*Article* **Physical Activity, Resilience, Sense of Coherence and Coping in People with Multiple Sclerosis in the Situation Derived from COVID-19**

**María Mercedes Reguera-García 1 , Cristina Liébana-Presa 1,\* , Lorena Álvarez-Barrio <sup>2</sup> , Lisa Alves Gomes <sup>3</sup> and Elena Fernández-Martínez <sup>1</sup>**


Received: 2 October 2020; Accepted: 4 November 2020; Published: 6 November 2020

**Abstract:** The confinement forced by COVID-19 can have repercussions on the health of people diagnosed with multiple sclerosis. The objective of this study is to analyze the relationships between physical activity, a sense of coherence, resilience and coping among people diagnosed with Multiple Sclerosis during the health emergency situation. To achieve this goal, this transversal descriptive study included 84 patients that belonged to multiple sclerosis associations during the period of confinement. Participants filled out the Physical Activity (IPAQ-SF), Sense of Coherence (SOC-13), Resilience Scale (ER-14) and coping (COPE-28) questionnaires. The results showed that the average age was 46.9 and that 67.9% had Relapsing Remittent Multiple Sclerosis diagnosed on average 13.9 years ago. They had a high degree (33.3%) and moderate degree (34.5%) of physical activity, high levels of resilience, while the level of a sense of coherence was average and the most commonly used strategies for coping were active confrontation and religion. Physical activity was not related to the rest of the studied variables, but there were correlations between the other variables. The people with multiple sclerosis who belong to patient associations have remained physically active during the obligatory confinement period and have elevated degrees of resilience and an average sense of coherence, as well as using suitable coping strategies, which is why the social-health resource of belonging to a patient association could be boosting these variables that are beneficial to their health.

**Keywords:** multiple sclerosis; COVID-19; physical activity; resilience; sense of coherence; coping

### **1. Introduction**

The world is currently going through a pandemic due to a new strain of coronavirus, the severe acute respiratory syndrome (SARS-CoV-2). There are many aspects that remain unknown about this illness. At the moment there are no treatment strategies, nor is there a vaccine [1], and many countries have adopted social distancing and/or quarantining to break the chain of transmission of the COVID-19 illness. In Spain, a state of alarm was declared in order manage the health crisis situation brought about by COVID-19 in March 2020, the Royal Decree 463/2020 of 14 March [2], in response to the growing number of cases of coronavirus in Spain; it imposed a national quarantine as a health measure. On 21 June 2020, the country entered the dubbed "new normality", introducing health measures such as the use of facemasks and social distancing. Current research [3] referring to the psychological impact of quarantine suggests that it is necessary to take into account the stress factors resulting from the situation (duration, fear of infection, frustration, fear, boredom, anger, insufficient information, lack of

basic supplies, financial losses and stigma) in order to ensure that the experience of quarantining be as tolerable as possible for the population [3,4].

It is not known how duration might affect the different collectives of people who already suffer from prior illnesses such as multiple sclerosis (MS). MS is chronic and autoimmune, affecting the central nervous system [5] and with an unstable clinical course with a wide range of cognitive and neurological effects [6]. It affects women more than men (3:1) [7] and is usually diagnosed between 20 and 40 years of age. Certain characteristics of MS, such as its unknown origin, its unpredictable course, its multiple symptoms and the current nonexistence of a definitive cure, affect the adherence to the treatment as well as the quality of life [8]. MS patients suffer functional deterioration, fatigue, psychological anxiety and limitations in their social relationships, all of which leads to a lesser quality of life for these people at both the physical and psychological level [9,10], giving rise to them experiencing a low self-efficacy due to the limitations of the illness itself [8]. Furthermore, this population of MS sufferers is more sensitive to suffering cognitive dysfunctions and mental health problems, especially depression followed by anxiety, since this affects them at a larger scale than the general population (more than 20% of the population) [11,12]. Those people who suffered problems of mental health before the quarantine and social distancing may be more vulnerable to adverse psychological effects [1]. Recent research indicates that exposure risks are the cause of COVID-19 infection in the MS population. Younger people with a lower socioeconomic status will have a higher risk of exposure and are potential targets for educational intervention and medical support to limit exposure. Delays in therapies and the interruption of MS care have also been observed [13].

There is a lack of previous research that includes the consequences deriving from the COVID-19 virus pandemic at the physical and psychological level in people suffering from MS. This is why research into physical activity, resilience, coping and a sense of coherence has been selected, as these are the variables related to well-being and the promotion of health.

In people with MS, the degree of physical activity is usually reduced, making them less physically active than the general population [14]. It is related to the degree of general incapacity and the limitation for walking in people with MS [15]. In these patients, vigorous activities are reduced, leading to the predominance of light activities and the fact that they spend quite a lot of time being inactive during the day [16]. The reduced participation, the limitation of movement and the degree of incapacity factors mean that the physical activity of the MS population is less than that of the general population, and consequently they do not obtain the benefits that come from carrying out physical exercise, leading to a greater propensity for their suffering certain illnesses, especially cardiovascular ones [17]. It is necessary to promote physical activity and include it in intervention programs, since a greater degree of physical activity is related to personal factors such as sociodemographics, the level of education, their work situation, mechanisms for self-regulation and the perception of barriers, especially those related to physical activity [16].

Resilience is characterized by the ability to achieve, retain or recover a degree of physical or emotional health after adversity [17]. In people with MS, disability is associated with success in adapting and recuperation. Greater levels of resilience are associated with a reduction in fatigue (20% less), greater participation and exercise, greater social and financial support, a healthier diet and better psychological health [17]. People with MS have reported that higher levels of resilience are associated with healthy behaviors and a more successful adaptation to changes in the life of the patients that result from their illness [18]. This variable has been studied in different populations [19], but rarely in patients with MS.

The sense of coherence is understood as a way of perceiving the world that allows stressful factors to be faced [20]. MS is a chronic illness with an unpredictable evolution [21], which includes suffering physical limitations and psychosocial difficulties that affect the quality of life of these people, threatening their sense of cohesion [8,22]. A greater sense of cohesion gives rise to better mental health and a lower negative effect, as well as warding off depression [8,23].

People with MS use different coping strategies in the different stages of the illness. In the first stages, emotional and avoidance-type strategies predominate; meanwhile, in the intermediate stages of the illness, the use of more active coping strategies predominate [24]. On certain occasions, the use of emotional-type strategies are related to a better psychological adaptation and those of avoidance are related to the prevention of a depressive reaction through the rejection of negative thoughts about the future progression of the illness when they are used in the first stages following the diagnosis of the illness [23]. Psychological interventions centered on one's resources to cope more effectively with the MS are necessary to bring a positive adaptation to the adverse situation [25].

Based on the aforementioned points, the following study has been carried out to obtain greater knowledge on the influence of this pandemic on the health/illness of people with MS. The objective will be to discover and analyze the relationships between physical activity, a sense of coherence, resilience and coping in people diagnosed with MS during the emergency health situation brought about by COVID-19.

### **2. Materials and Methods**

A cross-sectional, descriptive and correlational study was conducted.

### *2.1. Participants*

The population is made up of 1236 people with MS belonging to multiple sclerosis associations in Castilla y León (Spain) (See Table 1). Through a nonprobabilistic convenience sampling, 84 participants were selected.


**Table 1.** Multiple sclerosis associations of the Castilla y León region (Spain).

### *2.2. Instruments Used to Collect Data*

The short-format of the (IPAQ-SF) international questionnaire, validated in Spanish [26], is used with young adults and middle-aged people (15–69). It consists of four generic items to obtain information on physical activity related to health. It provides separate results for the three types of activity: vigorous intensive activities, activities of moderate intensity and walking. Obtaining the final result requires the totaling of the duration (in minutes) and frequency (in days) of these three types of activity, from which the physical activity is categorized in three levels: low, moderate and high. The "sitting time" is also registered. This is time dedicated to carrying out a sedentary activity [27].

