Occupational Therapy Interventions in Adults with Multiple Sclerosis or Amyotrophic Lateral Sclerosis: A Scoping Review

This scoping review aims to describe occupational therapy interventions carried out with multiple sclerosis (MS) and amyotrophic lateral sclerosis (ALS) patients in occupational therapy. A peer review of the literature was conducted in different databases: Pubmed, Scopus, Web of Science and Embase, and in some occupational therapy journals. A search of the literature published was carried out before December 2019. The inclusion criteria were as follows: (1) articles evaluating the intervention of occupational therapy in MS or ALS including experimental, randomized, nonrandomized and exploratory studies; (2) written in English or Spanish; (3) adult population (over 18 years old). The initial search identified 836 articles of which we included 32 divided into four areas of intervention: fatigue-targeted interventions, cognitive interventions, physical interventions and others. Only 16 studies were carried out exclusively by occupational therapists. Most occupational therapy interventions are aimed at fatigue and physical rehabilitation. The majority of the studies in our review included MS patients, with little representation from the ALS population. These interventions have shown an improvement in perceived fatigue, manual dexterity, falls prevention and improvement in cognitive aspects such as memory, communication, depression and quality of life in the MS and ALS populations.


Introduction
Multiple sclerosis (MS) and amyotrophic lateral sclerosis (ALS) are neurodegenerative diseases of the nervous system [1,2]. These diseases have a medium to high prevalence, but recently published epidemiological studies have shown an increasing incidence and prevalence of MS and ALS in different populations worldwide [3][4][5]. Globally, in the case of MS, the incidence is 2.5 per 100,000 inhabitants, while in ALS it is between 0.6 and 3.8 per 100,000 inhabitants, although the age of onset is later than in MS [3,6]. Though the origin of the diseases is unknown, previous studies have ruled out risk factors such as geographical latitude or ethnicity [7], while other studies suggest that having a family member with these diseases can increase the risk of developing them [8]. However, the mechanisms and causes for their development are not completely understood.
These diseases affect not only quality of life but also physical and cognitive aspects, increasing fatigue and the probability of suffering from depression [9]. They can include multiple symptoms such as muscle stiffness, paralysis of the lower and upper limbs, sensory dysfunction, visual problems, ataxia, dysarthria or dysphagia [10] as well as cognitive impairment and psychological problems in the affected persons [2,11,12]. These diseases have a multidimensional impact on a person s life, and their symptoms imply a significant loss of autonomy which greatly affects their occupational performance [11].
The treatment of these diseases is carried out by multidisciplinary teams [11,13] and it can be pharmacological [14] and/or nonpharmacological. In nonpharmacologi- Table 1. Database and search strategy.

Database Strategy
Pubmed "occupational therapy" [All Fields] AND ("methods" [MeSH Terms] OR "methods" [All Fields] OR "intervention" [All Fields]) AND "sclerosis" [All Fields] Scopus TITLE-ABS-KEY ("occupational therapy" AND intervention AND "sclerosis") Embase ('occupational therapy'/exp OR 'occupational therapy') AND ('intervention'/exp OR intervention) AND ('sclerosis'/exp OR sclerosis) Web of Science ("occupational therapy" AND intervention AND sclerosis) Teseo ("occupational therapy" AND intervention AND sclerosis) Journal of Occupational Rehabilitation '"occupational therapy" AND intervention AND sclerosis' Physical & Occupational Therapy In Pediatrics The inclusion criteria were as follows: (1) articles evaluating the intervention of occupational therapy in MS or ALS including experimental, randomized, nonrandomized and exploratory studies; (2) written in English or Spanish; (3) adult population (over 18 years old). We excluded the following: (1) qualitative studies; (2) studies with no abstract, no full text or not available.
Study selection and data extraction of the information were carried out independently. We migrated the results from the databases to a Microsoft Excel spreadsheet where inclusion and exclusion decisions were recorded. The two reviewers (LB and LT) independently selected articles based on the selection criteria. Any disagreement between them regarding possible inclusion/exclusion criteria was resolved by a third reviewer (MG). LB and LT only had discrepancies regarding the inclusion of one article, and with the intervention of a third reviewer we decided to exclude it.

