Abstract
Objective
Health care has increasingly expanded into a hybrid in-person/telehealth model. Patients with a variety of health conditions, including cerebellar ataxia, have received virtual health evaluations; however, it remains unknown whether some outcome measures that clinicians utilize in the telehealth setting are reliable and valid. The goal of this project is to evaluate the psychometric properties of the Scale for Assessment and Rating of Ataxia (SARA) for patients with cerebellar ataxia in the telehealth setting.
Methods
Nineteen individuals with cerebellar impairments were recruited on a voluntary basis. Participants completed 2 30-minute testing sessions during which a clinical examination and the SARA were performed. One session was performed in person, and the other session was assessed remotely. Outcome measure performance was video recorded in both environments and independently scored by 4 additional raters with varying levels of clinical experience (ranging from 6 months to 29 years). Concurrent validity was assessed with the Spearman rank order correlation coefficient (α < .05), comparing the virtual SARA scores to their gold standard in-person scores. Interrater reliability was evaluated with the intraclass correlation coefficient (ICC) (2,4) (α < .05).
Results
Fourteen of the 19 participants completed both in-person and telehealth SARA evaluations. We found that the in-person SARA and the telehealth SARA have large concurrent validity (Spearman rho significant at the 2-tailed α of .01 = 0.90; n = 14). Additionally, raters of varying years of experience had excellent interrater reliability for both the in-person SARA (ICC [2,4] = 0.97; n = 19) and the telehealth SARA (ICC [2,4] = 0.98; n = 14).
Conclusion
Our results show that the telehealth SARA is comparable to the in-person SARA. Additionally, raters of varying years of clinical experience were found to have excellent interrater reliability scores for both remote and in-person SARA evaluations.
Impact
Our study shows that the SARA can be used in the telehealth setting for patients with ataxia.
Keywords: Ataxia, Reliability, Scale for Assessment and Rating of Ataxia, Telemedicine, Validity
Introduction
Cerebellar damage may occur from multiple etiologies such as congenital malformations, hereditary syndromes, and acquired injuries. The prognosis for functional mobility often depends on the cause of the impairment. Congenital malformations occur during development and are not progressive; however, patients may appear to have movement changes as they transition through developmental stages. Hereditary syndromes are progressive, and individuals often require compensatory strategies such as external assistive devices and/or physical support to complete mobility tasks over time. The prognosis for acquired cerebellar ataxia is varied; for example, an individual with damage involving a portion of a cerebellar hemisphere may have a more positive prognosis than someone who has damage to cerebellar nuclei, as nuclei are the output stations for cerebellar signals.1–3 The prevalence of hereditary ataxia is 5.6 in 100,000,4 while the prevalence of acquired ataxia is 27 in 100,000.5
Cerebellar pathologies can lead to a number of motor impairments, including ataxia, hypotonia, and/or action tremors.6 These impairments often lead to dysfunction in balance and/or coordination,7,8 which may produce movement decomposition9 and ataxic gait.7,10–14 In addition to balance and coordination, the cerebellum is responsible for a form of motor learning called adaptation, and when impaired can impact rehabilitation and activities of daily living.3,11
Activities of daily living include such things as grooming, bathing, eating, and working. When an individual cannot perform activities of daily living independently, or without assistance, they often limit or eliminate their participation in personal and social experiences. Decreased participation can lead to negative health conditions such as depression, anxiety, and deconditioning, which can negatively impact an individual’s physical and mental health.15,16 A variety of physical therapist interventions, including balance and coordination training,17–23 axial weighting,24–26 and ambulation training27–30 have been found to provide recovery and/or compensatory benefits to promote improvement in functional mobility and increase participation in activities of daily living for individuals with cerebellar impairments. To assess whether physical therapist treatment plans are improving patient function, physical therapists rely on outcome measures to track patient progress and intervention efficacy. Outcome measures address patient performance with a combination of the aspects of the International Classification of Functioning, Disability, and Health model.31
