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Beyond Disease: Happiness, Goals, and Meanings among Persons with Multiple Sclerosis and Their Caregivers

Beyond Disease: Happiness, Goals, and Meanings among Persons with Multiple Sclerosis and Their... ORIGINAL RESEARCH published: 20 December 2017 doi: 10.3389/fpsyg.2017.02216 Beyond Disease: Happiness, Goals, and Meanings among Persons with Multiple Sclerosis and Their Caregivers 1 2 3 4 5 Antonella Delle Fave *, Marta Bassi , Beatrice Allegri , Sabina Cilia , Monica Falautano , 6 7 5 3 Benedetta Goretti , Monica Grobberio , Eleonora Minacapelli , Marianna Pattini , 8 7 6 9 Erika Pietrolongo , Manuela Valsecchi , Maria Pia Amato , Alessandra Lugaresi and Francesco Patti 1 2 Department of Pathophysiology and Transplantation, Università degli Studi di Milano, Milan, Italy, Department of Biomedical and Clinical Sciences “L.Sacco”, Università degli Studi di Milano, Milan, Italy, Neurology Unit, Multiple Sclerosis 4 5 Centre, Ospedale di Vaio-Fidenza, Fidenza, Italy, Ospedale Policlinico Universitario G. Rodolico, Catania, Italy, Neurological Department, San Raffaele Hospital (IRCCS), Milan, Italy, Department of NEUROFARBA, University of Florence, Florence, 7 8 Italy, Laboratory of Clinical Neuropsychology, ASST Lariana, Como, Italy, Department of Neurosciences, Imaging and Clinical Science, Università degli Studi ‘G. d’Annunzio’ Chieti - Pescara, Chieti, Italy, Department of Biomedical and Neuromotor Sciences, Università di Bologna, Bologna, Italy Edited by: Francisco José Eiroa-Orosa, The experience of persons with multiple sclerosis (MS) and their caregivers is usually University of Barcelona, Spain investigated in terms of emotional distress and health-related quality of life, while Reviewed by: David John Oliver, well-being indicators remain largely underexplored. In addition, findings are often University of Kent, United Kingdom interpreted from the clinical perspective, neglecting socio-cultural aspects that may Anna Fogdell-Hahn, crucially contribute to individuals’ functioning. At the methodological level, most Karolinska Institute (KI), Sweden Karin Zebenholzer, studies rely on scaled instruments, not allowing participants to freely express their Medical University of Vienna, Austria needs and resources. Based on the bio-psycho-social perspective endorsed by *Correspondence: the International Classification of Functioning, well-being indicators were investigated Antonella Delle Fave antonella.dellefave@unimi.it among 62 persons with MS (PwMS), their 62 caregivers and two control groups, matched by age and gender. Participants completed the Positive Affect Negative Specialty section: Affect Schedule (PANAS), the Satisfaction with Life Scale (SWLS), and the Eudaimonic This article was submitted to Clinical and Health Psychology, and Hedonic Happiness Investigation instrument (EHHI). EHHI provides information on a section of the journal participants’ happiness, goals and meanings through scaled and open-ended questions, Frontiers in Psychology contextualized within major life domains. No relevant differences emerged among Received: 30 September 2017 PwMS and caregivers, compared with the respective control groups, as concerns life Accepted: 07 December 2017 Published: 20 December 2017 domains associated with happiness, goals and meaning. Participants across groups Citation: prominently mentioned family, highlighting its intrinsic value and its relevance as a Delle Fave A, Bassi M, Allegri B, sharing context; health did not represent a major theme for PwMS; community, society Cilia S, Falautano M, Goretti B, Grobberio M, Minacapelli E, Pattini M, and religion/spirituality issues were substantially neglected by all participants. PwMS Pietrolongo E, Valsecchi M, and caregivers reported lower levels of positive affect than their control groups, while Amato MP, Lugaresi A and Patti F no substantial differences emerged for negative affect, happiness and meaningfulness (2017) Beyond Disease: Happiness, Goals, and Meanings among Persons levels in life and across most domains. Results suggest that the experience of MS is with Multiple Sclerosis and Their associated with well-being in relevant life domains, such as family and close relationships. Caregivers. Front. Psychol. 8:2216. doi: 10.3389/fpsyg.2017.02216 Although PwMS and caregivers identified a lower number of goals and meaning-related Frontiers in Psychology | www.frontiersin.org 1 December 2017 | Volume 8 | Article 2216 Delle Fave et al. Psychosocial Well-Being in Multiple Sclerosis 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 as well as material, social and institutional barriers. Despite these disadvantages, the mobilization of personal, relational The technological advancements in the medical domain and environmental resources allows these people to attain high fostered the representation and interpretation of health-related levels of mental health (Arnold et al., 2005; Andrykowski et al., phenomena through rigorously quantitative approaches, 2008; Delle Fave et al., 2015). Similarly, caregivers experience leading to the affirmation of the biomedical model. Despite the coexistence of caring burden and limitations in daily the shortcomings entailed by this narrow perspective, and the opportunities with psychological and social resources (Song and classical definition of health as “a state of complete physical, Singer, 2006; Olsson et al., 2008; Fianco et al., 2015). mental and social well-being” (World Health Organization, 1946, The study of mental health and well-being has received p. 1), both research and clinical practice are still permeated by a great impulse during the last two decades, through theories strictly biological view of human functioning. and empirical models deriving from two complementary A laudable effort to overcome this limitation and to perspectives. Within the hedonic perspective, well-being is promote the adoption of a bio-psycho-social view of health operationalized as the predominance of positive over negative and disease is represented by the International Classification affect in daily experience and a globally positive life evaluation, of Functioning, Disability and Health (ICF; World Health defined as satisfaction with life (Ryan and Deci, 2001). From Organization, 2001). ICF is aimed at comprehensively assessing the eudaimonic perspective well-being is instead conceptualized the physical, mental and social functioning of any individual, as a dynamic growth process, that includes a wide range including persons with diseases and disabilities. It presents a of constructs such as goal setting and pursuit, meaning- marked shift in terminology, from focusing on the consequences making, self-expressiveness, self-determination, self-acceptance, of disease (impairments, disabilities, handicaps) to investigating skill development and mastery, trust in relationships, and the components of health and functioning, classified as structures social integration (Huta and Waterman, 2014). Both these and functions, daily activities, and social participation. Far from approaches proved to be useful in exploring protective resources, endorsing a pure linguistic convention, the ICF stems from a adaptation processes and adjustment outcomes that allow relevant conceptual change, leading to the evaluation of health persons with chronic diseases and their caregivers to attain conditions from a constructive perspective. It is based on the positive functioning (Cummins, 2005; Diener and Chan, 2011). assumption of a dynamic interplay between individual features This avenue of research is however characterized by an (body functions, activities, and participation) and environmental emphasis on individual processes, while the societal and cultural aspects that can facilitate or hinder the person’s functioning factors influencing the person’s daily functioning are substantially (Dixon et al., 2008; Bodde and Seo, 2009). However, the neglected (Di Martino et al., 2017). Demographic and contextual implementation of this model into practice is problematic, features, when investigated, are treated as components of the as it requires a multidisciplinary integration effort involving person’s stable identity, despite the changes that both the researchers and practitioners from the healthcare, education and environment and the individual ceaselessly undergo (Slife and policy domains (Stucki et al., 2017). In addition, the assessment Richardson, 2008). of psychosocial functioning through the current version of ICF poses several challenges. Psychological and environmental The Individual and Family Experience of dimensions are collapsed into the heterogeneous domain of Multiple Sclerosis: Challenges and “contextual factors”; the psychological aspects are not explicitly identified through a checklist; and the components of community Resources participation are difficult to evaluate (Chang et al., 2013). Multiple sclerosis (MS) is a chronic neurodegenerative disease, Despite these limitations, only the ICF currently offers an characterized by largely unpredictable symptoms and course, integrated approach to human functioning which can be used by and currently lacking of curative treatment. Therefore, besides professionals working in different sectors. physical impairments, MS poses a number of psychological, From the ICF perspective, it is not surprising that people behavioral and social challenges to both affected persons and with chronic diseases or disabilities describe themselves as their caregivers (Dennison et al., 2009; Ackroyd et al., 2011). ordinary persons who cope with extraordinary circumstances The progressive course of the disease requires a constant re- (Saravanan et al., 2001) that include biological impairments adjustment over time (Bogosian et al., 2017), leading the person Frontiers in Psychology | www.frontiersin.org 2 December 2017 | Volume 8 | Article 2216 Delle Fave et al. Psychosocial Well-Being in Multiple Sclerosis to gradually scale back or adapt daily activities to the new reflect a condition of less severe illness (Dennison et al., condition, find new occupations and interests, but also disengage 2009). The assessment of demographic and clinical dimensions from the adjustment process and withdraw from active life. however entails methodological and interpretive challenges, While the negative consequences of MS were extensively as showed by the contrasting findings obtained in different explored, research on the positive aspects is still limited. Studies studies. For example, in some studies demographic variables have been conducted to investigate the eudaimonic process such as education and employment emerged as the strongest of meaning making, a key individual resource to attain well- predictors of health-related quality of life (HR-QoL), together being under unfavorable and irreversible conditions (Hicks and with clinical ones like depression and disability (Patti et al., King, 2009). Meaning making allows individuals to integrate 2003, 2007b). In other studies, HR-QoL itself emerged as an the problematic condition into a global and constructive view independent predictor of disability progression (Benito-Léon of their present and future life. The qualitative exploration of et al., 2013). Satisfaction with life was primarily predicted meaning making among persons with MS shed light on its by disease severity and social support in one study (Ryan components, which include disease acceptance, personal and et al., 2007), while in another study age and education level relationship growth, and positive lifestyle changes (Pakenham, emerged as the strongest predictors of hedonic and eudaimonic 2008a). Quantitative studies highlighted the positive association well-being (Bassi et al., 2014). Finally, a comparative study of sense making with life satisfaction, and its negative association did not highlight significant differences in personal growth (a with depression. Sense making was identified as a key predictor eudaimonic dimension) between persons with MS and healthy of positive adjustment also among caregivers (Pakenham, 2008b). participants, while life satisfaction (a hedonic dimension) was Similar findings were detected as concerns perceived illness significantly lower among the former (Barak and Achiron, 2011). coherence, a construct closely related to meaning making; The investigation of well-being among caregivers of persons illness coherence was positively associated with eudaimonic and with MS is even less frequent, as studies are primarily focused hedonic well-being indicators in both persons with MS and their on the emotional impact of caring-related burden and stress caregivers (Bassi et al., 2016). (Patti et al., 2007a; Rivera-Navarro et al., 2009), and on the Benefit finding, consisting in the ability to identify positive daily choices and free time restrictions imposed by the caring consequences in an otherwise negative situation (Lechner et al., role (Becker, 2011). Studies investigating satisfaction with life 2009), was also investigated among persons with MS and did not detect differences between caregivers of persons with their family caregivers. Cross-sectional and longitudinal studies MS and samples derived from the general population or carers highlighted its positive association with meaning-based coping of persons with other chronic diseases (Waldron-Perrine et al., strategies, and its direct effects on positive adjustment outcomes, 2009; Bassi et al., 2014). Eudaimonic well-being dimensions such such as good dyadic relationships and subjective well-being as meaning making, benefit finding and illness coherence were (Pakenham, 2005, 2007, 2008b; Pakenham and Cox, 2009). more often investigated among caregivers, as reported in the Related findings support the idea of a “communal search for previous paragraphs (Pakenham, 2007; Pakenham and Cox, 2009; meaning” where persons with MS and their caregivers experience Bassi et al., 2016). In a recent qualitative study involving Turkish the trauma of a chronic illness and subsequently find positive caregivers (Topcu et al., 2016), participants mentioned both aspects together (Pakenham, 2005). Further evidence of this individual and social resources, such as motivation to care and dyadic process emerged from the investigation of post-traumatic perceived support. growth and illness perceptions among persons with MS and their Acknowledging the existence and adaptive role of well- partners (Ackroyd et al., 2011). being dimensions in the experience of MS surely represents These studies also showed that the disruption of life goals an advancement. At the same time, most studies suffer from caused by MS progression can be counterbalanced by the two conceptual limitations. The first one is the lack of a development of new meanings and life purposes (Joseph comprehensive bio-psycho-social perspective, and especially the and Linley, 2006). This process entails a complex interplay neglect of the societal context surrounding persons with MS between disengagement from previous goals and reengagement and their caregivers. Only few studies were conducted within in new ones (Neter et al., 2009). It also requires the the ICF framework. One of them was aimed at identifying implementation of adaptive tasks (Bensing et al., 2002), such as a core set of ICF categories specific to MS that could be defining new challenges, acknowledging one’s limits, maintaining used in healthcare practice (Khan and Pallant, 2007). Another emotional balance and self-esteem, facing uncertain future, study proposed the Multiple Sclerosis Impact Profile (MSIP; cultivating social relationships, and looking at the bright Wynia et al., 2008) an instrument designed to evaluate the side of life. Besides these individual resources, other factors perceived impact of MS on functioning. A recent review (Dorstyn located at the intersection between persons and their context et al., 2017) highlighted the association between depressive may foster positive adaptation; they include facilitators of symptoms and reduced social participation in persons with mobility/independence, social support, and social comparisons MS. A qualitative study exploring the impact of ICF contextual (Dilorenzo et al., 2008). factors on the daily functioning of Jordanian persons with MS A frequent problem in studies investigating well-being and (Hamed et al., 2012) identified adequacy of financial and medical perceived quality of life among persons with MS is the lack of resources, religion and community awareness as facilitators, objective measures of disease severity; this limitation prevents and social stigma as a barrier. Finally, the environmental and researchers to exclude that positive adjustment may simply personal facilitators of social participation and satisfaction with Frontiers in Psychology | www.frontiersin.org 3 December 2017 | Volume 8 | Article 2216 Delle Fave et al. Psychosocial Well-Being in Multiple Sclerosis parenting were investigated among mothers with MS (Farber functioning (World Health Organization, 2002; Martins, 2015). et al., 2015). Findings suggested the importance of incorporating The level of disability was also taken into consideration in the both categories of facilitators into treatment. The relevance analysis of data collected among persons with MS. of environmental factors also emerged from the investigation Based on the available literature, some hypotheses were of unmet needs among persons with MS. These needs most formulated. In line with studies on adjustment to MS (Pakenham, often refer to contextual dimensions, such as family and social 2008a; Bassi et al., 2016) we expected that, when describing support, healthcare services, everyday life management, and the happiness, goals and meaningful things, a significantly higher relationship with physicians (Galushko et al., 2014). Interestingly, percentage of persons with MS would refer to health compared representations of the unmet needs of persons with MS differ with a control group. We also expected persons with MS to among health professionals (Golla et al., 2012): while physicians associate health with significantly lower levels of happiness and emphasize limitations in access to care due to poor financial higher levels of meaningfulness, compared with a control group. resources, nurses and social workers are more aligned with As concerns caregivers, based on previous evidence (Pakenham, patients’ perceptions, quoting family support, social relationships 2008b; Becker, 2011; Mausbach et al., 2011) we expected that and daily life management. a significantly higher percentage of participants would refer A second limitation characterizing the literature on MS to family and a lower percentage to leisure, compared with a is the emphasis on disease and related adjustment processes: control group. We also expected caregivers to report significantly participants and caregivers are rarely solicited to freely reflect lower levels of happiness with leisure, and higher levels of on aspects of their life and daily experience potentially unrelated meaningfulness in relation to family. As concerns ratings of to disease or caregiving respectively. This limitation emerged in affect and life satisfaction (the two components of hedonic well- one of the studies