The experience of persons with multiple sclerosis (MS) and their caregivers is usually investigated in terms of emotional distress and health-related quality of life, while well-being indicators remain largely underexplored. In addition, findings are often interpreted from the clinical perspective, neglecting socio-cultural aspects that may crucially contribute to individuals' functioning. At the methodological level, most studies rely on scaled instruments, not allowing participants to freely express their needs and resources. Based on the bio-psycho-social perspective endorsed by the International Classification of Functioning, well-being indicators were investigated among 62 persons with MS (PwMS), their 62 caregivers and two control groups, matched by age and gender. Participants completed the Positive Affect Negative Affect Schedule (PANAS), the Satisfaction with Life Scale (SWLS), and the Eudaimonic and Hedonic Happiness Investigation instrument (EHHI). EHHI provides information on participants' happiness, goals and meanings through scaled and open-ended questions, contextualized within major life domains. No relevant differences emerged among PwMS and caregivers, compared with the respective control groups, as concerns life domains associated with happiness, goals and meaning. Participants across groups prominently mentioned family, highlighting its intrinsic value and its relevance as a sharing context; health did not represent a major theme for PwMS; community, society and religion/spirituality issues were substantially neglected by all participants. PwMS and caregivers reported lower levels of positive affect than their control groups, while no substantial differences emerged for negative affect, happiness and meaningfulness levels in life and across most domains. Results suggest that the experience of MS is associated with well-being in relevant life domains, such as family and close relationships. Although PwMS and caregivers identified a lower number of goals and meaning-related opportunities compared to control groups, they showed a positive adjustment to disease through the development of personal and family resources. These assets are often undervalued by health professionals and social institutions, while they could be fruitfully exploited through the active involvement of PwMS and their families as expert and exemplary informants in initiatives aimed at promoting the well-being of individuals and communities

Keywords: multiple sclerosis (MS), well-being, caregiving, daily living, meaning-making, goals, mixed method, psychosocial interventions

#### INTRODUCTION

The technological advancements in the medical domain fostered the representation and interpretation of health-related phenomena through rigorously quantitative approaches, leading to the affirmation of the biomedical model. Despite the shortcomings entailed by this narrow perspective, and the classical definition of health as "a state of complete physical, mental and social well-being" (World Health Organization, 1946, p. 1), both research and clinical practice are still permeated by a strictly biological view of human functioning.

A laudable effort to overcome this limitation and to promote the adoption of a bio-psycho-social view of health and disease is represented by the International Classification of Functioning, Disability and Health (ICF; World Health Organization, 2001). ICF is aimed at comprehensively assessing the physical, mental and social functioning of any individual, including persons with diseases and disabilities. It presents a marked shift in terminology, from focusing on the consequences of disease (impairments, disabilities, handicaps) to investigating the components of health and functioning, classified as structures and functions, daily activities, and social participation. Far from endorsing a pure linguistic convention, the ICF stems from a relevant conceptual change, leading to the evaluation of health conditions from a constructive perspective. It is based on the assumption of a dynamic interplay between individual features (body functions, activities, and participation) and environmental aspects that can facilitate or hinder the person's functioning (Dixon et al., 2008; Bodde and Seo, 2009). However, the implementation of this model into practice is problematic, as it requires a multidisciplinary integration effort involving researchers and practitioners from the healthcare, education and policy domains (Stucki et al., 2017). In addition, the assessment of psychosocial functioning through the current version of ICF poses several challenges. Psychological and environmental dimensions are collapsed into the heterogeneous domain of "contextual factors"; the psychological aspects are not explicitly identified through a checklist; and the components of community participation are difficult to evaluate (Chang et al., 2013). Despite these limitations, only the ICF currently offers an integrated approach to human functioning which can be used by professionals working in different sectors.

From the ICF perspective, it is not surprising that people with chronic diseases or disabilities describe themselves as ordinary persons who cope with extraordinary circumstances (Saravanan et al., 2001) that include biological impairments as well as material, social and institutional barriers. Despite these disadvantages, the mobilization of personal, relational and environmental resources allows these people to attain high levels of mental health (Arnold et al., 2005; Andrykowski et al., 2008; Delle Fave et al., 2015). Similarly, caregivers experience the coexistence of caring burden and limitations in daily opportunities with psychological and social resources (Song and Singer, 2006; Olsson et al., 2008; Fianco et al., 2015).

The study of mental health and well-being has received great impulse during the last two decades, through theories and empirical models deriving from two complementary perspectives. Within the hedonic perspective, well-being is operationalized as the predominance of positive over negative affect in daily experience and a globally positive life evaluation, defined as satisfaction with life (Ryan and Deci, 2001). From the eudaimonic perspective well-being is instead conceptualized as a dynamic growth process, that includes a wide range of constructs such as goal setting and pursuit, meaningmaking, self-expressiveness, self-determination, self-acceptance, skill development and mastery, trust in relationships, and social integration (Huta and Waterman, 2014). Both these approaches proved to be useful in exploring protective resources, adaptation processes and adjustment outcomes that allow persons with chronic diseases and their caregivers to attain positive functioning (Cummins, 2005; Diener and Chan, 2011).

This avenue of research is however characterized by an emphasis on individual processes, while the societal and cultural factors influencing the person's daily functioning are substantially neglected (Di Martino et al., 2017). Demographic and contextual features, when investigated, are treated as components of the person's stable identity, despite the changes that both the environment and the individual ceaselessly undergo (Slife and Richardson, 2008).

#### The Individual and Family Experience of Multiple Sclerosis: Challenges and Resources

Multiple sclerosis (MS) is a chronic neurodegenerative disease, characterized by largely unpredictable symptoms and course, and currently lacking of curative treatment. Therefore, besides physical impairments, MS poses a number of psychological, behavioral and social challenges to both affected persons and their caregivers (Dennison et al., 2009; Ackroyd et al., 2011). The progressive course of the disease requires a constant readjustment over time (Bogosian et al., 2017), leading the person to gradually scale back or adapt daily activities to the new condition, find new occupations and interests, but also disengage from the adjustment process and withdraw from active life.

While the negative consequences of MS were extensively explored, research on the positive aspects is still limited. Studies have been conducted to investigate the eudaimonic process of meaning making, a key individual resource to attain wellbeing under unfavorable and irreversible conditions (Hicks and King, 2009). Meaning making allows individuals to integrate the problematic condition into a global and constructive view of their present and future life. The qualitative exploration of meaning making among persons with MS shed light on its components, which include disease acceptance, personal and relationship growth, and positive lifestyle changes (Pakenham, 2008a). Quantitative studies highlighted the positive association of sense making with life satisfaction, and its negative association with depression. Sense making was identified as a key predictor of positive adjustment also among caregivers (Pakenham, 2008b). Similar findings were detected as concerns perceived illness coherence, a construct closely related to meaning making; illness coherence was positively associated with eudaimonic and hedonic well-being indicators in both persons with MS and their caregivers (Bassi et al., 2016).

Benefit finding, consisting in the ability to identify positive consequences in an otherwise negative situation (Lechner et al., 2009), was also investigated among persons with MS and their family caregivers. Cross-sectional and longitudinal studies highlighted its positive association with meaning-based coping strategies, and its direct effects on positive adjustment outcomes, such as good dyadic relationships and subjective well-being (Pakenham, 2005, 2007, 2008b; Pakenham and Cox, 2009). Related findings support the idea of a "communal search for meaning" where persons with MS and their caregivers experience the trauma of a chronic illness and subsequently find positive aspects together (Pakenham, 2005). Further evidence of this dyadic process emerged from the investigation of post-traumatic growth and illness perceptions among persons with MS and their partners (Ackroyd et al., 2011).

These studies also showed that the disruption of life goals caused by MS progression can be counterbalanced by the development of new meanings and life purposes (Joseph and Linley, 2006). This process entails a complex interplay between disengagement from previous goals and reengagement in new ones (Neter et al., 2009). It also requires the implementation of adaptive tasks (Bensing et al., 2002), such as defining new challenges, acknowledging one's limits, maintaining emotional balance and self-esteem, facing uncertain future, cultivating social relationships, and looking at the bright side of life. Besides these individual resources, other factors located at the intersection between persons and their context may foster positive adaptation; they include facilitators of mobility/independence, social support, and social comparisons (Dilorenzo et al., 2008).

A frequent problem in studies investigating well-being and perceived quality of life among persons with MS is the lack of objective measures of disease severity; this limitation prevents researchers to exclude that positive adjustment may simply reflect a condition of less severe illness (Dennison et al., 2009). The assessment of demographic and clinical dimensions however entails methodological and interpretive challenges, as showed by the contrasting findings obtained in different studies. For example, in some studies demographic variables such as education and employment emerged as the strongest predictors of health-related quality of life (HR-QoL), together with clinical ones like depression and disability (Patti et al., 2003, 2007b). In other studies, HR-QoL itself emerged as an independent predictor of disability progression (Benito-Léon et al., 2013). Satisfaction with life was primarily predicted by disease severity and social support in one study (Ryan et al., 2007), while in another study age and education level emerged as the strongest predictors of hedonic and eudaimonic well-being (Bassi et al., 2014). Finally, a comparative study did not highlight significant differences in personal growth (a eudaimonic dimension) between persons with MS and healthy participants, while life satisfaction (a hedonic dimension) was significantly lower among the former (Barak and Achiron, 2011).

The investigation of well-being among caregivers of persons with MS is even less frequent, as studies are primarily focused on the emotional impact of caring-related burden and stress (Patti et al., 2007a; Rivera-Navarro et al., 2009), and on the daily choices and free time restrictions imposed by the caring role (Becker, 2011). Studies investigating satisfaction with life did not detect differences between caregivers of persons with MS and samples derived from the general population or carers of persons with other chronic diseases (Waldron-Perrine et al., 2009; Bassi et al., 2014). Eudaimonic well-being dimensions such as meaning making, benefit finding and illness coherence were more often investigated among caregivers, as reported in the previous paragraphs (Pakenham, 2007; Pakenham and Cox, 2009; Bassi et al., 2016). In a recent qualitative study involving Turkish caregivers (Topcu et al., 2016), participants mentioned both individual and social resources, such as motivation to care and perceived support.

Acknowledging the existence and adaptive role of wellbeing dimensions in the experience of MS surely represents an advancement. At the same time, most studies suffer from two conceptual limitations. The first one is the lack of a comprehensive bio-psycho-social perspective, and especially the neglect of the societal context surrounding persons with MS and their caregivers. Only few studies were conducted within the ICF framework. One of them was aimed at identifying a core set of ICF categories specific to MS that could be used in healthcare practice (Khan and Pallant, 2007). Another study proposed the Multiple Sclerosis Impact Profile (MSIP; Wynia et al., 2008) an instrument designed to evaluate the perceived impact of MS on functioning. A recent review (Dorstyn et al., 2017) highlighted the association between depressive symptoms and reduced social participation in persons with MS. A qualitative study exploring the impact of ICF contextual factors on the daily functioning of Jordanian persons with MS (Hamed et al., 2012) identified adequacy of financial and medical resources, religion and community awareness as facilitators, and social stigma as a barrier. Finally, the environmental and personal facilitators of social participation and satisfaction with

parenting were investigated among mothers with MS (Farber et al., 2015). Findings suggested the importance of incorporating both categories of facilitators into treatment. The relevance of environmental factors also emerged from the investigation of unmet needs among persons with MS. These needs most often refer to contextual dimensions, such as family and social support, healthcare services, everyday life management, and the relationship with physicians (Galushko et al., 2014). Interestingly, representations of the unmet needs of persons with MS differ among health professionals (Golla et al., 2012): while physicians emphasize limitations in access to care due to poor financial resources, nurses and social workers are more aligned with patients' perceptions, quoting family support, social relationships and daily life management.

A second limitation characterizing the literature on MS is the emphasis on disease and related adjustment processes: participants and caregivers are rarely solicited to freely reflect on aspects of their life and daily experience potentially unrelated to disease or caregiving respectively. This limitation emerged in one of the studies investigating unmet needs of persons with MS (Galushko et al., 2014): participants expressed the need to be viewed as distinct individuals, not constrained by and identified through their health conditions.