There is a limited selection of outcomes that physical therapists can rely on to measure performance for patients with ataxia. Two primary outcome measures used to track body function and structure impairments are the International Cooperative of Ataxia Rating Scale and a refined version of that scale, the Scale for the Assessment and Rating of Ataxia (SARA). The Friedreich Ataxia Inventory Scale is a subjective outcome measure that addresses participation for people with ataxia.32–37 For the activity component of the International Classification of Functioning, Disability, and Health, the Dynamic Gait Index has been shown to effectively evaluate balance and ambulation, and the Action Research Arm Test has been used to assess functional reach for patients with ataxia.38,39 The psychometric properties of the above outcome measures have been evaluated in the in-person clinical environment.32–39
In particular, the SARA examines a wide range of movement impairments, such as dysmetria, decomposition, and ataxia, and the test has a number of markers to guide therapists in evaluations. For example, there are a variety of minimal detectable change and cutoff scores on the SARA that are relevant to several different ataxic populations, and the SARA has shown excellent interrater reliability,40–42 concurrent validity,43 and construct validity.36 People with spinocerebellar ataxia, the minimal detectable change for the SARA was found to be 3.5.37 Cutoff scores for the SARA for patients with ataxia secondary to cerebellar stroke have been identified as follows: patients who ambulate independently have scores of 8 or lower, patients who ambulate with a quad cane have scores of 11.5 or lower, and patients who ambulate with a walker have scores of 12.25 or lower. Additionally, for patients with ataxia from cerebellar stroke, cutoff SARA scores for the level of dependence in activities of daily living have been found to be as follows: people with mild dependence have scores of 5.5 or lower, people with moderate dependence have scores of 10.0 or lower, people with severe dependence have scores of 14.25 or lower, and people with total dependence have scores of 23 or higher.44 However, despite the above properties, there are no studies that assess the reliability and validity of the SARA for patients with cerebellar dysfunction in the virtual telehealth rehabilitation setting.
Although telehealth medicine has been in existence for many years, current changes in global health, prompted by the coronavirus (COVID-19) pandemic, have highlighted the advantages of telehealth for patients with a variety of health conditions, including patients with cerebellar impairments.45 In the USA, telehealth rehabilitation increased 100-fold and became the primary means of care for clinicians during the COVID-19 pandemic.46 Previous telehealth care research has focused predominantly on musculoskeletal examinations and treatments.47,48 It is important that clinicians understand the psychometric properties of their standardized in-person outcome measures, such as the SARA, in this novel virtual environment, to ensure that they are developing and implementing appropriate virtual physical therapist goals and treatment plans. Thus, the goal of this study is to assess if the SARA is a reliable and valid outcome measure in the remote telehealth setting for patients with cerebellar ataxia. As the SARA is scored predominantly through movement observation, we hypothesize that the SARA is a valid and reliable outcome measure for quantifying cerebellar impairments for patients with cerebellar dysfunction in the telehealth setting.
Methods
Participants
Participants were recruited on a voluntary basis from the Johns Hopkins Hospital Ataxia Clinic, the Johns Hopkins Hospital Outpatient Physical Therapy Clinic, and the Kennedy Krieger Institute Motion Analysis Laboratory. All participants provided written consent per the Johns Hopkins School of Medicine Institutional Review Board. Participants were included if they had cerebellar damage from cerebellar stroke, tumor, or degeneration and were 20 to 80 years old. Participants were excluded if they had extrapyramidal symptoms, peripheral vestibular loss, uncontrolled hypertension, weakness, peripheral sensory loss, dementia, aphasia, orthopedic or pain conditions, and/or were pregnant. Additionally, all participants were required to have internet and camera access. Presence of family and or caregivers was not required for inclusion criteria.
Participants completed 2 30-minute testing sessions during which a neurologic examination and the SARA were performed to assess the impact of cerebellar damage.36 One session was performed in person, and the other session was assessed remotely. The order of remote and in-person sessions varied, with the aim of 50% of participants completing the in-person assessment first and the other 50% completing the telehealth session first, to control for an effect of order. Clinical in-person assessments and virtual telehealth assessments were completed no more than 1 week apart to reduce performance variability.