investigating unmet needs of persons with MS being), the contrasting findings detected across studies (Barak (Galushko et al., 2014): participants expressed the need to be and Achiron, 2011; Diener et al., 2017) did not allow us to viewed as distinct individuals, not constrained by and identified formulate specific hypotheses. Finally, based on the literature through their health conditions. highlighting the relevant role of education and employment in predicting individual well-being (Keyes, 2007; Patti et al., The Present Study 2007b; Diener et al., 2017), we hypothesized that these two The first aim of the present study was to investigate different demographic features would provide a specific contribution to dimensions of well-being among persons with MS and their hedonic and eudaimonic well-being values across all groups. The caregivers through a mixed method approach and from a same hypothesis was formulated as concerns the role of severity bio-psycho-social, ICF informed perspective. Answers obtained disease among persons with MS. through open-ended and scaled questions were jointly analyzed, in the attempt to contextualize findings within participants’ MATERIALS AND METHOD global outlook of their own life and environmental opportunities. The well-being dimensions investigated in the study included Participants happiness (its definition and recent related situations), hedonic The study involved 248 Italian adults divided into four groups, well-being (positive and negative affect, satisfaction with life) each of them comprising 62 participants: persons with MS and eudaimonic well-being (perceived meaningful things and (PwMS), their caregivers, and two control groups of adults with goals). In addition, information was collected on happiness no history of chronic disease or caregiving experience, selected and meaningfulness levels in life in general, as well as in the from a larger study aimed at investigating well-being in the specific domains of work, family, standard of living, interpersonal general population. Inclusion criteria for PwMS were being at relations, health, personal growth, leisure, spirituality/religion, least 18 years of age, having a clinically definite MS diagnosis community, and society. Overall, the mixed method potentials for at least 3 years and having a caregiver; exclusion criteria are still underexploited in the psychological literature; moreover, comprised the presence of additional neurological or psychiatric to the best of our knowledge no studies have yet been conducted disorders, severe cognitive impairment, MS in the active phase, on these topics and through this methodological approach in the and a condition of very severe disability, corresponding to a MS domain. score above 8 on the Extended Disability Status Scale (EDSS; The second aim was to compare the findings collected among Kurtzke, 1983). The majority of PwMS involved in the study persons with MS and their caregivers with those obtained from (59.68%) showed an EDSS score between 3.5 and 8 (indicating two groups of participants, matched by age and gender, but with increasing levels of motor impairment), while 40.32% scored no history of chronic disease or caregiving experience. From an below 3.5 (indicating a high level of autonomy). No significant ICF informed perspective, this comparison was expected to shed differences were detected between PwMS with low and high levels light on group differences in perceived personal and contextual of disability for any of the variables examined in this study; facets of well-being, including daily activities and opportunities, therefore disability level will not be further considered in the relational networks, and social participation. Attention was also following sections. paid to the role of employment status and education level Caregivers were predominantly partners (59.67%) or parents in predicting well-being dimensions. These two demographic (22.57%) of PwMS, siblings or children (each accounting for features represent crucial indicators of participation, classified 6.46%), friends (3.23%), or professional caregivers (1 participant, in the ICF as contextual personal factors influencing human 1.61%). Participants in the control groups were randomly Frontiers in Psychology | www.frontiersin.org 4 December 2017 | Volume 8 | Article 2216 Delle Fave et al. Psychosocial Well-Being in Multiple Sclerosis extracted from a sample of 691 healthy Italian adults; age, gender associated to 10 major life domains (work, family, standard of and education level were used as filters to match the control living, interpersonal relations, health, personal growth, leisure, groups with the respective comparison groups – PwMS and spirituality/religion, community, and society) and to life in caregivers. It was however not possible to obtain a complete general. match between caregivers and their control group as concerns Satisfaction with Life Scale (SWLS; Diener et al., 1985; education level. The decision to include two different control Goldwurm et al., 2004). This widely used measure of well-being groups was based on the significant age difference between PwMS comprises five items on scales ranging from 1 (strongly disagree) and their caregivers (t = 2.85, p < 0.005). The demographic to 7 (strongly agree). The items investigate the individuals’ features of the four groups of participants are reported in Table 1. overall cognitive evaluation of their global life conditions and No differences emerged between PwMS and their control achievements. group (control 1) for age, gender, marital status and education, Positive Affect and Negative Affect Schedule (PANAS; Watson while a significantly higher percentage of PwMS was unemployed et al., 1988; Terracciano et al., 2003). The instrument assesses the (χ = 21.81, p < 0.001). As concerns caregivers and their control overall perceived intensity of positive and negative affect during group (control 2), differences were detected for employment and daily life, through 10 items measuring components of positive education, both higher in the control group (χ = 7.52, p < 0.01, affect (PA) and 10 items measuring components of negative and χ = 4.61, p < 0.05 respectively). affect (NA). Scales range from 1 (very slightly or not at all) to 5 (extremely). Materials Procedure Data were collected through the following instruments: This study involved seven different academic and clinical A socio-demographic questionnaire provided information on institutions; therefore, the protocol was submitted to the participants’ age, employment status, education level, and civil ethical committees of each institution. After approval from all status. Clinical information including time from disease onset, committees, participants with MS and their caregivers were level of disability, type of treatment and co-morbidities was recruited at six MS centers in different Italian regions, in the collected for PwMS. Caregivers also reported their type of bond context of a broader project aimed at investigating clinical, with PwMS. psychological and relational aspects of MS (Bassi et al., 2014, Eudaimonic and Hedonic Happiness Investigation inventory 2016). PwMS were contacted by the centers’ personnel during (EHHI; Delle Fave et al., 2011, 2016). This mixed-method check-ups or by phone, and were invited to identify their primary instrument allows researchers to collect qualitative and caregiver. Data from participants in the two control groups were quantitative information on different components of well-being. selected from a larger study that had been approved by the ethical Through open-ended questions, participants are asked to define committee of the first author’s institution. happiness in their own words; to list the three future goals they Upon their expression of interest in joining the study, consider most important, and the three things they consider participants received detailed information on the project from most meaningful in their present life; and to briefly describe a researcher involved in the study. They signed an informed three situations associated with intense happiness during the consent in compliance with Italian privacy rules, and were last 6 months. In addition, participants are asked to rate on provided with the battery of questionnaires. They could inspect two sets of scaled items, ranging from 1 (extremely low) to 7 the questionnaires, pose general and specific questions to the (extremely high), the levels of happiness and meaningfulness 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 TABLE 1 | Demographic characteristics of the four groups. place convenient to them. They could hand in their responses, or PwMS Control 1 Caregivers Control 2 send them via mail. Before data processing researchers removed (N = 62) (N = 62) (N = 62) (N = 62) the consent form from each battery of questionnaires, thus guaranteeing participants’ anonymity throughout the phases of Age 40.1 ± 9.7 40.5 ± 10.9 45.7 ± 12.0 45.7 ± 11.4 data coding, storing and analysis. Data were stored in password Age range 21–63 20–60 19–81 22–81 protected computers. Participation to both the MS related study Gender (% Female) 69.35 69.35 58.06 56.45 and the general survey was voluntary, and participants were EDUCATION (%) free to leave the study at any time. Persons with MS were High school or less 82.26 75.81 85.48 69.35 explicitly assured that refusal to participate or withdrawal from University 17.74 24.19 14.52 30.65 participation in the study would in no way interfere with the Work/study (%) 57.38 93.55 75.81 93.55 long-term healthcare services they were receiving at the MS CIVIL STATUS (%) center. Married/cohabiting 66.13 51.61 77.42 72.13 Single/divorced/widowed 33.87 48.39 22.58 27.87 Coding Procedure Answers to the open-ended questions of the EHHI required an Means and Standard Deviations are reported. b accurate coding work, using a coding system originally developed Education was dichotomized: “High school or less” includes elementary, middle, and high school; “University” includes graduation, post-graduation and PhD. by Delle Fave et al. (2011), and gradually expanded through Frontiers in Psychology | www.frontiersin.org 5 December 2017 | Volume 8 | Article 2216 Delle Fave et al. Psychosocial Well-Being in Multiple Sclerosis additions from various studies, in line with the bottom-up represent the positive and meaningful sides of this life domain approach guiding the EHHI research project. In this coding (Delle Fave et al., 2013a). system, answers are grouped into broad categories corresponding to the major life domains: work, family, standard of living, Statistical Analyses interpersonal relationships, health, personal/psychological life, The analysis of qualitative data collected through the EHHI was leisure, spirituality/religion, society and community issues, and aimed at identifying the life domains predominantly mentioned more general/unspecific life aspects. Multifaceted categories by participants across groups in their descriptions of happiness, such as work, family, relationships, leisure, spirituality/religion, major goals, meaningful things, and recent sources of intense community/society and personal life are further subdivided happiness. Results were compared between groups (respectively into more fine-grained sub-categories (Delle Fave et al., PwMS and control 1, and caregivers and control 2). Since a 2013a). The categories family and interpersonal relationships high percentage of participants across groups indicated family were organized into the subcategories intrinsic value (e.g., as prominent goal, meaningful thing, and source of recent having a family; a partner; children; friends), sharing (e.g., happiness, analyses were performed on family sub-categories. spending time with children; sharing life projects with partner; The same in-depth analysis was conducted for the psychological sharing good and bad experiences with friends), personal definitions of happiness. contribution (caring for elder parents; raising children; helping Coded answers were first grouped into the corresponding friends in need), family well-being/harmony, balance (e.g., categories and sub-categories; subsequently, considering that health of family members; children’s positive development; each participant provided more than one answer for each harmony in relationships) and personal reward (e.g., gratification question, the number of participants reporting at least one from partner, children; gratitude expressions from siblings; answer in each category and subcategory was calculated across satisfaction with relations). A similar partition characterizes groups. This approach allowed us to compare the percentage of community/society issues. In the psychological/personal life participants referring to each answer category and subcategory category, some subcategories refer to eudaimonic well-being between groups through 2 × 2 frequency tables by means of the dimensions (growth/engagement, purpose, competence/mastery, χ procedure. The procedure was not considered as reliable when autonomy, self-actualization, meaning/value, harmony/balance, the number of participants in one or more cells was below 5. fullness/awareness, optimism), and others to hedonic well- Through Spearman coefficients, correlations were then calculated being ones (satisfaction/achievement, positive emotions, and between participants’ distribution in answer categories and their absence of negative feelings); a less specific subcategory, labeled demographic and group features. Logistic regression analyses allowed us to verify whether demographic or group features as “positive experiences/states”, includes answers generically referring to happiness as “inner well-being”, “a stable state”, or predicted a specific pattern of answers. “a way of being.” Quantitative data were first analyzed using descriptive The coding procedure for each answer comprises first the statistics. Correlations of hedonic and eudaimonic dimensions identification of a category in which the answer can be of well-being with participants’ group and demographic features included, and then a specific numeric item to which the were calculated through Pearson and Spearman coefficients. answer can correspond, if available. If an answer content does Hierarchical regression analyses provided information on the not fit any available item, a new item will be added to the specific contribution of participants’ group and demographic category. As concerns happiness definitions, participants often features to well-being dimensions. report complex descriptions including different components of Given the large number of group comparisons performed happiness. Each of these facets is treated as a specific semantic on quantitative and qualitative variables, we took a Bonferroni unit and coded separately; up to six answer units are retained approach, adjusting the critical alpha value for significance to the for each participant. Based on the specific formulation of number of t-tests and χ comparisons performed on the same the questions concerning goals, meaningful things, and recent dataset (25 and 61 respectively). More specifically, to achieve α < situations of most intense happiness, three answer units are 0.05 with 25 t-tests, the alphas obtained from each data set had retained for each question. In the present study, reliability in the to score below 0.002 (α < 0.05/25, two-tailed); in order to reject coding process was established through the involvement of two a null hypothesis, the test statistic had to exceed critical t = 3.16, expert coders providing independent ratings for each answer; corresponding to p < 0.002 with 122 degrees of freedom (for two divergences were clarified through a subsequent discussion; groups, df = N participants-2). unsolved discrepancies and answers potentially requiring the The same approach was used for frequency table comparisons inclusion of new items in the coding system were further (N = 61 in each data set). To achieve α < 0.05 with 61 discussed with the first author. comparisons, the alphas obtained from the data set had to As concerns the scaled questions included in the EHHI, and score below 0.0008 (α < 0.05/61); in order to reject a null the data collected through PANAS and SWLS, the numeric hypothesis, the test statistic had to exceed critical χ = 11.34, values corresponding to the perceived levels of each variable corresponding to p < 0.0008 with 1 degree of freedom, since df = were reported. The EHHI items assessing the levels of happiness (N columns-1) (N rows-1). As for correlations, regardless of the and meaningfulness associated with personal/psychological life statistical significance only coefficient values equal to or higher are labeled as “personal growth”; this decision was originally than 0.30 were considered as adequate to interpret associations based on the assumption that growth and development could best as meaningful (Hinkle et al., 2003). More specifically, values Frontiers in Psychology | www.frontiersin.org 6 December 2017 | Volume 8 | Article 2216 Delle Fave et al. Psychosocial Well-Being in Multiple Sclerosis TABLE 2 | Definitions of happiness: percentage of participants mentioning each between 0.30 and 0.50 indicated low correlation, values between category and subcategory by group, and comparisons between groups. 0.50 and 0.70 moderate correlation, and values above 0.70 high correlation. 2 2 PwMS Control 1 χ Caregivers Control 2 χ Psychological definitions 54.72 93.33 22.51** 66.67 80.00 2.61 RESULTS Harmony/balance 68.97 60.71 0.56 63.89 72.92 0.78 Other eudaimonic 24.14 39.29 1.95 25.00 41.67 2.52 This section illustrates the qualitative and quantitative findings Hedonic 41.38 46.43 0.19 27.78 33.33 0.30 presented for each group separately, in order to allow for Positive states 10.34 10.71 2.78 8.33 comparisons. Family 45.28 15.00 12.48* 51.85 28.33 6.58 Intrinsic value 28.00 11.11 13.79 11.76 Qualitative Findings Sharing 48.00 55.56 0.69 44.83 58.82 0.83 The findings obtained from the open-ended questions of the Personal contribution 4.00 11.11 10.34 0 EHHI are presented as percentages of participants across groups Family well-being 12.00 22.22 34.48 23.53 Personal reward 28.00 22.22 20.69 23.53 who provided at least one answer in the different categories and Interpersonal relations 15.09 23.33 1.22 12.96 30.00 4.82 subcategories. The answers referring to psychological dimensions Health 11.32 8.33 0.29 12.96 15.00 0.10 are grouped into the category labeled as “psychological Work 11.32 1.67 5.56 8.33 definitions” for the definitions of happiness, and into the category Standard of living 1.89 1.67 7.41 3.33 “personal life” for goals, meaningful things and recent situations Leisure 5.66 0 0 1.67 of intense happiness. Spirituality, Religion 3.77 1.67 3.70 6.67 Community, Society 7.55 5.00 0.00 3.33 Happiness Definitions Life in general 0 13.33 3.70 8.33 Participants were invited to answer the question “What is N participants 53 60 54 60 happiness for you?” in their own words, without specific Each participant could provide more than one answer; Bonferroni adjusted alpha constraints. It is worth noticing that 9 PwMS and 8 caregivers *p < 0.05; **p < 0.01; χ -values are not reported if the no. of participants in one or either did not provide any answer, or stated that “happiness more cells falls below 5. 