#### The Present Study

The first aim of the present study was to investigate different dimensions of well-being among persons with MS and their caregivers through a mixed method approach and from a bio-psycho-social, ICF informed perspective. Answers obtained through open-ended and scaled questions were jointly analyzed, in the attempt to contextualize findings within participants' global outlook of their own life and environmental opportunities. The well-being dimensions investigated in the study included happiness (its definition and recent related situations), hedonic well-being (positive and negative affect, satisfaction with life) and eudaimonic well-being (perceived meaningful things and goals). In addition, information was collected on happiness and meaningfulness levels in life in general, as well as in the specific domains of work, family, standard of living, interpersonal relations, health, personal growth, leisure, spirituality/religion, community, and society. Overall, the mixed method potentials are still underexploited in the psychological literature; moreover, to the best of our knowledge no studies have yet been conducted on these topics and through this methodological approach in the MS domain.

The second aim was to compare the findings collected among persons with MS and their caregivers with those obtained from two groups of participants, matched by age and gender, but with no history of chronic disease or caregiving experience. From an ICF informed perspective, this comparison was expected to shed light on group differences in perceived personal and contextual facets of well-being, including daily activities and opportunities, relational networks, and social participation. Attention was also paid to the role of employment status and education level in predicting well-being dimensions. These two demographic features represent crucial indicators of participation, classified in the ICF as contextual personal factors influencing human functioning (World Health Organization, 2002; Martins, 2015). The level of disability was also taken into consideration in the analysis of data collected among persons with MS.

Based on the available literature, some hypotheses were formulated. In line with studies on adjustment to MS (Pakenham, 2008a; Bassi et al., 2016) we expected that, when describing happiness, goals and meaningful things, a significantly higher percentage of persons with MS would refer to health compared with a control group. We also expected persons with MS to associate health with significantly lower levels of happiness and higher levels of meaningfulness, compared with a control group. As concerns caregivers, based on previous evidence (Pakenham, 2008b; Becker, 2011; Mausbach et al., 2011) we expected that a significantly higher percentage of participants would refer to family and a lower percentage to leisure, compared with a control group. We also expected caregivers to report significantly lower levels of happiness with leisure, and higher levels of meaningfulness in relation to family. As concerns ratings of affect and life satisfaction (the two components of hedonic wellbeing), the contrasting findings detected across studies (Barak and Achiron, 2011; Diener et al., 2017) did not allow us to formulate specific hypotheses. Finally, based on the literature highlighting the relevant role of education and employment in predicting individual well-being (Keyes, 2007; Patti et al., 2007b; Diener et al., 2017), we hypothesized that these two demographic features would provide a specific contribution to hedonic and eudaimonic well-being values across all groups. The same hypothesis was formulated as concerns the role of severity disease among persons with MS.

### MATERIALS AND METHOD

#### Participants

The study involved 248 Italian adults divided into four groups, each of them comprising 62 participants: persons with MS (PwMS), their caregivers, and two control groups of adults with no history of chronic disease or caregiving experience, selected from a larger study aimed at investigating well-being in the general population. Inclusion criteria for PwMS were being at least 18 years of age, having a clinically definite MS diagnosis for at least 3 years and having a caregiver; exclusion criteria comprised the presence of additional neurological or psychiatric disorders, severe cognitive impairment, MS in the active phase, and a condition of very severe disability, corresponding to a score above 8 on the Extended Disability Status Scale (EDSS; Kurtzke, 1983). The majority of PwMS involved in the study (59.68%) showed an EDSS score between 3.5 and 8 (indicating increasing levels of motor impairment), while 40.32% scored below 3.5 (indicating a high level of autonomy). No significant differences were detected between PwMS with low and high levels of disability for any of the variables examined in this study; therefore disability level will not be further considered in the following sections.

Caregivers were predominantly partners (59.67%) or parents (22.57%) of PwMS, siblings or children (each accounting for 6.46%), friends (3.23%), or professional caregivers (1 participant, 1.61%). Participants in the control groups were randomly extracted from a sample of 691 healthy Italian adults; age, gender and education level were used as filters to match the control groups with the respective comparison groups – PwMS and caregivers. It was however not possible to obtain a complete match between caregivers and their control group as concerns education level. The decision to include two different control groups was based on the significant age difference between PwMS and their caregivers (t = 2.85, p < 0.005). The demographic features of the four groups of participants are reported in **Table 1**.

No differences emerged between PwMS and their control group (control 1) for age, gender, marital status and education, while a significantly higher percentage of PwMS was unemployed (χ <sup>2</sup> = 21.81, p < 0.001). As concerns caregivers and their control group (control 2), differences were detected for employment and education, both higher in the control group (χ <sup>2</sup> = 7.52, p < 0.01, and χ <sup>2</sup> = 4.61, p < 0.05 respectively).

#### Materials

Data were collected through the following instruments:

A socio-demographic questionnaire provided information on participants' age, employment status, education level, and civil status. Clinical information including time from disease onset, level of disability, type of treatment and co-morbidities was collected for PwMS. Caregivers also reported their type of bond with PwMS.

Eudaimonic and Hedonic Happiness Investigation inventory (EHHI; Delle Fave et al., 2011, 2016). This mixed-method instrument allows researchers to collect qualitative and quantitative information on different components of well-being. Through open-ended questions, participants are asked to define happiness in their own words; to list the three future goals they consider most important, and the three things they consider most meaningful in their present life; and to briefly describe three situations associated with intense happiness during the last 6 months. In addition, participants are asked to rate on two sets of scaled items, ranging from 1 (extremely low) to 7 (extremely high), the levels of happiness and meaningfulness

<sup>a</sup>Means and Standard Deviations are reported.

<sup>b</sup>Education was dichotomized: "High school or less" includes elementary, middle, and high school; "University" includes graduation, post-graduation and PhD.

associated to 10 major life domains (work, family, standard of living, interpersonal relations, health, personal growth, leisure, spirituality/religion, community, and society) and to life in general.

Satisfaction with Life Scale (SWLS; Diener et al., 1985; Goldwurm et al., 2004). This widely used measure of well-being comprises five items on scales ranging from 1 (strongly disagree) to 7 (strongly agree). The items investigate the individuals' overall cognitive evaluation of their global life conditions and achievements.

Positive Affect and Negative Affect Schedule (PANAS; Watson et al., 1988; Terracciano et al., 2003). The instrument assesses the overall perceived intensity of positive and negative affect during daily life, through 10 items measuring components of positive affect (PA) and 10 items measuring components of negative affect (NA). Scales range from 1 (very slightly or not at all) to 5 (extremely).

#### Procedure

This study involved seven different academic and clinical institutions; therefore, the protocol was submitted to the ethical committees of each institution. After approval from all committees, participants with MS and their caregivers were recruited at six MS centers in different Italian regions, in the context of a broader project aimed at investigating clinical, psychological and relational aspects of MS (Bassi et al., 2014, 2016). PwMS were contacted by the centers' personnel during check-ups or by phone, and were invited to identify their primary caregiver. Data from participants in the two control groups were selected from a larger study that had been approved by the ethical committee of the first author's institution.

Upon their expression of interest in joining the study, participants received detailed information on the project from a researcher involved in the study. They signed an informed consent in compliance with Italian privacy rules, and were provided with the battery of questionnaires. They could inspect the questionnaires, pose general and specific questions to the researcher, and express their doubts and concerns. PwMS and their caregivers were free to decide whether to complete the questionnaires immediately at the MS center, or at a time and place convenient to them. They could hand in their responses, or send them via mail. Before data processing researchers removed the consent form from each battery of questionnaires, thus guaranteeing participants' anonymity throughout the phases of data coding, storing and analysis. Data were stored in password protected computers. Participation to both the MS related study and the general survey was voluntary, and participants were free to leave the study at any time. Persons with MS were explicitly assured that refusal to participate or withdrawal from participation in the study would in no way interfere with the long-term healthcare services they were receiving at the MS center.

#### Coding Procedure

Answers to the open-ended questions of the EHHI required an accurate coding work, using a coding system originally developed by Delle Fave et al. (2011), and gradually expanded through additions from various studies, in line with the bottom-up approach guiding the EHHI research project. In this coding system, answers are grouped into broad categories corresponding to the major life domains: work, family, standard of living, interpersonal relationships, health, personal/psychological life, leisure, spirituality/religion, society and community issues, and more general/unspecific life aspects. Multifaceted categories such as work, family, relationships, leisure, spirituality/religion, community/society and personal life are further subdivided into more fine-grained sub-categories (Delle Fave et al., 2013a). The categories family and interpersonal relationships were organized into the subcategories intrinsic value (e.g., having a family; a partner; children; friends), sharing (e.g., spending time with children; sharing life projects with partner; sharing good and bad experiences with friends), personal contribution (caring for elder parents; raising children; helping friends in need), family well-being/harmony, balance (e.g., health of family members; children's positive development; harmony in relationships) and personal reward (e.g., gratification from partner, children; gratitude expressions from siblings; satisfaction with relations). A similar partition characterizes community/society issues. In the psychological/personal life category, some subcategories refer to eudaimonic well-being dimensions (growth/engagement, purpose, competence/mastery, autonomy, self-actualization, meaning/value, harmony/balance, fullness/awareness, optimism), and others to hedonic wellbeing ones (satisfaction/achievement, positive emotions, and absence of negative feelings); a less specific subcategory, labeled as "positive experiences/states", includes answers generically referring to happiness as "inner well-being", "a stable state", or "a way of being."

The coding procedure for each answer comprises first the identification of a category in which the answer can be included, and then a specific numeric item to which the answer can correspond, if available. If an answer content does not fit any available item, a new item will be added to the category. As concerns happiness definitions, participants often report complex descriptions including different components of happiness. Each of these facets is treated as a specific semantic unit and coded separately; up to six answer units are retained for each participant. Based on the specific formulation of the questions concerning goals, meaningful things, and recent situations of most intense happiness, three answer units are retained for each question. In the present study, reliability in the coding process was established through the involvement of two expert coders providing independent ratings for each answer; divergences were clarified through a subsequent discussion; unsolved discrepancies and answers potentially requiring the inclusion of new items in the coding system were further discussed with the first author.

As concerns the scaled questions included in the EHHI, and the data collected through PANAS and SWLS, the numeric values corresponding to the perceived levels of each variable were reported. The EHHI items assessing the levels of happiness and meaningfulness associated with personal/psychological life are labeled as "personal growth"; this decision was originally based on the assumption that growth and development could best represent the positive and meaningful sides of this life domain (Delle Fave et al., 2013a).

#### Statistical Analyses

The analysis of qualitative data collected through the EHHI was aimed at identifying the life domains predominantly mentioned by participants across groups in their descriptions of happiness, major goals, meaningful things, and recent sources of intense happiness. Results were compared between groups (respectively PwMS and control 1, and caregivers and control 2). Since a high percentage of participants across groups indicated family as prominent goal, meaningful thing, and source of recent happiness, analyses were performed on family sub-categories. The same in-depth analysis was conducted for the psychological definitions of happiness.

Coded answers were first grouped into the corresponding categories and sub-categories; subsequently, considering that each participant provided more than one answer for each question, the number of participants reporting at least one answer in each category and subcategory was calculated across groups. This approach allowed us to compare the percentage of participants referring to each answer category and subcategory between groups through 2 × 2 frequency tables by means of the χ <sup>2</sup> procedure. The procedure was not considered as reliable when the number of participants in one or more cells was below 5. Through Spearman coefficients, correlations were then calculated between participants' distribution in answer categories and their demographic and group features. Logistic regression analyses allowed us to verify whether demographic or group features predicted a specific pattern of answers.

Quantitative data were first analyzed using descriptive statistics. Correlations of hedonic and eudaimonic dimensions of well-being with participants' group and demographic features were calculated through Pearson and Spearman coefficients. Hierarchical regression analyses provided information on the specific contribution of participants' group and demographic features to well-being dimensions.

