless. Socially, they felt isolated, as people did not make them understand their family situation, and that it was difficult to talk to other people, including doctors and nurses about the child.

These results are consistent with those of other studies, such as that of ^{Hatzmann, Heymans, Ferrer-i-Carbonell, Van Praag and Grootenhuis (2008)}, in which parents of children with chronic diseases reported significant problems with daily living, vitality, sleep, difficulties in making family decisions compared to parents with healthy children of school age, evidencing the disparity in relation to quality of life and vision about their physical and mental health.

In addition to an unfavorable sociodemographic picture, the fact that the caregiver is frequently witnessing the conditions of suffering of whatever the deficiencies of their children, may lead to a process of physical and mental exhaustion, and these individuals internalize a thought and self-perception of complete impotence in relation to the reality that they experience. In many cases, the dedication is so intense that these caregivers abdicate their social life, family, school, leisure programs, church etc., thus restricting their lives exclusively to care, which, by the way, is usually a painful and lonely task.

^{Keller and Honig (2004)} reported that parents play an important role in the task of caring for children with special needs, and this requires increased attention. However, in many cases, they fail to notice their own existence, possibilities and potential. Some authors (^{Vieira, Mendes, Frota, & Frota, 2008}; ^{Castro & Piccinini, 2002}) have observed that social isolation is a frequent occurrence in the lives of caregivers of people with disabilities. Perhaps, this is also a preponderant factor for the caregivers of children with disabilities and so they perceive their health negatively.

Even for these caregivers of children or adolescents with disabilities who dedicate their time to care, believing that they are strong enough to overcome the difficulties faced in their daily lives, there is an arduous pilgrimage of family members potentially exposed to constant fatigue and stress, who cannot perceive their health in a positive way and end up incorporating for themselves the reality lived by the individual with disability. In this perspective, it is not uncommon to find high levels of tension in caregivers of children with special needs that result in depression, fatigue, lack of physical health and social isolation (^{Carnevale, Alexander, Davis, Rennick, & Troini, 2006}; ^{Cousino & Hazen, 2013}).

The lack of social support, as a possible agent that can prevent the onset of diseases, and social isolation, can also reinforce the negative health perception of the main caregivers of children or adolescents with disabilities, given the great dependence on family members, friends, school, and the health system for the treatment or follow-up medication of children with disabilities. ^{Ribeiro (1999)} describe social support as a set of functions performed by people who may be members of the nuclear family and friends who offer instrumental, informational or emotional assistance.

For these and other reasons, ^{Lima (2006)} described some difficulties faced by mothers of people with disabilities: intense emotional overload of mothers due to their own difficulties, discrimination, exclusion and social reaction to the person with disability and often to the lack of or little support received; exhaustion and abandonment of other family members, difficulties in moving their children with disabilities and other issues. This could explain the more negative health perceptions of caregivers of children and adolescents with disabilities in this study.

4.2 CHILD’S FOOD RESTRICTION

The child/adolescent food restriction was the risk factor associated to the presence of disability among the investigated subjects. Probably the major risk of food restriction in child/adolescent with disabilities is due to the diseases that they have. This finding may confirm to the theory that children with disabilities present, in addition to their deficiency, other health complications that consequently restrict their feeding.

That association indicates a worrisome situation in the life of subjects with disability as well as in the routine of their caregivers. It is necessary to take into account that children with disability are more susceptible to the diseases that might cause any food restriction. This generates an extra demand for family members at the time of purchase, preparation and provision of adequate food for children/adolescents with disabilities. The financial hardship experienced for many families can be a major obstacle in this process, given the high costs of maintaining a varied diet. Consequently, these children/adolescents tend to be more exposed to the appearance of other diseases acquired by malnutrition and by the inherent immunological fragility in relation to the condition of disability.

It should also be noted that the low socioeconomic status of families makes access and maintenance of a healthy diet difficult. ^{Rose-Jacobs et al (2016)} reported that, in 2014, 3.4 million (19.9%) of North American families with children under six years of age presented family food insecurity, and 1.6 million (9.1%) reported that children have food insecurity. Food insecurity refers to the inability to pay enough to have quality food for an active and healthy life for all members. Thus, it could be concluded that food restriction of children with disabilities does not come from financial difficulties, but it can negatively impact something that comes from the condition imposed by the disability itself.

Since disabilities involve specific conditions in relation to health care, food restriction has a major impact on their lives and also on the lives of family members, especially caregivers, who need to control the feeding of these children/adolescents. Food restrictions require controlling the labeling of foods and/or beverages, often necessitating the manipulation of certain products to meet the quantity and quality of nutrients needed for these children and adolescents. Often, products that are free of substances such as gluten, lactose or sugar are more expensive, and as such their access is much more restricted, such as those living in counties far away from large urban centers as are the cities eligible for this study.

In this sense, caring for children with special needs in the long term may entail direct and indirect financial consequences, since all the family resources may be managed in favor of the care of the child with disability. This significantly affects low-income families, with direct consequences to the subjects’ health. Yet, it also prevents the ability of the family to meet the needs of having quality basic food items in their household (^{Rose-Jacobs et al, 2016}).

Costs associated with access to complex health and/or educational services can challenge the financial capacity of low-income families to meet basic needs. In addition, caregivers in their routine still live with the possibility of observing the dietary restrictions of children and adolescents with disabilities and receive guidance through a differentiated food schedule, but not being able to maintain this food schedule. Thus, these caregivers often need to change working hours, work more flexible hours (even with a lower salary), or give up work to devote exclusively to maintaining the medical, educational and food needs of the child/adolescent with disability.