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 Goals and Meaningful Things (2.65 and 2.80 respectively) compared with PwMS and caregivers While all participants in the control groups identified some (2.25 and 2.19 respectively). These differences were however important future goals, 2 PwMS and 4 caregivers did not. In not statistically significant. Table 2 shows for each group the addition, 59 participants in control 1 reported up to three goals percentage distribution of the participants who provided at least compared with 45 PwMS (χ = 11.68, p < 0.05); similarly, 59 one answer in the different categories and subcategories. Results participants in control 2 reported up to three goals compared of group comparisons are also reported. with 44 caregivers (χ = 12.89, p < 0.05). Table 3 depicts the Overall, a substantial similarity emerged between groups percentage distribution of the participants who provided at least across most categories. Only two significant differences were one answer across categories in each group. detected between PwMS and control 1: a lower percentage The majority of participants in all groups mentioned family, of PwMS reported psychological definitions of happiness, specifically referring to its intrinsic value (having a family; and a lower percentage of controls reported family-related having or adopting children; having grandchildren; finding ones. Despite these overarching differences, the percentage the right partner) and to the well-being of family members distribution of participants across subcategories of both (physical health, self-actualization or goal achievement of psychological and family-related definitions did not differ children, grandchildren, partner, siblings, and parents). Health, between the two groups. Among psychological definitions, work, and personal life were mentioned by lower percentages participants predominantly referred to eudaimonic constructs, of participants; spirituality/religion and community/society were such as harmony/balance, self-actualization, personal growth, marginally represented, together with interpersonal relations. and optimism. Since a remarkably high percentage of participants The only significant difference concerned the higher percentage specifically mentioned harmony/balance, related findings are of PwMS mentioning health, compared to control 1. reported separately in Table 2. As concerns family, most As regards the most meaningful things in the present participants in both groups provided answers related to sharing life, 7 caregivers did not identify any, compared to 4 PwMS (experiences, activities, projects). Personal rewards followed and no participants in the control groups (χ = 13.22, as subcategory; answers included receiving love expressions, p < 0.05). Moreover, 60 participants in control 2 reported up support, acknowledgment from family members, and satisfaction to three meaningful things, compared with only 46 caregivers with family. Fewer participants quoted the other domains, and (χ = 12.74, p < 0.05). Nevertheless, as illustrated in Table 4, a negligible percentage in both groups mentioned leisure, participants’ percentage distribution across answer categories was spirituality/religion, community/society issues and standard of largely overlapping across groups. living. A similar answer pattern was detected among caregivers Participants in all groups almost unanimously quoted and control 2, but no significant group differences emerged. family as one of the most meaningful things in their lives Frontiers in Psychology | www.frontiersin.org 7 December 2017 | Volume 8 | Article 2216 Delle Fave et al. Psychosocial Well-Being in Multiple Sclerosis TABLE 3 | The most important future goals: percentage of participants TABLE 4 | The most meaningful things in present life: percentage of participants mentioning each answer category by group and comparisons between groups. mentioning each answer category by group and comparisons between groups. 2 2 2 2 PwMS Control 1 χ Caregivers Control 2 χ PwMS Control 1 χ Caregivers Control 2 χ Personal life 26.67 38.71 2.00 15.52 20.97 0.59 Personal life 12.07 20.97 1.71 25.45 17.74 1.03 Family 68.33 67.74 0.004 79.31 64.52 3.23 Family 91.38 79.03 3.58 83.64 82.26 0.04 Intrinsic value 51.22 52.38 0.01 39.13 40.00 0.007 Intrinsic value 86.79 83.67 0.20 78.26 84.31 0.59 Sharing 14.63 4.76 10.87 25.00 2.96 Sharing 1.89 12.24 13.04 5.84 Personal contribution 12.20 23.81 1.89 23.91 12.50 1.84 Personal contribution 1.89 6.12 6.52 3.92 Family well-being 31.71 35.71 0.15 43.48 45.00 0.02 Family well-being 7.55 0 6.52 7.84 Personal reward 4.88 0 0 2.50 Personal reward 7.55 2.04 2.17 1.96 Interpersonal relations 5.00 6.45 1.72 6.45 Interpersonal Relations 27.59 40.32 2.16 23.64 38.71 3.06 Health 53.33 22.58 12.28* 39.66 32.26 0.71 Health 48.28 30.65 3.90 32.73 33.87 0.02 Work 45.00 59.68 2.63 37.93 58.06 4.86 Work 43.10 30.65 2.00 43.64 50.00 0.47 Standard of living 6.67 16.13 2.69 29.31 22.58 0.71 Standard of living 5.17 12.90 9.09 14.52 Leisure 10.00 19.35 2.12 8.62 16.13 1.54 Leisure 5.17 17.74 3.64 9.68 Spirituality, Religion 3.33 0.00 3.45 6.45 Spirituality, Religion 8.62 6.45 5.45 9.68 Community, Society 3.33 3.23 0.00 14.52 Community, Society 5.17 11.29 3.64 1.61 Life in general 3.33 12.90 6.90 14.52 Life in general 0 13.33 5.45 4.84 0 a a N participants 60 62 58 62 N participants 58 62 55 62 a 2 Each participant could provide more than one answer; Bonferroni adjusted alpha Each participant could provide more than one answer; χ -values are not reported if the *p < 0.05; χ -values are not reported if the no. of participants in one or more cells falls no. of participants in one or more cells falls below 5. below 5. TABLE 5 | Recent situations of intense happiness: percentage of participants mentioning each answer category by group and comparisons between groups. (specifically referring to its intrinsic value); progressively 2 2 PwMS Control 1 χ Caregivers Control 2 χ lower percentages of participants mentioned work, health, interpersonal relationships and personal life, without significant Personal life 6.00 8.33 0 10.53 group differences. Spirituality/religion, leisure, standard of living Family 74.00 73.33 0.006 82.05 78.95 0.14 and community/society were mentioned by less than 10% of the Intrinsic value 10.81 22.73 1.99 12.50 15.56 participants across groups. Sharing 70.27 68.18 0.04 56.25 46.67 0.69 Personal contribution 0 4.55 6.25 0 Family well-being 35.14 29.55 0.29 50.00 55.56 0.23 Recent Situations of Intense Happiness Personal reward 13.31 13.64 3.13 11.11 As illustrated in Table 5, only 39 caregivers (62.9%) provided Interpersonal relations 22.00 36.67 2.79 20.51 22.81 0.07 Health 18.00 3.33 6.52 5.13 1.75 answers to this question, while the remaining 23 (37.10%) could Work 20.00 21.67 0.05 12.82 29.82 3.79 not remember any recent situation of intense happiness. This Standard of living 4.00 6.67 7.69 10.53 answer distribution pattern was significantly different from those 2 Leisure 34.00 31.67 0.08 38.46 33.33 0.27 detected for the other three groups (χ = 29.93, p < 0.001): only N participants 50 60 39 57 12 participants (19.35%) among PwMs, 5 (8.1%) in control 2 and a 2 2 (3.23%) in control 1 did not report recent situations of intense Each participant could provide more than one answer; χ -values were not reported if one or more cells included less than 5 participants; answer categories cited by less than happiness. 5 participants across groups were excluded from analysis. 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 Quantitative Findings participants across groups referred to sharing positive events and experiences, such as anniversaries and other celebrations. Ratings of Happiness, Meaningfulness, and Hedonic A lower percentage of participants mentioned events promoting Well-Being the well-being of family members (positive school and work Table 6 shows the mean ratings of happiness and meaningfulness achievements, disease recovery or health improvements). Leisure across groups, and the results of the comparisons between PwMS predominantly included the practice of arts and crafts, sports, and control 1, and between caregivers and control 2. travels and media fruition. Interpersonal relationships and Participants across groups reported the highest levels of work followed in rank across groups. A higher percentage of happiness and meaningfulness in the domains of family, health, PwMS referred to health related situations, but after Bonferroni interpersonal relations, life in general, and personal growth adjustment the difference with control 1 was not significant. (with slight variations in domain order across groups). In Spirituality/religion and community/society were again reported contrast, spirituality/religion, community and society issues were by a negligible percentage of participants across groups, together associated with the lowest levels of happiness and meaningfulness with personal life and standard of living. across groups. The only difference between PwMs and control Frontiers in Psychology | www.frontiersin.org 8 December 2017 | Volume 8 | Article 2216 Delle Fave et al. Psychosocial Well-Being in Multiple Sclerosis 1 concerned happiness with health, with the former reporting issues, and they attributed lower meaningfulness to the domain significantly lower values. The comparison between caregivers of leisure. and control 2 highlighted two significant differences: Caregivers More relevant differences were detected between groups reported higher values of happiness in relation to community 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 TABLE 6 | Levels of happiness and meaningfulness in life domains and among PwMS than among participants in control 1. No group comparisons between groups. differences were instead detected for negative affect. PwMS Control 1 t Caregivers Control 2 t Demographic and Group Predictors of Well-Being M (sd) M (sd) M (sd) M (sd) Dimensions HAPPINESS Correlations and regression models were finally calculated within Work 4.50 4.47 0.11 4.25 4.56 1.13 each group set-PwMS and control 1, and caregivers and control (1.96) (1.31) (1.72) (1.25) 2 respectively -, in order to investigate the role of group type and Family 5.90 5.60 1.18 5.56 5.84 1.13 demographic features as predictors of qualitative and quantitative (1.47) (1.34) (1.35) (1.20) evaluations of well-being. Age, gender and marital status were Standard liv. 4.74 4.90 0.69 4.41 4.88 2.00 not considered, as these characteristics did not differ between (1.34) (1.30) (1.28) (1.28) PwMS and control 1, and between caregivers and control 2. Relations 5.00 5.10 0.37 4.70 5.33 2.75 Attention was instead paid to education level and employment (1.55) (1.34) (1.42) (1.06) status, as group differences were detected for these two features, Health 4.37 5.45 4.17** 5.21 5.54 1.40 (1.72) (1.10) (1.14) (1.30) as reported in the comments on Table 1. Table 8 illustrates Personal Growth 5.05 4.92 0.52 4.87 5.18 1.31 Spearman correlations among predictors (demographics and (1.41) (1.35) (1.43) (1.16) group classification) and between predictors and well-being Leisure 4.15 4.66 1.96 3.98 4.41 1.41 dimensions. (1.48) (1.45) (1.72) (1.66) For the sake of synthesis, analyses were performed only for Spirituality 3.93 4.04 0.29 4.02 4.89 2.60 the dimensions showing significant differences between groups. (2.09) (1.70) (2.04) (1.56) As reported in the method section, correlations higher than Community 3.64 4.23 2.18 3.56 4.49 3.29* 0.30 were deemed as meaningful. Overall, group classification (1.41) (1.44) (1.67) (1.31) showed the most numerous and strongest correlations with Society 3.71 3.62 0.29 3.70 3.72 0.08 the well-being dimensions. In the PwMS/control 1 group set, (1.60) (1.43) (1.73) (1.20) only employment status showed a low positive correlation with Life in general 5.03 5.08 0.22 5.10 5.31 1.05 satisfaction with life; together with group classification, it showed (1.31) (1.12) (1.25) (0.93) a low positive correlation with happiness with health as well. MEANINGFULNESS No meaningful correlations were instead identified for education Work 5.28 5.21 0.24 5.36 5.66 1.12 (1.76) (1.29) (1.67) (1.23) level. In the caregivers/control 2 group set, group type showed Family 6.45 6.58 0.75 6.72 6.69 0.19 low to moderate correlations with most well-being dimensions, (1.08) (0.82) (0.77) (0.82) except for satisfaction with life and negative affect. A low positive Standard liv. 5.15 5.03 0.50 4.93 5.29 1.60 correlation emerged between education level and positive affect, (1.34) (1.19) (1.28) (1.19) while employment status did not show any relevant correlation Relations 5.56 6.02 2.13 5.34 5.97 3.10 with well-being dimensions. (1.35) (0.98) (1.28) (0.96) Health 6.32 6.44 0.67 6.53 6.81 2.19 (1.02) (0.84) (0.88) (0.44) Personal Growth 5.74 5.91 0.85 5.51 5.90 1.64 TABLE 7 | Levels of affective and cognitive dimensions of hedonic well-being, and (1.25) (1.08) (1.37) (1.31) their comparison between groups. Leisure 4.87 5.10 0.93 4.55 5.55 4.48** (1.52) (1.17) (1.42) (1.04) PwMS Control 1 t Caregivers Control 2 t Spirituality 4.01 4.00 0.04 4.61 5.08 1.35 M (sd) M (sd) M (sd) M (sd) (2.12) (2.12) (2.06) (1.79) Community 4.26 4.45 0.70 4.19 4.64 1.63 Positive affect 3.01 3.59 4.62** 3.01 3.87 7.83** (1.37) (1.67) (1.60) (1.45) (0.81) (0.57) (0.71) (0.48) Society 4.29 4.25 0.14 4.26 4.88 2.53 Negative affect 2.15 2.51 2.29 2.03 2.22 1.35 (1.54) (1.62) (1.68) (0.99) (0.93) (0.82) (0.89) (0.66) Life in general 5.98 6.08 0.57 5.90 6.27 1.93 Satisfaction with life 3.80 4.65 3.30* 4.14 4.87 3.06 (1.06) (0.80) (1.25) (0.85) (1.55) (1.30) (1.51) (1.15) N participants 62 62 62 62 N participants 62 62 62 62 Bonferroni adjusted alpha *p < 0.05; **p < 0.01. Bonferroni adjusted alpha *p < 0.05; **p < 0.01. Frontiers in Psychology | www.frontiersin.org 9 December 2017 | Volume 8 | Article 2216 Delle Fave et al. Psychosocial Well-Being in Multiple Sclerosis TABLE 8 | Correlations among demographic and group predictors, and between definitions was predicted by group type only (B = −1.27, OR = predictors and variables showing significant differences in the two group sets. 2 0.079, Wald χ = 22.63, p < 0.001; and B = 0.67, OR = 3.820, Wald χ = 9.04, p < 0.01 respectively). Group type was also the Employment Education Group sole predictor of health-related goals distribution (B = 0.66, OR PwMS/CONTROL 1 = 3.783, Wald χ = 11.20, p < 0.001). In the caregivers/control 2 group set, positive affect was Employment – 0.11 0.42*** significantly predicted by group type and education level, Education 0.11 – 0.08 together explaining 36.9% of the variable variance (r = 0.334, Positive affect 0.28** 0.22* 0.39*** F = 61.25, p < 0.001, and r = 0.035, F = 6.75, p < 0.05 Negative affect −0.004 −0.15 0.20* respectively); no significant predictors were instead identified Satisfaction with life 0.35*** 0.24** 0.29** for negative affect. Group type emerged as the only significant Happiness with health 0.31* 0.05 0.33* predictor for the three remaining variables: satisfaction with life Happiness def.—psychological 0.16 0.13 0.49*** (r = 0.071, F = 9.36, p < 0.01, explaining 2.2% of the variable Happiness def.—family −0.04 −0.09 −0.29** variance); happiness with community (r = 0.088, F = 10.80, Goals—health −0.19* −0.03 −0.30*** p < 0.01, explaining 2.6% of the variable variance) and leisure CAREGIVERS/CONTROL 2 meaningfulness (r = 0.141, F = 20.04, p < 0.001, explaining Employment – 0.12 0.25 4.2% of the variable variance). Education 0.12 – 0.19* Positive affect 0.26** 0.30*** 0.58*** Negative affect 0.11 0.03 0.12 DISCUSSION Satisfaction with life 0.16 0.15 0.27** Happiness with community 0.06 0.21* 0.30** This study aimed at investigating different facets of well-being Leisure meaningfulness 0.23** 0.12 0.38*** among persons with MS and their caregivers from an integrated perspective. At the conceptual level, the eudaimonic dimensions *p < 0.05; **p < 0.01; ***p < 0.001. “Group” is a dummy variable with 1 for PwMS or of goal pursuit and meaning-making were jointly investigated caregivers and 0 for the respective control groups. 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 Based on these results, linear regressions with stepwise life domains, and findings were compared with those reported by selection were carried out to investigate the role of employment two groups of participants sharing similar demographic features, status, education level and group classification as predictors but not exposed to experiences of chronic illness or caregiving. of the hedonic well-being dimensions: positive affect, negative At the empirical level, the adoption of a mixed method approach affect, and satisfaction with life. In addition, linear regressions gave participants the opportunity to freely describe their present (with scaled outcome variables) and binary logistic regressions evaluations and future expectations. (with categorical outcome variables) were performed to assess the role of demographic features in the findings obtained through The Private Context of Well-Being: A the EHHI for which group differences were identified. More specifically, for the PwMS/control 1 group set regressions were Shared Perspective conducted for happiness with health, psychological and family- The information collected through the EHHI allowed us to related definitions of happiness, and health-related goals. For the contextualize individuals’ perceived well-being within the major caregivers/control 2 group set regressions were conducted for life domains. Regardless of group inclusion, in both qualitative happiness with community and meaningfulness of leisure. and quantitative answers participants primarily referred to few As concerns PwMS and control 1, for the dependent variable life domains, basically circumscribed to the private sphere. positive affect the two predictors entered in the model were Family, personal life and health distinctly emerged across groups group type and education level, together explaining 18% of the as the predominant contexts of happiness, meaningfulness and 2 2 variable variance (r = 0.143, F = 20.13, p < 0.001, and r goal investment. A substantially lower relevance was attributed = 0.039, F = 5.41, p < 0.05 respectively). Negative affect was to the broader contexts of work and interpersonal relations; significantly predicted by group type (r = 0.041, F = 5.27, finally, the public domains of community and society, and the p < 0.05), however explaining only 4.4% of the variable variance. transcendent sphere of spirituality/religion were almost absent Satisfaction with life was significantly predicted by employment from participants’ qualitative answers, and they scored lowest status and education level (r = 0.121, F = 16.69, p < 0.001, and in rank on the scales. Overall, these findings largely confirmed r = 0.042, F = 5.82, p < 0.05 respectively), but not by group previous EHHI studies conducted across countries on adult type; the two significant predictors together explained 16.3% of samples belonging to the general population (Delle Fave et al., the variable variance. The level of happiness with health was 2011; Delle Fave et al., 2013b). predicted by group type (r = 0.126, F = 17.47, p < 0.001), and Across groups, participants who