Clinical Examination
All participants received an examination performed by a licensed physical therapist to assess baseline performance and to ensure that the individuals did not have any other impairments that would impact the outcome assessment. The examination consisted of range of motion of the major joints of the bilateral upper and lower extremities. Strength of the limbs was assessed in a seated position, by holding the limbs against gravity for approximately 5 seconds and included the following muscle groups: shoulder and hip flexors; elbow, wrist, finger, knee, and ankle flexors/extensors. Tone and sensation were not assessed secondary to the inability to assess in the virtual platform. Participants subjectively reported tone and sensory changes by answering the following question: “have you noticed abnormal sensations, like numbness and tingling or difficulty moving your arms and/or legs in the past month?” Additionally, participants reported the number of falls experienced in the past 3 months. Given the high report of falls, for the telehealth sessions, if participants did not have family members or caregivers present, we reconfirmed their location prior to testing. This was done in the event that an adverse event occurred, and we would need to contact emergent medical assistance. All participants were found to have intact strength, range of motion and, sensation. No adverse events occurred.
Outcome Measure
Scale for Assessment and Rating of Ataxia
The SARA is an 8-item measure that assesses sitting and standing balance, gait, and limb kinetic functions. The maximum score for the SARA is 40; higher numbers indicate more severe ataxia. All items were performed as described by the measure36 and took approximately 15 minutes to perform.
Telehealth SARA Modifications
For the telehealth SARA, participants used the technological device they had access to, which included, desktop computer, laptop computer, or phone. We did not standardize the distance to/from the camera. The only requirement was the participants’ body was able to be visualized by the examiner. When available, family/caregivers held the device as required; otherwise, devices were propped on furniture in the room to allow complete visualization.
Of note, the finger chase, finger-to-nose, and heel-to-shin items of the SARA required modification prior to the telehealth assessment. To successfully perform the finger chase, individuals were required to sit sufficiently far from their camera to permit viewing the upper extremity in its entirety. If individuals were too close to the screen, their finger would travel off the screen during the finger chase. As noted above, the examiner requested that participants make their video full screen to have increased visualization of the examiner’s finger, and cameras were modified to ensure that screens were mirrored and not adjacent. For ease of tracking, the examiner’s video was positioned vertically to the participants video, which aided in the ability to assess overshoot or undershoot of the finger chase. To make the finger-to-nose item compatible with the telehealth setting, participants were asked to place an object (eg, a cup) within arm’s reach and make reaching movements from the nose to a specific point on the object. Participants were positioned horizontally to the camera to provide the examiner with a lateral view of the arm. This enabled the examiner to visualize multi-joint movements of the upper extremity, as well as over or undershoots of reach endpoints. Finally, the heel-to-shin item, which requires an individual to lie supine, was modified as several individuals did not have a couch or bed within view of their camera. The heel-to-shin item was modified to have participants sitting in a chair with 1 lower extremity extended to maintain heel contact with the floor, while the contralateral limb translated the shin. In addition to the time to complete the SARA, approximately 10 minutes were required prior to the examination to ensure proper device setup and visualization.
Raters
The clinical examination and SARA were administered to all participants by a licensed physical therapist with more than 7 years of experience. The SARA performance was assessed in both the in-person clinical setting and the telehealth setting, and performance was video recorded in both environments, by the same single rater. Videos were uploaded to a secured drive and independently scored by 4 additional raters with varying levels of clinical experience (ranging from 6 months to 29 years). Prior to scoring, raters underwent a 30-minute group training session on performance of the SARA. During this training session, the raters were given written instructions detailing the performance and scoring of each item, as provided by the measurement. No additional instructions were provided as the goal was to ensure generalizability of the measure. Raters were blinded to the scores of other raters, and videos were randomly assigned/viewed by each rater to reduce impact of prior scores on future ratings.
Analysis
Sample Size
To determine sample size, a power analysis was assessed through G*Power software (Heinrich-Heine-Universität Düsseldorf, Düsseldorf, Germany). An a priori power analysis was used to determine the sample size: the power was set to 0.80, the alpha level was .05, and the effect size was established from previous work.38,39 The power analysis provided an estimated sample size of 17 participants; however, with the expected rate of attrition, we recruited a sample size of 20 participants.