The Resilience Scale, (ER-14), has 14 items to evaluate the degree of individual resilience [19]. The version was validated into Spanish by Sánchez–Teruel and Robles–Bello [28]. The ER-14 measures two factors: on the one hand, personal competence (11 items), and on the other hand, the acceptance of oneself and of life (three items). This scale presents a suitable internal consistency (α = 0.79) and validity of the calculated criterion with other measurements of general resilience (CD-RISC) (r = 0.87; *p* < 0.01). According to the scores: 98–82: very high resilience; 81–64: high resilience; 63–49: normal; 48–31: low; and between 30–14: very low. The reliability indices are suitable, demonstrating that it is a good instrument for measuring this variable [28].

The questionnaire on the Sense of Coherence (SOC 13) [20] is the Spanish-adapted version [29]. This scale consists of 13 items that are scored using a Likert-type scale of seven points. It contains three scales corresponding to the three dimensions of the construct: compression (α = 0.630), direction (α = 0.596) and significance (α = 0.594), and a total score (α = 0.799).

The questionnaire on coping COPE-28 [30] is the Spanish version (COPE-28) [31]. It is composed of 28 items scored using a Likert-type scale that goes from 0 (absolutely nothing) to 3 (a lot). These items are grouped together in pairs, producing 14 modes of coping: active coping (α = 0.452), instrumental support (α = 0.630), acceptance (α = 0.516), self-distraction (α = 0.482), venting (α = 0.302), planning (α = 0.585), religion (α = 0.880), denial (α = 0.458), self-blaming (α = 0.617), substance use, emotional support, positive reframing (α = 0.602), humor (α = 0.835) and behavioral disengagement (α = 0.654).

### *2.3. Procedure*

The data for the study was gathered online by means of Google Forms (https://forms.gle/ yu1gfoSX8h6L1HnV9). Prior to sending the questionnaire, all of the MS associations of Castilla y León (Spain) were contacted by telephone and e-mail. The members diagnosed with MS were requested to participate in the questionnaire using e-mail or WhatsApp. The gathering of the data took place between 28 March and 3 June 2020, during the period of confinement, due to the state of alarm imposed by the Spanish government for the management of the health crisis situation.

### *2.4. Data Analysis*

The analysis of the data was descriptive, analytical and correlational. It was checked that the adjustment did not comply with the normal distribution of the data by means of the Kolmogorov–Smirnov test with the Lilliefors significance correction. For this reason, nonparametric tests were carried out (U of Mann–Whitney) for the comparisons of averages between the groups. Nonparametric correlations were carried out in order to evaluate the associations between the quantitative variables, and the Spearman rho coefficients were obtained. A value of *p* < 0.05 was considered statistically significant.

The database and the statistical analyses cited were carried out using the SPSS 26.0 (Statistical Package for the Social Sciences) computer program (IBM, New York, NY, USA).

### *2.5. Ethical Considerations*

The people participating in the study were considered anonymous and the data confidential. Prior informed consent was requested for their collaboration. The data are collected and handled

respecting all of their rights and guarantees in accordance with those established in the UE2016/679 Regulation and in the Constitutional Law 3/2018 of 5 December on the Protection of Personal Data and digital rights guarantees. The study was approved by the ethics committee (ETICA-ULE-016-2020) of the University of León (Spain), which guarantees that the questions are both ethical and legal.

### **3. Results**

The sociodemographic characteristics of the participants are set out in Table 2. 84 people diagnosed with MS in the Spanish Autonomous Community of Castilla y León participated in the study. The results of the average and the standard deviation in terms of age were 46.9 ± 9.7 years old (M±SD). In relation to the clinical characteristics of the illness, the greatest part of the sample, 67.9%, was diagnosed with Relapsing Remittent Multiple Sclerosis (EMRR), followed by 25% diagnosed with its Secondary Progressive Multiple Sclerosis (EMPS). The descriptive values for the years of diagnosis of the MS were 13.9 ± 8.3 (average 15 years old, maximum 35 and minimum 1).


**Table 2.** Sample description.


**Table 2.** *Cont.*

The results obtained for the physical activity variable are set out in Table 3. The U Mann–Whitney test has been carried out, and no differences have been found between men and women. As regards the scores for the questions on physical activity, 28 participants (33.3%) have scored high, 18 (21.4%) low and 29 (34.5%) have achieved a moderate degree of physical activity.


**Table 3.** Descriptive statistics of physical activity in minutes per week.

Note: \* minutes/week. M; Mean. SD; Standard Deviation. Me; Median. IR; Interquartile Ranges.

The descriptive statistics of the resilience, sense of coherence and coping variables are set out in Table 4. No differences have been found in the sex of the participants for the scores of these variables. In the case of resilience, 30 (35.7%) participants were perceived as having a very high resilience, 37 (44%) were evaluated as having high levels, 14 (16.7%) scored within what is considered normal, and 2 (2.4%) and 1 (1.2%) obtained low values or very low values, respectively.

Table 5 details the correlations between the variables of the study. The results indicate that physical activity does not correlate significantly with any of the other studied variables; however, although in some cases they are weak, correlations are observed between resilience, sense of coherence and coping. Thus, the highest levels of personal competence (CP), acceptance of oneself and of life (De) for resilience are related to the highest scores of compression (CO), direction (Dir) and significance (Sig) for the sense of coherence. As regards coping, the strategies for active coping (A), acceptance (Ac) and positive reframing (Pr) are positively related to the total and to the two dimensions of resilience of personal competence (CP) and acceptance of oneself and of life (AV), as well as to the factor of significance (Sig) of the sense of coherence variable. The strategies of denial (D), self-blaming (Sb) and behavioral disengagement (Bd) show negative correlations with resilience and the sense of coherence which have been evaluated in the study. Substance use as a strategy (Su) correlates negatively with resilience, in particular with the dimension of accepting oneself and life (De).


**Table 4.** Descriptive statistics of resilience, sense of coherence and coping.

Note: ER-14: Resilience Scale. SOC-13: Sense of Coherence. COPE-28: Coping. SD: standard deviation. M: mean.



**Table 5.** Spearman's rho correlations of physical activity, resilience, sense of coherence and coping.

emotional support, Pr: Positive reframing, H: humor, Bd: behavioral disengagement.

### **4. Discussion**

The results revealed that the clinical and personal characteristics of the participants in the present study were similar to other studies conducted on patients with multiple sclerosis during the COVID-19 pandemic [1,13,32].

If we compare the time spent sitting down per week collected with the IPAQ of our sample with the study carried out by Moti et al. [33], which analyzed people with MS in America, we find that our population has a slightly higher value. Furthermore, our data show elevated levels of physical activity. These data seem to indicate a high participation in the activities proposed by the different associations and that they are concerned about their physical condition during this period of confinement. On the other hand, Hubbard et al. [34] observed that people with slight MS tended to do less physical activity in comparison with healthy people.

The results showed that people with multiple sclerosis who participate in associations presented high levels of resilience [19] overall and in the domains of personal competence and acceptance of oneself and of life. These values of adaptation to adverse situations are important for the well-being of people, as the studied sample went through 11 weeks of quarantine and time is considered as a predictive factor for acute stress disorder [3]. Furthermore, the effect of interventions that focus on promoting resilience and other personal resources are necessary to effectively cope with multiple sclerosis [25]. If the present study is compared to the work of Plowman et al. [17] in people with MS, the total resilience factors are very similar. On the contrary, the total resilience values and the I factor (personal competence) obtained in our sample are higher than those reached in the study carried out by Sánchez–Teruel et al. [28] in young healthy people. The case of populations with painful chronic pathologies such as fibromyalgia [35] highlights that our results are much higher (almost double) for this variable. It will be interesting to analyze the role of the associations in the promotion of the health of patients diagnosed with MS.