referred to family in their employment status (r = 0.048, F = 7.04, p < 0.01), together definition of happiness mainly focused on the dimension of explaining 17.4% of the variable variance. The distribution of sharing. This finding is consistent with the models emphasizing participants citing psychological and family-related happiness the primacy of relational connectedness in humans (Richardson, Frontiers in Psychology | www.frontiersin.org 10 December 2017 | Volume 8 | Article 2216 Delle Fave et al. Psychosocial Well-Being in Multiple Sclerosis 2012). As specifically concerns persons with MS and their followed across groups, while only a negligible percentage of caregivers, these results are also consistent with the evidence participants mentioned spirituality/religion, community/society, of a shared process of adaptation to illness, based on and leisure. A significantly higher percentage of caregivers communal growth and search for meaning (Pakenham, 2005; reported less than three meaningful things compared to control Pakenham and Cox, 2009; Ackroyd et al., 2011). Among the participants; this difference may be related to the narrower psychological definitions of happiness, eudaimonic dimensions range of daily opportunities that characterizes the caring role were predominant; inner harmony was reported by the highest (Mausbach et al., 2011). percentage of participants across groups, followed by self- When describing recent situations of intense happiness, most actualization and personal growth. This finding, consistent with participants across groups referred to events shared with the previous evidence (Delle Fave et al., 2016), provides further family: birthdays, marriages, holidays, but also receiving a good support to the view of happiness as connectedness, at both the news concerning family members. Leisure and interpersonal inner and relational levels (Kjell, 2011). Most of the research relationships, following in rank, were mentioned by a relatively hypotheses concerning happiness definitions were however high percentage of participants across groups only in the context not confirmed. Against expectations, the percentage of PwMS of this answer. Finally, and in line with expectations, health was referring to health and the percentage of caregivers referring to quoted almost exclusively by PwMS. It is also important to note family were not significantly higher, compared to their respective that over one third of the caregivers could not identify any recent control groups. Similarly, against expectations no difference was situation of intense happiness. detected between caregivers and control 2 in the percentage of participants quoting leisure. Only two unexpected differences Extraordinary Circumstances, Ordinary emerged between PwMS and control 1: a lower percentage of PwMS provided psychological definitions of happiness, and a Experiences of Well-Being higher percentage mentioned family related ones. In both cases, Research has repeatedly emphasized the negative psychological group classification emerged as the only significant predictor. consequences of living with MS as a person or a caregiver, The overall relevance of family has been widely documented especially at the emotional level. Our findings were only among persons with MS (Ryan et al., 2007; Pakenham, 2008a). partially consistent with the literature, rather emphasizing the It is however worth noting that in this study persons with “ordinariness” of PwMS and caregivers (Saravanan et al., 2001) MS, similarly to participants in the other groups, emphasized in their quantitative ratings of global and domain-related well- sharing rather than receiving support and rewards from family— being. as it could be expected from individuals facing a progressively As concerns domain-related happiness and hedonic increasing dependence on their caregivers. well-being (assessed as positive and negative affect, and Across groups, the majority of participants mentioned family satisfaction with life), PwMS scored significantly lower as a major future goal, primarily referring to its intrinsic value than control participants in happiness with health (thus and to the well-being of family members. Work followed in confirming expectations) and in positive affect. Although rank, while marginal relevance was attributed to extra-family participants’ group emerged as the strongest predictor of the two relations, and to social, community and spirituality issues. As variables, unemployment further contributed in predicting lower hypothesized, a significantly higher percentage of persons with happiness with health, and lower education level in predicting MS quoted health, compared to control 1; group classification lower positive affect. PwMS also reported significantly lower was the only predictor of this result. All the other hypotheses levels of life satisfaction, but employment status and education, were not confirmed. It is worth noticing that, compared to their rather than presence of MS, emerged as significant predictors respective control groups, a significantly higher percentage of of this result. No differences instead emerged between PwMS PwMS and caregivers reported less than three future goals. This and control participants for negative affect, despite the (weakly) result can be related to the perceived uncertainty highlighted significant predictive role of group classification. To this purpose, in the MS literature, and in studies involving people with it is worth mentioning that negative affect values were on average other chronic diseases (Bensing et al., 2002; Tams et al., 2016). higher in the control group. The comparison between caregivers Uncertainty leads individuals to focus on the present rather and their control group highlighted a significant difference in than on future planning; this aspect can be even more relevant positive affect, with the former scoring significantly lower. Group in diseases such as MS, which entails a higher margin of type was the strongest predictor of positive affect; the additional unpredictability, compared with other chronic and degenerative though limited contribution of lower education level replicated conditions (Alschuler and Beier, 2015). the findings detected between PwMS and control 1. Group type Family also emerged as a key meaningful thing; the vast emerged as significant predictor of satisfaction with life as well, majority of participants across groups referred to its intrinsic though with marginal explanatory relevance. value—having a family, a partner, children, siblings were The lower levels of positive affect reported by PwMS and identified as valuable components of life per se. The primacy caregivers were consistent with the literature highlighting the of intimate relationships in the meaning making process largely emotional burden of chronic disease; nevertheless, education confirmed previous evidence obtained with a variety of samples emerged in both cases as an additional environmental predictor, (Baumeister and Leary, 1995; Lambert et al., 2010; Taubman – suggesting that emotions are—at least partially—socially Ben-Ari et al., 2012). Work, health and interpersonal relations constructed. In particular, this finding provides support to the Frontiers in Psychology | www.frontiersin.org 11 December 2017 | Volume 8 | Article 2216 Delle Fave et al. Psychosocial Well-Being in Multiple Sclerosis role of education as a major objective indicator of hedonic or communal competences, rather than being considered as weak subjective well-being, regardless of health conditions (Kroll and and low-performing members of the society. Their psychological Delhey, 2013). The same consideration can be made as regards and relational competences, laboriously built over time, could satisfaction with life, whose levels were not predicted by group be rather shared to the benefit of others. Community Based type, but by the social opportunities derived from employment Rehabilitation (CBR) programs are rooted in this view, aimed status and education, thus confirming previous evidence (Oishi at empowering persons with disabilities through the promotion and Diener, 2014). The lack of group differences in negative and acknowledgment of their active community role (Khasnabis affect levels confirmed instead the conceptual and empirical et al., 2010). Across countries, people experiencing disease are independence of positive and negative affect (Seib-Pfeifer et al., often founders or active members of associations, promoters of 2017), as well as the importance to consider well-being and fundraising campaigns in support of biomedical research, civil ill-being as partially independent domains of experience, rather rights activists. Therefore, health professionals could approach than as opposite poles of a single continuum (Keyes, 2007). them as experts who can offer first-person knowledge of a In this study eudaimonic well-being was quantitatively specific condition, and not only as patients to treat and caregivers assessed as the level of meaningfulness associated with different to instruct (Greenhalgh, 2009). Their social involvement could life domains. Findings did not support the study hypotheses, be extended to educational programs and other community as PwMS and caregivers did not differ from control groups initiatives, allowing them to share their resources and enjoy in family and health related meaningfulness. An unexpected recognition as full-fledged members of the society. Although difference emerged between caregivers and control 2, with the this change implies an overall revision of the health culture, the former attributing significantly lower meaningfulness to the advantages would be remarkable, as efforts in this direction could leisure domain. This result, solely predicted by participants’ lead to a more inclusive and participative society. group, can be related to the restrictions in daily life opportunities experienced by family caregivers (Becker, 2011). Since under Study Strengths and Limitations these circumstances leisure activities get often sacrificed first, The major strength of the present study is the investigation downplaying their meaning and relevance can help caregivers of hedonic and eudaimonic dimensions of well-being among adjust to the related constraints (Pakenham and Cox, 2009). 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- Patients and Caregivers: Social Assets Gonzales et al., 2004), allows for contextualizing scaled ratings Beyond Clinical Labels within qualitative, semantically richer answers. In this study, the This study highlighted that overall persons with MS and their domain-related ratings of happiness and meaningfulness could caregivers do not differ from healthy people in their experience be combined with a fine grained description of the same domains, of hedonic and eudaimonic well-being. These findings are their present and future relevance, and their relation to well- consistent with the literature showing that individuals and being. The findings provided an integrated representation of the families mobilize a variety of resources in order to adjust daily activities, contexts and relationships in which participants’ to disease conditions. At the same time, they offer further meaning-making process, goal pursuit and happiness experiences suggestions. Not only have these people adjusted to disease; they took place. To our knowledge, no studies of this kind are available pursue value-driven goals, cultivate inner harmony and balance, in the psychological literature on MS, and more generally on invest their energies in meaningful activities and relationships. chronic diseases. Persons with MS do not seem to be primarily focused on their This study has limitations as well. First of all, the cross- own health and related needs; they are rather actively engaged sectional design prevented from identifying causal relationships in sharing experiences and collaborating to the promotion of among variables. Although the circumscribed observation field family well-being (Bogosian et al., 2017). As for caregivers, their allowed for an in-depth analysis of well-being, the negative life trajectory—although forcefully disengaged from extra-family impact of disease and caregiving was not explored. The sample socialization and leisure—is grounded in personal and relational sizes were relatively small. Disability levels of the persons with values, despite the costs emerging at the hedonic well-being level. MS did not reflect the whole range of progression stages: the In general, the present study did not highlight group differences inclusion of participants with very severe disability could lead in participants’ level of engagement in public roles and social to different results, at both the qualitative and quantitative activities, thus showing that active involvement in community levels. All participants were Italians, thus belonging to a and society is related to cultural dimensions rather than health specific socio-cultural context: this feature increases reliability conditions. in the comparison of results across samples, but it prevents Overall, these findings can be considered as a provocative from generalizing results to countries characterized by different claim for a change in perspective, as concerns the social healthcare, welfare and value systems. representation of health. This claim, consistent with the ICF model, is based on theoretical and empirical evidence. As Future Directions highlighted by studies investigating resilience (Walsh, 2015), The findings from this study shed light on participants’ individuals and families experiencing chronic diseases should be experience of well-being, in the context of their daily activities valued and appreciated for their ability to develop personal and and social roles. It is however important to consider that both Frontiers in Psychology | www.frontiersin.org 12 December 2017 | Volume 8 | Article 2216 Delle Fave et al. Psychosocial Well-Being in Multiple Sclerosis individuals and their contexts are dynamic entities, changing version to be published; and agreement to be accountable for over time while interacting with each other; this further level all aspects of the work in ensuring that questions related to the of complexity, endorsed by the ICF model, can be satisfactorily accuracy or integrity of any part of the work are appropriately evaluated only through the collaboration of researchers from investigated and resolved. All the other co-authors provided different disciplinary fields. As specifically concerns psychology, substantial contributions to the acquisition and interpretation of relevant contributions could derive from community and data; critical revision of the work at the conceptual level; final cultural psychology, with their focus on the interaction dynamics approval of the version to be submitted; and agreement to be between socio-cultural practices, individual experience and accountable for all aspects of the work in ensuring that questions collective behaviors (Christopher and Hickinbottom, 2008; Di related to the accuracy or integrity of any part of the work are Martino et al., 2017). appropriately investigated and resolved. A stronger interdisciplinary collaboration is especially needed in the light of a specific result emerged from this study: persons FUNDING with MS, their caregivers and the control groups reported low levels of happiness and meaningfulness in community This study was supported by FISM—Fondazione Italiana Sclerosi and society issues; in addition, these domains were almost Multipla [Grant Nr. 2011/R/5], with MB as principal investigator. absent from their lists of goals, meaningful things, and occasions for happiness. This finding may be interpreted as ACKNOWLEDGMENTS an alarming sign of civic disengagement. However, it confirms evidence obtained in other studies conducted in individualistic This study was made possible by the engagement and dedication societies (Delle Fave et al., 2011). We consider this result of many people. The authors would like to express their gratitude as a general warning for researchers, practitioners and policy to the persons with multiple sclerosis, their caregivers and the makers, highlighting the pressing need to promote a culture of participants in the control groups who carefully completed the interconnectedness (Prilleltensky, 2005), in order to contrast the questionnaires and scales used in this work. The complex data deterioration of community networks presently emerging across collection was conducted through the active collaboration of nations. 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 AUTHOR CONTRIBUTIONS (ASST Lariana, Como, Italy), Giovanna De Luca and Marco ADF and MB provided substantial contributions to the Onofri (University G. D’Annunzio, Chieti, Italy). 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Theory Psychol. 18, 699–723. Bayer, Biogen, Merck Serono, Novartis, Sanofi, and Teva. doi: 10.1177/0959354308093403 Song, L.-Y., and Singer, M. (2006). Life stress, social support, coping and depressive Copyright © 2017 Delle Fave, Bassi, Allegri, Cilia, Falautano, Goretti, Grobberio, symptoms: a comparison between the general population and family caregivers. Minacapelli, Pattini, Pietrolongo, Valsecchi, Amato, Lugaresi and Patti. This is an Int. J. Soc. Welfare 15, 172–180. doi: 10.1111/j.1468-2397.2006.00386.x open-access article distributed under the terms of the Creative Commons Attribution Stucki, G., Zampolini, M., Juocevicius, A., Negrini, S., and Christodoulou, N. License (CC BY). The use, distribution or reproduction in other forums is permitted, (2017). Practice, science and governance in interaction: European effort for the provided the original author(s) or licensor are credited and that the original system-wide implementation of the International Classification of Functioning, publication in this journal is cited, in accordance with accepted academic practice. Disability and Health (ICF) in Physical and Rehabilitation Medicine. Eur. J. No use, distribution or reproduction is permitted which does not comply with these Phys. Rehab. Med. 53, 299–307. doi: 10.23736/S1973-9087.16.04436-1 terms. Frontiers in Psychology | www.frontiersin.org 15 December 2017 | Volume 8 | Article 2216 http://www.deepdyve.com/assets/images/DeepDyve-Logo-lg.png Frontiers in Psychology Pubmed Central

Beyond Disease: Happiness, Goals, and Meanings among Persons with Multiple Sclerosis and Their Caregivers

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Abstract