Results
The search strategies identified a total of 836 articles and, after conducting the peer review, 58 articles were selected for their retrieval and evaluation of the full text. We excluded 26 articles, as they did not fulfil the inclusion criteria, leaving 32 articles for data analysis and extraction. The flow chart is shown in Figure 1.
The articles analyzed were classified into four clearly differentiated areas: (1) interventions for fatigue and energy conservation; (2) cognitive interventions; (3) physical interventions; and (4) other interventions. The fully description of OT intervention carried out in MS and ALS is shown in Table 3.  The 32 selected studies were conducted in different countries: in USA (n = 12), Belgium (n = 4), Netherlands (n = 3), Switzerland (n = 3), Spain (n = 2), Italy (n = 2), and the remaining 6 studies were conducted in Cuba, Ireland, United Kingdom, Israel, Iran and Austria. A total of 29 studies were carried out in MS patients and only three studies were carried out in ALS patients. A total of 16 studies were led exclusively by occupational therapists and the remaining studies were carried out by multidisciplinary teams, including neurologists, neuropsychologists, social workers or experts in certain fields such as assisted technology or mathematics.
The main limitations reported by included studies were small sample size, lack of long-term evaluation of the intervention, lack of randomization and low generalizability of the results. Table 2 presents the characteristics and information of the included studies: author, year, country where study was conducted, objective of study, sample, intervention, standardized proof used, main results and conclusions of study following Cochrane Manual recommendations [25].
We observed that intervention studies conducted in MS and ALS patients could be classified into four clearly differentiated areas: fatigue, physical rehabilitation, cognitive interventions and others. The full description of these interventions are presented in Table 3.
The articles analyzed were classified into four clearly differentiated areas: (1) interventions for fatigue and energy conservation; (2) cognitive interventions; (3) physical interventions; and (4) other interventions. The fully description of OT intervention carried out in MS and ALS is shown in Table 3.

Interventions in Fatigue and Energy Conservation
Twelve studies conducted interventions related to energy and impact on fatigue in people with MS. The results of these interventions are described in Table 2. Seven of these studies were based on the fatigue management program developed by Packer et al. [28], which consists of a 12 h intervention for people with MS and includes a balanced lifestyle, rest, posture and efficient communication, among other aspects [29][30][31][32][33][34][35]. Another study modified Packer s program and evaluated these interventions nonpresentially by monitoring patients either through teleconferences, applications or on the internet [36].
In addition, several authors examined the effectiveness of physiotherapy and diet interventions aimed at fatigue carried out by a multidisciplinary team, including occupational therapists [37,38]. Other authors proposed different intervention programs [39,40] based on changes in daily occupational performance and proposed strategies related to occupational balance, activity, fatigue, energy account, goals or effective communication.
We did not identify studies carried out in fatigue and energy conservation in ALS patients.

Cognitive Interventions
Six studies carried out cognitive interventions. A full description intervention appear in Table 3.
Of these studies, two included the use of technology to facilitate communication and automated control at home [19,41] to evaluated the functional performance which increased significantly with PDA use [18]. The remaining studies evaluated interventions related to improvement of memory, attention, processing speed and strategies to compensate these cognitive strategies [20,[42][43][44] with different results (Table 2) Only one study was carried out in people with ALS [41]. This study evaluated the feasibility and usability of an assistive technology prototype in users who have different degrees of muscular impairment to improve interaction with environment.