Given the large number of group comparisons performed on quantitative and qualitative variables, we took a Bonferroni approach, adjusting the critical alpha value for significance to the number of t-tests and χ 2 comparisons performed on the same dataset (25 and 61 respectively). More specifically, to achieve α < 0.05 with 25 t-tests, the alphas obtained from each data set had to score below 0.002 (α < 0.05/25, two-tailed); in order to reject a null hypothesis, the test statistic had to exceed critical t = 3.16, corresponding to p < 0.002 with 122 degrees of freedom (for two groups, df = N participants-2).

The same approach was used for frequency table comparisons (N = 61 in each data set). To achieve α < 0.05 with 61 comparisons, the alphas obtained from the data set had to score below 0.0008 (α < 0.05/61); in order to reject a null hypothesis, the test statistic had to exceed critical χ <sup>2</sup> = 11.34, corresponding to p < 0.0008 with 1 degree of freedom, since df = (N columns-1) (N rows-1). As for correlations, regardless of the statistical significance only coefficient values equal to or higher than 0.30 were considered as adequate to interpret associations as meaningful (Hinkle et al., 2003). More specifically, values between 0.30 and 0.50 indicated low correlation, values between 0.50 and 0.70 moderate correlation, and values above 0.70 high correlation.

### RESULTS

This section illustrates the qualitative and quantitative findings presented for each group separately, in order to allow for comparisons.

#### Qualitative Findings

The findings obtained from the open-ended questions of the EHHI are presented as percentages of participants across groups who provided at least one answer in the different categories and subcategories. The answers referring to psychological dimensions are grouped into the category labeled as "psychological definitions" for the definitions of happiness, and into the category "personal life" for goals, meaningful things and recent situations of intense happiness.

#### Happiness Definitions

Participants were invited to answer the question "What is happiness for you?" in their own words, without specific constraints. It is worth noticing that 9 PwMS and 8 caregivers either did not provide any answer, or stated that "happiness does not exist," while only 2 participants in each control group did so. Moreover, control groups 1 and 2 reported on average a higher number of answer units in their happiness definitions (2.65 and 2.80 respectively) compared with PwMS and caregivers (2.25 and 2.19 respectively). These differences were however not statistically significant. **Table 2** shows for each group the percentage distribution of the participants who provided at least one answer in the different categories and subcategories. Results of group comparisons are also reported.

Overall, a substantial similarity emerged between groups across most categories. Only two significant differences were detected between PwMS and control 1: a lower percentage of PwMS reported psychological definitions of happiness, and a lower percentage of controls reported family-related ones. Despite these overarching differences, the percentage distribution of participants across subcategories of both psychological and family-related definitions did not differ between the two groups. Among psychological definitions, participants predominantly referred to eudaimonic constructs, such as harmony/balance, self-actualization, personal growth, and optimism. Since a remarkably high percentage of participants specifically mentioned harmony/balance, related findings are reported separately in **Table 2**. As concerns family, most participants in both groups provided answers related to sharing (experiences, activities, projects). Personal rewards followed as subcategory; answers included receiving love expressions, support, acknowledgment from family members, and satisfaction with family. Fewer participants quoted the other domains, and a negligible percentage in both groups mentioned leisure, spirituality/religion, community/society issues and standard of living. A similar answer pattern was detected among caregivers and control 2, but no significant group differences emerged.

TABLE 2 | Definitions of happiness: percentage of participants mentioning each category and subcategory by group, and comparisons between groups.

<sup>a</sup>Each participant could provide more than one answer; Bonferroni adjusted alpha \*p < 0.05; \*\*p < 0.01; χ 2 -values are not reported if the no. of participants in one or more cells falls below 5.

#### Goals and Meaningful Things

While all participants in the control groups identified some important future goals, 2 PwMS and 4 caregivers did not. In addition, 59 participants in control 1 reported up to three goals compared with 45 PwMS (χ <sup>2</sup> = 11.68, p < 0.05); similarly, 59 participants in control 2 reported up to three goals compared with 44 caregivers (χ <sup>2</sup> = 12.89, p < 0.05). **Table 3** depicts the percentage distribution of the participants who provided at least one answer across categories in each group.

The majority of participants in all groups mentioned family, specifically referring to its intrinsic value (having a family; having or adopting children; having grandchildren; finding the right partner) and to the well-being of family members (physical health, self-actualization or goal achievement of children, grandchildren, partner, siblings, and parents). Health, work, and personal life were mentioned by lower percentages of participants; spirituality/religion and community/society were marginally represented, together with interpersonal relations. The only significant difference concerned the higher percentage of PwMS mentioning health, compared to control 1.

As regards the most meaningful things in the present life, 7 caregivers did not identify any, compared to 4 PwMS and no participants in the control groups (χ <sup>2</sup> = 13.22, p < 0.05). Moreover, 60 participants in control 2 reported up to three meaningful things, compared with only 46 caregivers (χ <sup>2</sup> = 12.74, p < 0.05). Nevertheless, as illustrated in **Table 4**, participants' percentage distribution across answer categories was largely overlapping across groups.

Participants in all groups almost unanimously quoted family as one of the most meaningful things in their lives

TABLE 3 | The most important future goals: percentage of participants mentioning each answer category by group and comparisons between groups.

<sup>a</sup>Each participant could provide more than one answer; Bonferroni adjusted alpha \*p < 0.05; χ 2 -values are not reported if the no. of participants in one or more cells falls below 5.

TABLE 4 | The most meaningful things in present life: percentage of participants mentioning each answer category by group and comparisons between groups.

<sup>a</sup>Each participant could provide more than one answer; χ 2 -values are not reported if the no. of participants in one or more cells falls below 5.

(specifically referring to its intrinsic value); progressively lower percentages of participants mentioned work, health, interpersonal relationships and personal life, without significant group differences. Spirituality/religion, leisure, standard of living and community/society were mentioned by less than 10% of the participants across groups.

#### Recent Situations of Intense Happiness

As illustrated in **Table 5**, only 39 caregivers (62.9%) provided answers to this question, while the remaining 23 (37.10%) could not remember any recent situation of intense happiness. This answer distribution pattern was significantly different from those detected for the other three groups (χ <sup>2</sup> = 29.93, p < 0.001): only 12 participants (19.35%) among PwMs, 5 (8.1%) in control 2 and 2 (3.23%) in control 1 did not report recent situations of intense happiness.

No group differences instead emerged in the percentage of participants who provided at least one answer across categories. Family ranked first again; within this category, the majority of participants across groups referred to sharing positive events and experiences, such as anniversaries and other celebrations. A lower percentage of participants mentioned events promoting the well-being of family members (positive school and work achievements, disease recovery or health improvements). Leisure predominantly included the practice of arts and crafts, sports, travels and media fruition. Interpersonal relationships and work followed in rank across groups. A higher percentage of PwMS referred to health related situations, but after Bonferroni adjustment the difference with control 1 was not significant. Spirituality/religion and community/society were again reported by a negligible percentage of participants across groups, together with personal life and standard of living.

TABLE 5 | Recent situations of intense happiness: percentage of participants mentioning each answer category by group and comparisons between groups.

<sup>a</sup>Each participant could provide more than one answer; χ 2 -values were not reported if one or more cells included less than 5 participants; answer categories cited by less than 5 participants across groups were excluded from analysis.

#### Quantitative Findings

#### Ratings of Happiness, Meaningfulness, and Hedonic Well-Being

**Table 6** shows the mean ratings of happiness and meaningfulness across groups, and the results of the comparisons between PwMS and control 1, and between caregivers and control 2.

Participants across groups reported the highest levels of happiness and meaningfulness in the domains of family, health, interpersonal relations, life in general, and personal growth (with slight variations in domain order across groups). In contrast, spirituality/religion, community and society issues were associated with the lowest levels of happiness and meaningfulness across groups. The only difference between PwMs and control Delle Fave et al. Psychosocial Well-Being in Multiple Sclerosis

1 concerned happiness with health, with the former reporting significantly lower values. The comparison between caregivers and control 2 highlighted two significant differences: Caregivers reported higher values of happiness in relation to community

issues, and they attributed lower meaningfulness to the domain of leisure.

More relevant differences were detected between groups as concerns the hedonic well-being dimensions. As illustrated in **Table 7**, both PwMS and caregivers scored significantly lower on positive affect compared with their respective control groups. Satisfaction with life ratings were significantly lower among PwMS than among participants in control 1. No group differences were instead detected for negative affect.

#### Demographic and Group Predictors of Well-Being Dimensions

Correlations and regression models were finally calculated within each group set-PwMS and control 1, and caregivers and control 2 respectively -, in order to investigate the role of group type and demographic features as predictors of qualitative and quantitative evaluations of well-being. Age, gender and marital status were not considered, as these characteristics did not differ between PwMS and control 1, and between caregivers and control 2. Attention was instead paid to education level and employment status, as group differences were detected for these two features, as reported in the comments on **Table 1**. **Table 8** illustrates Spearman correlations among predictors (demographics and group classification) and between predictors and well-being dimensions.

For the sake of synthesis, analyses were performed only for the dimensions showing significant differences between groups. As reported in the method section, correlations higher than 0.30 were deemed as meaningful. Overall, group classification showed the most numerous and strongest correlations with the well-being dimensions. In the PwMS/control 1 group set, only employment status showed a low positive correlation with satisfaction with life; together with group classification, it showed a low positive correlation with happiness with health as well. No meaningful correlations were instead identified for education level. In the caregivers/control 2 group set, group type showed low to moderate correlations with most well-being dimensions, except for satisfaction with life and negative affect. A low positive correlation emerged between education level and positive affect, while employment status did not show any relevant correlation with well-being dimensions.

TABLE 7 | Levels of affective and cognitive dimensions of hedonic well-being, and their comparison between groups.

Bonferroni adjusted alpha \*p < 0.05; \*\*p < 0.01.

Bonferroni adjusted alpha \*p < 0.05; \*\*p < 0.01.

TABLE 8 | Correlations among demographic and group predictors, and between predictors and variables showing significant differences in the two group sets.

\*p < 0.05; \*\*p < 0.01; \*\*\*p < 0.001. "Group" is a dummy variable with 1 for PwMS or caregivers and 0 for the respective control groups.

Based on these results, linear regressions with stepwise selection were carried out to investigate the role of employment status, education level and group classification as predictors of the hedonic well-being dimensions: positive affect, negative affect, and satisfaction with life. In addition, linear regressions (with scaled outcome variables) and binary logistic regressions (with categorical outcome variables) were performed to assess the role of demographic features in the findings obtained through the EHHI for which group differences were identified. More specifically, for the PwMS/control 1 group set regressions were conducted for happiness with health, psychological and familyrelated definitions of happiness, and health-related goals. For the caregivers/control 2 group set regressions were conducted for happiness with community and meaningfulness of leisure.

As concerns PwMS and control 1, for the dependent variable positive affect the two predictors entered in the model were group type and education level, together explaining 18% of the variable variance (r <sup>2</sup> = 0.143, F = 20.13, p < 0.001, and r 2 = 0.039, F = 5.41, p < 0.05 respectively). Negative affect was significantly predicted by group type (r <sup>2</sup> = 0.041, F = 5.27, p < 0.05), however explaining only 4.4% of the variable variance. Satisfaction with life was significantly predicted by employment status and education level (r <sup>2</sup> = 0.121, F = 16.69, p < 0.001, and r <sup>2</sup> = 0.042, F = 5.82, p < 0.05 respectively), but not by group type; the two significant predictors together explained 16.3% of the variable variance. The level of happiness with health was predicted by group type (r <sup>2</sup> = 0.126, F = 17.47, p < 0.001), and employment status (r <sup>2</sup> = 0.048, F = 7.04, p < 0.01), together explaining 17.4% of the variable variance. The distribution of participants citing psychological and family-related happiness definitions was predicted by group type only (B = −1.27, OR = 0.079, Wald χ <sup>2</sup> = 22.63, p < 0.001; and B = 0.67, OR = 3.820, Wald χ <sup>2</sup> = 9.04, p < 0.01 respectively). Group type was also the sole predictor of health-related goals distribution (B = 0.66, OR = 3.783, Wald χ <sup>2</sup> = 11.20, p < 0.001).