ORIGINAL RESEARCH published: 20 December 2017 doi: 10.3389/fpsyg.2017.02216 Beyond Disease: Happiness, Goals, and Meanings among Persons with Multiple Sclerosis and Their Caregivers 1 2 3 4 5 Antonella Delle Fave *, Marta Bassi , Beatrice Allegri , Sabina Cilia , Monica Falautano , 6 7 5 3 Benedetta Goretti , Monica Grobberio , Eleonora Minacapelli , Marianna Pattini , 8 7 6 9 Erika Pietrolongo , Manuela Valsecchi , Maria Pia Amato , Alessandra Lugaresi and Francesco Patti 1 2 Department of Pathophysiology and Transplantation, Università degli Studi di Milano, Milan, Italy, Department of Biomedical and Clinical Sciences “L.Sacco”, Università degli Studi di Milano, Milan, Italy, Neurology Unit, Multiple Sclerosis 4 5 Centre, Ospedale di Vaio-Fidenza, Fidenza, Italy, Ospedale Policlinico Universitario G. Rodolico, Catania, Italy, Neurological Department, San Raffaele Hospital (IRCCS), Milan, Italy, Department of NEUROFARBA, University of Florence, Florence, 7 8 Italy, Laboratory of Clinical Neuropsychology, ASST Lariana, Como, Italy, Department of Neurosciences, Imaging and Clinical Science, Università degli Studi ‘G. d’Annunzio’ Chieti - Pescara, Chieti, Italy, Department of Biomedical and Neuromotor Sciences, Università di Bologna, Bologna, Italy Edited by: Francisco José Eiroa-Orosa, The experience of persons with multiple sclerosis (MS) and their caregivers is usually University of Barcelona, Spain investigated in terms of emotional distress and health-related quality of life, while Reviewed by: David John Oliver, well-being indicators remain largely underexplored. In addition, findings are often University of Kent, United Kingdom interpreted from the clinical perspective, neglecting socio-cultural aspects that may Anna Fogdell-Hahn, crucially contribute to individuals’ functioning. At the methodological level, most Karolinska Institute (KI), Sweden Karin Zebenholzer, studies rely on scaled instruments, not allowing participants to freely express their Medical University of Vienna, Austria needs and resources. Based on the bio-psycho-social perspective endorsed by *Correspondence: the International Classification of Functioning, well-being indicators were investigated Antonella Delle Fave antonella.dellefave@unimi.it among 62 persons with MS (PwMS), their 62 caregivers and two control groups, matched by age and gender. Participants completed the Positive Affect Negative Specialty section: Affect Schedule (PANAS), the Satisfaction with Life Scale (SWLS), and the Eudaimonic This article was submitted to Clinical and Health Psychology, and Hedonic Happiness Investigation instrument (EHHI). EHHI provides information on a section of the journal participants’ happiness, goals and meanings through scaled and open-ended questions, Frontiers in Psychology contextualized within major life domains. No relevant differences emerged among Received: 30 September 2017 PwMS and caregivers, compared with the respective control groups, as concerns life Accepted: 07 December 2017 Published: 20 December 2017 domains associated with happiness, goals and meaning. Participants across groups Citation: prominently mentioned family, highlighting its intrinsic value and its relevance as a Delle Fave A, Bassi M, Allegri B, sharing context; health did not represent a major theme for PwMS; community, society Cilia S, Falautano M, Goretti B, Grobberio M, Minacapelli E, Pattini M, and religion/spirituality issues were substantially neglected by all participants. PwMS Pietrolongo E, Valsecchi M, and caregivers reported lower levels of positive affect than their control groups, while Amato MP, Lugaresi A and Patti F no substantial differences emerged for negative affect, happiness and meaningfulness (2017) Beyond Disease: Happiness, Goals, and Meanings among Persons levels in life and across most domains. Results suggest that the experience of MS is with Multiple Sclerosis and Their associated with well-being in relevant life domains, such as family and close relationships. Caregivers. Front. Psychol. 8:2216. doi: 10.3389/fpsyg.2017.02216 Although PwMS and caregivers identified a lower number of goals and meaning-related Frontiers in Psychology | www.frontiersin.org 1 December 2017 | Volume 8 | Article 2216 Delle Fave et al. Psychosocial Well-Being in Multiple Sclerosis 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 as well as material, social and institutional barriers. Despite these disadvantages, the mobilization of personal, relational The technological advancements in the medical domain and environmental resources allows these people to attain high fostered the representation and interpretation of health-related levels of mental health (Arnold et al., 2005; Andrykowski et al., phenomena through rigorously quantitative approaches, 2008; Delle Fave et al., 2015). Similarly, caregivers experience leading to the affirmation of the biomedical model. Despite the coexistence of caring burden and limitations in daily the shortcomings entailed by this narrow perspective, and the opportunities with psychological and social resources (Song and classical definition of health as “a state of complete physical, Singer, 2006; Olsson et al., 2008; Fianco et al., 2015). mental and social well-being” (World Health Organization, 1946, The study of mental health and well-being has received p. 1), both research and clinical practice are still permeated by a great impulse during the last two decades, through theories strictly biological view of human functioning. and empirical models deriving from two complementary A laudable effort to overcome this limitation and to perspectives. Within the hedonic perspective, well-being is promote the adoption of a bio-psycho-social view of health operationalized as the predominance of positive over negative and disease is represented by the International Classification affect in daily experience and a globally positive life evaluation, of Functioning, Disability and Health (ICF; World Health defined as satisfaction with life (Ryan and Deci, 2001). From Organization, 2001). ICF is aimed at comprehensively assessing the eudaimonic perspective well-being is instead conceptualized the physical, mental and social functioning of any individual, as a dynamic growth process, that includes a wide range including persons with diseases and disabilities. It presents a of constructs such as goal setting and pursuit, meaning- marked shift in terminology, from focusing on the consequences making, self-expressiveness, self-determination, self-acceptance, of disease (impairments, disabilities, handicaps) to investigating skill development and mastery, trust in relationships, and the components of health and functioning, classified as structures social integration (Huta and Waterman, 2014). Both these and functions, daily activities, and social participation. Far from approaches proved to be useful in exploring protective resources, endorsing a pure linguistic convention, the ICF stems from a adaptation processes and adjustment outcomes that allow relevant conceptual change, leading to the evaluation of health persons with chronic diseases and their caregivers to attain conditions from a constructive perspective. It is based on the positive functioning (Cummins, 2005; Diener and Chan, 2011). assumption of a dynamic interplay between individual features This avenue of research is however characterized by an (body functions, activities, and participation) and environmental emphasis on individual processes, while the societal and cultural aspects that can facilitate or hinder the person’s functioning factors influencing the person’s daily functioning are substantially (Dixon et al., 2008; Bodde and Seo, 2009). However, the neglected (Di Martino et al., 2017). Demographic and contextual implementation of this model into practice is problematic, features, when investigated, are treated as components of the as it requires a multidisciplinary integration effort involving person’s stable identity, despite the changes that both the researchers and practitioners from the healthcare, education and environment and the individual ceaselessly undergo (Slife and policy domains (Stucki et al., 2017). In addition, the assessment Richardson, 2008). of psychosocial functioning through the current version of ICF poses several challenges. Psychological and environmental The Individual and Family Experience of dimensions are collapsed into the heterogeneous domain of Multiple Sclerosis: Challenges and “contextual factors”; the psychological aspects are not explicitly identified through a checklist; and the components of community Resources participation are difficult to evaluate (Chang et al., 2013). Multiple sclerosis (MS) is a chronic neurodegenerative disease, Despite these limitations, only the ICF currently offers an characterized by largely unpredictable symptoms and course, integrated approach to human functioning which can be used by and currently lacking of curative treatment. Therefore, besides professionals working in different sectors. physical impairments, MS poses a number of psychological, From the ICF perspective, it is not surprising that people behavioral and social challenges to both affected persons and with chronic diseases or disabilities describe themselves as their caregivers (Dennison et al., 2009; Ackroyd et al., 2011). ordinary persons who cope with extraordinary circumstances The progressive course of the disease requires a constant re- (Saravanan et al., 2001) that include biological impairments adjustment over time (Bogosian et al., 2017), leading the person Frontiers in Psychology | www.frontiersin.org 2 December 2017 | Volume 8 | Article 2216 Delle Fave et al. Psychosocial Well-Being in Multiple Sclerosis to gradually scale back or adapt daily activities to the new reflect a condition of less severe illness (Dennison et al., condition, find new occupations and interests, but also disengage 2009). The assessment of demographic and clinical dimensions from the adjustment process and withdraw from active life. however entails methodological and interpretive challenges, While the negative consequences of MS were extensively as showed by the contrasting findings obtained in different explored, research on the positive aspects is still limited. Studies studies. For example, in some studies demographic variables have been conducted to investigate the eudaimonic process such as education and employment emerged as the strongest of meaning making, a key individual resource to attain well- predictors of health-related quality of life (HR-QoL), together being under unfavorable and irreversible conditions (Hicks and with clinical ones like depression and disability (Patti et al., King, 2009). Meaning making allows individuals to integrate 2003, 2007b). In other studies, HR-QoL itself emerged as an the problematic condition into a global and constructive view independent predictor of disability progression (Benito-Léon of their present and future life. The qualitative exploration of et al., 2013). Satisfaction with life was primarily predicted meaning making among persons with MS shed light on its by disease severity and social support in one study (Ryan components, which include disease acceptance, personal and et al., 2007), while in another study age and education level relationship growth, and positive lifestyle changes (Pakenham, emerged as the strongest predictors of hedonic and eudaimonic 2008a). Quantitative studies highlighted the positive association well-being (Bassi et al., 2014). Finally, a comparative study of sense making with life satisfaction, and its negative association did not highlight significant differences in personal growth (a with depression. Sense making was identified as a key predictor eudaimonic dimension) between persons with MS and healthy of positive adjustment also among caregivers (Pakenham, 2008b). participants, while life satisfaction (a hedonic dimension) was Similar findings were detected as concerns perceived illness significantly lower among the former (Barak and Achiron, 2011). coherence, a construct closely related to meaning making; The investigation of well-being among caregivers of persons illness coherence was positively associated with eudaimonic and with MS is even less frequent, as studies are primarily focused hedonic well-being indicators in both persons with MS and their on the emotional impact of caring-related burden and stress caregivers (Bassi et al., 2016). (Patti et al., 2007a; Rivera-Navarro et al., 2009), and on the Benefit finding, consisting in the ability to identify positive daily choices and free time restrictions imposed by the caring consequences in an otherwise negative situation (Lechner et al., role (Becker, 2011). Studies investigating satisfaction with life 2009), was also investigated among persons with MS and did not detect differences between caregivers of persons with their family caregivers. Cross-sectional and longitudinal studies MS and samples derived from the general population or carers highlighted its positive association with meaning-based coping of persons with other chronic diseases (Waldron-Perrine et al., strategies, and its direct effects on positive adjustment outcomes, 2009; Bassi et al., 2014). Eudaimonic well-being dimensions such such as good dyadic relationships and subjective well-being as meaning making, benefit finding and illness coherence were (Pakenham, 2005, 2007, 2008b; Pakenham and Cox, 2009). more often investigated among caregivers, as reported in the Related findings support the idea of a “communal search for previous paragraphs (Pakenham, 2007; Pakenham and Cox, 2009; meaning” where persons with MS and their caregivers experience Bassi et al., 2016). In a recent qualitative study involving Turkish the trauma of a chronic illness and subsequently find positive caregivers (Topcu et al., 2016), participants mentioned both aspects together (Pakenham, 2005). Further evidence of this individual and social resources, such as motivation to care and dyadic process emerged from the investigation of post-traumatic perceived support. growth and illness perceptions among persons with MS and their Acknowledging the existence and adaptive role of well- partners (Ackroyd et al., 2011). being dimensions in the experience of MS surely represents These studies also showed that the disruption of life goals an advancement. At the same time, most studies suffer from caused by MS progression can be counterbalanced by the two conceptual limitations. The first one is the lack of a development of new meanings and life purposes (Joseph comprehensive bio-psycho-social perspective, and especially the and Linley, 2006). This process entails a complex interplay neglect of the societal context surrounding persons with MS between disengagement from previous goals and reengagement and their caregivers. Only few studies were conducted within in new ones (Neter et al., 2009). It also requires the the ICF framework. One of them was aimed at identifying implementation of adaptive tasks (Bensing et al., 2002), such as a core set of ICF categories specific to MS that could be defining new challenges, acknowledging one’s limits, maintaining used in healthcare practice (Khan and Pallant, 2007). Another emotional balance and self-esteem, facing uncertain future, study proposed the Multiple Sclerosis Impact Profile (MSIP; cultivating social relationships, and looking at the bright Wynia et al., 2008) an instrument designed to evaluate the side of life. Besides these individual resources, other factors perceived impact of MS on functioning. A recent review (Dorstyn located at the intersection between persons and their context et al., 2017) highlighted the association between depressive may foster positive adaptation; they include facilitators of symptoms and reduced social participation in persons with mobility/independence, social support, and social comparisons MS. A qualitative study exploring the impact of ICF contextual (Dilorenzo et al., 2008). factors on the daily functioning of Jordanian persons with MS A frequent problem in studies investigating well-being and (Hamed et al., 2012) identified adequacy of financial and medical perceived quality of life among persons with MS is the lack of resources, religion and community awareness as facilitators, objective measures of disease severity; this limitation prevents and social stigma as a barrier. Finally, the environmental and researchers to exclude that positive adjustment may simply personal facilitators of social participation and satisfaction with Frontiers in Psychology | www.frontiersin.org 3 December 2017 | Volume 8 | Article 2216 Delle Fave et al. Psychosocial Well-Being in Multiple Sclerosis parenting were investigated among mothers with MS (Farber functioning (World Health Organization, 2002; Martins, 2015). et al., 2015). Findings suggested the importance of incorporating The level of disability was also taken into consideration in the both categories of facilitators into treatment. The relevance analysis of data collected among persons with MS. of environmental factors also emerged from the investigation Based on the available literature, some hypotheses were of unmet needs among persons with MS. These needs most formulated. In line with studies on adjustment to MS (Pakenham, often refer to contextual dimensions, such as family and social 2008a; Bassi et al., 2016) we expected that, when describing support, healthcare services, everyday life management, and the happiness, goals and meaningful things, a significantly higher relationship with physicians (Galushko et al., 2014). Interestingly, percentage of persons with MS would refer to health compared representations of the unmet needs of persons with MS differ with a control group. We also expected persons with MS to among health professionals (Golla et al., 2012): while physicians associate health with significantly lower levels of happiness and emphasize limitations in access to care due to poor financial higher levels of meaningfulness, compared with a control group. resources, nurses and social workers are more aligned with As concerns caregivers, based on previous evidence (Pakenham, patients’ perceptions, quoting family support, social relationships 2008b; Becker, 2011; Mausbach et al., 2011) we expected that and daily life management. a significantly higher percentage of participants would refer A second limitation characterizing the literature on MS to family and a lower percentage to leisure, compared with a is the emphasis on disease and related adjustment processes: control group. We also expected caregivers to report significantly participants and caregivers are rarely solicited to freely reflect lower levels of happiness with leisure, and higher levels of on aspects of their life and daily experience potentially unrelated meaningfulness in relation to family. As concerns ratings of to disease or caregiving respectively. This limitation emerged in affect and life satisfaction (the two components of hedonic well- one of the studies investigating unmet needs of persons with MS being), the contrasting findings detected across studies (Barak (Galushko et al., 2014): participants expressed the need to be and Achiron, 2011; Diener et al., 2017) did not allow us to viewed as distinct individuals, not constrained by and identified formulate specific hypotheses. Finally, based on the literature through their health conditions. highlighting the relevant role of education and employment in predicting individual well-being (Keyes, 2007; Patti et al., The Present Study 2007b; Diener et al., 2017), we hypothesized that these two The first aim of the present study was to investigate different demographic features would provide a specific contribution to dimensions of well-being among persons with MS and their hedonic and eudaimonic well-being values across all groups. The caregivers through a mixed method approach and from a same hypothesis was formulated as concerns the role of severity bio-psycho-social, ICF informed perspective. Answers