According to the data on the sense of coherence, those with multiple sclerosis who frequently attended associations in the Autonomous Community of Castilla y León had a total sense of coherence of 61.19 ± 12.252 during the confinement period. If we compare our data with the study of the sense of coherence in health personnel during the COVID-19 pandemic, we observe that our sample obtained higher mean values although they did not present symptoms of COVID [36]. Nonetheless, lower values than those of the present study are observed in populations with MS [37,38]. Conversely, persons with late effects of polio have obtained higher values, which could be due to their perception of the disability being less acute and to the fact that, upon having a later start, they have developed the capacity to understand, manage and be motivated when faced with stressful events and problems that come up during their lives [39]. If we compare our data with people without neurological problems and with a cardiac pathology, we observe that cardiac patients obtain worse values since they tend to be associated with other pathologies such as depression [40]. However, if we compare our sample with people older than 65–75, 76–80 and over 80, we see that all of the older people have higher scores in total (74 ± 3.44) and for decompression (27 ± 0.49), direction (23 ± 0.83) and significance (23 ± 1.09) [40].

The coping of the people with multiple sclerosis who participated in associations showed diverse levels according to the strategies used in comparison with people with multiple sclerosis who coped with the prognosis of their illness [29]; we observe that our population presented higher levels in terms of instrumental support 3.48 ± 1.52, substance use 3.52 ± 1.72, self-blaming 2.57 ± 2.16 and religion 4.55 ± 1.38. This leads us to think that they have less functional strategies when facing the COVID-19 situation due to their low self-esteem, perceived high levels of stress or psychological unease, as observed in French people facing serious illness or trauma [12]. In addition, people with MS can show increased discomfort due to having psychological problems due to alterations of cognitive dysfunctions, information processing, and immediate and episodic memory typical of MS [41]. Furthermore, the high values in religious coping lead us to think that they indicate a lower satisfaction with the decision that was taken to resolve the existence of stress [42]. Comparing our results on coping with values for healthy people, we find that emotional support, humor and positive revaluation present lower

values in our study [43]. These data are logical because people with MS offer to undertake maladaptive coping strategies [12].

Finally, the results of this study indicate that the level of physical activity of a person with MS, which is measured in terms of the mastery of activity (vigorous, moderate), the total and the time spent sitting down, is not related to resilience, coping or sense of coherence in the social confinement situation that derives from the COVID-19 virus. However, we have found theoretical studies which support that psychological aspects can be influenced by physical activity [9,44].

Regarding the relationships between resilience and sense of coherence, this study observed that there was a positive relationship between them. This means that the most resilient people present greater levels of a sense of coherence and vice versa. This could benefit patients with MS, since there is evidence that supports a relationship between positive dimension factors (resilience and/or optimism) and subjective emotional well-being [45]. Recent research on the impact of COVID-19 on German public health supports our data, proposing to improve the sense of coherence to improve resilience to stressors [46]. According to the results of the relationships between coping and resilience, we observe that if a patient has a higher score in the strategies of active coping, acceptance, emotional support and planning, he or she will present greater levels of resilience and will have a greater capacity for adapting to adverse situations; that is, strategies for positive coping or task orientation are related to a better mental health [47,48]. In the same way, coping strategies such as denial, self-blame, substance abuse, venting and behavioral disconnection are negatively related to resilience, since they are strategies that maintain stress, as has been observed by other researchers [43,49]. Concerning the relationships between sense of coherence and coping, they depend on the strategies, as not everybody behaves in the same way. However, we can highlight the negative relationship between both venting and behavioral disengagement and the dimension of compression of the sense of coherence. According to recent studies, the behavior of our sample is the same as in Bedouin women older than 61 with little education and few possibilities for finding a job [50]. In both populations, one can observe that when there is a sensation of there being no control over the results or of being at the mercy of luck, it is probable that they will have a sensation of impotence and use passive or evasive strategies such as behavioral disconnection or the strategy of venting, which consists in verbally expressing the negative parts of a stressful situation. For all of the points mentioned above in relation to the COVID-19 scenario, researchers such as Ornell et al. support three main factors for developing mental health strategies: (1) multidisciplinary teams; (2) regular and precise clear communications about the outbreak situation; and (3) psychological counseling services through electronic applications for the community in general and especially for vulnerable people [4].

This study presents limitations, such as the method of sampling and the selection of the participants, which may exclude MS patients who do not belong to associations; furthermore, other variables such as the level of education, degree of incapacity and the medication used could have been included. Furthermore, it would be desirable to carry out longitudinal studies during similar adverse situations in people with multiple sclerosis, which might include variables such as resilience, sense of cohesion and coping strategies, as well as other more specific physical capacities altered by the illness, such as balance, walking or strength.

### **5. Conclusions**

The majority of the participants in associations in Castilla y León (Spain) suffer from a type of Relapsing Remittent Multiple Sclerosis, followed by Secondary Progressive Multiple Sclerosis. In spite of the confinement/quarantine, the greatest part obtains high and moderate scores in physical activity. Although no relationship has been found between physical activity and the psychological variables, there is evidence of the influence of the latter on the well-being of the patients. It can be observed as those people who achieve high values in resilience, as well as in sense of coherence. Furthermore, the use of coping strategies in the sample confirms that active coping, acceptance, positive reframing, denial, self-blaming, behavioral disengagement and venting are negatively related to resilience and the sense of coherence.

This research may be useful in describing the degree of coping, sense of coherence, physical activity and coping strategies of people with multiple sclerosis in the confinement/quarantine situation deriving from the COVID-19 virus in Spain. Likewise, to detect possible problems and be able to establish psychological and or physical interventions for the associations of those affected with MS, this research might be repeated again in the future in order to promote health in similar situations.

**Author Contributions:** Conceptualization, M.M.R.-G., E.F.-M., C.L.-P. and L.Á.-B.; data curation, M.M.R.-G., L.Á.-B., C.L.-P. and E.F.-M.; formal analysis, E.F.-M., C.L.-P., M.M.R.-G. and L.Á.-B.; investigation, M.M.R.-G., L.Á.-B., E.F.-M. and C.L.-P.; methodology, E.F.-M., C.L.-P., L.Á.-B. and M.M.R.-G.; Project administration E.F.-M., C.L.-P., M.M.R.-G., L.Á.-B. and L.A.G.; resources, M.M.R.-G., E.F.-M., C.L.-P., L.Á.-B. and L.A.G.; software C.L.-P., E.F.-M., M.M.R.-G. and L.Á.-B.; supervision, M.M.R.-G., C.L.-P., E.F.-M., L.Á.-B. and L.A.G.; validation, E.F.-M., C.L.-P., M.M.R.-G., L.Á.-B. and L.A.G.; visualization, C.L.-P., E.F.-M., M.M.R.-G., L.Á.-B. and L.A.G.; writing—original draft, M.M.R.-G., L.Á.-B., C.L.-P. and E.F.-M.; writing—review and editing, E.F.-M., C.L.-P., M.M.R.-G., L.Á.-B. and L.A.G. All authors have read and agreed to the published version of the manuscript.

**Funding:** This research received no external funding.

**Conflicts of Interest:** The authors declare no conflict of interest.

### **References**


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International Journal of *Environmental Research and Public Health*

### *Article* **Daylight Saving Time and Spontaneous Deliveries: A Case–Control Study in Italy**

**Rosaria Cappadona 1,2,3 , Sara Puzzarini <sup>2</sup> , Vanessa Farinelli <sup>2</sup> , Piergiorgio Iannone <sup>1</sup> , Alfredo De Giorgi <sup>4</sup> , Emanuele Di Simone <sup>5</sup> , Roberto Manfredini 1,3,4,\* , Rosita Verteramo <sup>2</sup> , Pantaleo Greco 1,2, María Aurora Rodríguez Borrego 3,6 , Fabio Fabbian 1,3,4,\* and Pablo Jesús López Soto 3,6**


Received: 1 September 2020; Accepted: 25 October 2020; Published: 3 November 2020

**Abstract:** (1) Background: Although the current literature shows that daylight saving time (DST) may play a role in human health and behavior, this topic has been poorly investigated with reference to Obstetrics. The aim of this case–control study was to evaluate whether DST may influence the number of spontaneous deliveries. (2) Methods: A low-risk pregnancy cohort with spontaneous onset of labor (*n* = 7415) was analyzed from a single Italian region for the period 2016–2018. Primary outcome was the number of spontaneous deliveries. Secondary outcomes were: gestational age at delivery, type and time of delivery, use of analgesia, birth weight, and 5-min Apgar at delivery. We compared the outcomes in the two weeks after DST (cases) to the two weeks before DST (controls). (3) Results: Data showed no significant difference between the number of deliveries occurring before and after DST (Chi-square = 0.546, *p* = 0.46). Vaginal deliveries at any gestational age showed no statistical difference between the two groups (Chi-square = 0.120, *p* = 0.73). There were no significant differences in the secondary outcomes, as well. (4) Conclusions: DST has neither a significant impact on the number of deliveries nor on the obstetric variables investigated by this study.

