Abstract
Management of shoulder pain has been studied in people with many neurological conditions, including Parkinson’s disease, cerebrovascular accident, and traumatic brain injury (Coskun Benlidayi & Basaran, 2014; Davis, 2012; Koh et al., 2008; Leung et al., 2007; Manara et al., 2015; Rana et al., 2013). Inferior subluxation, spasticity, and adhesive capsulitis are known causes of shoulder pain in these patient populations (Manara et al., 2015). Other studies specific to neuromuscular disease have shown that shoulder pain is a contributing factor to negative quality of life (Jensen et al., 2005). In the Jensen et al. (2005) study, 39% reported that their pain was severe enough to interfere with their activities of daily living, recreational activities, sleep, and work-related activities. In their study of exercise among people with neuromuscular disease, Anziska and Inan (2014) reported on the importance of exercise and its benefits in maximizing functional ability and minimizing the development of contractures.
Evidence of shoulder pain specific to amyotrophic lateral sclerosis (ALS) has been summarized in four articles. A case study conducted by Bello-Haas et al. (1998) monitored a patient throughout her illness, assessing functionality and quality of life at various stages. Their study illustrated an overall decrease of force in the shoulder muscles, accompanied by pain and loss of shoulder range of motion (ROM) in all planes. A case study demonstrating the importance of rehabilitation for people with ALS noted that a common site for musculoskeletal pain was the shoulder region (Majmudar et al., 2014), and shoulder pain has been reported as a presenting complaint for diagnosis of ALS (Mitsumoto, 1997). Ho et al. (2011) conducted a retrospective study specifically to investigate the frequency of shoulder pain in patients with ALS; 23% of their patients reported having pain in this area. Factors such as poor mobility, suboptimal transfer techniques, spasticity, loss of ROM, difficulty with positioning in bed or in a wheelchair, and contractures are thought to contribute to this type of secondary musculoskeletal pain (Majmudar et al., 2014).
Scapular mobilization has been shown to play a role in prevention of shoulder pain in people with a variety of conditions. A review of the literature supports the efficacy of scapular mobilization in the reduction of pain in the shoulder complex, with multiple reports that this technique has provided relief of pain and returned mobility to patients with adhesive capsulitis, frozen shoulder, hemiplegic shoulder pain, and subacromial impingement (Davis, 2012; Dean et al., 2013; Donatelli et al., 2014; Kibler & Sciascia, 2016; Lucado, 2011; Manske, 2011; Paine & Voight, 2013). To our knowledge, no published studies have examined the efficacy of scapular mobilization of the painful shoulder among people with ALS. The aim of our retrospective case series was to investigate the efficacy of such an intervention to reduce pain and improve shoulder ROM for people with ALS.
Method
Study Patients and Data Collection
We reviewed the records of all patients with ALS who had documented shoulder pain and shoulder forward flexion ROM limitations and who had been seen by the same occupational therapist at the Mayo Clinic in Jacksonville, Florida, between October 1, 2014, and September 30, 2016, for inclusion in this retrospective case series. Patients were excluded if information on visual analog scale (VAS) pain score (Hawker et al., 2011) and shoulder ROM before and after mobilization treatment was not available. A diagnosis of ALS was provided by a neurologist using El Escorial criteria (Brooks, 1994).
After chart review, 28 patients met the inclusion criteria. Information was collected on age at visit, age at ALS onset, duration of ALS, sex, ALS onset site, hand dominance, and affected extremity. The two outcomes were VAS pain score regarding degree of shoulder pain (primary outcome) and shoulder forward flexion ROM (secondary outcome) using a standard shoulder goniometer (Patterson Medical, Warrenville, IL). The data for the primary and secondary outcome measures were collected via the occupational therapy evaluation and treatment notes, which were both located in the electronic health record. The evaluation and one-time treatment were performed by the same occupational therapist, and the two outcomes were assessed both before and after mobilization treatment.
Although normal scapular mechanics demonstrate that upward rotation is needed to improve forward flexion of the glenohumeral joint, the literature has reported that patients with adhesive capsulitis experience excessive upward rotation and anterior tilt of the scapula and would benefit from improvement in downward rotation and posterior tilt of the scapula (Ludewig & Reynolds, 2009). The patients in this study had in common excessive scapular upward rotation and anterior tilt, resulting in a similar pattern of reduced forward flexion glenohumeral ROM. The therapist performed scapular mobilization consisting of downward rotation and posterior tilt of the scapula with anterior glenohumeral support. Scapular mobilization was performed in the seated position for all patients because of limitations with functional mobility and endurance for transfers. Total treatment time for the mobilization was 15 min.
Detailed instructions for home were provided verbally and via video for mobilization in side-lying and sitting positions to accommodate patient and caregiver preferences. The frequency recommended for home was one time daily for 15 min with verbal feedback from the patient to the caregiver for correct positioning and pain relief. Caregiver education via return demonstration and a link to a video were provided to improve compliance and outcomes.
Ten patients had both their left and right shoulders affected and had outcomes assessed for both shoulders. The treatment provided by the occupational therapist was in addition to standard treatment of this patient population, including but not limited to evaluation and treatment of occupational performance deficits and recommendations for energy conservation, activity modifications, compensatory techniques, and adaptive equipment to improve occupational performance. To satisfy the statistical assumption of independent measurements, our primary analysis considered only measurements taken on the dominant arm for those 10 patients, resulting in one set of measurements for each of the 28 study patients. In a secondary analysis, we separately analyzed the data from the nondominant arm for the subset of 10 patients with both shoulders affected.