In the caregivers/control 2 group set, positive affect was significantly predicted by group type and education level, together explaining 36.9% of the variable variance (r <sup>2</sup> = 0.334, F = 61.25, p < 0.001, and r <sup>2</sup> = 0.035, F = 6.75, p < 0.05 respectively); no significant predictors were instead identified for negative affect. Group type emerged as the only significant predictor for the three remaining variables: satisfaction with life (r <sup>2</sup> = 0.071, F = 9.36, p < 0.01, explaining 2.2% of the variable variance); happiness with community (r <sup>2</sup> = 0.088, F = 10.80, p < 0.01, explaining 2.6% of the variable variance) and leisure meaningfulness (r <sup>2</sup> = 0.141, F = 20.04, p < 0.001, explaining 4.2% of the variable variance).

### DISCUSSION

This study aimed at investigating different facets of well-being among persons with MS and their caregivers from an integrated perspective. At the conceptual level, the eudaimonic dimensions of goal pursuit and meaning-making were jointly investigated with the hedonic ones of affect and life satisfaction. In addition, based on a bio-psycho-social and ICF informed perspective, the investigation of well-being was contextualized within the major life domains, and findings were compared with those reported by two groups of participants sharing similar demographic features, but not exposed to experiences of chronic illness or caregiving. At the empirical level, the adoption of a mixed method approach gave participants the opportunity to freely describe their present evaluations and future expectations.

### The Private Context of Well-Being: A Shared Perspective

The information collected through the EHHI allowed us to contextualize individuals' perceived well-being within the major life domains. Regardless of group inclusion, in both qualitative and quantitative answers participants primarily referred to few life domains, basically circumscribed to the private sphere. Family, personal life and health distinctly emerged across groups as the predominant contexts of happiness, meaningfulness and goal investment. A substantially lower relevance was attributed to the broader contexts of work and interpersonal relations; finally, the public domains of community and society, and the transcendent sphere of spirituality/religion were almost absent from participants' qualitative answers, and they scored lowest in rank on the scales. Overall, these findings largely confirmed previous EHHI studies conducted across countries on adult samples belonging to the general population (Delle Fave et al., 2011; Delle Fave et al., 2013b).

Across groups, participants who referred to family in their definition of happiness mainly focused on the dimension of sharing. This finding is consistent with the models emphasizing the primacy of relational connectedness in humans (Richardson, 2012). As specifically concerns persons with MS and their caregivers, these results are also consistent with the evidence of a shared process of adaptation to illness, based on communal growth and search for meaning (Pakenham, 2005; Pakenham and Cox, 2009; Ackroyd et al., 2011). Among the psychological definitions of happiness, eudaimonic dimensions were predominant; inner harmony was reported by the highest percentage of participants across groups, followed by selfactualization and personal growth. This finding, consistent with previous evidence (Delle Fave et al., 2016), provides further support to the view of happiness as connectedness, at both the inner and relational levels (Kjell, 2011). Most of the research hypotheses concerning happiness definitions were however not confirmed. Against expectations, the percentage of PwMS referring to health and the percentage of caregivers referring to family were not significantly higher, compared to their respective control groups. Similarly, against expectations no difference was detected between caregivers and control 2 in the percentage of participants quoting leisure. Only two unexpected differences emerged between PwMS and control 1: a lower percentage of PwMS provided psychological definitions of happiness, and a higher percentage mentioned family related ones. In both cases, group classification emerged as the only significant predictor. The overall relevance of family has been widely documented among persons with MS (Ryan et al., 2007; Pakenham, 2008a). It is however worth noting that in this study persons with MS, similarly to participants in the other groups, emphasized sharing rather than receiving support and rewards from family as it could be expected from individuals facing a progressively increasing dependence on their caregivers.

Across groups, the majority of participants mentioned family as a major future goal, primarily referring to its intrinsic value and to the well-being of family members. Work followed in rank, while marginal relevance was attributed to extra-family relations, and to social, community and spirituality issues. As hypothesized, a significantly higher percentage of persons with MS quoted health, compared to control 1; group classification was the only predictor of this result. All the other hypotheses were not confirmed. It is worth noticing that, compared to their respective control groups, a significantly higher percentage of PwMS and caregivers reported less than three future goals. This result can be related to the perceived uncertainty highlighted in the MS literature, and in studies involving people with other chronic diseases (Bensing et al., 2002; Tams et al., 2016). Uncertainty leads individuals to focus on the present rather than on future planning; this aspect can be even more relevant in diseases such as MS, which entails a higher margin of unpredictability, compared with other chronic and degenerative conditions (Alschuler and Beier, 2015).

Family also emerged as a key meaningful thing; the vast majority of participants across groups referred to its intrinsic value—having a family, a partner, children, siblings were identified as valuable components of life per se. The primacy of intimate relationships in the meaning making process largely confirmed previous evidence obtained with a variety of samples (Baumeister and Leary, 1995; Lambert et al., 2010; Taubman – Ben-Ari et al., 2012). Work, health and interpersonal relations followed across groups, while only a negligible percentage of participants mentioned spirituality/religion, community/society, and leisure. A significantly higher percentage of caregivers reported less than three meaningful things compared to control participants; this difference may be related to the narrower range of daily opportunities that characterizes the caring role (Mausbach et al., 2011).

When describing recent situations of intense happiness, most participants across groups referred to events shared with the family: birthdays, marriages, holidays, but also receiving a good news concerning family members. Leisure and interpersonal relationships, following in rank, were mentioned by a relatively high percentage of participants across groups only in the context of this answer. Finally, and in line with expectations, health was quoted almost exclusively by PwMS. It is also important to note that over one third of the caregivers could not identify any recent situation of intense happiness.

## Extraordinary Circumstances, Ordinary Experiences of Well-Being

Research has repeatedly emphasized the negative psychological consequences of living with MS as a person or a caregiver, especially at the emotional level. Our findings were only partially consistent with the literature, rather emphasizing the "ordinariness" of PwMS and caregivers (Saravanan et al., 2001) in their quantitative ratings of global and domain-related wellbeing.

As concerns domain-related happiness and hedonic well-being (assessed as positive and negative affect, and satisfaction with life), PwMS scored significantly lower than control participants in happiness with health (thus confirming expectations) and in positive affect. Although participants' group emerged as the strongest predictor of the two variables, unemployment further contributed in predicting lower happiness with health, and lower education level in predicting lower positive affect. PwMS also reported significantly lower levels of life satisfaction, but employment status and education, rather than presence of MS, emerged as significant predictors of this result. No differences instead emerged between PwMS and control participants for negative affect, despite the (weakly) significant predictive role of group classification. To this purpose, it is worth mentioning that negative affect values were on average higher in the control group. The comparison between caregivers and their control group highlighted a significant difference in positive affect, with the former scoring significantly lower. Group type was the strongest predictor of positive affect; the additional though limited contribution of lower education level replicated the findings detected between PwMS and control 1. Group type emerged as significant predictor of satisfaction with life as well, though with marginal explanatory relevance.

The lower levels of positive affect reported by PwMS and caregivers were consistent with the literature highlighting the emotional burden of chronic disease; nevertheless, education emerged in both cases as an additional environmental predictor, suggesting that emotions are—at least partially—socially constructed. In particular, this finding provides support to the role of education as a major objective indicator of hedonic or subjective well-being, regardless of health conditions (Kroll and Delhey, 2013). The same consideration can be made as regards satisfaction with life, whose levels were not predicted by group type, but by the social opportunities derived from employment status and education, thus confirming previous evidence (Oishi and Diener, 2014). The lack of group differences in negative affect levels confirmed instead the conceptual and empirical independence of positive and negative affect (Seib-Pfeifer et al., 2017), as well as the importance to consider well-being and ill-being as partially independent domains of experience, rather than as opposite poles of a single continuum (Keyes, 2007).

In this study eudaimonic well-being was quantitatively assessed as the level of meaningfulness associated with different life domains. Findings did not support the study hypotheses, as PwMS and caregivers did not differ from control groups in family and health related meaningfulness. An unexpected difference emerged between caregivers and control 2, with the former attributing significantly lower meaningfulness to the leisure domain. This result, solely predicted by participants' group, can be related to the restrictions in daily life opportunities experienced by family caregivers (Becker, 2011). Since under these circumstances leisure activities get often sacrificed first, downplaying their meaning and relevance can help caregivers adjust to the related constraints (Pakenham and Cox, 2009).

### Patients and Caregivers: Social Assets Beyond Clinical Labels

This study highlighted that overall persons with MS and their caregivers do not differ from healthy people in their experience of hedonic and eudaimonic well-being. These findings are consistent with the literature showing that individuals and families mobilize a variety of resources in order to adjust to disease conditions. At the same time, they offer further suggestions. Not only have these people adjusted to disease; they pursue value-driven goals, cultivate inner harmony and balance, invest their energies in meaningful activities and relationships. Persons with MS do not seem to be primarily focused on their own health and related needs; they are rather actively engaged in sharing experiences and collaborating to the promotion of family well-being (Bogosian et al., 2017). As for caregivers, their life trajectory—although forcefully disengaged from extra-family socialization and leisure—is grounded in personal and relational values, despite the costs emerging at the hedonic well-being level. In general, the present study did not highlight group differences in participants' level of engagement in public roles and social activities, thus showing that active involvement in community and society is related to cultural dimensions rather than health conditions.

Overall, these findings can be considered as a provocative claim for a change in perspective, as concerns the social representation of health. This claim, consistent with the ICF model, is based on theoretical and empirical evidence. As highlighted by studies investigating resilience (Walsh, 2015), individuals and families experiencing chronic diseases should be valued and appreciated for their ability to develop personal and communal competences, rather than being considered as weak and low-performing members of the society. Their psychological and relational competences, laboriously built over time, could be rather shared to the benefit of others. Community Based Rehabilitation (CBR) programs are rooted in this view, aimed at empowering persons with disabilities through the promotion and acknowledgment of their active community role (Khasnabis et al., 2010). Across countries, people experiencing disease are often founders or active members of associations, promoters of fundraising campaigns in support of biomedical research, civil rights activists. Therefore, health professionals could approach them as experts who can offer first-person knowledge of a specific condition, and not only as patients to treat and caregivers to instruct (Greenhalgh, 2009). Their social involvement could be extended to educational programs and other community initiatives, allowing them to share their resources and enjoy recognition as full-fledged members of the society. Although this change implies an overall revision of the health culture, the advantages would be remarkable, as efforts in this direction could lead to a more inclusive and participative society.

### Study Strengths and Limitations

The major strength of the present study is the investigation of hedonic and eudaimonic dimensions of well-being among persons with MS, their caregivers and two control groups through a mixed method approach. The complexity of this research design, too often neglected by researchers (Morales-Gonzales et al., 2004), allows for contextualizing scaled ratings within qualitative, semantically richer answers. In this study, the domain-related ratings of happiness and meaningfulness could be combined with a fine grained description of the same domains, their present and future relevance, and their relation to wellbeing. The findings provided an integrated representation of the daily activities, contexts and relationships in which participants' meaning-making process, goal pursuit and happiness experiences took place. To our knowledge, no studies of this kind are available in the psychological literature on MS, and more generally on chronic diseases.

This study has limitations as well. First of all, the crosssectional design prevented from identifying causal relationships among variables. Although the circumscribed observation field allowed for an in-depth analysis of well-being, the negative impact of disease and caregiving was not explored. The sample sizes were relatively small. Disability levels of the persons with MS did not reflect the whole range of progression stages: the inclusion of participants with very severe disability could lead to different results, at both the qualitative and quantitative levels. All participants were Italians, thus belonging to a specific socio-cultural context: this feature increases reliability in the comparison of results across samples, but it prevents from generalizing results to countries characterized by different healthcare, welfare and value systems.

### Future Directions

The findings from this study shed light on participants' experience of well-being, in the context of their daily activities and social roles. It is however important to consider that both individuals and their contexts are dynamic entities, changing over time while interacting with each other; this further level of complexity, endorsed by the ICF model, can be satisfactorily evaluated only through the collaboration of researchers from different disciplinary fields. As specifically concerns psychology, relevant contributions could derive from community and cultural psychology, with their focus on the interaction dynamics between socio-cultural practices, individual experience and collective behaviors (Christopher and Hickinbottom, 2008; Di Martino et al., 2017).