**Keywords:** daylight saving time (DST); desynchronization; circadian rhythm; chronobiology; nursing; spontaneous delivery; midwifery; obstetrics

### **1. Introduction**

The mechanism that starts the spontaneous onset of labor still remains an unanswered question in the current literature [1]. In the obstetric field, scientific research is rich in studies, conducted both in vitro and in vivo, explaining the action of hormones in the onset and maintenance of labor during birth by correlating it with the circadian cycle [2]. Cortisol is the main stress hormone responsible for the normal adaptation of the neonate to extrauterine life. Placental corticotrophin releasing hormone (CRH) is active from the early weeks of pregnancy and determines its duration, the time of onset of labor, and the timing of delivery; moreover, its plasma levels increase rapidly near the beginning of spontaneous labor. Data suggest that CRH may have a pivotal role in activating the mechanism of delivery [3–5]. In addition, type of delivery, e.g., vaginal or caesarean, may determine a different stress response. Recently, a study compared the activity of 11beta-hydroxysteroid dehydrogenase type 2 (11β-HSD 2) in the placenta and the umbilical cord blood cortisol level between caesarean sections, with or without uterine contraction, and vaginal delivery groups [6]. No statistically significant differences in the activity of 11β-HSD 2 were found in placentas delivered via caesarean sections compared to vaginal deliveries, while umbilical cord blood cortisol in the elective caesarean sections group was significantly lower compared to the vaginal deliveries and intrapartum caesarean sections [6]. Sleep disturbances and alterations in the circadian rhythm can lead to an increase in the levels of stress hormones and the activation of a pro-inflammatory condition can trigger and enhance the activity of prostaglandins and oxytocin, by promoting uterine contractions, the dynamic phenomena of labor, and consequently, the onset of labor [4–7]. These alternations can also be induced by shift work; in fact, women who experience an alteration of their circadian rhythm due to work are significantly more exposed to a reduction in birth rate and a lower birth weight of their newborns [8]. The purpose of the so-called "Summer time" was to capitalize on natural daylight: by turning the clock one hour forward as the days get longer in Spring, sunset is delayed by this same hour, until the clock is set back again in Autumn. After a first phase of Daylight Saving Time (DST) policy, where single countries decided their own standards, not regulated, harmonization attempts began in the 1970s to facilitate the effective operation of the internal market, and this practice is applied in over 60 countries worldwide [9]. Since light–dark alternation influences the synchronization of circadian rhythms of most human systems, DST may cause a chrono-disruption by significantly altering circadian rhythm and negatively influencing sleep quality [10]. A growing amount of evidence showed that DST transition may have negative effects on health [11–16]. Thus, based on these premises, an international consensus statement suggested that DST cannot be encouraged and therefore, should be discontinued [17].

As for female reproduction, disruptions of circadian rhythms, e.g., shift work, jet lag, and DST, have been associated with poorer fertility and early pregnancy outcomes [18]. However, very few data are available on the effect of DST on the onset of spontaneous labor. Only one recent published study showed no significant association between DST circadian rhythm and the number of spontaneous deliveries [19]. Based on this hypothesis, we evaluated whether DST might affect the number of spontaneous deliveries in a single Italian region.

### **2. Materials and Methods**

A cohort of 7415 deliveries from low risk pregnancies with spontaneous onset of labor was enrolled in this case–control study. We used the Certificate of Childbirth Assistance (CedAP) data collection from one single Italian region, Emilia-Romagna, in the period 2016–2018. We identified the exact dates of DST during each year analyzed (Figure 1). Age and gestational age were evaluated. We defined the 2 weeks after DST as the exposure period and the 2 weeks before DST as the control period.

Exclusion criteria are shown in Table 1.

The primary outcome was defined as the number of deliveries, in particular the number of non-operative vaginal deliveries.

The rationale underlying the choice of the latter outcome was to exclude, as far as possible, the conditioning acted out by the health workers and the healthcare organizational system that weighs on the birth path, in order to bring out the influences that the endocrine and central nervous systems have on childbirth.

Secondary outcomes were:


operative vaginal deliveries.

Secondary outcomes were:

and post term beyond 41 weeks;

have on childbirth.

**Figure 1.** The two weeks belonging to the control period and the exposure period identified by daylight saving time (DST) during each year analyzed (2016–2018) are reported (grey is related with 2016, orange with 2017 and light blue with 2018). **Figure 1.** The two weeks belonging to the control period and the exposure period identified by daylight saving time (DST) during each year analyzed (2016–2018) are reported (grey is related with 2016, orange with 2017 and light blue with 2018).

> **Table 1.** The table shows the exclusion criteria applied on the sample. **Table 1.** The table shows the exclusion criteria applied on the sample.


Elective Cesarean section The primary outcome was defined as the number of deliveries, in particular the number of non-We also analyzed local data relating to individual provinces. In particular, the number of at term deliveries that occurred in the three-year period was considered in the various Emilia-Romagna

on the birth path, in order to bring out the influences that the endocrine and central nervous systems

• Gestational age delivery, defining preterm before 37 weeks, at term between 37 and 41 weeks,

• Type of delivery: vaginal delivery, cesarean section, or operative delivery;

provinces, in order to verify whether there were any differences related to DST. The statistical analysis of the data was performed with the "chi-square" test.

We considered a 5% error with a confidence interval of 95%, standard deviation of 0.5, and z-score of 1.96.

The differences in the number of deliveries across the Spring and Autumn shifts were assessed for significance using the Poisson Means Test.

Annual birth rate was calculated as the ratio between the number of births during the two weeks pre and post DST, and the mean value of births obtained by the sum of births in the previous and the considered year divided by 2. The ratio was then multiplied by 10<sup>3</sup> . This calculation was performed for any year considered in this study.

$$N(\mathbf{x}) = \frac{N(\mathbf{x})}{\left[\frac{\frac{\mathbf{B}(\mathbf{x}-1)}{2}}{2} + \frac{\mathbf{B}(\mathbf{x})}{2}\right]} \* 1000 \tag{1}$$

N = number of births during the index 2 weeks;

X = year considered;

x−1 = previous year;

B = total births in the Emilia-Romagna region of Italy.

In addition, we calculated the age and gestational age of participants. A logistic regression analysis was carried out, where the period including the two weeks pre and post DST was the dependent variable and the other parameters investigated in this study the independent ones. A 2-sided *p*-value < 0.05 was considered statistically significant. IBM Statistical Package for Social Science (SPSS 13.0 for Windows, SPSS Inc., Chicago, IL, USA) was used.

### **3. Results**

The mean age of the 7415 women was 31.4 ±5 years, and mean gestational age was 39.3 ± 1.4 weeks. Italian patients totaled 4801 (64.7%), whilst 35.3% were classified as non-Italian. The mean age of the women who delivered during the two weeks before DST were older than those who delivered during the two weeks post DST (31.6 ± 5.4 vs. 31.3 ± 5.3 years, *p* = 0.033); on the contrary, gestational age was not different in the two groups (39.3 ± 1.4 vs. 39.3 ± 1.4 weeks, *p* = ns). As regarding time of delivery, 2708 births occurred in the morning (36.5%), 2022 (27.3%) in the afternoon, and 2685 (36.2%) at night. Birth rate during the Spring and Autumn shifts in the three different years of the study are shown in Table 2.


**Table 2.** Birth rates in the years 2016, 2017, and 2018 are shown.

Data are expressed as \* 10 <sup>3</sup> .