Statistical Analysis
To evaluate the primary aim of our study, we compared VAS pain scores and shoulder forward flexion ROM before and after mobilization treatment using a paired Wilcoxon signed-rank test. In our secondary analysis, we evaluated both the correlation between change in VAS pain score and that between change in shoulder forward flexion ROM from before to after treatment using Spearman rank order correlations; p values of .025 or lower were considered statistically significant after applying a Bonferroni correction for the two statistical tests that were performed in evaluating the primary aim of the study. All statistical tests were two sided. All statistical analysis was performed using R statistical software (Version 3.1.1; R Foundation for Statistical Computing, Vienna, Austria).
Results
A summary of patient characteristics and outcomes is provided in Table 1. Median age at ALS onset was 59.8 yr, and median age at the occupational therapy visit was 61.5 yr. Approximately two-thirds of patients (67.9%) were men, and the majority of patients had limb onset (82.1%).
Patient Characteristics and Outcomes (N = 28)
Note. Percentages may not total 100 because of rounding. ALS = amyotrophic lateral sclerosis; Mdn = median; ROM = range of motion; VAS = visual analog scale.
Continuous variables are summarized using sample median (range). For the 10 patients who had both their left and right shoulders affected, with outcomes (VAS pain score and shoulder ROM) assessed for both shoulders, only measurements taken on the dominant arm were used in these analyses to satisfy the statistical assumption of independent measurements.
In the primary analysis including all patients (and using only measurements taken from the dominant arm for the 10 patients with both shoulders affected), the median VAS pain score was 2 (range = 0–10) before treatment and 0 (range = 0–3) after treatment, resulting in a significant median reduction in VAS pain score of 2 (range = 0–10, p < .001; Table 1). Similarly, median shoulder forward flexion ROM was 100° (range = 45°–150°) before mobilization treatment and 130° (range = 65°–160°) after treatment, with a significant median increase of 25° (range = 10°–40°, p < .001; Table 1). There was no significant correlation between either change in VAS pain score or change in shoulder forward flexion ROM from before to after treatment (Spearman’s r = −.12, p = .55).
In our secondary analysis, we analyzed the data from the nondominant arm for the subset of 10 patients with both shoulders affected. These 10 patients had a significant reduction in VAS pain score (median = 2 vs. 0, respectively; p = .008) and a significant increase in shoulder forward flexion ROM (median = 115° vs. 135°, respectively; p = .002) from before to after treatment. Again, there was no notable correlation between change in VAS pain score and change in shoulder ROM (Spearman r = −.09, p = .80).
Discussion
Lack of evidence supporting treatment of painful shoulder in patients with motor neuron disease presents a challenge for occupational therapists. The clinician treating patients with progressive motor neuron disease must carefully consider avoidance of pain and complications such as a painful shoulder. Patients with shoulder pain demonstrate a statistically significant increased incidence of upper extremity weakness (Ho et al., 2011).
A barrier that we encountered in the process of this study was how to transition our treatment from the institutional environment to the patient’s personal environment. Education was a key component of improving compliance. Part of the education process for the caregiver and the patient was to determine the most effective schedule for the patient’s specific needs. Incorporating the new exercise recommendation into the patient’s and caregiver’s existing daily routine allowed them to determine the best long-term schedule.
Improvement is needed in the standard of care for the patient with a motor neuron disease. Manual therapy, specifically scapular mobilization, to improve scapulohumeral rhythm of the affected upper extremity among people with ALS is a valuable tool. For the 28 patients discussed in the case series, scapular mobilization resulted in a decrease in pain in the affected shoulder and significant improvement in shoulder forward flexion ROM, which has been identified as a complication of upper and lower motor neuron weakness in people with ALS. Increased awareness of scapular mobilization to address the specific needs of the affected upper extremity can improve management of complications, quality of life, and occupational performance of these people.
Implications for Occupational Therapy Practice
A focus on self-management and maintaining routines that promote health is at the core of occupational therapy practice. Techniques that decrease pain and subsequently improve quality of life are vital in occupational therapy practice with patients with ALS. The importance of intervention is supported by evidence of its psychological and physical effects on people with ALS and their caregivers (Simmons, 2005). When not properly managed, pain can have detrimental effects on all aspects of daily activity and quality of life (Katz, 2002). According to the World Health Organization’s (2001) International Classification of Functioning, Disability and Health, body structure and function affect people’s activities and participation. The pain of ALS affects people’s participation in self-care activities and increases the risk for isolation resulting from pain behaviors (Simons et al., 2014). This study has the following implications for occupational therapy practice:
Reducing pain is an important goal to improve quality of life and increase activity participation for people with ALS.
Occupational therapists can effectively promote shoulder care techniques such as scapular mobilization to both patients and care providers to reduce shoulder pain.
Reducing pain for patients with ALS programs might help them to retain functional independence for longer, allowing for continued participation in activities of daily living.
Conclusion
Our results provide strong evidence that VAS pain score and shoulder forward flexion ROM both improve significantly after mobilization treatment. Limitations include the relatively small sample size, retrospective design, lack of long-term follow-up, and measurement of home compliance. A prospective study, including details of home compliance and long-term follow-up to continually identify and validate the optimal application of scapular mobilization for shoulder pain in ALS care, would further validate our findings.
Footnotes
Acknowledgments
None of the authors received any funding from any funding agencies in the public, commercial, or not-for-profit sectors. We thank Kevin Boylan for his collaboration and leadership on the multidisciplinary ALS team at the Mayo Clinic in Jacksonville, FL. Dr. Boylan provided the diagnosis using El Escorial criteria for each patient included in this retrospective review.