A stronger interdisciplinary collaboration is especially needed in the light of a specific result emerged from this study: persons with MS, their caregivers and the control groups reported low levels of happiness and meaningfulness in community and society issues; in addition, these domains were almost absent from their lists of goals, meaningful things, and occasions for happiness. This finding may be interpreted as an alarming sign of civic disengagement. However, it confirms evidence obtained in other studies conducted in individualistic societies (Delle Fave et al., 2011). We consider this result as a general warning for researchers, practitioners and policy makers, highlighting the pressing need to promote a culture of interconnectedness (Prilleltensky, 2005), in order to contrast the deterioration of community networks presently emerging across nations.

#### AUTHOR CONTRIBUTIONS

ADF and MB provided substantial contributions to the conception and design of the work; the acquisition, analysis, and interpretation of data; drafting the work and revising it critically for important intellectual content; final approval of the

#### REFERENCES

version to be published; and agreement to be accountable for all aspects of the work in ensuring that questions related to the accuracy or integrity of any part of the work are appropriately investigated and resolved. All the other co-authors provided substantial contributions to the acquisition and interpretation of data; critical revision of the work at the conceptual level; final approval of the version to be submitted; and agreement to be accountable for all aspects of the work in ensuring that questions related to the accuracy or integrity of any part of the work are appropriately investigated and resolved.

### FUNDING

This study was supported by FISM—Fondazione Italiana Sclerosi Multipla [Grant Nr. 2011/R/5], with MB as principal investigator.

#### ACKNOWLEDGMENTS

This study was made possible by the engagement and dedication of many people. The authors would like to express their gratitude to the persons with multiple sclerosis, their caregivers and the participants in the control groups who carefully completed the questionnaires and scales used in this work. The complex data collection was conducted through the active collaboration of Beatrice Biolzi (Hospital of Vaio-Fidenza, Italy), Claudia Niccolai (University of Florence, Italy), Clara Chisari (University Hospital G. Rodolico, Catania, Italy), Miriam Benin and Samuela Turati (ASST Lariana, Como, Italy), Giovanna De Luca and Marco Onofri (University G. D'Annunzio, Chieti, Italy). Raffaela Sartori and Luca Negri (University of Milano) actively contributed to the recruitment of the participants in the control groups, and to the coding and storing of the data collected with the EHHI.

**Conflict of Interest Statement:** There was no involvement of study sponsors in the study design; collection, analysis and interpretation of data; writing of the manuscript; decision to submit the manuscript for publication. ADF, MB, BA, MP, SC, MF, EM, MG, MV, MPA, and EP declare that the research was conducted in the absence of any commercial or financial relationships that could be construed as a potential conflict of interest. BG received personal grant for speaking and advisor activities from Biogen and TEVA. FP received personal grant for speaking and advisor activities from Almirall, Bayer, Biogen, Celgene, Merck, Novartis, Roche, Sanofi, TEVA. MPA received research grants and honoraria as a speaker and member of advisory boards from Bayer, Biogen, Merck, Novartis, Roche, Sanofi Genzyme and Teva. AL served as Advisory Board Member for Bayer, Biogen, Genzyme/Sanofi, Merck Serono, Novartis, Teva; this author also received travel grants and honoraria from Bayer, Biogen, Genzyme, Merck Serono, Novartis, Sanofi, and Teva, as well as travel and research grants from Associazione Italiana Sclerosi Multipla; this author's institution received research grants from Almirall, Bayer, Biogen, Merck Serono, Novartis, Sanofi, and Teva.

Copyright © 2017 Delle Fave, Bassi, Allegri, Cilia, Falautano, Goretti, Grobberio, Minacapelli, Pattini, Pietrolongo, Valsecchi, Amato, Lugaresi and Patti. This is an open-access article distributed under the terms of the Creative Commons Attribution License (CC BY). The use, distribution or reproduction in other forums is permitted, provided the original author(s) or licensor are credited and that the original publication in this journal is cited, in accordance with accepted academic practice. No use, distribution or reproduction is permitted which does not comply with these terms.

# Family Function and Self-esteem among Chinese University Students with and without Grandparenting Experience: Moderating Effect of Social Support

Jingyu Shi<sup>1</sup> \*, Lu Wang<sup>1</sup> , Yuhong Yao<sup>2</sup> , Na Su<sup>3</sup> , Xudong Zhao1,3,4 and Chenyu Zhan<sup>1</sup>

<sup>1</sup> East Hospital, Tongji University School of Medicine, Shanghai, China, <sup>2</sup> Student Counselling Center, Tongji University, Shanghai, China, <sup>3</sup> Medical School of Tongji University, Shanghai, China, <sup>4</sup> Pudong Mental Health Center, Shanghai, China

#### Edited by:

Francisco José Eiroa-Orosa, University of Barcelona, Spain

#### Reviewed by:

Lucia Romo, Paris West University Nanterre La Défense, France M<sup>a</sup> Angeles Gomez Martínez, Pontifical University of Salamanca, Spain

> \*Correspondence: Jingyu Shi<EMAIL_ADDRESS>
#### Specialty section:

This article was submitted to Psychology for Clinical Settings, a section of the journal Frontiers in Psychology

Received: 20 September 2016 Accepted: 15 May 2017 Published: 30 May 2017

#### Citation:

Shi J, Wang L, Yao Y, Su N, Zhao X and Zhan C (2017) Family Function and Self-esteem among Chinese University Students with and without Grandparenting Experience: Moderating Effect of Social Support. Front. Psychol. 8:886. doi: 10.3389/fpsyg.2017.00886 This study examines the association between family function and self-esteem of Chinese university students with grandparenting experience, and explores the moderating effects of social support in this link. Two thousand five hundred thirty university students (1372 males and 1158 females) from a Chinese university completed the Perceived Social Support Scale, the Rosenberg's Self-esteem Scale, and the Family Assessment Device (FAD). Six hundred and forty-five (25.69%) students reported grandparenting experience and they reported lower scores on self-esteem and social support than the students raised only by their parents. The grandparenting group scored higher on such dimensions of family functioning as Communication, Role, Affective Involvement, Affective Responsiveness, and General Family Function (GF) than their counterpart group. For both groups, self-esteem scores were positively correlated with social support scores, while negatively correlated with FAD all sub-scale scores. Hierarchical regression analysis showed that for the students with grandparenting experience the social support moderated the relationship between GF and self-esteem. When students reported a high level of social support, those with low GF score reported higher scores in self-esteem than those with low self-esteem. However, in case of low social support, there were no differences in self-esteem between groups with high and low GF scores. These findings suggest that social support plays a positive role to relieve the adverse impact of poor family function on self-esteem of the adolescents with grandparenting experience. In addition, the significance and limitations of the results will be discussed.

Keywords: social support, self-esteem, family function, Chinese university students, grandparenting

## INTRODUCTION

Since the 1980s, due to the radical development of the society resulting from China's "reform and opening," there were many significant changes within the Chinese family. One of the major changes was the introduction of grandparenting which became an important family rearing pattern. Grandparenting refers to the situation of grandparents rearing and educating their grandchildren.

A nationwide survey in China has shown that in the largest cities, such as Beijing, Shanghai, and Guangzhou, among children under the age of six those accepting grandparenting accounted for more than 50% (Wu, 2010). Therefore, grandparenting has become an important regular condition of children's growth in China. In the European countries and United States of America, the reasons for grandparenting are mainly based on the parents' marital status (divorce, separation, premarital pregnancy, etc.) and economic reasons, while in China there are unique reasons for this phenomenon: first of all, Chinese traditional culture has formed strong and unique family ethics, as part of this moral code of conduct, grandparents view raising grandchildren as their responsibilities, and through this it is considered to obtain happiness and satisfaction. In addition, the young parents spend most of the time and energy on working or further studying, which means that they cannot fully take care of their children, therefore the children are handed over to the grandparents to be raised.  Other research is focusing on the influence of grandparenting on the psychological and social development of the children, reporting a negative effect on their self-esteem. The children being raised by their grandparents show more psychological problems and difficulties with social adaptation in family and school life (Qi, 2011; Edwards and Daire, 2006; Li and Li, 2006; Zhang and Lu, 2017). Compared with children who were raised by their parents, they showed more emotional and behavior problems (Edwards, 2006; Edwards and Daire, 2006; Wang, 2007; Qi, 2011; Zhang and Lu, 2017), they were shy and had little skills in communicating with others (Fuller-Thomson and Minkler, 2005; Xin, 2010; Han and Guo, 2016). However, the main research objects in this study were pre-school or primary school children who currently accept grandparenting. Due to a lack of retrospective studies on the influence of grandparenting on adolescents and young adults, we raised the following question: Do children with a background of being brought up by their grandparents up to adolescence or adulthood show different levels of selfesteem compared to their peers without the experience of grandparenting?

Self-esteem refers to the evaluation and experience related to self-value, the perception of self ability as well as the acceptance of the whole self, which an individual obtains during the process of socialization (Rosenberg, 1965; Leary and MacDonald, 2005). According to Maslow (1987) a person with high selfesteem feels more confident, is more competent, and hence more productive; while an individual with low self-esteem often feels inferior, hopeless, even desperate and tends to get neurosis. An individual with high self-esteem has a high level of mental health status and self-harmony (Peng et al., 2013). Moreover, self-esteem could positively affect the general wellbeing (Shim et al., 2013; Tian et al., 2013). Self-esteem might also contribute to a higher success motivation (Pang et al., 2011). In addition, it might affect the ability to achieve goals or increase the ability to cope with problems, while low selfesteem may lead to avoidance (Hendy et al., 2003). Self-esteem can also affect decision-making, which may have an impact on the individual's entire life (D'Amico and Cardaci, 2003). Selfesteem is one of the basic needs for all individuals especially in adolescence. College Students are in the transitional period from adolescents to adults, they inevitably experience great changes during the shift from middle school students to college freshmen. They should independently manage their college life and cope with challenges. Both family and society put high expectations on them, and at the same time they must adjust to the new environment. Although their intellectual and physical development has already been completed, their psychological development is not yet fully mature, and in addition their selfconsciousness is still developing. Therefore the college students are easily to experience inner conflicts and ambivalence. Selfesteem as an important trait of the self-system has a positive impact on the mental health, personality development and social adaptation of the individual (Leary and MacDonald, 2005).

The formation and development of one's self-esteem is not only the result of the socialization process, but also the product of the interaction with one's sociocultural and familial environment as well as school education. Most of the research findings demonstrated that children's self-concepts are related to parentchild relationships, and family characteristics (Brown et al., 2009). Brown et al. (2009) found that the interplay of familial relationships has an impact on a child's developing sense of self: In the families that show more harmonious interactions, the children describe themselves as being more adventurous. However, it is more likely that in the families which experience high levels of discord the children view themselves as more fearful and less agreeable. The results of another study showed that the parents of the children with low self-esteem tend to punish the children severely, whereas in the group of the children with high self-esteem the family exhibited a more democratic way of interaction (Peng and Fan, 2007; Heaven and Ciarrochi, 2008). Various researchers examined the relationship between family function and self-esteem, they consistently found that there is a close relationship between self-esteem and family function, and the results have cross-cultural consistency: Wang et al. (2010) for example found that there is a significant correlation between the development of adolescent self-esteem and parental rearing styles. Other data showed that there is a significant positive correlation between a high level of family intimacy as well as emotional expression and a high level of self-esteem and self achievement of adolescents (Li et al., 2006); the extent of closeness of family members and the parental expectations might affect the self-esteem and self-concept of the college freshmen (Wu and Ye, 2009). However, grandparenting experience might affect the parent-child relationship. Li (2010) examined the parent-child relationship of college students with grandparenting experience during childhood and found that intimacy between parents and children was lower compared to those with nongrandparenting experience; the students felt distant from their parents and the parents did not trust in their children's capability due to a lack of initiative communication between parents and

children. Hence, it is necessary to explore whether and how the family function of the students with grandparenting experience influences their self-esteem? Compared with students solely raised by their parents, in case of existing factors, which ones do have a moderating effect on the relationship between family function and self-esteem of the students with grandparenting experience?