Physical Interventions
Of a total of ten articles describing interventions in relation to physical condition, nine focused on MS patients and one on ALS patients, only 2 were led by occupational therapists. The results and the details of interventions are shown in Tables 2 and 3. In the MS focused studies, we identified three categories: upper limb recovery, physical rehabilitation and falls prevention.
In the first category, four of the studies focused on upper limb recovery, both at the level of sensory re-education and at that of improvement in manual dexterity in MS [45][46][47][48]. Another study assessed an intervention program to improve the physical resistance of MS patients [49].
In the second category, two other MS focused studies aimed at physical rehabilitation were carried out using new technologies such as virtual reality [50] or images and videos [51].
Finally, the third category included two intervention studies which evaluated programs to decrease falls risk in MS patients, by sending them information related to falls and how to avoid them [52] or by giving them tape training sessions in order to improve balance reactions [53].
In relation to the ALS focused study, Gómez-Fernández et al., assessed the effectiveness of a multifactorial program by working on different aspects such as breathing, posture control or transfers using a multidisciplinary approach [54] which showed that ALS patients improved significantly on forced vital capacity. There was no improvement in disability, participation and autonomy in IG.
Eyssen et al. [56], 2014, Netherlands To check whether client-centred practice spends more time on assessment than on intervention.

269, MS
Client-centred occupational therapy The results showed a significant increase in time dedication on the diagnostic process in the IG.
The client-centred practice devotes too much time to the evaluation process with no improvements.

2009, United States
To evaluate the effectiveness of the development of capacities and the health promotion in self-efficacy and ability to achieve objectives.

35, MS
Health promotion in self-efficacy and empowerment The results showed significant improvements in self-efficacy and ability to achieve objectives.
The program could took action in multiple areas of intrapersonal, interpersonal, and behavioral functioning.

30, MS
Physical intervention with the "Safe at Home BAASE" program.
Significant improvements in knowledge, prevention and manage of falls risk with 5:6 sessions.
The program has potential to improve knowledge, skills and behavior associated with reduced personal fall risk.
Ortiz et al. [50], 2013, Spain To examine postural control and balance with a virtual reality telerehabilitation program.

50, MS
Physical intervention with a telerrehabilitation program to improve balance and postural control.
Significant improvement in balance, visual preference, the contribution of vestibular information, mean response time and Tinetti test yielded.
The rehabilitation program with virtual reality could be an alternative to standard rehabilitation programmes.
Waliño-Paniagua et al. [47], 2019, Spain To compare the conventional occupational therapy treatment by virtual reality in manual dexterity training.

16, MS
Physical intervention with virtual reality training in manual dexterity.
Program showed no significant differences in manual dexterity. Improvements were found in precision, execution times, and the efficiency of functional tasks.
This therapy with virtual reality can be complementary to conventional intervention.
Bovend Eerdt et al. [51], 2010, United Kingdom To evaluate the effectiveness of a motor imagery program compared with OT.

30, MS
Physical intervention with a motor imagery program.
Compliance with advised treatment was poor in 85% of the therapists and in 72% of the patients.
Therapist and patient compliance was low, restricting the conclusions of the effectiveness of the imagery program Kalron et al. [48], 2013, Israel To evaluate the effectiveness of a sensory home-based hand re-education and manual dextery program.

18, MS
Physical intervention with a sensory hand re-education and manual dextery program. To evaluate the effectiveness of an energy conservation program on fatigue.

37, MS Energy conservation program
Fatigue was and physical, cognitive, and psychosocial measures were improved.
The program was effective and reduced levels of fatigue in people with moderate-severe MS. There were significant improvements in fatigue in both groups. The IEME alone resulted in significant improvements in self-efficacy regarding energy conservation strategies.
The IEME program was effective at improving self-efficacy in performance and fatigue management strategies.