Concurrent Validity
Given the noncontinuous ordinal nature of the data, concurrent validity was assessed with the Spearman rank order correlation coefficient (α < 0.05), comparing the telehealth SARA scores to their gold standard in-person scores. Correlations of <0.3 were considered to indicate small relationships, those of 0.3 to 0.7 indicated moderate relationships, and those of >0.7 indicated large relationships.49
Interrater Reliability
Interrater reliability was evaluated using the intraclass coefficient (ICC) (2,k) (α < .05). ICC values of <0.5 were considered to indicate poor reliability, ICC values between 0.5 and 0.75 were indicative of moderate reliability, ICC values of >0.75 to 0.9 indicated good reliability, and ICC values of >0.9 represented excellent reliability.50
Post Hoc Analysis
To assess if there was an order effect between individuals who completed the in-person SARA first and those who had the telehealth SARA first, we ran a post hoc Wilcoxon signed rank test using the difference in the SARA scores (calculation: telehealth SARA score minus clinic SARA score for participants assessed in the remote format first; clinic SARA score minus telehealth SARA score for participants assessed in the clinic first). Additionally, a Wilcoxon signed rank test was used to assess the magnitude of the effect between the in-person and the telehealth SARA scores. The Statistical Package for the Social Sciences , version 28.0.0.0 (IBM SPSS, Chicago, IL, USA), was used for all statistical analyses, and an alpha level of <.05 was used for all analyses.
Role of the Funding Source
The funder played no role in the design, conduct, or reporting of this study.
Results
Participants
Twenty individuals with cerebellar impairments were recruited; however, 6 dropped out (19 individuals completed the in-person SARA, and 14 of the 19 completed both the telehealth SARA and the in-person SARA). One individual declined to continue after the clinical examination. The Figure shows the SARA score distribution from the in-person and virtual sessions.
Figure.
Distribution of Scale for Assessment and Rating of Ataxia (SARA) scores for 19 participants. X-axis: participants; y-axis: SARA scores.
Given the dropout rate, we utilized the data from our first 13 participants to perform a post hoc power analysis to reassess sample size: the power was set to 0.80, and the alpha level was .05. The post hoc analysis established the need for 11 participants to meet a power of 0.80.
Table 1 shows the complete participant demographic information.
Table 1.
Demographic Information and SARA Scoresa
Participant | Sex | Age (y) b | Diagnosis | Clinic SARA Score c | Virtual SARA Score d | Time Between Sessions (d) e | First Session |
---|---|---|---|---|---|---|---|
1 | F | 85 | A | 12.5 | 11 | 4 | C |
2 | F | 64 | A | 7 | 5 | 6 | C |
3 | F | 63 | IA | 10.5 | C | ||
4 | F | 41 | MS | 18.5 | 14.5 | 2 | V |
5 | M | 56 | Ac | 24 | 23 | 5 | V |
6 | F | 56 | A | 19 | 14.5 | 5 | C |
7 | F | 73 | FA | 13 | 13.5 | 5 | C |
8 | F | 75 | A | 6.5 | C | ||
9 | F | 66 | A | 17.5 | 30 | 13 | C |
10 | F | 46 | A | 7 | 12 | 5 | C |
11 | F | 72 | A | 9.5 | 7 | 8 | C |
12 | M | 70 | CVA | 14 | C | ||
13 | F | 20 | FA | 22 | 20 | 1 | V |
14 | F | 31 | SCA28 | 11 | 13 | 2 | C |
15 | M | 75 | A | 5 | C | ||
16 | M | 63 | A | 2.5 | 2 | 4 | C |
17 | M | 44 | SCA3 | 2 | 2 | 3 | C |
18 | F | 67 | LoA | 8.5 | C | ||
19 | M | 57 | Sp | 20.5 | 19 | 5 | V |
A = ataxia; Ac = acquired ataxia; C = clinic; CVA = cerebrovascular; F = female; FA = Friedreich ataxia; IA = iatrogenic ataxia; LoA = late-onset ataxia; M = male; MS = multiple sclerosis; SARA = Scale for Assessment and Rating of Ataxia; SCA28 = spinocerebellar ataxia type 28; SCA3 = spinocerebellar ataxia type 3; Sp = spontaneous ataxia; V = virtual.
Mean = 59.16 (SD = 16.48).
Mean = 12.13 (SD = 6.60).
Mean = 13.32 (SD = 7.96).
Mean = 4.79 (SD = 3.22).