### *3.1. Primary Outcome*

Our results showed a difference in the number of deliveries between the exposed and the control groups, although it did not reach statistical significance (Table 3).

No statistical difference has been observed in the analysis of the number of deliveries (*p* = 0.46), including mode of delivery (vaginal delivery, cesarean section, or operative delivery) and gestational age (term, preterm, post term). The same result was obtained analyzing the number of spontaneous deliveries (*p* = 0.73).

The differences in the number of deliveries and number of spontaneous deliveries across the Spring and Autumn shifts assessed by the Poisson Means Test were not significant (*p* = 0.37, *p* = 0.92, *p* = 0.6, and *p* = 0.65 respectively).


**Table 3.** Summarized data of results of primary outcome.

### *3.2. Secondary Outcome*

There were no significant differences in the secondary outcomes analyzed, between what was detected during the exposure period and what was found in the control period (Table 4).


**Table 4.** Summarized data of results of secondary outcomes.

Women's age was independently associated with delivery during the two weeks post DST (OR 1.010, 95% Confidence intervals 1.002–1.019, *p* = 0.021). All the other investigated parameters were not associated with the period of the DST. Therefore, every year of increasing age of the delivering woman increased the risk of event occurrence during the two weeks after DST of 1.0% (Table 5).

Finally, we assessed the percentages of type and time of delivery during the Spring and Autumn shifts, but we could not find any difference (data not shown).


**Table 5.** Logistic regression analysis considering the period including the two weeks pre and post DST as the dependent variable and the other parameters investigated the independent ones.

### **4. Discussion**

The present study found no statistically significant differences in the number of deliveries that occurred during the two weeks following DST compared to those occurring prior to the DST shifts. The same result was obtained for each secondary outcome analyzed. These data confirm the findings by Laszlo et al. [19] also for the latitudes and climatic conditions of the Emilia-Romagna region of Italy, despite the difference in daily average hours of sunlight (approximately 180 and 50 monthly sun-hours in March and October, respectively, in Sweden, vs. 190 and 100 in March and October, respectively, in Italy). The Swedish study, in fact, showed that the number of daily and weekly deliveries in the case and control group was similar, and statistically significant (IR 1.005, 95% CI 0.990–1.019), underlining the validity of the result only for latitudes, exposure to light, and climatic conditions to which the people included in the sample are subjected. Although the characteristics just stated for Sweden and the Emilia-Romagna region are extremely different, in both studies, it is confirmed that the transition from standard time to summer time and vice versa does not impact on the number of deliveries.

These results confirm data from Roizen et al. [20] regarding the role of oxytocin in the onset and completion of childbirth and its rhythmicity. Hormone secretion, in fact, has its own circadian rhythm, not influenced by the alternation of dark and light and, therefore, not affected by the change of time. This may explain why the number of deliveries in the two weeks following the transition from standard time to summer time, and vice versa, is almost superimposable to that found in the two weeks preceding it.

The analysis of type of delivery was based on the hypothesis of a possible influence of DST on the secretion of melatonin [21–24], through the indirect evaluation of the outcome of contractile activity. In this case, we could expect to observe a greater number of operative deliveries and cesarean sections in the weeks following DST shifts, caused by dynamic or mechanical dystocia induced by anomaly or stop of contractions. This difference was more marked in the Spring period as the transition from winter time to summer time leads to the loss of one hour of darkness, with a consequent increase in light exposure and reduction in serum melatonin levels and, presumably, also in valid contractions. However, our data do not allow confirmation of this hypothesis; both the statistical analysis performed on the number of cesarean sections (*p* = 0.08) and that relating to the operative deliveries (*p* = 0.99) do not allow us to exclude that what has been detected is not attributable to simple chance.

Another relevant finding in the secondary outcome analysis was the confirmation of previous data collected in Texas, USA, by Mancuso et al. [25]. In our sample also, relating to the Emilia-Romagna region, the transition from standard time to summer time does not significantly affect the time of delivery, regardless of the gestational period and the way it occurs.

Neonatal birth weight was also investigated as a secondary outcome, to test the hypothesis of a short-term, acute effect of rhythm disruption. This is because the available data in the literature refer to studies performed on shift worker women [8], and shift work represents a chronic, long-term condition of rhythm desynchronizing effect. According to this hypothesis, the expected result could be, therefore, an increase in newborns with a low birth weight (<2500 g) in the weeks following the time change. However, the data obtained allow us to affirm that the transition from solar time to summer time and vice versa does not have a statistically significant effect on the increase in the incidence of newborns born with a low birth weight (<2500 g) (*p* = 0.97). It is possible to extend the same statement to the other investigated weight classes (2500–4000 g: *p*-value = 0.45; >4000 g: *p* = 0.98). Thus, we can conclude that DST shift does not have a statistically significant impact on newborn birth weight.

The rationale for the analysis of the use of epidural analgesia in periods of exposure and control was to assess whether the desynchronization induced by DST that shifts the transition could play a role in the perception of pain. In this case also, the results obtained do not allow us to exclude that the differences found are attributable to chance (*p* = 0.64).

We took into consideration age and gestational age of this low risk population and we found that the mean age of women who delivered during the two weeks before DST was older than those who delivered during the two weeks post DST and that women's age was independently associated with delivery during the two weeks post DST. We do not think that these findings could be of any relevant clinical significance due to the very small difference in age between the two groups. Moreover, these results could be related to the selection criteria aiming at evaluating only very low risk pregnancies.

We are aware of several limitations: (i) retrospective observational study; (ii) lack of perinatal complications—however, we selected only very low risk pregnancies; (iii) three years represent a short period of observation; (iv) unfortunately, we did not apply the methods used by László et al. [19] due to the different statistical package used for analysis—in fact, we used SPSS instead of SAS. Finally, (v) possible differences by race and ethnicity, since 35% of patients were not Italian. However, according to the last report of the Ministry for Health (year 2017) [26], in Italy, 21% of the births were related to mothers of non-Italian citizenship. This phenomenon is more widespread in the areas of the country with a greater foreign presence, i.e., in the Center–North, where more than 25% of births are from non-Italian mothers; in particular, in Emilia-Romagna and Lombardy, approximately 31% of births refer to foreign mothers. The most represented geographical areas of origin are Africa (27.7%), European Union (24.4%), Asia (18.1%), and South America (7.5%) [26].

However, the present study has also several strengths: (i) the large sample size, (ii) the quality of the data, and (iii) the rigorous methodology. The sample analyzed is representative of women characterized by a very low risk pregnancy, and the Emilia-Romagna region is characterized by one the best regional health systems in Italy. The choice of performing the study in a specific Italian region allowed us to obtain a reliable, good, complete, uniform, and coded source of data. The Childbirth Assistance Certificate is unique for the entire regional territory, and its compilation is mandatory following each birth in the region, independent of the structure in which it takes place.

### **5. Conclusions**

This study shows no differences in the number of deliveries in the weeks following the change of time compared to those that precede it, neither during the Spring nor in the Autumn. This finding provides further confirmation to previous data obtained at quite different conditions of latitude, climate, and light exposure. It is possible that the multihormonal etiology of labor may explain this phenomenon. It would be interesting to extend the study to different latitudes and ethnicities, in order to verify whether our findings could be generalized or not.

**Author Contributions:** Conceptualization: R.C., R.V., P.G., R.M. and F.F.; methodology: R.C., S.P., V.F., A.D.G., E.D.S. and R.V.; data curation: S.P., V.F., A.D.G., P.I. and R.V.; formal analysis: S.P., V.F., P.I., A.D.G., E.D.S. and R.V.; writing—original draft preparation: R.C., S.P., V.F. and R.V.; writing—review and editing, S.P., P.I., E.D.S., P.G., R.M., M.A.R.B., F.F. and P.J.L.S.; supervision: P.G., R.M., M.A.R.B., F.F. and P.J.L.S.; project administration: R.C., S.P., E.D.S., and R.V.; funding acquisition: P.G. and R.M. All authors have read and agreed to the published version of the manuscript.

**Funding:** This study is supported by a scientific grant by the University of Ferrara (Fondo Incentivazione Ricerca-FIR- 2020, Roberto Manfredini).