Other Interventions
Four articles did not fit into any of the previous categories. Of these, three studies focused on MS patients.
Two of the studies focusing on MS assessed client-centered practice intervention in people with MS patients to evaluate disability, autonomy and participation in daily life with no significant effects in these outcomes [55,56]. Another of these studies, led by Block et al., assessed the effectiveness of health promotion in people with MS which worked on different aspects such as the empowerment of the person [57]. This study showed significant improvements in self-efficacy and ability to achieve objectives [57].
The one study focusing in ALS evaluated the impact of music therapy programs on psychological aspects such as depression and anxiety [58]. In this study, occupational therapists stimulates patients to communicate using instruments and express emotions with positive results in quality of life.

Discussion
This scoping review describes different occupational therapy interventions carried out in MS and ALS patients. These interventions were mainly focused on physical rehabilitation, cognitive rehabilitation and reducing fatigue. Although some of the interventions included in this review were not exclusively led by occupational therapists, they can use these interventions to facilitate the occupational therapy evidence-based interventions.
This review shows that the majority of occupational therapy interventions are performed on MS patients while there is little information about ALS patients. In fact, few intervention studies led by occupational therapists have been found.

Fatigue Interventions and Energy Conservation
This study shows that certain occupational therapy interventions for MS and ALS patients could be effective in improving different outcomes. The majority of the studies identified in this scoping review were fatigue interventions carried out in MS patients. In our search we found that studies principally focused on fatigue are based on the Packer et al. program [28]. This fatigue program is a six-week energy conservation course, which was designed for adults suffering from fatigue as a symptom of chronic disease [28]. In this program occupational therapists educated participants in the benefits of breaking up high-energy tasks by incorporating rest periods into their daily activities [28]. In addition, we observed several studies that included the Packer et al. energy conservation course with some adaptations. Lamb et al. found that patients using self-study material in nonpresential sessions, and who had missed some sessions, obtained similar benefits regarding energy and fatigue management to those whose sessions were guided by a professional and who fully completed the intervention [31]. Similarly, Sauter et al., adapted the fatigue management program to the German population and showed improvements in users physical and mental fatigue. Subsequent studies [33,34] modified the Packer et al. fatigue management course [28] so that it could be delivered by teleconference and online for people with MS who had problems accessing treatment, leading to an improvement in fatigue and quality of life [33,34]. The therapy showed significant improvements in fatigue management even when participants were guided via technological devices [29][30][31][32][33][34][35]. In a similar way, D hooghe et al., developed a course related to a fatigue management program using a smartphone to provide monitoring, motivational messaging and the establishment of objectives [36]. The results showed that this type of intervention can be complementary to conventional treatment to reduce fatigue [36]. Overall, according to the previous evidence, the use of new technologies seems to be a good treatment option.
Conversely, other studies explored fatigue management using different multidisciplinary interventions related to personal care. Rietberg et al. evaluated an intervention carried out by multidisciplinary professions including physical therapy, social work and occupational therapy which applied fatigue management strategies and personal care as compared with only nurse consultation and found that multidisciplinary rehabilitation did not lead to a more effective reduction of self-reported fatigue [37]. In the same line, Kos et al. evaluated a multidisciplinary fatigue management program intervention comparing it with an intervention program based on sleep advice and relaxation exercises [38]. It should be pointed out that neither of the two multidisciplinary interventions explored by these authors showed significant results. This could be because chronic fatigue does not improve significantly over time in MS patients only with personal care advice [37]. All interventions were conducted solely by occupational therapists, except for the multidisciplinary interventions and the D hooghe et al. program [36], in which technology experts collaborated with occupational therapists.
Other authors have proposed other intervention programs [39,40] based on changes in daily occupational performance such as rest management and the proposal of strategies in relation to the management of instrumental activities such as childcare or shopping, that suggest significant improvements in performance, perceived fatigue and individual satisfaction [39,40].
It must be emphasized that fatigue is one of the most frequently reported symptoms in MS patients and can affect their occupational performance [59]. In this sense, a recent review [60] showed that patient-reported outcomes (PROs) are increasingly used in MS treatment. PROs not only describes symptoms, function and health status in MS patients but also evaluates the impact of this disease and assess the concerns on MS patient s life [60].
Fatigue intervention in the included studies was found to be effective in reducing fatigue, managing fatigue symptoms and improving different aspects such as health-related quality of life [12,[29][30][31][32][33][34][35]39,40]. Thus, it is essential that occupational therapy interventions should include fatigue intervention in daily practice with MS and ALS patients.

