Diagnosis and therapeutic landscape of MS

Revised diagnostic criteria for MS complemented by the results of magnetic resonance imagining (MRI) tests allow for a breakthrough in management of this incurable disease [1]. Clinical diagnosis can be accelerated, and early introduction of immunomodulatory therapy constitutes grounds for prevention of the subsequent consequences of the disease. Continuous treatment of MS has been present and widely available for more than 20 years. Immunomodulatory treatment changes the natural course of the disease and, ideally, stops its progression [2,3,4]. Nowadays, there are a number of therapeutic options available for patients, particularly in the relapsing-remitting course of MS (RR-MS). Selection of the best therapeutic strategy depends on the disease activity. In Poland, therapeutic modalities relying on the use of immunomodulatory injections are widely used and constitute a foundation of treatment.

Socio-economic impact of MS

According to “Ready to Work” Report of 2011, 80% of patients leave employment within 15 years after the diagnosis [5]. More than 4,600 MS patients from over 100 countries considered the possibility of continuing education and employment as the most important issue for young MS patients [6]. Chronic diseases negatively affect the patients’ productivity. Over the course of the disease, the situation is further aggravated by the changing employment status of the caregivers. Half of the caregivers are forced to modify their professional life in order to provide care to the family members affected by MS [5]. In this respect, MS is one of the most cost-intensive diseases [7,8,9].

Predictors of employment in MS patients

There are factors known from previous studies that facilitate staying in employment such as: higher educational attainment, low severity of fatigue, lower level of physical disability and cognitive disorders [10,11,12,13]. However, it should be noted that therapeutic strategies that prevent from uncontrolled disease progression should be considered as one more predictor of employment [14]. It was documented in the 11-year-long observation of early beta-interferon treatment—the BENEFIT trial (Betaferon treatment in newly diagnosed MS), that 73% of patients undergoing continuous treatment were in employment. This result accounts for most of the study group, as 81% of the patients were professionally active at the introduction of the treatment [15].

Role of self-motivation in employment status of MS patients

Only a few studies concern the influence of personality or self-motivation as an independent factor for staying in employment [16,17,18,19]. Therefore, the present study includes the assessment of the hope for success among MS patients–the notion of believing in having a strong will and in one’s ability to find solutions [20,21]. The possibility to assess motivation, including the subjective as well as objective evaluation of factors which facilitate being professionally active, provides a broad view on the issue of employment, from the perspective of both the patient and the therapist.

Statistical analysis

The statistical analysis was performed using R (The R Project for Statistical Computing)- version 3.2.3. In descriptive statistics the following tests were used: Fisher’s exact test to establish differences in proportions, T-test to establish differences in quantitative data (for normally distributed data), Wilcoxon test to establish differences in quantitative data (for non-normally distributed data), Anderson-Darling test to establish the normality of given quantitative data. In order to determine the influence of main neurological rates (EDSS, FSS, PQD5 and KNS) on unemployment status, the univariate and multivariate logistic regressions were performed.

Employment status

The study group comprised patients aged from 18 to 72 (mean age 40.6 years), with a mean EDSS score of 2.37. 60% (n = 90) of the participants were professionally active. Within this group, 51% (n = 46) performed physical work and 49% (n = 44) were white collar workers, including full-time students and pupils (n = 6). Among professionally active patients, 14 employees were entitled to a retirement or disability pension. The group of professionally inactive patients (n = 60) comprised patients who reached the retirement age (n = 5), were entitled to a disability pension (n = 43), a rehabilitation benefit (n = 3), as well as family dependents (n = 9). Disability pensions or rehabilitation benefits for all the patients were awarded due to MS.

Demographic variables

Patients professionally inactive were older (p<0.001), with the mean difference from the employed group of 8.19 years. The majority of the participants were female, yet no difference was observed as to remaining in employment. Unemployed patients were less educated (p = 0.007). The number of children was higher in the unemployed group (p<0.01). The employment status was not determined by the place of residence.

Disease duration and treatment delay

Progressive forms of MS were present in 28% of the unemployed group and led more frequently to unemployment (p<0,01). The patients who left employment had been showing symptoms for more years (p = 0.042) and the treatment had been initiated later (p = 0.054). A mean disease duration in the unemployed patients (40% of all patients) was almost 5 years. Although the disease duration was similar in both groups, the unemployed patients were currently less frequently undergoing immunomodulatory treatment (p<0,01). We didn’t find any difference regarding the age at diagnosis (p = 0.08), delay in treatment initiation (p = 0.748) or duration of treatment (p = 0.879) between both analysed groups—Table 1.

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Table 1. Socio-demographic and clinical characteristic of MS patients.

https://doi.org/10.1371/journal.pone.0194117.t001

Taking into account young population with major clinical presentation of RRMS and rather short disease duration, we further analysed the clinical differences between both groups.