**Acknowledgments:** We thank the Emilia-Romagna region, Direzione Generale 'Cura della persona, salute e welfare', for valuable and precious collaboration.

**Conflicts of Interest:** The authors declare no conflict of interest.

### **References**


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### *Article* **Implementing a Care Pathway for Complex Chronic Patients from a Nursing Perspective: A Qualitative Study**

**Rosario Fernández-Peña 1,2,3,\* , Carmen Ortego-Maté 1,2 , Francisco José Amo-Setién 1,2, Tamara Silió-García 1,2 , Antoni Casasempere-Satorres <sup>4</sup> and Carmen Sarabia-Cobo 1,2**

	- 2 IDIVAL Nursing Research Group, 39011 Santander, Spain
	- <sup>3</sup> SALBIS Research Group, University of León, 24400 León, Spain

**Abstract:** A care pathway constitutes a complex care strategy for decision-making and the organization of processes in the care of complex chronic patients, avoiding the fragmentation of care. Health professionals play a decisive role in the implementation, development, and evaluation of care pathways. This study sought to explore nurses' opinions on the care pathway for complex chronic patients three years after its implementation. The study participants were thirteen nurses with different roles who were involved in the care pathway. Thematic content analysis of the semi-structured interviews resulted in four major themes: (a) the strengths of the route; (b) the impact of the route on caregivers; (c) the weaknesses of the route; and (d) the future of the route. Overall, the pathway was positively valued for the benefits it provides to patients, the caregiver, and the administration of professional health care. Participants voiced their concerns regarding: communication and coordination difficulties among professionals across the different levels of care, the need for improved teamwork and consensus among professionals at the same center, and human and material resources. The ongoing evaluation and monitoring of facilitators and barriers is necessary throughout the implementation process, to ensure continuity and quality of care in the health system.

**Keywords:** care pathway; integrated health care; long-term care; quality of health care; Spain; qualitative research

### **1. Introduction**

Currently, our society is facing demographic, political, social, and economic changes that highlight the need to have health services that guarantee that the population's health needs are met, especially concerning chronic processes. Progressive population ageing, together with the increasing prevalence of chronic diseases, are the main challenges faced by health systems across Europe [1].

Integrated care is ideally suited to respond to care needs in this context via proactive and well-coordinated patient-centered multidisciplinary care, using new technologies to support patient self-management and to improve the collaboration between caregivers [1]. This transformation requires a multi-level strategy with the simultaneous and synergistic implementation of various interventions within the same territory and population [2].

Following global trends, in recent years, mortality and disability in Spain are related to NCDs (Non-Communicable Diseases), which, in 2016, according to the WHO (World Health Organization), represented up to 91% of total deaths [3]. This reality has led to a redirection in the organizational approach of the health system as well as the design of specific policies for the care of these diseases [4].

The European Pathway Association defines the care pathway as a complex intervention for the mutual decision-making and organization of care processes for a well-defined

**Citation:** Fernández-Peña, R.; Ortego-Maté, C.; Amo-Setién, F.J.; Silió-García, T.; Casasempere-Satorres, A.; Sarabia-Cobo, C. Implementing a Care Pathway for Complex Chronic Patients from a Nursing Perspective: A Qualitative Study. *Int. J. Environ. Res. Public Health* **2021**, *18*, 6324. https://doi.org/10.3390/ijerph18126324

Academic Editor: Paul B. Tchounwou

Received: 6 May 2021 Accepted: 1 June 2021 Published: 11 June 2021

**Publisher's Note:** MDPI stays neutral with regard to jurisdictional claims in published maps and institutional affiliations.

**Copyright:** © 2021 by the authors. Licensee MDPI, Basel, Switzerland. This article is an open access article distributed under the terms and conditions of the Creative Commons Attribution (CC BY) license (https:// creativecommons.org/licenses/by/ 4.0/).

group of patients during a well-defined period, although a variety of terms have been used interchangeably in the international literature without a clear differentiation, such as critical pathway, clinical pathway, integrated care pathway, or care map [5,6]. Different studies have highlighted the strengths of implementing the care pathway, revealing an overall positive impact on the organization, coordination, and monitoring of care processes, the involvement of professionals and the improvement of process indicators [7–13]. Health professionals play a decisive role in the implementation, development, and evaluation of chronic care strategies [14,15]. Specifically, nurses carry out an important task, both as coordinators for patient care and as care liaisons between health levels, supported by their own experience in the care path and managing patients with complex chronic needs [16–18].

Adopting the Chronic Care Model as a framework for the reorientation of the health care system [15,19], chronicity management models in Spain have been developed via plans or strategies at a national and regional level in different waves from 2006 to 2015. Most of these plans are based on population models focused on the analysis of the needs of people with chronic diseases, according to the risk stratification based on the Kaiser Permanente Pyramid [20] and the model of the British Kings Fund [21]. The Strategy for Addressing Chronicity of the Spanish National Health System [22] contemplates management by integrated care processes and the definition of care pathways for the different chronic health conditions that are included in most plans or strategies for the care of chronicity across different regions in Spain [21,23].

The autonomous community of Cantabria, located in the north of Spain, has a population of 584,308 inhabitants in 2020, with an aging rate of 22.5% (population aged 65 and over, compared to the total population) [24,25].

Currently, in the community of Cantabria, there are 5802 patients identified as pluripathological. Following the guidelines of the Chronicity Care Plan in Cantabria [26], in 2016, the "Chronic Patient Care Pathway" project was created, which was aimed at all chronic patients, seeking to address different organizational models for providing care to people with different chronic diseases. In the framework of this broader project, the Pluripathological or Complex Chronic Patient Care Pathway was implemented, designed for adult patients with two or more chronic pathologies and a high level of complexity. Its objectives were to modify the natural course of their diseases, delaying their progression and improving their health by maintaining patients in their usual environment. Three nursing roles are involved in the care of the complex chronic patient included in the pathway. Firstly, the hospital liaison nurse is present in the four community hospitals with the main role of ensuring continuity of care and coordination between the different levels of care. Secondly, due to their position of leadership among other health professionals, nurse managers are essential to strengthen the quality, coordination, and integration of care [27]. Lastly, community primary care nurses are considered well-positioned regarding the nursing care of patients included in the care pathways as well as for their insights regarding the evaluation of the care pathways [28].

Although the assessment of the chronic care strategy in Cantabria has been carried out in recent years from a quantitative approach, the assessment of the Complex Chronic Patient Care Pathway has not been studied from a qualitative approach, or, more concretely, from the point of view of nursing professionals.

The aim of this study was to explore nurses' opinions on the care pathway for complex chronic patients in Cantabria, three years after its implementation, and on the different nursing roles involved in the complex chronic care pathway, specifically, the primary care nurse, the hospital liaison nurse and the nurse managers.

### **2. Materials and Methods**

### *2.1. Design*

A qualitative phenomenological descriptive design was conducted using semi- structured interviews. The analysis of the phenomena as they emerge from the point of view of nursing professionals' experience is necessary for a deeper understanding of their meaning [29,30]. This study adhered to the Consolidated Criteria for Reporting on Qualitative Research (COREQ) guidelines developed to evaluate qualitative research reports [31].

### *2.2. Research Team*

The research team was formed by PhD university teachers with a background in Nursing, Psychology (CO and CS), and Anthropology (RF). In addition, a sociologist, expert in qualitive data analysis and with a PhD in Education, participated in this research. All researchers had experience in research in health sciences and none were involved in professional activity associated with the participants.

This study was part of a larger project primarily aimed at studying the impact of nursing care in the complex chronic patient on dependency, perceived satisfaction, and caregiver burden in Cantabria and the Balearic Islands. The design of this mixed methods study included participants who were patients in the Complex Chronic Patient Care Pathway, together with their caregivers [32,33]. During the fieldwork phase and after the collection of the initial quantitative and qualitative data, the research team was involved in two work sessions, in which they raised and discussed the need to include the nursing professionals involved in the complex chronic patient pathway as participants in the project. In these sessions, the research team reflected on the beliefs and motivations for this study, considering that the inclusion of this type of participants would enable the possibility of gathering a broader and more profound view of nursing care in the care pathway for complex chronic patients.