Social support can be defined as an individual's perception or experience that their social network will provide effective emotional and substantial support during the times of need (Wills, 1991). The consistent finding in many researches show that social support is positively related to self-esteem (e.g., Barrera and Garrison-Jones, 1992; Wentzel, 1994; Sweeting and West, 1995; Goodwin et al., 2004; Birndorf et al., 2005; Zhang, 2007; Denissen et al., 2008). In adolescence significant changes to social relationships will happen. In particular, the transition to the university needs more and more independence from the family and the development of a new social network. Data showed that there was a positive relationship between the utilization of social support and self-esteem of university students (Peng et al., 2003; Ma, 2005). Moreover, it is suggested by some authors that during early adolescence the peer relationships take on increased importance (Furman and Buhrmester, 1992). The results of previous studies (Fan and Fang, 2004; Lian et al., 2016) showed that support from the peer group significantly influences the self-esteem of adolescents, especially the emotional support positively predicted the level of selfesteem.

Further data demonstrated that adolescents' social relations outside the family might be a moderator for the connection of relationships with parents and adolescent adjustment. For example, Beyers and Seiffge-Krenke (2007) longitudinal study found that adolescents' relationships with parents influenced later problems of behavior when interacting with friends and romantic partners. To our knowledge, no studies exist which examine the moderator effect of social support on the relationship between family function and self-esteem. Therefore, in the current study the relationship between the three factors was explored. The aim of this study is to examine the difference in family functioning and self-esteem between university students with and without grandparenting experience and to investigate the moderating effect of social support in the relationship between family functioning and selfesteem based on retrospective data from a group of university students.

### Hypothesis

First, we assumed that grandparenting experience would have influence on family functioning and level of selfesteem. Second, we assumed that social support would have moderating effect in the relationship between family functioning and self-esteem in the students with grandparenting experience; Third, from a perspective of moderation model, we hypothesized that in the relationship between family function and self-esteem it would show a significant difference between persons with high social support and low social support.

## MATERIALS AND METHODS

### Participants

A sample of 2530 freshmen and sophomores (1372 were males, 1158 were females) was recruited at a university in Shanghai, a large city in the eastern part of China. The students' ages ranged from 16 to 20 years, with an average of 18.79 years (SD = 0.02). Written informed consent was obtained from all the participants prior to the study. The written informed consent from the caretakers on behalf of the participants under 18 years was also obtained.

### Measures

#### Family Assessment Device (FAD)

The Family Assessment Device (FAD) is a well established measure to assess family functioning based on the theoretical concept of the McMaster Model of Family Functioning (MMFF). The term "family functioning" refers to the ability of the family to work together as a unit to satisfy the basic needs of its members (Ryan and Keitner, 2009). The MMFF emphasizes the interrelatedness of the family members and the family system, and represents six dimensions of family functioning that are considered of high relevance in clinical practice. It is made up of seven scales that assess six dimensions of the MMFF as well as the General Family Functioning. Among the six dimensions there are Problem Solving (PS), Communication (CM), Roles (RL), Affective Involvement (AI), Affective Responsiveness (AR), and Behavioral Control (BC) (Shek, 2001): (1) problem solving (the capability of the family to cope with problems in order to keep effective family functioning); (2) CM (the way of exchanging information between family members); (3) RL (whether the family assign certain tasks to guarantee implementation of family functions); (4) AR (to which extent the family members emotionally react to stimulation); (5) AI (to which extent the family members show concern to each other); and (6) behavior control (the behavioral models that the family establishes to cope with stressful situations). The instrument consists of 60 statements about a family; the requirement is to rate to which extent the description of each statement accords with their own family. All items are rated on a 4-point Likert scale, with answer choices from "1 = strongly agree" to "4 = strongly disagree." Sample items for example include, "You can't tell how a person is feeling from what they are saying," "If someone is in trouble, the others become too involved," "Making decisions is a problem in our family," and, "We confront problems involving feelings." The higher scores the respondents rate, the worse levels of family functioning indicate. The validity and reliability of the Chinese FAD has been demonstrated by Shek (2001, 2002). The test-retest reliability is 0.53–0.81, and coefficient alpha ranges from 0.53 to 0.94.

#### Rosenberg Self-esteem Scale

The Rosenberg Self-esteem Scale (SES; Rosenberg, 1965) was applied to assess self-esteem. This instrument consists of 10 items. It is a self-report measure of global self-esteem. All items are rated on a 4-point Likert scale with answer choices from "1 = strongly disagree" to "4 = strongly agree." Sample items for example include "I am able to do things as well as most other people." and "I take a positive attitude toward myself." The total scores range from 10 (low level of self-esteem) to 40 (high level of selfesteem). Good levels of reliability and validity of the Chinese RSES have been demonstrated in the previous studies (Zhao et al., 2012; Kong and You, 2013). In this study, the Cronbach alpha coefficient for the RSES was 0.84.

#### Perceived Social Support Scale

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The Perceived Social Support Scale (PSSS) was developed by Zimet et al. (1988). The scale consists of 12 items designed to assess perceived social support from three sources: Family, Friends, and Significant Others. Items are scored on a 7-point rating scale ranging from 1 (very strongly disagree) to 7 (very strongly agree) with possible scores ranging from 12 to 84. The current total perceived social support status can be measured according to the total score for the 12 items. The Chinese version of this scale has been widely used and exhibited good level of validity and reliability (Wang et al., 1999; Kong and You, 2013). In this study, the Cronbach's a coefficient for the PSSS was 0.72.

#### Grandparenting Experience

The definition of grandparenting experience in this study refers to that individual during their childhood left their parents and was raised by grandparents for at least 1 year. The grandparenting experience was evaluated using a questionnaire designed by the authors that included sex, age, whether left parents and was raised by others in the childhood, the duration and caregiver. The students who reported that they left their parents and were raised by grandparents for at least 1 year during their childhood were defined as grandparenting group.

### Procedure

All of the participants volunteered to participate at the end of the mental health education course. They signed an informed consent form at first. Participants were instructed to complete a questionnaire survey including measures of family functions, self-esteem, and perceived social support as well as grandparenting information in a quiet classroom environment after informing consent. It took approximately 20 min for the students to complete all the instruments.

### Data Analysis

All statistical procedures were conducted using the Statistical Package for the Social Sciences (SPSS 17.0). Independent sample t-test was used to compare the level of self-esteem, family function score and social support score between the two groups with and without grandparenting experience; Pearson correlation was calculated to explore how levels of self-esteem and family functions as well as perceived social support were related; Hierarchical regression analysis was applied to examine the moderating effect of social support on the relationship between family function and selfesteem.

## RESULTS

## Demographic Background of the Participants

Six hundred and forty-five (25.69%) students reported that they left their parents and were raised by grandparents for at least 1 year during their childhood; 1764 (70.25%) students were brought up only by their parents; and 121 (4.06%) students reported that they had experience of being raised by other persons, who were not their own parents or grandparents. Among the students who were only raised by their parents the percentage of only children was higher compared to the grandparenting group (80% vs. 74.1%; X <sup>2</sup> = 9.648, P = 0.002); there was no statistic difference in proportion of gender between two groups (Parents-raising: male 53.5% vs. Grandparenting: male 57.1%; X <sup>2</sup> = 2.463, P = 0.117).

### Comparison of the Level of Self-esteem, Family Function Scores and Social Support Scores between the Two Groups with and without Grandparenting Experience

The students with grandparenting experience scored lower on self-esteem and total score of social support as well as support from family compared to the students raised only by their parents, while on the sub-scales of family function such as CM, RL, AR, AI, and GF the grandparenting group scored higher than their counterpart group (see **Table 1**).

### Correlation between Family Function, Social Support and Self-Esteem

In both of the two groups there are obviously significant positive correlations between self-esteem and social support and obviously significant negative correlations between self-esteem and all the sub-scales of family function. The sub-scales of family function are positively inter-correlated (see **Table 2**).

### Hierarchical Regression Analysis on the Moderating Effect of Social Support on the Relationship between Family Function and Self-esteem

In order to test the moderating effects of social support on the relationships between family function and selfesteem, hierarchical regression procedures were performed as recommended by Baron and Kenny (1986). Before testing the moderating effects, the two predictor variables (social support and family function) were standardized to reduce problems associated with multicollinearity between the interaction term and the main effects (Frazier et al., 2004). Thus, z-scores were calculated for social support and family function.

The hierarchical multiple regression analyses were conducted in each group. In the hierarchical regression model, the order of entry was as follows. At step 1, the control variables (age, gender, and "only child status") were entered into the regression

TABLE 1 | Comparison of the level of self-esteem, social support and family function scores between two groups (x¯ ± s).

PS, problem solving; CM, communication; RL, role; AR, affective responsiveness; AI, affective involvement; BC, behavioral control; GF, general functioning.

equation. At step 2, the predictor variable (all sub-scales of family function) was entered with the method of stepwise (α = 0.05) into the regression equation. At step 3, the moderator variable (social support) was entered into the regression equation. At step 4, the interaction of each dimension of family function × social support was added with the method of stepwise (α = 0.05). Significant change in R 2 for the interaction term indicates a significant moderator effect.

The results exploring predictors of self-esteem showed that for the grandparenting group three variables – RL (β = −0.170, P < 0.01), social support (β = 0.243, P < 0.01), and BC (β = −0.078, P < 0.05) had a significant effect on the self-esteem. In this model, lower family function score and higher social support score were associated with higher self-esteem score. Most importantly, there was a significant interaction between general family functioning and social support (β = −0.123, P < 0.01). These results suggest that social support moderated the relationship between general family functioning and self-esteem. In the parent-raising group, six significant predictors of self-esteem were found, these were social support (β = 0.212, P < 0.01), RL (β = −0.115, P < 0.01), CM (β = −0.111, P < 0.01), AI (β = −0.080, P < 0.01), BC (β = −0.065, P < 0.01), and PS (β = −0.062, P < 0.05). However, there was no significant interaction between family function and social support. Results of these analyses are presented in **Table 3**.

To illustrate the general family functioning × social support interaction for self-esteem in the grandparenting group, we plotted the regression of self-esteem on general family functioning at high and low levels of social support (see **Figure 1**). According to the procedures posed by Aiken and West (1991), we used the simple slope for the regression of self-esteem on general family function by using the high (one standard deviation above the mean) and low (one standard deviation below the mean) values for social support. There was a significant negative relation between general family functioning and self-esteem at high levels of social support (β = −0.452, P < 0.01). However, at low levels of social support, the relation between general family functioning and self-esteem was non-significant (β = −0.096, P > 0.05). Thus, among students with high social support, general family functioning was a significant determinant of self-esteem. In contrast, at low levels of social support, general family function did not influence self-esteem.

### DISCUSSION

The current study was designed to examine the difference in family functioning and self-esteem between university students with and without grandparenting experience and the important role of social support on the relationship between family function and self-esteem in a sample of Chinese university students. We found that the students who have the experience of grandparenting showed a lower level of self-esteem than the students raised by their parents. Grandparents raising children is a common phenomenon in China, which is related to the country's social and cultural background. The parents are both too busy to take care of the child or they are working in another city far away from their hometown, so the child is sent back to the grandparents to be raised until he or she goes to kindergarten or school (Wang, 2012). Chinese culture is collectivist and deeply influenced by Confucianism, while many western cultures are more individualistic. Collectivism emphasizes common interests, conformity, cooperation, and interdependence. These cultural traits strengthen extended families and collectives where each person takes responsibility for fellow members of their group (Hofestede, 2001). Therefore, in China many grandparents in retirement take over the responsibility of raising the grandchildren by their own choice. Song (2004) found that for late adolescents, parental attachment contributed significantly to both self-liking and self-competence. According to Bowlby (1982), the period from birth to 3 years old is crucial to establish attachment between the infant and main caregiver; individual's fundamental self-perception and

self-evaluation derive from the experience of interaction between the child and caregiver in the early childhood. The stable and health parent-child relationship makes the child feel that he or she is a favorite and capable person, and otherwise the unstable parent-child relationship makes the child feel unwelcome and worthless. The grandparenting experience could result in failure of establishment of secure attachment, and hence exert a negative influence on the self-esteem development. The children who left their parents and were raised by grandparents in infancy fail to establish an attachment relationship with their parents in their early childhood; instead they may become more attached to their grandparents. However, the lack of parental care and companionship means that when they came back to live with their parents, both the parents and the children would feel distant from each other, and the children could fail to gain strong sense of security and belonging, which might affect their self-awareness and self-evaluation (Song, 2004). Furthermore, the rearing style of grandparents is different from the parents; the grandparents may look after the grandchildren too carefully to give them enough exploration space; according to Erikson's psychosocial stages, it would hinder their development of sense of autonomy and self-worth.