Disease severity and self-motivation

The level of disability, considered to be indicative of disease advancement, was significantly higher in patients with progressive types of MS with mean EDSS score 5.425. Whereas patients with RR-MS were characterised by lower level of disability with mean EDSS score 1.719. The average EDSS score for the unemployed MS patients was twice as high as that of the employed (p<0.001). The analysis of FSS showed a higher level of fatigue in the unemployed group (p<0.001). More severe subjective cognitive problems in daily living (PDQ5) were found in the unemployed MS patients (p = 0.004). Moreover, motivation measured using the KNS scale was less prominent in this group (p = 0.003)—Table 2.

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Table 2. Main predictors of unemployment.

https://doi.org/10.1371/journal.pone.0194117.t002

Factors influencing employment

Patients who withdrew from employment had higher EDSS scores. The cut-off EDSS > 3 was established with OR 13.227, (p<0.01). Also the progressive type of disease was a strong predictor of unemployment OR 11.465, (p<0.01). Similarly, the patients suffered from more severe fatigue, the cut-off FSS>4 was connected with OR 4, (p<0.01), they showed lower self-motivation (KNS) (OR = 0.941, p<0.01). Intellectual deficits experienced by these patients interfered to a greater extent with daily life activities (PDQ5) (OR = 1.115, p<0.01) Table 3.

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Table 3. Univariate logistic regression analysis of factors influencing employment.

https://doi.org/10.1371/journal.pone.0194117.t003

Multivariate analysis revealed that moderate level of neurological disability (EDSS>3) (OR = 11.089, p< 0.01), more severe fatigue (FSS>4) (OR = 2.65, p = 0.048) and less present self-motivation (KNS) (OR = 0.947, p = 0.042) were independently associated with unemployment Table 4.

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Table 4. Multivariate logistic regression analysis of factors influencing employment.

https://doi.org/10.1371/journal.pone.0194117.t004

The employment situation of a young adult diagnosed with an incurable disease has been widely discussed in the literature on the subject in many countries [25,26,27,28] Therefore, this paper provides data on factors determining unemployment among MS patients in Poland. To our knowledge, this is the first report about demographic and clinical factors leading to unemployment in Polish MS patients who undergo immunomodulatory therapy. The report indicates that demographic factors such as higher age at the moment of participation in the research study and on the onset of first symptoms have a negative influence on the employment status. We found the number of children to be significantly higher among unemployed women. It may potentially indicate a decision to withdraw from professional activity due to having many children, not because of severity of symptoms or disease advancement only.

Despite higher educational attainment, only some of the participants were in employment. Less educated MS patients are more likely to withdraw from employment. These findings are consistent with the results obtained in other countries [29,30,31]. It is often the case that patients suffering from MS take less well-paid jobs so as to stay in employment [8,32], while their healthy counterparts enjoy their career opportunities with the perspective of an income increase [9].

In terms of the disease duration, 40% of the patients from the cohort were unemployed approximately after 5 years from the diagnosis despite the fact that most of them were in treatment. Flachenecker et al.[26] reported the same proportion of unemployment even after 3 years of disease duration.

The most significant determinant of unemployment in our study was EDSS >3 which increased the possibility of unemployment by 13 times even though the patients were not severely disabled, having a mean EDSS score of 2.37 From the cut of EDSS 3.0, day-to-day functioning is gradually limited and the conversion to progressive disability became more probable. Therefore the higher disability levels as well as being diagnosed with progressive types of MS, increase the risk of unemployment by several times. This correlation is a natural result of limited abilities of the patients both factors are strongly interlinked.

MS, as a chronic central nervous system (CNS) disease, affects not only motor functions, which is well represented by EDSS scores, but also leads to impaired cognition and general fatigue [24,33, 34,35,36].

The studies by O’Connor et al. [37] and Simmons et al. [8] indicate that chronic fatigue, impaired memory and concentration are important factors limiting employment. In our study, more than 66% of the unemployed patients experienced fatigue to the point of limiting activities of daily living (FSS score >4.0). This factor increases the risk of unemployment by 4 times. Additionally, self-reported problems with concentration, assessed with the PDQ5 scale, were more prominent in the unemployed group.

The analysis of independent factors leading to unemployment identified the following factors: moderate disability, severe fatigue and lower level of self-motivation.

The newly analysed factor of self-motivation seems to have been neglected in terms of its relation to long term influence on employment. In our cohort there were 14 still professionally active patients who were entitled to a retirement or disability pension. The fact that these patients continued to be employed while the majority withdrew from employment is worth studying.

Having low hopes for success is associated with an attitude of avoidance, i.e. passive attitude [38,39]. Motivation to achieve success, analysed in this paper according to Snyder, constitutes a learned thinking pattern which serves as an adaptive mechanism to change the life situation, allowing the person to be more flexible and persistent [40]. Moreover, it is linked with high self-esteem and a higher life satisfaction level, which provides more flexible approach to difficult life events and facilitates finding alternative solutions.

Assessment of self-motivation may allow identification of the patients who engage in preventive actions and show a greater perseverance in terms of treatment adherence. Higher level of hope for success corresponds with better adaptation strategies, higher level of general self-esteem and the ability to endure physical pain [41,42].

Both physical disability as well as non-motor symptoms of MS greatly affect motivation and work ability of patients. The focus of interdisciplinary preventive therapeutic teams should be on establishing long-term motivation and non-motor MS symptoms.