Positive aspects of caregiving and influential variables
This study shows that 84 % of the caregiving sample was represented by mothers and that few mothers admitted negative feelings in assisting their child, as expected taking into account Italian social customs.
The results of the study suggest that most key relatives consider that their caregiving experience has had a positive impact on their lives, despite the practical difficulties of caring for patients with MD.
In line with findings from other studies [
14,
16,
20,
23], 88 % of key relatives reported that they had gotten something positive out of the situation. In particular, 72 % of key relatives mentioned having changed their perception of the meaning of life’s values, while 18 % mentioned an increased sense of strength and courage against adversities.
These findings can be interpreted within the framework of Lazarus and Folkman’s [
35] transactional model, which postulates that an individual’s adaptation to an event is a process based on primary and secondary cognitive appraisal. In regards to MD, primary appraisal refers to the realization of what the illness is, while secondary appraisal implies the development of emotional and problem-oriented strategies to cope with the difficulties of caring for patients with MD. In this model, adaptation is significantly influenced by internal factors, such as the key relative’s attitude toward the patient, and external resources, such as the availability of social and professional support [
36‐
38].
Key relatives’ intentions to be engaged in caregiving may be due to several factors, such as: (a) the high social acceptance of MD, resulting in the valorization of the caregiver role; (b) the awareness of poor alternatives to family care, which become increasingly necessary while treating a long-term illness [
39]; (c) the availability of welfare benefits due to family assistance [
29]; (d) the innate love and sense of responsibility toward a child affected by a disabling illness with an unfavorable outcome [
40,
41]; and (e) the process of adapting to an illness in a loved one, which involves lowered use of emotion-focused coping strategies, such as avoidance, which proves ineffective for managing long-term stress [
35,
42,
43]. Among the 26 (5 %) caregivers stating that they had considered parting from their patients, 23 (92 %) were mothers and 16 (61 %) were employed, and they were relatives of patients with average duration of illness of 10.1 (6.9 SD) years. Furthermore, they were relatives of patients with higher levels of dependency (69.1 ± 31.0 versus 54.4 ± 34.1,
F = 5.4, df 1,500;
p < 0.02), and they received lower psychological support from their friends (2.7 ± 0.6 versus 2.3 ± 0.3,
F = 6.5, df 1,500;
p < 0.01) and/or partners (3.1 ± 0.7 versus 2.7 ± 0.7,
F = 5.5, df 1,454;
p < 0.02), compared with relatives who did not report these feelings.
Key relatives who reported a higher objective burden were more convinced that the situation had positive consequences for their lives. This finding shows that, when relatives feel they can manage the practical difficulties of caring for patients with MD, their tolerance threshold for stress is not exceeded, thus they can consider positive aspects of caregiving despite the burden [
16,
19]. Key relatives’ long-term adaptation to caring for patients with MD was also confirmed by statistics showing that, the longer key relatives have been involved in caregiving, the higher the positive evaluation they give to the experience.
Though this study does not examine caregiving for later, more severe stages of MD, it is likely that in such cases, when tasks become too demanding for key relatives, their perceptions of positive aspects may decline, while the psychological consequences of caregiving may increase [
9,
39].
Our study further finds that key relatives who had more positive attitudes toward their patients were more likely to identify the positive aspects of the situation. This finding suggests the importance of helping key relatives to identify “the person beyond the illness” as a strategy to valorize their lived experience and to support their patient’s adaptation to the illness [
31].
In this study, intrafamily factors were shown to be very significant and related to key relatives’ acknowledgement of positive aspects of caregiving. In particular, key relatives who felt supported by their spouses or partners were more likely to identify positive aspects of caregiving. This finding is in line with data from previous studies [
7,
26,
41] in which the occurrence of a severe disease was found to strengthen the bond between the parents of child patients. Strengthened partnership bonds generally appear in our study, in which more than 70 % of key relatives considered themselves totally understood and helped by their partner.
Psychological support from social networks and professionals were both related to key relatives’ perceptions of caregiving as a positive experience. These results support research evidence that suggest that social networks are critical factors in reducing the detrimental effects of stress and serve as a buffer between coping with an event and stress [
26,
34,
44]. This finding was also supported by the inverse correlation found between key relatives’ sense of family burden and sense of support from the social network [
32]. Furthermore, though investigation of the impact of social resources on caregiver’s negative feelings was not among the aims of this study, it is likely that relatives with low social support and those unemployed are potentially at risk to perceive anger and injustice.
Methodological considerations
To our knowledge, this is the first study of the condition of families of young patients with MD (1) to be performed on a large, national sample of key relatives of patients with different types of severe MD; (2) to use well-validated assessment tools already available in several languages; (3) to examine the positive aspects of caregiving in relation to burden of care, as well as key relatives’ personal factors, or attitudes toward the patient, and social and professional resources; and (4) to examine the positive experience of caregiving by means of quantitative measures and key relatives’ subjective descriptions.
Due to its cross-sectional design, this study does not allow inferences regarding the evolution of positive aspects over time or whether external resources and attitudes toward the patient influence the key relative’s perception of positive aspects or vice versa.
The survey focused on the experience lived by relatives of patients with DMD, BMD or LGMD aged 4–25 years. Therefore, its findings cannot be generalized to families of older patients or patients suffering from other types of MD. These aspects will be specifically addressed in further studies, which are now in the planning stage.
Practical implications
The results of this study may be useful for clinicians engaged with families of persons with MD to better understand the complexity of the caregiving process and to learn how to support caregiving families. Clinicians and other professionals should help caregivers to develop not just practical but also cognitive competencies to deal with providing care for MD patients and to identify the benefits of the caregiving situation without denying its difficulties. In particular, professionals should: (a) provide families with information on the patient’s disease, according to a step-by-step approach; (b) teach relatives to reinforce their problem-oriented coping strategies; and (c) invite the relatives to share their experiences with other families through involvement with family associations and self-help groups [
45].
Furthermore, clinicians themselves should develop positive attitudes towards family caregiving. This is not always the case, as reported by Green [
16], a mother of a patient with cerebral palsy and a researcher, who stated that “...parents who hold positive attitudes toward raising a child with disability are often pathologized as being unrealistic, failing to accept their “tragic” circumstances, or being “in denial” of their children problems. Pathologizing, and thus discouraging, parental ability to find benefits in having a child with disability is potentially very problematic for parents.... If parents of children with disability are repeatedly discouraged from finding and acknowledging the positive aspects of caregiving, they may be denied the potentially positive consequences of doing so.”
Future studies
Further investigations are needed to specifically explore the following aspects of caregiving in MD: (a) key relatives’ adaptation at different stages of the illness, (b) the experience of caregiving in other relatives of the same family, (c) the effects of relatives’ attitudes on patients’ perceptions of their own experiences with illness, (d) the effects of supportive interventions to improve relatives’ coping strategies, and (e) the influence of acceptance and awareness of the disease on the well-being of relatives and patients.
Forthcoming studies by our research group on a larger sample of older patients with MD will provide a model for the negative/positive feelings in relation to progression of the illness, and a profile of the psychological perception of the disease over time.