Cognitive Interventions
With regard to cognitive interventions, we identified six studies in MS and ALS treatment of which three were conducted exclusively by occupational therapists. In MS interventions, Tony Gentry et al., evaluated a program with PDAs that resulted in an improvement of the person s functional performance and satisfaction using PDA as a compensation for cognitive deficits [19]. The remaining cognitive interventions [20,[42][43][44] were related to improving memory, attention, processing speed and strategies to compensate this deficit. Among them, Goverover et al., evaluated the effectiveness of a cognitive strategies program, through visual supports when memorizing words, which showed improvements in memory, learning, depressive symptomatology and quality of life [20]. Only one cognitive intervention was identified for ALS. Schettini et al., evaluated the reliability of an assistive technology device for home automation control and communication, and there were no significant improvements. This could be due to the fact that the sample included only eight people, which may be too small to provide strong evidence [41].
Overall, although cognitive interventions in MS and ALS have scarcely been analyzed, these studies show that cognitive interventions in this type of population have significant beneficial effects in functional performance, depression and quality of life [12,19,20,[42][43][44]. However, these results should be interpreted with caution because the samples in most of the studies described were small and there is no evidence regarding their long-term effects on functional performance [41][42][43]. In addition, it also should have taken into account that there is a lack of information about the effectiveness of these interventions in the progressive forms of MS [61], and there is no evidence about therapeutic intervention to enhance cognitive performances in MS patients [62]. Thus, more studies are needed.

Physical Interventions
Evidence based on different physical therapeutic modalities suggested that interventions improve different functional outcomes (manual dexterity), reduce fatigue and improve quality of life [12,23]. All the studies focused on upper limb recovery were carried out in MS patients. Lamers et al., evaluated the ideal intensity in an upper limb recovery program, showing a positive result, although no overall intensity was established [45]. Kamm et al., conducted a program to improve manual dexterity with exercises using fingers, coins, paper and pencil, and clay, showing improvements in fine motor skills in the experimental group [46]. In the same way Waliño-Paniagua et al., evaluated manual dexterity with virtual reality games in comparison with conventional occupational therapy, showing significant differences. These interventions could also be used as complementary activities in occupational therapy [47]. Finally, Kalron et al., conducted a sensory re-education with tubes of different textures and thickness, showing an improvement in manual dexterity and, although sensitivity did not improve, this program may help to prevent deterioration in manual dexterity in early stages of rehabilitation [48]. It should be pointed out that evidence-based rehabilitation for upper limb recovery are essential for improving performance in daily tasks [23]. Thus, occupational therapists could carry out this evidence-based intervention in MS and ALS patients.
With respect to physical rehabilitation interventions, Bansi et al., evaluated physical rehabilitation with cycle-ergometers or aquatic-bikes in two groups, showing an improvement in quality of life and fatigue [49]. Another study examined virtual reality rehabilitation with strength and proprioception exercises on unstable surfaces and muscletendon stretching, showing significant improvements at the motor level, which suggests that it could be an alternative treatment [50]. In the same line, Bovend Eerdt et al., assessed a film and image presentation program with exercise information and guided rehabilitation strategies [51]. This intervention did not provide valid results because participants did not perform the program in the established time, although there were significant differences after the intervention [51].
We only identified one study in ALS patients, where Gómez-Fernández et al., examined a multifactorial program in ALS, through postural control exercises, exercises with lips, breathing, walking or psycho-emotional support that showed significant improvements in forced vital capacity [54]. However, the sample is very small, which could cast doubt on the results [54].
Regarding the interventions aimed at falls prevention, previous studies carried out programs either by receiving information about falls, strategies and changes in the environment [52], or by treadmill with caused imbalance [53]. Both studies showed a decrease in falls in MS patients.