obtained disease among persons with MS. through open-ended and scaled questions were jointly analyzed, in the attempt to contextualize findings within participants’ MATERIALS AND METHOD global outlook of their own life and environmental opportunities. The well-being dimensions investigated in the study included Participants happiness (its definition and recent related situations), hedonic The study involved 248 Italian adults divided into four groups, well-being (positive and negative affect, satisfaction with life) each of them comprising 62 participants: persons with MS and eudaimonic well-being (perceived meaningful things and (PwMS), their caregivers, and two control groups of adults with goals). In addition, information was collected on happiness no history of chronic disease or caregiving experience, selected and meaningfulness levels in life in general, as well as in the from a larger study aimed at investigating well-being in the specific domains of work, family, standard of living, interpersonal general population. Inclusion criteria for PwMS were being at relations, health, personal growth, leisure, spirituality/religion, least 18 years of age, having a clinically definite MS diagnosis community, and society. Overall, the mixed method potentials for at least 3 years and having a caregiver; exclusion criteria are still underexploited in the psychological literature; moreover, comprised the presence of additional neurological or psychiatric to the best of our knowledge no studies have yet been conducted disorders, severe cognitive impairment, MS in the active phase, on these topics and through this methodological approach in the and a condition of very severe disability, corresponding to a MS domain. score above 8 on the Extended Disability Status Scale (EDSS; The second aim was to compare the findings collected among Kurtzke, 1983). The majority of PwMS involved in the study persons with MS and their caregivers with those obtained from (59.68%) showed an EDSS score between 3.5 and 8 (indicating two groups of participants, matched by age and gender, but with increasing levels of motor impairment), while 40.32% scored no history of chronic disease or caregiving experience. From an below 3.5 (indicating a high level of autonomy). No significant ICF informed perspective, this comparison was expected to shed differences were detected between PwMS with low and high levels light on group differences in perceived personal and contextual of disability for any of the variables examined in this study; facets of well-being, including daily activities and opportunities, therefore disability level will not be further considered in the relational networks, and social participation. Attention was also following sections. paid to the role of employment status and education level Caregivers were predominantly partners (59.67%) or parents in predicting well-being dimensions. These two demographic (22.57%) of PwMS, siblings or children (each accounting for features represent crucial indicators of participation, classified 6.46%), friends (3.23%), or professional caregivers (1 participant, in the ICF as contextual personal factors influencing human 1.61%). Participants in the control groups were randomly Frontiers in Psychology | www.frontiersin.org 4 December 2017 | Volume 8 | Article 2216 Delle Fave et al. Psychosocial Well-Being in Multiple Sclerosis extracted from a sample of 691 healthy Italian adults; age, gender associated to 10 major life domains (work, family, standard of and education level were used as filters to match the control living, interpersonal relations, health, personal growth, leisure, groups with the respective comparison groups – PwMS and spirituality/religion, community, and society) and to life in caregivers. It was however not possible to obtain a complete general. match between caregivers and their control group as concerns Satisfaction with Life Scale (SWLS; Diener et al., 1985; education level. The decision to include two different control Goldwurm et al., 2004). This widely used measure of well-being groups was based on the significant age difference between PwMS comprises five items on scales ranging from 1 (strongly disagree) and their caregivers (t = 2.85, p < 0.005). The demographic to 7 (strongly agree). The items investigate the individuals’ features of the four groups of participants are reported in Table 1. overall cognitive evaluation of their global life conditions and No differences emerged between PwMS and their control achievements. group (control 1) for age, gender, marital status and education, Positive Affect and Negative Affect Schedule (PANAS; Watson while a significantly higher percentage of PwMS was unemployed et al., 1988; Terracciano et al., 2003). The instrument assesses the (χ = 21.81, p < 0.001). As concerns caregivers and their control overall perceived intensity of positive and negative affect during group (control 2), differences were detected for employment and daily life, through 10 items measuring components of positive education, both higher in the control group (χ = 7.52, p < 0.01, affect (PA) and 10 items measuring components of negative and χ = 4.61, p < 0.05 respectively). affect (NA). Scales range from 1 (very slightly or not at all) to 5 (extremely). Materials Procedure Data were collected through the following instruments: This study involved seven different academic and clinical A socio-demographic questionnaire provided information on institutions; therefore, the protocol was submitted to the participants’ age, employment status, education level, and civil ethical committees of each institution. After approval from all status. Clinical information including time from disease onset, committees, participants with MS and their caregivers were level of disability, type of treatment and co-morbidities was recruited at six MS centers in different Italian regions, in the collected for PwMS. Caregivers also reported their type of bond context of a broader project aimed at investigating clinical, with PwMS. psychological and relational aspects of MS (Bassi et al., 2014, Eudaimonic and Hedonic Happiness Investigation inventory 2016). PwMS were contacted by the centers’ personnel during (EHHI; Delle Fave et al., 2011, 2016). This mixed-method check-ups or by phone, and were invited to identify their primary instrument allows researchers to collect qualitative and caregiver. Data from participants in the two control groups were quantitative information on different components of well-being. selected from a larger study that had been approved by the ethical Through open-ended questions, participants are asked to define committee of the first author’s institution. happiness in their own words; to list the three future goals they Upon their expression of interest in joining the study, consider most important, and the three things they consider participants received detailed information on the project from most meaningful in their present life; and to briefly describe a researcher involved in the study. They signed an informed three situations associated with intense happiness during the consent in compliance with Italian privacy rules, and were last 6 months. In addition, participants are asked to rate on provided with the battery of questionnaires. They could inspect two sets of scaled items, ranging from 1 (extremely low) to 7 the questionnaires, pose general and specific questions to the (extremely high), the levels of happiness and meaningfulness 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 TABLE 1 | Demographic characteristics of the four groups. place convenient to them. They could hand in their responses, or PwMS Control 1 Caregivers Control 2 send them via mail. Before data processing researchers removed (N = 62) (N = 62) (N = 62) (N = 62) the consent form from each battery of questionnaires, thus guaranteeing participants’ anonymity throughout the phases of Age 40.1 ± 9.7 40.5 ± 10.9 45.7 ± 12.0 45.7 ± 11.4 data coding, storing and analysis. Data were stored in password Age range 21–63 20–60 19–81 22–81 protected computers. Participation to both the MS related study Gender (% Female) 69.35 69.35 58.06 56.45 and the general survey was voluntary, and participants were EDUCATION (%) free to leave the study at any time. Persons with MS were High school or less 82.26 75.81 85.48 69.35 explicitly assured that refusal to participate or withdrawal from University 17.74 24.19 14.52 30.65 participation in the study would in no way interfere with the Work/study (%) 57.38 93.55 75.81 93.55 long-term healthcare services they were receiving at the MS CIVIL STATUS (%) center. Married/cohabiting 66.13 51.61 77.42 72.13 Single/divorced/widowed 33.87 48.39 22.58 27.87 Coding Procedure Answers to the open-ended questions of the EHHI required an Means and Standard Deviations are reported. b accurate coding work, using a coding system originally developed Education was dichotomized: “High school or less” includes elementary, middle, and high school; “University” includes graduation, post-graduation and PhD. by Delle Fave et al. (2011), and gradually expanded through Frontiers in Psychology | www.frontiersin.org 5 December 2017 | Volume 8 | Article 2216 Delle Fave et al. Psychosocial Well-Being in Multiple Sclerosis additions from various studies, in line with the bottom-up represent the positive and meaningful sides of this life domain approach guiding the EHHI research project. In this coding (Delle Fave et al., 2013a). system, answers are grouped into broad categories corresponding to the major life domains: work, family, standard of living, Statistical Analyses interpersonal relationships, health, personal/psychological life, The analysis of qualitative data collected through the EHHI was leisure, spirituality/religion, society and community issues, and aimed at identifying the life domains predominantly mentioned more general/unspecific life aspects. Multifaceted categories by participants across groups in their descriptions of happiness, such as work, family, relationships, leisure, spirituality/religion, major goals, meaningful things, and recent sources of intense community/society and personal life are further subdivided happiness. Results were compared between groups (respectively into more fine-grained sub-categories (Delle Fave et al., PwMS and control 1, and caregivers and control 2). Since a 2013a). The categories family and interpersonal relationships high percentage of participants across groups indicated family were organized into the subcategories intrinsic value (e.g., as prominent goal, meaningful thing, and source of recent having a family; a partner; children; friends), sharing (e.g., happiness, analyses were performed on family sub-categories. spending time with children; sharing life projects with partner; The same in-depth analysis was conducted for the psychological sharing good and bad experiences with friends), personal definitions of happiness. contribution (caring for elder parents; raising children; helping Coded answers were first grouped into the corresponding friends in need), family well-being/harmony, balance (e.g., categories and sub-categories; subsequently, considering that health of family members; children’s positive development; each participant provided more than one answer for each harmony in relationships) and personal reward (e.g., gratification question, the number of participants reporting at least one from partner, children; gratitude expressions from siblings; answer in each category and subcategory was calculated across satisfaction with relations). A similar partition characterizes groups. This approach allowed us to compare the percentage of community/society issues. In the psychological/personal life participants referring to each answer category and subcategory category, some subcategories refer to eudaimonic well-being between groups through 2 × 2 frequency tables by means of the dimensions (growth/engagement, purpose, competence/mastery, χ procedure. The procedure was not considered as reliable when autonomy, self-actualization, meaning/value, harmony/balance, the number of participants in one or more cells was below 5. fullness/awareness, optimism), and others to hedonic well- Through Spearman coefficients, correlations were then calculated being ones (satisfaction/achievement, positive emotions, and between participants’ distribution in answer categories and their absence of negative feelings); a less specific subcategory, labeled demographic and group features. Logistic regression analyses allowed us to verify whether demographic or group features as “positive experiences/states”, includes answers generically referring to happiness as “inner well-being”, “a stable state”, or predicted a specific pattern of answers. “a way of being.” Quantitative data were first analyzed using descriptive The coding procedure for each answer comprises first the statistics. Correlations of hedonic and eudaimonic dimensions identification of a category in which the answer can be of well-being with participants’ group and demographic features included, and then a specific numeric item to which the were calculated through Pearson and Spearman coefficients. answer can correspond, if available. If an answer content does Hierarchical regression analyses provided information on the not fit any available item, a new item will be added to the specific contribution of participants’ group and demographic category. As concerns happiness definitions, participants often features to well-being dimensions. report complex descriptions including different components of Given the large number of group comparisons performed happiness. Each of these facets is treated as a specific semantic on quantitative and qualitative variables, we took a Bonferroni unit and coded separately; up to six answer units are retained approach, adjusting the critical alpha value for significance to the for each participant. Based on the specific formulation of number of t-tests and χ comparisons performed on the same the questions concerning goals, meaningful things, and recent dataset (25 and 61 respectively). More specifically, to achieve α < situations of most intense happiness, three answer units are 0.05 with 25 t-tests, the alphas obtained from each data set had retained for each question. In the present study, reliability in the to score below 0.002 (α < 0.05/25, two-tailed); in order to reject coding process was established through the involvement of two a null hypothesis, the test statistic had to exceed critical t = 3.16, expert coders providing independent ratings for each answer; corresponding to p < 0.002 with 122 degrees of freedom (for two divergences were clarified through a subsequent discussion; groups, df = N participants-2). unsolved discrepancies and answers potentially requiring the The same approach was used for frequency table comparisons inclusion of new items in the coding system were further (N = 61 in each data set). To achieve α < 0.05 with 61 discussed with the first author. comparisons, the alphas obtained from the data set had to As concerns the scaled questions included in the EHHI, and score below 0.0008 (α < 0.05/61); in order to reject a null the data collected through PANAS and SWLS, the numeric hypothesis, the test statistic had to exceed critical χ = 11.34, values corresponding to the perceived levels of each variable corresponding to p < 0.0008 with 1 degree of freedom, since df = were reported. The EHHI items assessing the levels of happiness (N columns-1) (N rows-1). As for correlations, regardless of the and meaningfulness associated with personal/psychological life statistical significance only coefficient values equal to or higher are labeled as “personal growth”; this decision was originally than 0.30 were considered as adequate to interpret associations based on the assumption that growth and development could best as meaningful (Hinkle et al., 2003). More specifically, values Frontiers in Psychology | www.frontiersin.org 6 December 2017 | Volume 8 | Article 2216 Delle Fave et al. Psychosocial Well-Being in Multiple Sclerosis TABLE 2 | Definitions of happiness: percentage of participants mentioning each between 0.30 and 0.50 indicated low correlation, values between category and subcategory by group, and comparisons between groups. 0.50 and 0.70 moderate correlation, and values above 0.70 high correlation. 2 2 PwMS Control 1 χ Caregivers Control 2 χ Psychological definitions 54.72 93.33 22.51** 66.67 80.00 2.61 RESULTS Harmony/balance 68.97 60.71 0.56 63.89 72.92 0.78 Other eudaimonic 24.14 39.29 1.95 25.00 41.67 2.52 This section illustrates the qualitative and quantitative findings Hedonic 41.38 46.43 0.19 27.78 33.33 0.30 presented for each group separately, in order to allow for Positive states 10.34 10.71 2.78 8.33 comparisons. Family 45.28 15.00 12.48* 51.85 28.33 6.58 Intrinsic value 28.00 11.11 13.79 11.76 Qualitative Findings Sharing 48.00 55.56 0.69 44.83 58.82 0.83 The findings obtained from the open-ended questions of the Personal contribution 4.00 11.11 10.34 0 EHHI are presented as percentages of participants across groups Family well-being 12.00 22.22 34.48 23.53 Personal reward 28.00 22.22 20.69 23.53 who provided at least one answer in the different categories and Interpersonal relations 15.09 23.33 1.22 12.96 30.00 4.82 subcategories. The answers referring to psychological dimensions Health 11.32 8.33 0.29 12.96 15.00 0.10 are grouped into the category labeled as “psychological Work 11.32 1.67 5.56 8.33 definitions” for the definitions of happiness, and into the category Standard of living 1.89 1.67 7.41 3.33 “personal life” for goals, meaningful things and recent situations Leisure 5.66 0 0 1.67 of intense happiness. Spirituality, Religion 3.77 1.67 3.70 6.67 Community, Society 7.55 5.00 0.00 3.33 Happiness Definitions Life in general 0 13.33 3.70 8.33 Participants were invited to answer the question “What is N participants 53 60 54 60 happiness for you?” in their own words, without specific Each participant could provide more than one answer; Bonferroni adjusted alpha constraints. It is worth noticing that 9 PwMS and 8 caregivers *p < 0.05; **p < 0.01; χ -values are not reported if the no. of participants in one or either did not provide any answer, or stated that “happiness more cells falls below 5. 