In addition, compared to their counterpart group the grandparenting group reported worse family function in terms of role, CM, AI as well as AR and general family function. In nongrandparenting families, the children and parents can develop a close parent-children relationship and the children can establish a good sense of trust and security in the core family, whereas in grandparenting families, the parents may be both too busy to take care of the child or they are working in another city far away from their hometown, they cannot fully take over the responsibilities of rearing the child. In that case the grandparents instead bear a certain responsibility of raising and educating the grandchildren, it is therefore possible that the children fail to develop a close attachment relationship to their parents. As a result, this could lead to less emotional exchange and low confidence between parents and children when the children came back to their core family. Li (2010) has investigated the parent-child relationship in the college students with grandparenting experience, it was found poor intimacy, lack of trust on each other and deficient initiative CM. Therefore, worse family function might be exhibited within the grandparenting family compared to the families where the children are raised by their parents.

The students with grandparenting experience also reported lower perceived social support than the students raised by their parents. Perceived social support can be understood as a personal subjective evaluation that he or her social network will provide effective help when needed (Lakey and Scoboria, 2005). It can be distinguished from the received support, which means that actual support is provided within a specific time frame (Uchino, 2009). Family support is an important part of social support. Compared to the students raised by their parents, the perceived family support of students with grandparenting experience could be generally lower due to their distant parent-child relationship.

As we expected, the results of correlative analysis showed that self-esteem had a significant positive relationship with social support and a negative relationship with family function.

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TABLE 3 | Hierarchical regression analysis predicting self-esteem from family function and social support.

<sup>∗</sup> P < 0.05, ∗∗ P < 0.01.

SS, social support; PS, problem solving; CM, communication; RL, role; AR, affective responsiveness; AI, affective involvement; BC, behavioral control; GF, general functioning.

These results accord with previous studies on the relationships between self-esteem and social support (e.g., Goodwin et al., 2004; Birndorf et al., 2005; Zhang, 2007; Denissen et al., 2008; Lian et al., 2016; Chen et al., 2017) and family function (e.g., Heaven and Ciarrochi, 2008; An et al., 2010; Guo, 2012; Yen et al., 2013).

The most important finding of this study is that social support moderated the influence of general family function on selfesteem of the students with grandparenting experience. When these students reported a high level of social support, those with a low score of general family function reported a higher level in self-esteem. However, there were no differences in selfesteem between high and low general family function scores when they were confronted by a low level of social support. These results suggest that among the students who perceived great social support, more positive the family functioning were evaluated, higher level of self-esteem the students showed; whereas among the students who perceived poor social support, family functioning exert slight influence on the level of selfesteem. It seems that in the current study for the first time to report that social support influences on the association between general family function and self-esteem of students with grandparenting experience. We suggest that the results of our study can be ascribed to the self-development connected to the extent of early attachment. According to attachment theory, the children start to establish their basic self representation in reaction to the accessibility and sensitivity of parents and other caregivers in childhood, and these concepts are revised in the whole life (Bowlby, 1982; Bretherton, 1991). Therefore, if the caregiver is sensitive and accessible to the child, the child's self model is valuable and worthy of being loved. On the contrary, if the parents are not available and sensitive to the child, the child's self model is invaluable and is unworthy of being loved. Studies generally supports the idea that positive self-concept, including high levels of self-esteem and self-efficacy are associated with secure attachment to parents in infancy, childhood, and adolescence (Thompson, 1999; Arbona and Power, 2003). In Zhao's (2011) study it was found that the proportion of secure attachment among the children with grandparenting experience was less than that of the children raised by their parents, instead the disorganized relationship was more. Those children who were often separated from their parents in early childhood to be raised by their grandparents, missed this crucial period during childhood to build a long-lasting attachment with their parents. Although the children may establish secure attachment with the grandparents, when they came back to their parents, they tended to feel a lack of care from their parents, they did not feel

loved, and this kind of thought probably made the children feel unworthy (Li, 2010).

It is supposed that support from family, peer groups, and significant others, like teachers, or romantic partners for example is especially important for fostering identity and selfdevelopment of the college students who exhibit a lack of secure attachment to their parents (Allen and Land, 1999; Hazan and Zeifman, 1999; Fass and Tubman, 2002; Furman et al., 2002). The senses of self-importance, achievement and power gaining from the life experience are necessary for the construction of self-esteem (Yang and Zhang, 2003). If the college students are exposed to stressful situations, support from others might enable them to feel acceptance and strength, and thus enhance self-confidence and eventually lead to a higher self-esteem. Therefore, those students with a perceived lower social support as a result for being raised by their grandparents for at least 1 year, though reporting greater family function, will have difficulties in enhancing selfesteem.

Social support didn't show moderating effect in the relationship between family function and self-esteem among the students without grandparenting experience, the possible reason could be that in the parents-raising family the influence of the parents may play more crucial role in the development of self-esteem of the children across the lifespan from infancy to adolescence than that of other family members as well as significant others outside of the family.

### CONCLUSION

In the current study some limitations should be mentioned. Firstly, in this study we used self-report measures to collect data, so it could have a threat to internal validity. We suggest using multiple methods for evaluation (e.g., peer reports) in the future study, which in contrast might reduce the effect of subjectivity. The second limitation lies in quantitative methodology of the study, in which it is difficult to make deep analysis of the impact of grandparenting experience on attachment and hence on self-esteem. In future research we suggest using experimental studies or qualitative studies to test the moderating model. The third limitation is that this sample came from a university population, so it limits the extent to which these findings could be generalized to other educational environments and geographic locations in China. Despite these limitations, there are important contributions of this study.

In conclusion, all the three hypotheses in this study are fully supported. The current study makes a significant contribution to the study of the family functioning and self-esteem in youth with

#### REFERENCES

grandparenting experience. It provides an empirical framework for researchers through testing the moderating effects of social support between family function and self-esteem in a sample of Chinese college students. The findings show that, social support is an important contributor in the relationship between family functioning and self-esteem in the students with grandparenting experience. Support from family members and significant others outside of the family are especially important for fostering identity and self-development of the college students who exhibit a lack of secure attachment to their parents. In future studies, first, more exploration may be needed of how the quality of early attachment and the time of the grandparenting (e.g., infancy, toddlerhood, early childhood, or middle childhood) influence the self-esteem of the students with grandparenting experience; second, further investigation may be need of how they develop friendships with peers and significant others that helps them improve self-esteem and efficacy. In addition, these findings may provide empirical reference to help to establish effective psychosocial interventions with the aim of improving social support and family function in university students with low self-esteem, and hence improving mental health status.

## ETHICS STATEMENT

This study was approved by the Institutional Review Board of Tongji University. All participants provided written informed consent prior to the study. For the participants under 18 years old, one parent provides written consent.

### AUTHOR CONTRIBUTIONS

JS contributed to study design, recruitment of participants, data analysis and interpretation and writing of the manuscript. LW contributed to recruitment of participants and interpretation of results. YY and NS contributed to recruitment of participants. XZ contributed to study design and interpretation of results. CZ contributed to recruitment of participants. All authors have approved the final manuscript.

#### ACKNOWLEDGMENTS

We wish to thank all of the authors, researchers, and significant others who contributed to this study. This study was supported by Shanghai Pujiang Program (Grant no. 14PJ1408500) and sponsored by National Natural Science Foundation of China (Grant no. 31600892).

longitudinal analysis. Aging Ment. Health 16, 1047–1057. doi: 10.1080/ 13607863.2012.692766

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**Conflict of Interest Statement:** The authors declare that the research was conducted in the absence of any commercial or financial relationships that could be construed as a potential conflict of interest.

Copyright © 2017 Shi, Wang, Yao, Su, Zhao and Zhan. This is an open-access article distributed under the terms of the Creative Commons Attribution License (CC BY). The use, distribution or reproduction in other forums is permitted, provided the original author(s) or licensor are credited and that the original publication in this journal is cited, in accordance with accepted academic practice. No use, distribution or reproduction is permitted which does not comply with these terms.

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# Systemic Therapy for Youth at Clinical High Risk for Psychosis: A Pilot Study

#### *Jingyu Shi <sup>1</sup> \*, Lu Wang1 , Yuhong Yao2 , Chenyu Zhan1 , Na Su3 and Xudong Zhao1 \**

*1East Hospital, School of Medicine, Tongji University, Shanghai, China, 2Students Counseling Center, Tongji University, Shanghai, China, 3School of Medicine, Tongji University, Shanghai, China*

Psychosocial intervention trials for youth at clinical high risk (CHR) for psychosis have shown promising effects on treating psychotic symptoms but have not focused on psychosocial functional outcomes, and those studies have been conducted among help-seeking patients; there is a lack of research on non-clinical young CHR individuals. Systemic therapy (ST) is grounded in systemic-constructivist and psychosocial resilience theories. It has a number of advantages that makes it attractive for use with CHR individuals in non-clinical context. The present study evaluated the effect of ST for students at CHR on reducing symptoms and enhancing psychosocial function. This was a single-blind randomized controlled trial for CHR young people comparing ST to supportive therapy with a 6-month treatment. Psychotic and depressive symptoms (DS) as well as self-esteem and social support (SS) were assessed at pre- and posttreatment. 26 CHR individuals were randomly divided into intervention group (*n* = 13) and control group (*n* = 13). There were no significant differences in severity of symptoms, level of SS and self-esteem at baseline between the two groups (*P* > 0.05). At posttreatment, significant improvements in positive and DS as well as SS and self-esteem were observed in the ST group (*P* < 0.05); in the control group, these improvements were not significant (*P* > 0.05). The findings indicated that systemic intervention for university students at CHR for psychosis may have a positive effect on symptoms and self-esteem as well as SS in short term. More long-term research is needed to further evaluate this intervention.

Keywords: systemic therapy, intervention, clinical high risk, psychosis, youth

### INTRODUCTION

Young people are at greater risk of developing mental illness as they transition from childhood to adulthood (1). Psychosis typically emerges in late adolescence or early adulthood and can disrupt social and psychological development, including the attainment of educational goals and relationship skills, thus seriously impairing their quality of life. Prior to the first onset of psychotic disorder, 80–90% of individuals experience attenuated psychotic symptoms, and this stage was conceptualized as a prodromal phase, or at clinical high risk (CHR) (2, 3). The CHR for psychosis state is characterized by the presence of low intensity/frequency psychotic symptoms, brief limited psychotic episodes, and/or familial risk and/or schizotypal personality disorder in the presence of psychosocial functional decline, and with increased risk of developing psychosis (4). The CHR criteria provide an important opportunity for early intervention in preventing or delaying the onset of psychosis and reducing the social and economic burden associated with long-term mental health problems.