Other Interventions
Evidence for other interventions was limited. We included four articles in this category because they did not fit into any of the previous categories. In this category, Eyssen et al., explored the effectiveness of client centered practice, comparing it with a control group that received conventional occupational therapy practice [55,56]. The results showed no significant improvements in participants, possibly because more time was spent on evaluation than on intervention, resulting in a less effective recovery. Therefore, this type of practice is not recommended [55,56].
There is currently only limited evidence for the effectiveness of the role of environment in the experience of disability. However, the project Shake-it-up explored the effectiveness of health promotion which works on aspects such as self-efficacy and empowerment among others and found a significant improvement in these aspects [57]. These results could be useful for occupational therapists in their routine work in order to improve the independence, community access and participation of MS and ALS patients.
Finally, we also found one study which assessed the impact of music therapy in ALS, participants interacted with different instruments to express their emotions and communicate, showing an improvement in their quality of life [58]. These findings suggest that there is a need for better designed intervention studies which explore the impact of music therapy on other symptoms in ALS and MS patients.

Study Limitations
This study has a number of limitations. First, regarding inclusion criteria, we only included studies published in English or Spanish and those with full text available. Second, the articles included in this review were experimental studies and might contain biases associated with the experimental study design. Furthermore, the heterogeneity of the included studies meant that they were not comparable in terms of sessions, hours and study objectives. The generally limited study sample size of some included studies means that the results should be interpreted with caution. Third, like other authors in their respective scoping reviews, we did not critically assess the quality of the included studies, because this is not the role of a scoping review [63]. However, we mentioned the limitations of some of the studies in the discussion section. Finally, it should be pointed out that some studies did not clearly specify which professionals participated in the intervention or what their role in the study was. Thus, more studies are needed that specify the role of the researchers in the interventions, including those which are led by occupational therapists.
However, this review also has several strengths. To the best of our knowledge, it is the first study with the aim of describing the main occupational therapy interventions carried out in MS and ALS. In addition, this scoping review highlights the gaps in our knowledge: (i) there is no evidence regarding occupational therapy interventions carried out in Spain; (ii) most of the studies had small sample sizes and a lack of randomization; (iii) there is little evidence about long-terms interventions; and (iv) there is a need to determine the role of the different professionals in the multidisciplinary teams. These identified gaps of knowledge might be dealt with in future research.
This study provides the professionals with a description of therapies in MS and ALS that can support the use of early therapeutic interventions aimed at optimizing outcomes in this population. The included studies in this review showed that occupational therapists can not only collaborate in the multidisciplinary intervention but can also lead different interventions in MS and ALS. This review suggests that occupational therapy is a relevant discipline for MS and ALS patients' rehabilitation. The main intervention led by occupational therapists is fatigue management, which showed beneficial effects in MS patients, but occupational therapists could also carry out psychosocial, physical and emotional interventions in this population. In addition, we would like to underline that the updated summary of previous evidence carried out in this scoping review provides knowledge to facilitate occupational therapy evidence-based interventions.
Finally, our findings add new insights about the potentially beneficial role of physical rehabilitation, fatigue and cognitive interventions, and could inform future evidence-based guidelines for ML and ALS patients.

Conclusions
In conclusion, most studies were conducted in the MS population, with little representation from the ALS population. The main interventions in occupational therapy were those aimed at fatigue, cognitive interventions and physical rehabilitation. These interventions have shown an improvement in perceived fatigue, manual dexterity, falls prevention and in cognitive aspects such as memory, communication, depression and quality of life in the MS and ALS population. It should be pointed out that some of the interventions included in this review are not exclusive to occupational therapy practice. However, occupational therapy professionals can use these interventions in patients with MS and ALS, and they can help patients to incorporate activities and occupations into their intervention patterns.