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 Goals and Meaningful Things (2.65 and 2.80 respectively) compared with PwMS and caregivers While all participants in the control groups identified some (2.25 and 2.19 respectively). These differences were however important future goals, 2 PwMS and 4 caregivers did not. In not statistically significant. Table 2 shows for each group the addition, 59 participants in control 1 reported up to three goals percentage distribution of the participants who provided at least compared with 45 PwMS (χ = 11.68, p < 0.05); similarly, 59 one answer in the different categories and subcategories. Results participants in control 2 reported up to three goals compared of group comparisons are also reported. with 44 caregivers (χ = 12.89, p < 0.05). Table 3 depicts the Overall, a substantial similarity emerged between groups percentage distribution of the participants who provided at least across most categories. Only two significant differences were one answer across categories in each group. detected between PwMS and control 1: a lower percentage The majority of participants in all groups mentioned family, of PwMS reported psychological definitions of happiness, specifically referring to its intrinsic value (having a family; and a lower percentage of controls reported family-related having or adopting children; having grandchildren; finding ones. Despite these overarching differences, the percentage the right partner) and to the well-being of family members distribution of participants across subcategories of both (physical health, self-actualization or goal achievement of psychological and family-related definitions did not differ children, grandchildren, partner, siblings, and parents). Health, between the two groups. Among psychological definitions, work, and personal life were mentioned by lower percentages participants predominantly referred to eudaimonic constructs, of participants; spirituality/religion and community/society were such as harmony/balance, self-actualization, personal growth, marginally represented, together with interpersonal relations. and optimism. Since a remarkably high percentage of participants The only significant difference concerned the higher percentage specifically mentioned harmony/balance, related findings are of PwMS mentioning health, compared to control 1. reported separately in Table 2. As concerns family, most As regards the most meaningful things in the present participants in both groups provided answers related to sharing life, 7 caregivers did not identify any, compared to 4 PwMS (experiences, activities, projects). Personal rewards followed and no participants in the control groups (χ = 13.22, as subcategory; answers included receiving love expressions, p < 0.05). Moreover, 60 participants in control 2 reported up support, acknowledgment from family members, and satisfaction to three meaningful things, compared with only 46 caregivers with family. Fewer participants quoted the other domains, and (χ = 12.74, p < 0.05). Nevertheless, as illustrated in Table 4, a negligible percentage in both groups mentioned leisure, participants’ percentage distribution across answer categories was spirituality/religion, community/society issues and standard of largely overlapping across groups. living. A similar answer pattern was detected among caregivers Participants in all groups almost unanimously quoted and control 2, but no significant group differences emerged. family as one of the most meaningful things in their lives Frontiers in Psychology | www.frontiersin.org 7 December 2017 | Volume 8 | Article 2216 Delle Fave et al. Psychosocial Well-Being in Multiple Sclerosis TABLE 3 | The most important future goals: percentage of participants TABLE 4 | The most meaningful things in present life: percentage of participants mentioning each answer category by group and comparisons between groups. mentioning each answer category by group and comparisons between groups. 2 2 2 2 PwMS Control 1 χ Caregivers Control 2 χ PwMS Control 1 χ Caregivers Control 2 χ Personal life 26.67 38.71 2.00 15.52 20.97 0.59 Personal life 12.07 20.97 1.71 25.45 17.74 1.03 Family 68.33 67.74 0.004 79.31 64.52 3.23 Family 91.38 79.03 3.58 83.64 82.26 0.04 Intrinsic value 51.22 52.38 0.01 39.13 40.00 0.007 Intrinsic value 86.79 83.67 0.20 78.26 84.31 0.59 Sharing 14.63 4.76 10.87 25.00 2.96 Sharing 1.89 12.24 13.04 5.84 Personal contribution 12.20 23.81 1.89 23.91 12.50 1.84 Personal contribution 1.89 6.12 6.52 3.92 Family well-being 31.71 35.71 0.15 43.48 45.00 0.02 Family well-being 7.55 0 6.52 7.84 Personal reward 4.88 0 0 2.50 Personal reward 7.55 2.04 2.17 1.96 Interpersonal relations 5.00 6.45 1.72 6.45 Interpersonal Relations 27.59 40.32 2.16 23.64 38.71 3.06 Health 53.33 22.58 12.28* 39.66 32.26 0.71 Health 48.28 30.65 3.90 32.73 33.87 0.02 Work 45.00 59.68 2.63 37.93 58.06 4.86 Work 43.10 30.65 2.00 43.64 50.00 0.47 Standard of living 6.67 16.13 2.69 29.31 22.58 0.71 Standard of living 5.17 12.90 9.09 14.52 Leisure 10.00 19.35 2.12 8.62 16.13 1.54 Leisure 5.17 17.74 3.64 9.68 Spirituality, Religion 3.33 0.00 3.45 6.45 Spirituality, Religion 8.62 6.45 5.45 9.68 Community, Society 3.33 3.23 0.00 14.52 Community, Society 5.17 11.29 3.64 1.61 Life in general 3.33 12.90 6.90 14.52 Life in general 0 13.33 5.45 4.84 0 a a N participants 60 62 58 62 N participants 58 62 55 62 a 2 Each participant could provide more than one answer; Bonferroni adjusted alpha Each participant could provide more than one answer; χ -values are not reported if the *p < 0.05; χ -values are not reported if the no. of participants in one or more cells falls no. of participants in one or more cells falls below 5. below 5. TABLE 5 | Recent situations of intense happiness: percentage of participants mentioning each answer category by group and comparisons between groups. (specifically referring to its intrinsic value); progressively 2 2 PwMS Control 1 χ Caregivers Control 2 χ lower percentages of participants mentioned work, health, interpersonal relationships and personal life, without significant Personal life 6.00 8.33 0 10.53 group differences. Spirituality/religion, leisure, standard of living Family 74.00 73.33 0.006 82.05 78.95 0.14 and community/society were mentioned by less than 10% of the Intrinsic value 10.81 22.73 1.99 12.50 15.56 participants across groups. Sharing 70.27 68.18 0.04 56.25 46.67 0.69 Personal contribution 0 4.55 6.25 0 Family well-being 35.14 29.55 0.29 50.00 55.56 0.23 Recent Situations of Intense Happiness Personal reward 13.31 13.64 3.13 11.11 As illustrated in Table 5, only 39 caregivers (62.9%) provided Interpersonal relations 22.00 36.67 2.79 20.51 22.81 0.07 Health 18.00 3.33 6.52 5.13 1.75 answers to this question, while the remaining 23 (37.10%) could Work 20.00 21.67 0.05 12.82 29.82 3.79 not remember any recent situation of intense happiness. This Standard of living 4.00 6.67 7.69 10.53 answer distribution pattern was significantly different from those 2 Leisure 34.00 31.67 0.08 38.46 33.33 0.27 detected for the other three groups (χ = 29.93, p < 0.001): only N participants 50 60 39 57 12 participants (19.35%) among PwMs, 5 (8.1%) in control 2 and a 2 2 (3.23%) in control 1 did not report recent situations of intense Each participant could provide more than one answer; χ -values were not reported if one or more cells included less than 5 participants; answer categories cited by less than happiness. 5 participants across groups were excluded from analysis. 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 Quantitative Findings participants across groups referred to sharing positive events and experiences, such as anniversaries and other celebrations. Ratings of Happiness, Meaningfulness, and Hedonic A lower percentage of participants mentioned events promoting Well-Being the well-being of family members (positive school and work Table 6 shows the mean ratings of happiness and meaningfulness achievements, disease recovery or health improvements). Leisure across groups, and the results of the comparisons between PwMS predominantly included the practice of arts and crafts, sports, and control 1, and between caregivers and control 2. travels and media fruition. Interpersonal relationships and Participants across groups reported the highest levels of work followed in rank across groups. A higher percentage of happiness and meaningfulness in the domains of family, health, PwMS referred to health related situations, but after Bonferroni interpersonal relations, life in general, and personal growth adjustment the difference with control 1 was not significant. (with slight variations in domain order across groups). In Spirituality/religion and community/society were again reported contrast, spirituality/religion, community and society issues were by a negligible percentage of participants across groups, together associated with the lowest levels of happiness and meaningfulness with personal life and standard of living. across groups. The only difference between PwMs and control Frontiers in Psychology | www.frontiersin.org 8 December 2017 | Volume 8 | Article 2216 Delle Fave et al. Psychosocial Well-Being in Multiple Sclerosis 1 concerned happiness with health, with the former reporting issues, and they attributed lower meaningfulness to the domain significantly lower values. The comparison between caregivers of leisure. and control 2 highlighted two significant differences: Caregivers More relevant differences were detected between groups reported higher values of happiness in relation to community 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 TABLE 6 | Levels of happiness and meaningfulness in life domains and among PwMS than among participants in control 1. No group comparisons between groups. differences were instead detected for negative affect. PwMS Control 1 t Caregivers Control 2 t Demographic and Group Predictors of Well-Being M (sd) M (sd) M (sd) M (sd) Dimensions HAPPINESS Correlations and regression models were finally calculated within Work 4.50 4.47 0.11 4.25 4.56 1.13 each group set-PwMS and control 1, and caregivers and control (1.96) (1.31) (1.72) (1.25) 2 respectively -, in order to investigate the role of group type and Family 5.90 5.60 1.18 5.56 5.84 1.13 demographic features as predictors of qualitative and quantitative (1.47) (1.34) (1.35) (1.20) evaluations of well-being. Age, gender and marital status were Standard liv. 4.74 4.90 0.69 4.41 4.88 2.00 not considered, as these characteristics did not differ between (1.34) (1.30) (1.28) (1.28) PwMS and control 1, and between caregivers and control 2. Relations 5.00 5.10 0.37 4.70 5.33 2.75 Attention was instead paid to education level and employment (1.55) (1.34) (1.42) (1.06) status, as group differences were detected for these two features, Health 4.37 5.45 4.17** 5.21 5.54 1.40 (1.72) (1.10) (1.14) (1.30) as reported in the comments on Table 1. Table 8 illustrates Personal Growth 5.05 4.92 0.52 4.87 5.18 1.31 Spearman correlations among predictors (demographics and (1.41) (1.35) (1.43) (1.16) group classification) and between predictors and well-being Leisure 4.15 4.66 1.96 3.98 4.41 1.41 dimensions. (1.48) (1.45) (1.72) (1.66) For the sake of synthesis, analyses were performed only for Spirituality 3.93 4.04 0.29 4.02 4.89 2.60 the dimensions showing significant differences between groups. (2.09) (1.70) (2.04) (1.56) As reported in the method section, correlations higher than Community 3.64 4.23 2.18 3.56 4.49 3.29* 0.30 were deemed as meaningful. Overall, group classification (1.41) (1.44) (1.67) (1.31) showed the most numerous and strongest correlations with Society 3.71 3.62 0.29 3.70 3.72 0.08 the well-being dimensions. In the PwMS/control 1 group set, (1.60) (1.43) (1.73) (1.20) only employment status showed a low positive correlation with Life in general 5.03 5.08 0.22 5.10 5.31 1.05 satisfaction with life; together with group classification, it showed (1.31) (1.12) (1.25) (0.93) a low positive correlation with happiness with health as well. MEANINGFULNESS No meaningful correlations were instead identified for education Work 5.28 5.21 0.24 5.36 5.66 1.12 (1.76) (1.29) (1.67) (1.23) level. In the caregivers/control 2 group set, group type showed Family 6.45 6.58 0.75 6.72 6.69 0.19 low to moderate correlations with most well-being dimensions, (1.08) (0.82) (0.77) (0.82) except for satisfaction with life and negative affect. A low positive Standard liv. 5.15 5.03 0.50 4.93 5.29 1.60 correlation emerged between education level and positive affect, (1.34) (1.19) (1.28) (1.19) while employment status did not show any relevant correlation Relations 5.56 6.02 2.13 5.34 5.97 3.10 with well-being dimensions. (1.35) (0.98) (1.28) (0.96) Health 6.32 6.44 0.67 6.53 6.81 2.19 (1.02) (0.84) (0.88) (0.44) Personal Growth 5.74 5.91 0.85 5.51 5.90 1.64 TABLE 7 | Levels of affective and cognitive dimensions of hedonic well-being, and (1.25) (1.08) (1.37) (1.31) their comparison between groups. Leisure 4.87 5.10 0.93 4.55 5.55 4.48** (1.52) (1.17) (1.42) (1.04) PwMS Control 1 t Caregivers Control 2 t Spirituality 4.01 4.00 0.04 4.61 5.08 1.35 M (sd) M (sd) M (sd) M (sd) (2.12) (2.12) (2.06) (1.79) Community 4.26 4.45 0.70 4.19 4.64 1.63 Positive affect 3.01 3.59 4.62** 3.01 3.87 7.83** (1.37) (1.67) (1.60) (1.45) (0.81) (0.57) (0.71) (0.48) Society 4.29 4.25 0.14 4.26 4.88 2.53 Negative affect 2.15 2.51 2.29 2.03 2.22 1.35 (1.54) (1.62) (1.68) (0.99) (0.93) (0.82) (0.89) (0.66) Life in general 5.98 6.08 0.57 5.90 6.27 1.93 Satisfaction with life 3.80 4.65 3.30* 4.14 4.87 3.06 (1.06) (0.80) (1.25) (0.85) (1.55) (1.30) (1.51) (1.15) N participants 62 62 62 62 N participants 62 62 62 62 Bonferroni adjusted alpha *p < 0.05; **p < 0.01. Bonferroni adjusted alpha *p < 0.05; **p < 0.01. Frontiers in Psychology | www.frontiersin.org 9 December 2017 | Volume 8 | Article 2216 Delle Fave et al. Psychosocial Well-Being in Multiple Sclerosis TABLE 8 | Correlations among demographic and group predictors, and between definitions was predicted by group type only (B = −1.27, OR = predictors and variables showing significant differences in the two group sets. 2 0.079, Wald χ = 22.63, p < 0.001; and B = 0.67, OR = 3.820, Wald χ = 9.04, p < 0.01 respectively). Group type was also the Employment Education Group sole predictor of health-related goals distribution (B = 0.66, OR PwMS/CONTROL 1 = 3.783, Wald χ = 11.20, p < 0.001). In the caregivers/control 2 group set, positive affect was Employment – 0.11 0.42*** significantly predicted by group type and education level, Education 0.11 – 0.08 together explaining 36.9% of the variable variance (r = 0.334, Positive affect 0.28** 0.22* 0.39*** F = 61.25, p < 0.001, and r = 0.035, F = 6.75, p < 0.05 Negative affect −0.004 −0.15 0.20* respectively); no significant predictors were instead identified Satisfaction with life 0.35*** 0.24** 0.29** for negative affect. Group type emerged as the only significant Happiness with health 0.31* 0.05 0.33* predictor for the three remaining variables: satisfaction with life Happiness def.—psychological 0.16 0.13 0.49*** (r = 0.071, F = 9.36, p < 0.01, explaining 2.2% of the variable Happiness def.—family −0.04 −0.09 −0.29** variance); happiness with community (r = 0.088, F = 10.80, Goals—health −0.19* −0.03 −0.30*** p < 0.01, explaining 2.6% of the variable variance) and leisure CAREGIVERS/CONTROL 2 meaningfulness (r = 0.141, F = 20.04, p < 0.001, explaining Employment – 0.12 0.25 4.2% of the variable variance). Education 0.12 – 0.19* Positive affect 0.26** 0.30*** 0.58*** Negative affect 0.11 0.03 0.12 DISCUSSION Satisfaction with life 0.16 0.15 0.27** Happiness with community 0.06 0.21* 0.30** This study aimed at investigating different facets of well-being Leisure meaningfulness 0.23** 0.12 0.38*** among persons with MS and their caregivers from an integrated perspective. At the conceptual level, the eudaimonic dimensions *p < 0.05; **p < 0.01; ***p < 0.001. “Group” is a dummy variable with 1 for PwMS or of goal pursuit and meaning-making were jointly investigated caregivers and 0 for the respective control groups. 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 Based on these results, linear regressions with stepwise life domains, and findings were compared with those reported by selection were carried out to investigate the role of employment two groups of participants sharing similar demographic features, status, education level and group classification as predictors but not exposed to experiences of chronic illness or caregiving. of the hedonic well-being dimensions: positive affect, negative At the empirical level, the adoption of a mixed method approach affect, and satisfaction with life. In addition, linear regressions gave participants the opportunity to freely describe their present (with scaled outcome variables) and binary logistic regressions evaluations and future expectations. (with categorical outcome variables) were performed to assess the role of demographic features in the findings obtained through The Private Context of Well-Being: A the EHHI for which group differences were identified. More specifically, for the PwMS/control 1 group set regressions were Shared Perspective conducted for happiness with health, psychological and family- The information collected through the EHHI allowed us to related definitions of happiness, and health-related goals. For the contextualize individuals’ perceived well-being within the major caregivers/control 2 group set regressions were conducted for life domains. Regardless of group inclusion, in both qualitative happiness with community and meaningfulness of leisure. and quantitative answers participants primarily referred to few As concerns PwMS and control 1, for the dependent variable life domains, basically circumscribed to the private sphere. positive affect the two predictors entered in the model were Family, personal life and health distinctly emerged across groups group type and education level, together explaining 18% of the as the predominant contexts of happiness, meaningfulness and 2 2 variable variance (r = 0.143, F = 20.13, p < 0.001, and r goal investment. A substantially lower relevance was attributed = 0.039, F = 5.41, p < 0.05 respectively). Negative affect was to the broader contexts of work and interpersonal relations; significantly predicted by group type (r = 0.041, F = 5.27, finally, the public domains of community and society, and the p < 0.05), however explaining only 4.4% of the variable variance. transcendent sphere of spirituality/religion were almost absent Satisfaction with life was significantly predicted by employment from participants’ qualitative answers, and they scored lowest status and education level (r = 0.121, F = 16.69, p < 0.001, and in rank on the scales. Overall, these findings largely confirmed r = 0.042, F = 5.82, p < 0.05 respectively), but not by group previous EHHI studies conducted across countries on adult type; the two significant predictors together explained 16.3% of samples belonging to the general population (Delle Fave et al., the variable variance. The level of happiness with health was 2011; Delle Fave et al., 2013b). predicted by group type (r = 0.126, F = 17.47, p < 0.001), and Across groups, participants who referred to family in their employment status (r = 0.048, F = 7.04, p < 0.01), together definition of happiness mainly focused on the dimension of explaining 17.4% of the variable variance. The distribution of sharing. This finding is consistent with the models emphasizing participants citing psychological and family-related happiness the primacy of relational connectedness in humans (Richardson, Frontiers in Psychology | www.frontiersin.org 10 December 2017 | Volume 8 | Article 2216 Delle Fave et al. Psychosocial Well-Being in Multiple Sclerosis 2012). As specifically concerns persons with MS and their followed across groups, while only a negligible percentage of caregivers, these results are also consistent with the evidence participants mentioned spirituality/religion, community/society, of a shared process of adaptation to illness, based on and leisure. A significantly higher percentage of caregivers communal growth and search for meaning (Pakenham, 2005; reported less than three meaningful things