#### *Edited by:*

*Francisco José Eiroa-Orosa, University of Barcelona, Spain*

#### *Reviewed by:*

*Anthony William Love, Victoria University, Australia Xiao Zhou, Tel Aviv University, Israel*

#### *\*Correspondence:*

*Xudong Zhao<EMAIL_ADDRESS>Jingyu Shi<EMAIL_ADDRESS>
#### *Specialty section:*

*This article was submitted to Public Mental Health, a section of the journal Frontiers in Psychiatry*

*Received: 08 June 2017 Accepted: 05 October 2017 Published: 20 October 2017*

#### *Citation:*

*Shi J, Wang L, Yao Y, Zhan C, Su N and Zhao X (2017) Systemic Therapy for Youth at Clinical High Risk for Psychosis: A Pilot Study. Front. Psychiatry 8:211. doi: 10.3389/fpsyt.2017.00211*

Systemic therapy (ST) has shown particular promise in improving adolescent and adult mental health problems (5–7). ST is based on system theory and controlling theory. It emphasizes on viewing problems in developing, comprehensive, positive, and diverse ways and focuses on understanding the individual symptoms within the system of interpersonal relationships. In ST, the function and significance of the symptoms is much valued. Attention will be paid to the interaction between individual and his/her environment. System therapy is suitable for individuals and families. Its value orientation is positive psychology, namely, resource orientation, treating the patients/ clients as experts of solving their problems; it is assumed that the patients/clients do have the resource for solving the problem. ST emphasizes and explores the individual strengths, ability, ideas, and social resources, focusing on extending the space beyond the problem, to bring new and diverse perspectives for individuals and families, and thus to promote changes from the inner and interpersonal levels (8). Previous research reported that ST had shown positive effects on improving the symptoms and psychosocial functioning of schizophrenia patients (9). In addition, ST considers changes outside of the treatment; all in all, it is an efficient, economical, short-term treatment with a long interval.

To our knowledge, there was no specific manual for the treatment of clinical risk of psychosis available at the time of research, therefore a manual of ST was developed by the experts for ST, integrating a broad range of systemic methods, for the CHR university students.

We reviewed ST manuals for psychotic disorders in adult psychotherapy (10) and well-established ST manuals for various disorders (11–13). We used general ST concepts (8), integrating constructivist, solution-oriented methods (14), in addition to paying attention to the disorder-specific relational systemic dynamics (15, 16). According to the literature and to our experiences of treating patients, the aim of ST is to contextualize attenuated psychotic symptoms by addressing an individual's social system to which he/she attaches importance. Although the social system members could not attend the meeting physically, circular questions included them on a cognitive level. The analysis of transgenerational relationships and of past and present interpersonal interactions help to develop a new understanding of the important roles, places, and resources of all system members. Under the systemic model, the therapy sessions alike are held individually but with a strong focus on relationship issues. The ST manual differentiates between four phases, which are described in detail in the Section "Materials and Methods."

We found only seven systemic intervention RCTs for psychotic disorder (17–23), and only one for patients at CHR state (22). This study (*n*= 40, China) compared only medication treatment with 10 sessions of structural family therapy (one school of ST) plus medication. The outcome relied on the family function, severity of prodromal symptoms, and treatment compliance. This trial demonstrated the structural family therapy group with a higher reduction of severity of symptoms, higher treatment compliance, and a higher improvement in family function than the control group (*P* < 0.05).

To our knowledge, there is a lack of studies, which explore the effectiveness of ST for CHR individuals among general populations. Most studies focus on the primary outcome as a dichotomous one of transition to psychosis, rather than the dimensional domains of psychosocial functioning. Previous studies indicate that the adolescents and young adults at CHR might show poor psychosocial functioning. In some CHR studies, it was reported that the subjects had shown a lower level of self-esteem and social support (SS) in comparison to healthy controls (24, 25), and the levels of self-esteem and SS were negatively associated with the severity of the prodromal psychotic symptoms (25–28). However, many studies indicated that such psychosocial factors could play a positive role in mental health (29–31). Moreover, it is considered as an important resource for coping with psychotic symptoms (32). Thus, a positive self-perception and a strong sense of control would prevent a negative perception of daily stress; self-esteem also has a stress-buffering effect, which protects youth from the harmful effects of stress on mental health (33). In psychotic disorders, low self-esteem has been demonstrated in both the development of delusions (34) as well as in the maintenance of psychotic symptoms (35). SS as another important personal resource has been improved, having positive effects on mental health by either directly enhancing self-esteem or indirectly by protecting individuals against the adverse impact of exposure to stress and trauma (36). SS may exert such positive effects both prior to and at the onset, as well as during the course of disorder, operating to reduce both risks of onset and relapse.

Therefore, this study was designed to investigate the effectiveness of ST for CHR individuals among general youth populations on both symptom and psychosocial functioning outcome. We hypothesize that compared to the control group, ST would reduce symptom severity while improving self-esteem as well as SS.

#### MATERIALS AND METHODS

#### Study Design

This study was a single-blind RCT of ST compared to supportive therapy conducted at university with a 6-month treatment. The treatment began within 2 weeks of completion of the baseline assessment and was available for up to 10 sessions over a 6-month period. Assessments were conducted after the treatment. Clinical raters were blind to treatment groups.

#### Participants

In the study participated 26 university students at CHR (12 males and 14 females; age 18.85 ± 1.120). The 26 CHR subjects were screened out from 2,800 students of the first and second grades. The status of CHR was diagnosed through a two-stage assessment, consisting of screening with the self-report Chinese version of 16-item Prodromal Questionnaire (CPQ-16) (37) and an assessment interview according to the Structured Interview for Psychosis-Risk Syndromes (SIPS). The criteria of prodromal symptoms require that individuals meet at least one of the three clinical criteria: (1) attenuated positive symptom prodromal syndrome, defined as recent occurrence of attenuated positive psychotic symptoms with sufficient frequency or severity; (2) brief intermittent psychosis prodromal syndrome, defined as recent presenting of psychotic symptoms with spontaneous remission within 1 week; and (3) genetic risk and deterioration prodromal syndrome, defined as coexisting of genetic risk and recent functional decline (38). This study had the ethics approval from the institutional review board of Tongji University. Prior to the study, each participant provided written informed consent.

#### Procedure

In order to select the potential individuals at CHR for psychosis, at first stage, the CPQ-16 was given to 2,800 university students. In the current study, the positive threshold for the CPQ-16 was set at 6, according to the study conducted by Ising et al. (39). Totally, 611 students reached this cut-off score, and they were invited to take part in our second stage for SIPS. We received agreement from 529 participants who accepted the SIPS, which was operated by trained psychiatrists. Of the 529 students referred to the trial, 32 were screened and 26 were randomized for treatment, 13 to the ST group and 13 to the control group. The 26 students at CHR completed the 6-month treatment.

### Measures

#### Symptoms

The severity of positive and negative symptoms (NS) was measured with the Scale of Prodromal Syndromes from 0 (absent) to 6 (psychotic) and a total symptom score was created (38). The Montgomery–Åsberg Depression Rating Scale was used to assess depressive symptoms (DS) (40). The overall functioning was rated by the Global Assessment of Functioning scale (GAF) (41).

#### Self-Esteem

Self-esteem was rated with the self-reported Rosenberg Self-esteem Scale (SES) (42), which includes 10 items with a measure of global self-esteem. Each item is rated on a 4-point Likert scale ranging from 1 = strongly disagree to 4 = strongly agree. It has been demonstrated that the RSES has good reliability and validity (43, 44).

#### Social Support

Social support was measured with the Perceived Social Support Scale (45), which is based on the unique social and cultural conditions in China. The scale is made up by 12 items designed to assess subjective SS rated on a 7-point Likert scale ranging from 1 = strongly disagree to 7 = strongly agree. The higher scores reflect higher SS. The scale has been widely used among Chinese populations and proved to have a good validity and reliability (44, 45).

#### Interventions

Systemic therapy for CHR students followed the manual developed according to Carr (7) by experts on ST, ranging from 10 to 30 years of experience. It is grounded in systemic-constructivist (46) and psychosocial resilience theories (47) and based on a solution focused model that prioritizes a careful clarification of therapeutic goal. It is solution and resource oriented, reframing one's problem from functional and meaningful perspectives, using a variety of questioning techniques to enrich perspectives toward identified problems, exploring and strengthening the resource of the clients and creating more space and possibilities to solve the problem, homework tasks were usually given to help clients gain new insights and experiences. The treatment comprises four phases, each phase including 2 to 5 sessions. First phase: establishment of therapeutic relationship, collection of information, and clarification of therapeutic goals; second phase: understanding the context of the identified problem as well as interactive patterns around the identified problem, reconstruction of the problem, and exploring resources and solutions, making use of the resources and putting the solution into practice; third phase: reinforcement and deepening of changes; fourth phase: relapse prevention. Each session lasted 50 min. During the process of treatment, the treatment interval gradually extended from weekly to monthly. The details of sessions are listed in **Table 1**.

Supportive therapy is conducted only using general counseling techniques: warm, empathic, and non-judgmental face-to-face contact and supportive listening.

All therapy sessions including ST and supportive therapy were delivered by a qualified systemic therapist with 10 years experience. It has the advantage of controlling for non-specific aspects of treatment (e.g., therapist age, sex, personality, therapist experience). The therapist received expert and peer supervision regularly.

#### Data Analysis

All statistical procedures were conducted using the Statistical Package for the Social Sciences (SPSS 17.0). Chi-square tests were applied to compare categorical demographic variables between the two groups. *T*-tests for independent samples were used to assess differences in self-esteem score and SS score between the two groups. *T*-tests for paired samples as well as effect sizes were calculated to assess differences in self-esteem score, SS score, symptom score, and functioning (GAF) between baseline and posttreatment in two groups. End of intervention scores on various outcome measures between the ST group and the control groups were compared using ANOVA of repeated measures to explore the impact effects of time and intervention.

#### RESULTS

#### Social Demographic Characteristics and Baseline Data

There were no significant differences in age, gender, whether only child and family history between the two groups (*P* > 0.05). There were no significant differences in severity of psychotic symptoms, overall functioning, level of supports, and self-esteem at baseline between the two groups (*P* > 0.05). Social demographic characteristics and baseline data are presented in **Table 2**.

### Effectiveness of Systemic Intervention on Severity of Symptoms and GAF

Participants in the ST group demonstrated significant decreases in severity of positive symptoms (PS) and DS comparing to that at baseline (*P* = 0.005); however, the participants in the control group had shown no significant changes in severity of PS and DS (*P* > 0.05). Improvements were also observed in NS and GAF

Table 2 | Baseline clinical and social demographic characteristics of two groups.

*ST, systemic therapy; PS, positive symptoms; NS, negative symptoms; DS, depressive symptoms; SE, self-esteem; SS, social support.*

in both groups, but the changes were not significant (*P* > 0.05). The results are presented in **Table 3**. Furthermore, the statistic analysis results of ANOVA of repeated measures didn't show significant impact effects of the time factor, interaction between time and intervention, as well as intervention factor on the changes. These interactions are graphed in **Figure 1**. The statistic results are presented in **Table 4**.

#### Effectiveness of ST on Level of Self-Esteem and SS

Obvious improvements in level of SS and self-esteem in participants in ST were evident at posttreatment (*P* = 0.013, *P* = 0.011); the level of SS as well as self-esteem of the individuals in the control group had no significant changes at posttreatment (*P* > 0.05). The results are presented in **Table 3**. Furthermore, the statistic analysis results of ANOVA of repeated measures didn't show significant impact effects of the time factor, interaction between time and intervention, as well as intervention factor on the changes. These interactions are graphed in **Figure 2**. The statistic results are presented in **Table 4**.

#### Clinical Significance

At posttreatment, attenuated psychotic symptoms in eight participants (61.5%) in the ST group and six participants (46.2%) in the control group were reduced to a level of remission from an initial CHR status as defined by the SIPS. However, the difference was not significant (*X*<sup>2</sup> = 0.619, *P* = 0.431). One CHR individual in the control group developed an onset of the psychotic manic episode of bipolar I disorder during the follow-up period. The conversion rate was 3.8% (1/26).

#### DISCUSSION

The goals of this study were (1) to evaluate the effectiveness of systemic family therapy focusing specifically on the change in youth-attenuated psychotic symptoms and improvements in psychosocial functioning, and (2) to evaluate the effectiveness of systemic family therapy for CHR individuals in a non-clinical context. Most previous systemic intervention studies evaluated its impact on symptoms and treatment compliance. Although in clinical context to reduce the symptoms and enhance the

#### Table 3 | Comparison of changes between groups.

treatment compliance has a significant benefit for youth and families, it is important to know if ST enhances the psychosocial functioning level and the feasibility in non-clinical context. These issues provided the impetus for the present study. As far as we know, this trial is the first RCTs of ST for young people at CHR for psychosis. The trial targeted a non-clinical young sample, evaluated the treatment effect of ST, and employed an active control condition.