Concurrent Validity
Concurrent validity was assessed for the SARA scores. We found a large positive relationship between the clinic SARA and telehealth SARA scores (Spearman rho significant at the 2-tailed α of <.01 = 0.90; n = 14).
Interrater Reliability
Raters were found to have excellent interrater reliability for both the in-person SARA (ICC [2,4] = 0.97; n = 19) and the telehealth SARA (ICC [2.4] = 0.98; n = 14) (Tab. 2).
Table 2.
ICC (2,4) of Reliability for All Four Raters’ Scores for Clinic SARA and Virtual SARAa
SARA (No. of Participants) | ICC (2,4) | 95% CI Lower Bound | 95% CI Upper Bound | df |
---|---|---|---|---|
Clinic (19) | 0.97 | 0.95 | 0.99 | 18 |
Virtual (14) | 0.98 | 0.94 | 0.99 | 13 |
SARA = Scale for Assessment and Rating of Ataxia.
Post Hoc Analysis
A Wilcoxon signed rank test did reveal a significant difference for the SARA scores of those who completed the in-person SARA first versus those who completed the telehealth SARA first (P = .04; significance level = .05). All participants who completed the telehealth SARA first were found to have lower SARA scores (ie, higher function during the remote session) than those who completed the telehealth SARA second. Using a z value of −2.04, we found the effect size to be −0.39, indicating a moderate order effect. However, a significant difference was not identified when we compared the in-person SARA scores and the telehealth SARA scores (P = .40; significance level = .05). Using a z value of −0.84, we calculated an effect size of −0.16, indicating a small testing location effect.
Discussion
Our study found that the telehealth SARA had large concurrent validity and excellent interrater reliability. Our results allow both clinicians and researchers to use the SARA in the telehealth setting, with the modifications described above. Our findings of the in-person SARA scores are similar to those reported in prior studies completed in the in-person setting, as they all show high reliability and validity.36–44 Additionally, we found a small effect size between the telehealth and in-person SARA scores, thus supporting our large concurrent validity result.
Our results are clinically meaningful, as understanding the reliability and validity of outcome measures allows clinicians to select measures that are salient to their patient population. In this case, clinicians treating patients with ataxia may now confidently use the SARA in the telehealth setting, given its excellent validity and reliability. As patients have reported decreased certainty in communication with clinicians in the remote environment,51,52 using our study results, clinicians can educate their patients that the tools they are using have been found to be effective for use in the telehealth environment and equate to those scales in the in-person environment, which may promote ease and confidence in provider/patient communication.
Of note, 3 participants had a difference between in-person and telehealth scores of >4 points. Anecdotally, these patients either reported more confidence in their home setting or reported increased fatigue due to daily events that may have affected their performance.
Although we attempted to control for confounding variables, our study did have several limitations. First, we aimed to have half of the participants complete the in-person SARA first, with the other half completing the telehealth SARA first; however, given the dropout rate, we were limited in the number of individuals that completed the telehealth SARA first. We also were limited in our recruitment, as many participants were recruited from a 1-time, in-person ataxia clinic, meaning their in-person assessment was completed first. Our results show that there was a moderate negative order effect, with all participants who received remote assessment first having lower SARA scores in the telehealth setting. This cannot be explained by familiarization as this remote session was provided first. However, our study was powered to evaluate reliability and validity, not to assess differences between means; thus, although our results indicate that there was a significant order effect, future studies would need to be performed to confirm this statement.
Second, the telehealth assessment required modifying 3 test items based on the participants’ home environments. The examiner attempted to control for this factor; for example, if the remote setting was performed first and the heel-to-shin item was performed while the participant was sitting, then the heel-to-shin item was performed in the sitting position in the in-person setting. However, these controls were not applied to cases where the in-person assessment was performed first. Thus, future studies comparing the clinic and telehealth environments would benefit from selecting standard modifications to be used with all participants in both clinic and virtual settings.
Future Directions
It would be beneficial for future studies to assess outcome measures that represent other aspects of the International Classification of Functioning, Disability, and Health model in the telehealth setting for patients with ataxia. For example, the Action Research Arm Test and the Dynamic Gait Index have been found to be reliable and valid in the in-person clinical setting for patients with ataxia; however, it is unknown whether these tools would be useful in the telehealth setting. Additionally, although the majority of the participants in our study reported preference of the remote setting, secondary to ease and convenience of not traveling to a large metropolitan city, we did not objectively quantify patient satisfaction. Thus, the addition of patient satisfaction surveys to future research would provide clinicians with valuable information about their patients’ perspective.