compared to control Pakenham and Cox, 2009; Ackroyd et al., 2011). Among the participants; this difference may be related to the narrower psychological definitions of happiness, eudaimonic dimensions range of daily opportunities that characterizes the caring role were predominant; inner harmony was reported by the highest (Mausbach et al., 2011). percentage of participants across groups, followed by self- When describing recent situations of intense happiness, most actualization and personal growth. This finding, consistent with participants across groups referred to events shared with the previous evidence (Delle Fave et al., 2016), provides further family: birthdays, marriages, holidays, but also receiving a good support to the view of happiness as connectedness, at both the news concerning family members. Leisure and interpersonal inner and relational levels (Kjell, 2011). Most of the research relationships, following in rank, were mentioned by a relatively hypotheses concerning happiness definitions were however high percentage of participants across groups only in the context not confirmed. Against expectations, the percentage of PwMS of this answer. Finally, and in line with expectations, health was referring to health and the percentage of caregivers referring to quoted almost exclusively by PwMS. It is also important to note family were not significantly higher, compared to their respective that over one third of the caregivers could not identify any recent control groups. Similarly, against expectations no difference was situation of intense happiness. detected between caregivers and control 2 in the percentage of participants quoting leisure. Only two unexpected differences Extraordinary Circumstances, Ordinary emerged between PwMS and control 1: a lower percentage of PwMS provided psychological definitions of happiness, and a Experiences of Well-Being higher percentage mentioned family related ones. In both cases, Research has repeatedly emphasized the negative psychological group classification emerged as the only significant predictor. consequences of living with MS as a person or a caregiver, The overall relevance of family has been widely documented especially at the emotional level. Our findings were only among persons with MS (Ryan et al., 2007; Pakenham, 2008a). partially consistent with the literature, rather emphasizing the It is however worth noting that in this study persons with “ordinariness” of PwMS and caregivers (Saravanan et al., 2001) MS, similarly to participants in the other groups, emphasized in their quantitative ratings of global and domain-related well- sharing rather than receiving support and rewards from family— being. as it could be expected from individuals facing a progressively As concerns domain-related happiness and hedonic increasing dependence on their caregivers. well-being (assessed as positive and negative affect, and Across groups, the majority of participants mentioned family satisfaction with life), PwMS scored significantly lower as a major future goal, primarily referring to its intrinsic value than control participants in happiness with health (thus and to the well-being of family members. Work followed in confirming expectations) and in positive affect. Although rank, while marginal relevance was attributed to extra-family participants’ group emerged as the strongest predictor of the two relations, and to social, community and spirituality issues. As variables, unemployment further contributed in predicting lower hypothesized, a significantly higher percentage of persons with happiness with health, and lower education level in predicting MS quoted health, compared to control 1; group classification lower positive affect. PwMS also reported significantly lower was the only predictor of this result. All the other hypotheses levels of life satisfaction, but employment status and education, were not confirmed. It is worth noticing that, compared to their rather than presence of MS, emerged as significant predictors respective control groups, a significantly higher percentage of of this result. No differences instead emerged between PwMS PwMS and caregivers reported less than three future goals. This and control participants for negative affect, despite the (weakly) result can be related to the perceived uncertainty highlighted significant predictive role of group classification. To this purpose, in the MS literature, and in studies involving people with it is worth mentioning that negative affect values were on average other chronic diseases (Bensing et al., 2002; Tams et al., 2016). higher in the control group. The comparison between caregivers Uncertainty leads individuals to focus on the present rather and their control group highlighted a significant difference in than on future planning; this aspect can be even more relevant positive affect, with the former scoring significantly lower. Group in diseases such as MS, which entails a higher margin of type was the strongest predictor of positive affect; the additional unpredictability, compared with other chronic and degenerative though limited contribution of lower education level replicated conditions (Alschuler and Beier, 2015). the findings detected between PwMS and control 1. Group type Family also emerged as a key meaningful thing; the vast emerged as significant predictor of satisfaction with life as well, majority of participants across groups referred to its intrinsic though with marginal explanatory relevance. value—having a family, a partner, children, siblings were The lower levels of positive affect reported by PwMS and identified as valuable components of life per se. The primacy caregivers were consistent with the literature highlighting the of intimate relationships in the meaning making process largely emotional burden of chronic disease; nevertheless, education confirmed previous evidence obtained with a variety of samples emerged in both cases as an additional environmental predictor, (Baumeister and Leary, 1995; Lambert et al., 2010; Taubman – suggesting that emotions are—at least partially—socially Ben-Ari et al., 2012). Work, health and interpersonal relations constructed. In particular, this finding provides support to the Frontiers in Psychology | www.frontiersin.org 11 December 2017 | Volume 8 | Article 2216 Delle Fave et al. Psychosocial Well-Being in Multiple Sclerosis role of education as a major objective indicator of hedonic or communal competences, rather than being considered as weak subjective well-being, regardless of health conditions (Kroll and and low-performing members of the society. Their psychological Delhey, 2013). The same consideration can be made as regards and relational competences, laboriously built over time, could satisfaction with life, whose levels were not predicted by group be rather shared to the benefit of others. Community Based type, but by the social opportunities derived from employment Rehabilitation (CBR) programs are rooted in this view, aimed status and education, thus confirming previous evidence (Oishi at empowering persons with disabilities through the promotion and Diener, 2014). The lack of group differences in negative and acknowledgment of their active community role (Khasnabis affect levels confirmed instead the conceptual and empirical et al., 2010). Across countries, people experiencing disease are independence of positive and negative affect (Seib-Pfeifer et al., often founders or active members of associations, promoters of 2017), as well as the importance to consider well-being and fundraising campaigns in support of biomedical research, civil ill-being as partially independent domains of experience, rather rights activists. Therefore, health professionals could approach than as opposite poles of a single continuum (Keyes, 2007). them as experts who can offer first-person knowledge of a In this study eudaimonic well-being was quantitatively specific condition, and not only as patients to treat and caregivers assessed as the level of meaningfulness associated with different to instruct (Greenhalgh, 2009). Their social involvement could life domains. Findings did not support the study hypotheses, be extended to educational programs and other community as PwMS and caregivers did not differ from control groups initiatives, allowing them to share their resources and enjoy in family and health related meaningfulness. An unexpected recognition as full-fledged members of the society. Although difference emerged between caregivers and control 2, with the this change implies an overall revision of the health culture, the former attributing significantly lower meaningfulness to the advantages would be remarkable, as efforts in this direction could leisure domain. This result, solely predicted by participants’ lead to a more inclusive and participative society. group, can be related to the restrictions in daily life opportunities experienced by family caregivers (Becker, 2011). Since under Study Strengths and Limitations these circumstances leisure activities get often sacrificed first, The major strength of the present study is the investigation downplaying their meaning and relevance can help caregivers of hedonic and eudaimonic dimensions of well-being among adjust to the related constraints (Pakenham and Cox, 2009). 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- Patients and Caregivers: Social Assets Gonzales et al., 2004), allows for contextualizing scaled ratings Beyond Clinical Labels within qualitative, semantically richer answers. In this study, the This study highlighted that overall persons with MS and their domain-related ratings of happiness and meaningfulness could caregivers do not differ from healthy people in their experience be combined with a fine grained description of the same domains, of hedonic and eudaimonic well-being. These findings are their present and future relevance, and their relation to well- consistent with the literature showing that individuals and being. The findings provided an integrated representation of the families mobilize a variety of resources in order to adjust daily activities, contexts and relationships in which participants’ to disease conditions. At the same time, they offer further meaning-making process, goal pursuit and happiness experiences suggestions. Not only have these people adjusted to disease; they took place. To our knowledge, no studies of this kind are available pursue value-driven goals, cultivate inner harmony and balance, in the psychological literature on MS, and more generally on invest their energies in meaningful activities and relationships. chronic diseases. Persons with MS do not seem to be primarily focused on their This study has limitations as well. First of all, the cross- own health and related needs; they are rather actively engaged sectional design prevented from identifying causal relationships in sharing experiences and collaborating to the promotion of among variables. Although the circumscribed observation field family well-being (Bogosian et al., 2017). As for caregivers, their allowed for an in-depth analysis of well-being, the negative life trajectory—although forcefully disengaged from extra-family impact of disease and caregiving was not explored. The sample socialization and leisure—is grounded in personal and relational sizes were relatively small. Disability levels of the persons with values, despite the costs emerging at the hedonic well-being level. MS did not reflect the whole range of progression stages: the In general, the present study did not highlight group differences inclusion of participants with very severe disability could lead in participants’ level of engagement in public roles and social to different results, at both the qualitative and quantitative activities, thus showing that active involvement in community levels. All participants were Italians, thus belonging to a and society is related to cultural dimensions rather than health specific socio-cultural context: this feature increases reliability conditions. in the comparison of results across samples, but it prevents Overall, these findings can be considered as a provocative from generalizing results to countries characterized by different claim for a change in perspective, as concerns the social healthcare, welfare and value systems. representation of health. This claim, consistent with the ICF model, is based on theoretical and empirical evidence. As Future Directions highlighted by studies investigating resilience (Walsh, 2015), The findings from this study shed light on participants’ individuals and families experiencing chronic diseases should be experience of well-being, in the context of their daily activities valued and appreciated for their ability to develop personal and and social roles. It is however important to consider that both Frontiers in Psychology | www.frontiersin.org 12 December 2017 | Volume 8 | Article 2216 Delle Fave et al. Psychosocial Well-Being in Multiple Sclerosis individuals and their contexts are dynamic entities, changing version to be published; and agreement to be accountable for over time while interacting with each other; this further level all aspects of the work in ensuring that questions related to the of complexity, endorsed by the ICF model, can be satisfactorily accuracy or integrity of any part of the work are appropriately evaluated only through the collaboration of researchers from investigated and resolved. All the other co-authors provided different disciplinary fields. As specifically concerns psychology, substantial contributions to the acquisition and interpretation of relevant contributions could derive from community and data; critical revision of the work at the conceptual level; final cultural psychology, with their focus on the interaction dynamics approval of the version to be submitted; and agreement to be between socio-cultural practices, individual experience and accountable for all aspects of the work in ensuring that questions collective behaviors (Christopher and Hickinbottom, 2008; Di related to the accuracy or integrity of any part of the work are Martino et al., 2017). appropriately investigated and resolved. A stronger interdisciplinary collaboration is especially needed in the light of a specific result emerged from this study: persons FUNDING with MS, their caregivers and the control groups reported low levels of happiness and meaningfulness in community This study was supported by FISM—Fondazione Italiana Sclerosi and society issues; in addition, these domains were almost Multipla [Grant Nr. 2011/R/5], with MB as principal investigator. absent from their lists of goals, meaningful things, and occasions for happiness. This finding may be interpreted as ACKNOWLEDGMENTS an alarming sign of civic disengagement. However, it confirms evidence obtained in other studies conducted in individualistic This study was made possible by the engagement and dedication societies (Delle Fave et al., 2011). We consider this result of many people. The authors would like to express their gratitude as a general warning for researchers, practitioners and policy to the persons with multiple sclerosis, their caregivers and the makers, highlighting the pressing need to promote a culture of participants in the control groups who carefully completed the interconnectedness (Prilleltensky, 2005), in order to contrast the questionnaires and scales used in this work. The complex data deterioration of community networks presently emerging across collection was conducted through the active collaboration of nations. 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 AUTHOR CONTRIBUTIONS (ASST Lariana, Como, Italy), Giovanna De Luca and Marco ADF and MB provided substantial contributions to the Onofri (University G. D’Annunzio, Chieti, Italy). 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Predictors of subjective well-being among individuals with Multiple manuscript; decision to submit the manuscript for publication. ADF, MB, BA, MP, Sclerosis. Clin. Neuropsychol. 21, 239–262. doi: 10.1080/13854040600582460 SC, MF, EM, MG, MV, MPA, and EP declare that the research was conducted in Ryan, R. M., and Deci, E. L. (2001). On happiness and human potentials: a review the absence of any commercial or financial relationships that could be construed of research on hedonic and eudaimonic well-being. Ann. Rev. Psychol. 52, as a potential conflict of interest. BG received personal grant for speaking and 141–166. doi: 10.1146/annurev.psych.52.1.141 advisor activities from Biogen and TEVA. FP received personal grant for speaking Saravanan, B., Manigandam, C., Macaden, A., Tharion, G., and Bhattacharji, and advisor activities from Almirall, Bayer, Biogen, Celgene, Merck, Novartis, S. (2001). Re-examining the psychology of spinal cord injury: a meaning Roche, Sanofi, TEVA. MPA received research grants and honoraria as a speaker and centered approach from a cultural perspective. Spinal Cord 39, 323–326. member of advisory boards from Bayer, Biogen, Merck, Novartis, Roche, Sanofi doi: 10.1038/sj.sc.3101149 Genzyme and Teva. AL served as Advisory Board Member for Bayer, Biogen, Seib-Pfeifer, L.-E., Pugnaghi, G., Beauducel, A., and Leue, A. (2017). On the Genzyme/Sanofi, Merck Serono, Novartis, Teva; this author also received travel replication of factor structures of the Positive and Negative Affect Schedule grants and honoraria from Bayer, Biogen, Genzyme, Merck Serono, Novartis, (PANAS). Pers. Indiv. Diff. 107, 201–207. doi: 10.1016/j.paid.2016.11.053 Sanofi, and Teva, as well as travel and research grants from Associazione Italiana Slife, B. D., and Richardson, F. C. (2008). Problematic ontological underpinnings of Sclerosi Multipla; this author’s institution received research grants from Almirall, positive psychology: a strong relational alternative. Theory Psychol. 18, 699–723. Bayer, Biogen, Merck Serono, Novartis, Sanofi, and Teva. doi: 10.1177/0959354308093403 Song, L.-Y., and Singer, M. (2006). Life stress, social support, coping and depressive Copyright © 2017 Delle Fave, Bassi, Allegri, Cilia, Falautano, Goretti, Grobberio, symptoms: a comparison between the general population and family caregivers. Minacapelli, Pattini, Pietrolongo, Valsecchi, Amato, Lugaresi and Patti. This is an Int. J. Soc. Welfare 15, 172–180. doi: 10.1111/j.1468-2397.2006.00386.x open-access article distributed under the terms of the Creative Commons Attribution Stucki, G., Zampolini, M., Juocevicius, A., Negrini, S., and Christodoulou, N. License (CC BY). The use, distribution or reproduction in other forums is permitted, (2017). Practice, science and governance in interaction: European effort for the provided the original author(s) or licensor are credited and that the original system-wide implementation of the International Classification of Functioning, publication in this journal is cited, in accordance with accepted academic practice. Disability and Health (ICF) in Physical and Rehabilitation Medicine. Eur. J. No use, distribution or reproduction is permitted which does not comply with these Phys. Rehab. Med. 53, 299–307. doi: 10.23736/S1973-9087.16.04436-1 terms. Frontiers in Psychology | www.frontiersin.org 15 December 2017 | Volume 8 | Article 2216

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