### *2.3. Participants*

The inclusion criteria consisted of nurses working in the Cantabrian Health Service as nurse practitioners with care and management experience in handling patients labeled as complex chronic patients and included in the route, and who occupied managerial positions in the implementation of the pathway. The exclusion criterion was nurses without professional experience in the care of these patients.

In total, 13 nursing professionals participated in this qualitative study, with different nursing roles: two nurse managers, four hospital liaison nurses and seven primary care nurses, none of whom withdrew from the study. Four primary care nurses were not included in the study because they lacked experience in the management of patients included in the complex chronic patient pathway.

Sample selection was performed using purposive sampling. Key informants from the Cantabrian Health Service who held management positions were contacted to participate in the study in this professional role. These participants provided us with access to primary care nurses. For the selection of the primary care nurses, the segmentation criteria included nurses belonging to the four health areas of the public health service. The snowball technique was used to access new primary care nurses working in the different health areas of the community. Finally, we contacted the four liaison nurses of the Cantabrian Health Service working in the public hospitals of the community. Participants were contacted by telephone at their workplaces inviting them to participate in the study and informing them of the study aims and procedures.

### *2.4. Ethical Considerations*

This project was approved by the Clinical Research Ethics Committee of University of Cantabria (Internal Code 2017.049) and was authorized for its implementation in the Cantabrian Health Service. Prior to the study, the research aims and procedures were once again informed verbally and, in writing, and informed consent was obtained from each participant. The data were treated anonymously and confidentially, conforming to the Spanish legislation and the principles of the Declaration of Helsinki.

### *2.5. Data Collection*

The semi-structured interviews were conducted following a script and field notes were gathered to record observations related to the data collection phase. Table 1 presents the topics and questions that were asked to the participants to obtain information based on their specific nursing role.


**Table 1.** Semi-structured interview guide.

Data collection was carried out during the year 2019. The face-to-face interviews were conducted by two members of the research team (RF and CS) with experience in qualitative research and with the sole presence of the participant. The interviews were mostly conducted at the hospitals and primary care centers where the nursing professionals worked, and, in four cases, they were held on university premises. The interviews were conducted and analyzed in Spanish, lasted an average of 25 min, and were audio recorded and fully transcribed by two researchers from the research team. Finally, the interview extracts shown in this article were translated into English by expert translators. All information in the transcripts regarding the participants' names and locations that could facilitate their identification was anonymized. Personal data were kept in a separate register accessible only to the project's principal investigators and those involved in data collection (CO, CS, and RF). The interviews were stored and managed in MAXQDA software (VERBI Software—Consult—Sozialforschung GmbH, Berlin, Germany), and were only accessible to researchers involved in coding and analysis.

The field work was completed when data saturation was reached, meaning that the information provided in the interviews was already collected in the agreed upon analysis categories or in the emerging categories and did not provide new concepts for analysis [34].

### *2.6. Data Analysis*

Once transcribed, the interviews were exported to qualitative software analysis MAXQDA 2020. An inductive thematic content analysis was carried out [35,36] by four researchers from the group with the aim of enabling the emergence of categories and subcategories. Although the thematic structure served to initially organize the findings, the emergence of ideas from the data was what shaped the study themes via the appropriate analytical categories. For the analysis of the qualitative content, several analytical cycles were carried out [37–39]. First, an initial substantive coding was developed close to the data, which was complemented by in-vivo coding to track metaphors in the participants' discourse or significant terms. Subsequently, a process of categorization and recoding was performed which integrated the indicators into different categories or topics while reviewing and refining the coding system. The analysis was performed during the main data collection phase; however, it was considered mandatory to return to the field to perform more interviews to verify whether the categories raised were properly saturated. The analysis of the new data showed that no further relevant categories emerged from the data.

COREQ guidelines were followed [31], and different techniques were used to establish the trustworthiness of the data by reviewing issues concerning data credibility, transferability, dependability, and confirmability [40]. Researcher triangulation was achieved via team meetings that took place during the data collection and analysis phases, and participant triangulation was ensured by including different nursing professional roles involved in the

phenomenon under study. Additionally, during the data collection, participant validation was ensured by asking the nursing professional to confirm the data obtained. A thick description of the phenomenon under study was included, and the rationale for the study was described in the framework of the wider research project. In addition, regular quality checks were made to avoid researcher bias. Furthermore, the research team adopted a reflective attitude, together with systematic attention to the context of the construction of knowledge.

### **3. Results**

This section describes the characteristics of participants and presents the main thematic findings of our study.

In total, 13 nursing professionals (12 women, 1 man) participated in this qualitative study with a mean age of 45.9 years (range 37–54 years), and with a mean of 20.5 years professional experience as a nurse (range 12–29 years) (see Table 2). At the time of the study, all participants were working in different positions within the chronic patient care strategy of the Cantabrian Health Service: the four hospital liaison nurses in the community, primary care nurses from the different health areas, and two nurse managers.


M: Male, F: Female.

Thematic content analysis of the interviews resulted in four major themes: (a) the strengths of the route; (b) the impact of the route on caregivers; (c) the weaknesses of the route; and (d) the future of the route. The main categories and subcategories are presented in Table 3.

**Table 3.** Distribution of the main categories and subcategories.


### *3.1. Strengths of the Route*

An emerging idea in this category that was highly valued by nursing professionals was related to the benefits of the route for patients and their families. Among the objectives that were positively evaluated as strengths, the professionals highlighted that the care pathway avoids or reduces hospital admissions, the number of visits to the hospital emergency department, as well as the number of days spent in hospital. As a result, patients spend more time in their usual social and family environment, leading to a substantial improvement in their quality of life. In addition, it favors a more individualized health care, suited to the patients' needs, and prevents them from becoming unstable.

*The route increases the quality of life of patients within their pathological process (...) We are able to manage all resources better. We avoid unnecessary admissions, and therefore, at the hospital level there will be fewer admissions, which means less nosocomial diseases derived from the admissions. The greatest beneficiary is the patient, after that I think it is the family, and society. Regarding admissions, I believe there is good management, of course, it is more efficient to keep patients who are at home always well cared for, to avoid unnecessary admissions*. (Informant 10)

*The route is a tool that makes it possible to organize the path of these chronic patients throughout their disease process. We try to establish a connection between primary and specialized care, because what we want is for these patients to remain in their environment as long as possible, because that is what will give them stability*. (Informant 7)

Secondly, participants stressed that the route is a strategy that significantly improves the professional care provided to patients and also adds satisfaction in their work as professionals. Once patients are labeled as being pluripathological, this helps alert nurses to the need to provide health care adapted to the patients' needs. Thus, patients are kept more stable, by providing care that is individualized and appropriate for their needs, without having to enter the usual care path. Moreover, the route facilitates the continuity of health care between primary care and the hospital.

*Well, a care pathway, a guide, that serves us to refer patients and not to lose them in this maelstrom that we can call the health system. To have them under control in primary care and that when they get out of control of primary care, which we understand is where the patients are, and they have to go to hospital, well, they must do so without losing continuity of care and without disassociating themselves from primary care*. (Informant 1)

*The pluripathological label is a very important sign of alarm that puts us nurses with patients at risk, something that previously you could ignore [* . . . *]. Whereas now, you continue to take care of them and follow their progress*. (Informant 8)

### *3.2. Impact of the Route on Caregivers*

The emerging ideas in relation to caregivers are related to aspects of the route that translate into improved care performance. Firstly, the route speeds up the procedures and facilitates the patient's transit between the different institutions of the health system, which avoids overloading the caregiver and represents a benefit in the care provided.