4.3 USE OF CONTROLLED MEDICATION

This study found that most case children use controlled medications, whose data refer to the very fragile condition of the child with disabilities, since they need continuous and long-term specialized care to maintain their state of health. The drug therapy of these children is frequent. ^{Elias, Murphy and Council on children with disabilities (2012)} point out that children with special needs usually receive five times more medication than children in general. In view of this finding, the use of controlled drugs would lead to more diseases, greater search for health services and greater exposure to intoxication. In addition, the susceptibility of pediatric patients to adverse drug reactions may change during the various stages of their development and growth, especially in cases of chronic treatments (^{Caicedo, 2015}).

^{Santos, Barreto and Coelho (2009)} point out that continued use of medications, besides being an important indicator of health problems, also reflects social inequalities, deficiencies, among other factors. In this context, ^{Silva and Tuleski’s (2015)} study indicates that complaints related to mental suffering at different levels of severity have been increasingly perceived, and this reality being treated in a biological and pharmacological way. Mental disorders are responsible for five of the ten major causes of disability in the labor market, manifested through depression, bipolar disorder, schizophrenia, alcoholism and obsessive-compulsive disorder, diseases that cause drug dependence to reduce the effects of these risk factors.

Another important aspect, in addition to the intake of drugs by children with disabilities, is the regularity of medication administration, which is a challenge that increases maternal overload, since the mother usually becomes the main person responsible for the care and management of the therapy. Due to predominantly cultural and certainly sexist reasons, the role of the woman caregiver is still an attribution expected by society in the face of the problem of maternal care and even so more when the child has a disability (^{Karsch, 2003}).

^{Okido, Cunha, Neves, Dupas, & Lima (2016)} point out that these mothers present emotional, social and physical problems, such as difficulty sleeping at night, because they need to wake up to check the child’s condition or to administer medication. Thus, it is important to strengthen the link with the health system’s social network with the aim of promoting the effective and integral follow-up of these mothers who are vulnerable to psychic diseases. This denotes how caregivers and the one being cared for are immersed in the use of drugs to solve problems in the physical and/or mental health of both. Although this study found no association between positive CMD among caregivers and the children/adolescent deficiency condition, it is worth mentioning that the prevalence of CMD was higher among these caregivers (58.6%), when compared to caregivers of children/adolescents without disability (41.4%). These results should not be disregarded in the context of sociodemographic risk factors presented by pairs of caregiver-children/adolescents with disabilities.

It is worth mentioning that the low socioeconomic status hinders access to specialized medicines and services. According to ^{Gaiva, Neves and Siqueira (2009)}, the financial difficulty is present in the lives of the study sample, mainly because the majority of them have low socioeconomic status and part of the family income is used for curative activities and rehabilitation of children.

4.4 CHILD RECEIVING GOVERNMENT BENEFIT

The fact that most children with disabilities receive a Continuous Care Benefit (CCB), provided for by specific Brazilian legislation (Lei nº 8.742, 1993) allows us to identify an improvement in the welfare conditions of people with disabilities after receiving the CCB, in addition to the increase and security of family income. According to the Organic Law of Social Assistance (called LOAS) (Lei nº 8.742, 1993), people with disabilities who carry out follow-ups with rehabilitation services promoted by specialized institutions have the right to this benefit, which is equivalent to the value of the national minimum wage, and must be used for the cost of treatment, for items such as food, transportation and medications.

According to ^{Santos (2011)}, CCB is one of the most important public policies of combating inequalities in the country. CCB is an unconditional income transfer equivalent to a minimum wage, for poor people with disabilities and also for older adults over 65 years of age, which is a benefit of social assistance guaranteed in the Federal Constitution of 1988, regulated by Law No. 8,742, of December 7, 1993 Organic Law of Social Assistance (LOAS).

Across the country, children and adolescents are the age group with the highest incidence of people with disabilities receiving CCB: one in five CCBs granted in the country is for children and adolescents up to eighteen years of age. Thus, CCB was created as a social policy aimed at addressing the citizenship rights of poor people with disabilities who need social assistance (^{Santos, 2011}).

In addition to the fact that the benefit is mainly focused on providing minimum survival needs, the importance of the benefit as a social protection is also revealed when the role of CCB in the composition of the family income of persons with disabilities is established.

Significant changes in the notions of well-being have been observed because of access to the benefit, since the CCB target public is composed of very poor and very socially vulnerable people, and allows the monthly guarantee of a minimum wage to change the standards of consumption of families of people with disabilities even if this consumption is of basic goods such as food, health care and household expenses (^{Santos, 2011}).

Contrary to this, the lack of knowledge about these rights characterizes the non-exercise of the citizenship of these children and, in this case, it is the moral duty and obligation of health professionals to provide the necessary information. We point out that in many families, the total income they have comes from this resource which, in turn, should be to care for the person with disability. In fact, this benefit turns out to be the only financial source of family members who do not have the time to dedicate themselves to another work activity because they are involved in the care of child/adolescent with disability.

Another issue to be mentioned is the eligibility criteria for access to CCB, which relates to incapacity for work. This criterion is one of the great controversies for the granting of the benefit, since determining incapacity for work is not a simple task, and ends up restricting people with disabilities access to this social right. In addition, certain disabilities are not totally disabling for work; however, the experience of disability and the restriction of skills resulting from it disadvantages these people in the labor market because it is not well adapted to the characteristics of people with disabilities, preventing the promotion of autonomy and financial and social independence.