*Yes, I think it is very beneficial for the caregiver or family because when they go to the hospital they don't have to wait so long and then they're discharged because they're already on their way to where they're going: to the residence or home or they get help... or they go to the social worker. They connect them in another way, without the relative or caregiver being concerned about what they have to do, or where they have to go*. (Informant 13)

Secondly, the route makes it easier for caregivers to have a more accessible and closer relationship with health professionals and social workers at both the primary care and hospital levels. Likewise, this closeness makes caregivers feel more supported, with care adapted to their availability, feeling more confident with the care they provide to the patient and assume a more active role in care:

*For the caregivers, I think they perceive important support. In the end, what they see is that they truly have an avenue of help, a resource that is close to primary care*. (Informant 7)

*There is a very important part which is also the relevance given to the caregiver in this program. Of course, the improvement in the quality of life is evident. (...) the support that they have, the security that it gives them to have you as a reference. For example, in the hospital, they know that they can call you directly [...], it gives the caregivers security and saves them a lot of paperwork*. (Informant 5)

### *3.3. Weaknesses of the Route*

On this subject, the informants underlined the lack of relationship and coordination between primary care and hospital nursing professionals, as a weakness in the process of implementing the care pathway. The elements involved in this lack of relationship refer to an already traditional separation between these two levels of care, difficulties in making contact between the reference nurses in primary care and the liaison nurse in the same health area as well as the lack of common spaces for contact between nurses of both care levels.

*To me, it seems essential for a connection to exist between hospital and primary care nurses. A true connection, not only in times of instability to avoid an admission, and not just between primary care physicians and specialized internists*. (Informant 7)

Another point that stood out was the idea of insufficient joint work and consensus on the criteria and decision-making regarding complex chronic patients included in the route between professionals of the same center, especially in primary care centers, in addition to individual differences in terms of their involvement in the route as professionals. Specifically, referring to the control and power of decision that medical professionals have in the process, both in the labelling of patients and in the access to patient information during hospital visits:

*All patient information reaches the doctors so when a patient has been discharged, my doctor has to let me know that he has been discharged, otherwise I don't know*. (Informant 1)

Thirdly, the participants highlighted a weakness concerning implementation of the route, specifically, some aspects related to human and material resources that make it difficult for the operation of the route to achieve its objectives. Thus, the high turnover of health personnel, both across centers and between levels of care, due to changes in their employment situation, interferes with continuity in knowledge and in managing the route as professionals in the same centers. Moreover, communication difficulties were emphasized between hospital and primary care professionals, as the systems used in these different centers were often different.

*The computer applications should make it easier: if I, as a nurse, have a patient who is admitted and discharged, I should receive a discharge notice and a continuity of care report*. (Informant 7)

### *3.4. Future of the Route*

The need for a change of model in the approach to chronicity, the benefits of the route for patients and caregivers, and the conviction of their advantages as nursing professionals, makes the participants value their future in a positive way:

*[* . . . *] it's the trend in health care right now [* . . . *].. It's a patient-centered model. These programs must succeed either way*. (Informant 5)

*We are convinced that this is really good*. (Informant 4)

The success of the route requires the design and implementation of certain areas of improvement. The need to improve communication and coordination between primary care nurses and hospital nurses was emphasized, by using improved resources for telematic communication, increasing the provision of nurses involved in the route, especially at the hospital level, as well as by providing continuous training on the route. Likewise, the need to redirect the role of nurses in the route by giving it more prominence and leadership was highlighted.

### **4. Discussion**

The qualitative results of our study show that, overall, nurses have a positive assessment of the care pathway for both patients and caregivers. However, three years after its implementation, nurses stress the need for strategies to further improve in certain areas.

These areas include the need for more fluid communication and coordination channels and mechanisms between primary care nurses and hospital liaison nurses, teamwork, and consensus among professionals of the same center, as well as the improvement of human and material resources involved in the route.

The collaboration between clinical professionals from two different care levels has been carried out in Cantabria in recent years through the d'Amour questionnaire [41]. According to our findings, the quantitative results obtained from the questionnaire showed a mean score of 2.51 out of 5 in the years 2016–2019, thus suggesting equally improvable aspects in this area.

In this line, the care pathway should be considered as a process that represents a path of both success and challenges for professionals and managers, as shown in previous studies [42]. Given their dynamic nature over time, these new strategies essentially require periodic review and evaluation of their impact and assessment by both patients and professionals [11], considering the significant changes that their implementation may entail in the organizational culture and the possible need for support mechanisms to ensure their implementation in practice [7].

Inter-professional collaboration is a key element in efforts to increase the effectiveness of current health services, especially in the face of complex health problems, for which there are different conceptualizations, theoretical models [43] and assessment tools [41]. Regarding the care pathway of the complex chronic patient, the inter-professional collaboration between primary care and hospital care constitutes the vertebral axis upon which the comprehensive care between the health providers is based, thus avoiding fragmentation of care [44,45].

Other studies have focused on the role of the relational network between health professionals or health organizations from a structural point of view, in health settings and for the implementation of programs [46–50]. Thus, the multilevel model proposed by Van Houdt et al. [51] highlights the need for improved communication and coordination in the implementation of a care pathway between primary and hospital care professionals. These changes include improvements at the level of inter-organizational networks through the exchange of information and development of new communication channels, shared objectives, knowledge of the roles and competencies of the different professionals, and improved relationships. Secondly, changes in the inter-organizational mechanisms are required to change the structures of the organizations before and after the implementation of the care pathway in addition to the development of facilitating strategies to favor the creation of interpersonal networks between professionals, together with the transfer of information by electronic means [50].

The design and implementation of a new approach to health service delivery represents a challenge for managers and professionals, beginning with the preliminary planning of the implementation process, by considering the contextual, organizational, and cultural characteristics of the organizations and the intricacies of the care provided [42,52].

### **5. Conclusions**

The care pathway of the complex chronic patient in today's healthcare reality constitutes a valuable care strategy which responds to the needs of these patients and their caregivers in complex settings, by avoiding fragmentation of care. Due to their leadership roles, nurse managers are in an ideal position to ensure the correct implementation of these care pathways. A permanent evaluation and monitoring of facilitators and barriers to the implementation process is necessary to guarantee the continuity and quality of care in the health system.

**Author Contributions:** Conceptualization, R.F.-P., C.S.-C., C.O.-M., F.J.A.-S. and T.S.-G.; methodology, R.F.-P., C.S.-C., C.O.-M., F.J.A.-S. and T.S.-G., software, A.C.-S., R.F.-P., C.S.-C. and C.O.-M.; validation, R.F.-P.,C.S.-C., C.O.-M. and A.C.-S.; formal analysis, A.C.-S., R.F.-P., C.S.-C. and C.O.-M.; investigation, R.F.-P., C.S.-C., C.O.-M., F.J.A.-S., A.C.-S. and T.S.-G.; resources R.F.-P., C.S.-C., C.O.-M., F.J.A.-S., A.C.-S. and T.S.-G.; data curation, A.C.-S., R.F.-P., C.S.-C. and C.O.-M.; writing—original draft preparation, R.F.-P., C.S.-C., C.O.-M., A.C.-S., F.J.A.-S. and T.S.-G.; writing—review and editing, R.F.-P., C.S.-C., C.O.-M., A.C.-S., F.J.A.-S. and T.S.-G.; project administration, C.O.-M. and C.S.-C.; funding acquisition, C.O.-M. and C.S.-C. All authors have read and agreed to the published version of the manuscript.

**Funding:** This work was funded by the University of Cantabria (Spain), budget implementation 28.VU08.64662. The funders had no role in study design, data collection and analysis, decision to publish, or preparation of the manuscript.

**Institutional Review Board Statement:** The study was conducted according to the guidelines of the Declaration of Helsinki, and approved by the Clinical Research Ethics Committee of University of Cantabria (Internal Code 2017.049).

**Informed Consent Statement:** Informed consent was obtained from all subjects involved in the study.

**Data Availability Statement:** The data presented in this study are available on request from the corresponding author. The data are not publicly available due to privacy and ethical considerations.

**Acknowledgments:** We thank all the nursing professionals who agreed to participate in this work for their availability, time, and contribution to this study.

**Conflicts of Interest:** The authors declare no conflict of interest. The funders had no role in the design of the study; in the collection, analyses, or interpretation of data; in the writing of the manuscript, or in the decision to publish the results.

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