Additionally, as our study was designed to be completed by the patient without assistance, however, some participants had family members present who assisted with technology and others did not. Thus, it would be beneficial for future research to establish the impact of caregiver presence on participant performance during telehealth sessions. On the other hand, our inclusion criteria did require that patients have the cognitive capability to perform our measures independently; thus, future research on the impact of cognition on the performance of the remote SARA would be useful.
Conclusion
Clinicians and researchers who use the SARA for assessment of patients with ataxia, can use the SARA in the telehealth environment. The telehealth SARA has excellent validity and reliability. Continued research is warranted for the evaluation of additional outcome measures in the telehealth setting.
Contributor Information
Rachel Reoli, Department of Physical Medicine and Rehabilitation, Johns Hopkins Hospital, Baltimore, Maryland, USA; Department of Rehabilitation Sciences, University of Maryland Baltimore, Baltimore, Maryland, USA.
Amanda Therrien, Moss Rehabilitation Research Institute, Thomas Jefferson University, Elkins Park, Pennsylvania, USA; Department of Rehabilitation Medicine, Sidney Kimmel Medical College, Thomas Jefferson University, Philadelphia, Pennsylvania, USA.
Jennifer Millar, Department of Physical Medicine and Rehabilitation, Johns Hopkins Hospital, Baltimore, Maryland, USA.
Nayo Hill, Department of Movement Studies, Kennedy Krieger Institute, Baltimore, Maryland, USA; Department of Neuroscience, Johns Hopkins School of Medicine, Baltimore, Maryland, USA.
Rini Varghese, Department of Movement Studies, Kennedy Krieger Institute, Baltimore, Maryland, USA; Department of Neuroscience, Johns Hopkins School of Medicine, Baltimore, Maryland, USA.
Ryan Roemmich, Department of Physical Medicine and Rehabilitation, Johns Hopkins Hospital, Baltimore, Maryland, USA; Department of Movement Studies, Kennedy Krieger Institute, Baltimore, Maryland, USA.
Jill Whitall, Department of Rehabilitation Sciences, University of Maryland Baltimore, Baltimore, Maryland, USA.
Amy Bastian, Department of Physical Medicine and Rehabilitation, Johns Hopkins Hospital, Baltimore, Maryland, USA; Department of Movement Studies, Kennedy Krieger Institute, Baltimore, Maryland, USA; Department of Neuroscience, Johns Hopkins School of Medicine, Baltimore, Maryland, USA; Department of Neurology, Johns Hopkins School of Medicine, Baltimore, Maryland, USA.
Jennifer Keller, Department of Movement Studies, Kennedy Krieger Institute, Baltimore, Maryland, USA; Department of Physical Medicine and Rehabilitation, Johns Hopkins School of Medicine, Baltimore, Maryland, USA.
Author Contributions
Rachel Reoli (Conceptualization [Equal], Data curation [Equal], Formal analysis [Equal], Investigation [Equal], Methodology [Equal], Supervision [Equal], Validation [Equal], Writing—original draft [Equal]), Amanda Therrien (Methodology [Supporting], Supervision [Supporting], Writing—review & editing [Supporting]), Jennifer Millar (Data curation [Supporting], Resources [Supporting], Writing—review & editing [Supporting]), Nayo Hill (Data curation [Supporting], Methodology [Supporting], Writing—review & editing [Supporting]), Rini Varghese (Data curation [Supporting], Methodology [Supporting], Writing—review & editing [Supporting]), Ryan T. Roemmich (Formal analysis [Supporting], Methodology [Supporting], Supervision [Supporting], Writing—review & editing [Supporting]), Jill Whitall (Methodology [Supporting], Supervision [Supporting], Writing—review & editing [Supporting]), Amy Bastian (Conceptualization [Supporting], Methodology [Supporting], Supervision [Supporting], Writing—review & editing [Supporting]), Jennifer Keller (Data curation [Supporting], Methodology [Supporting], Supervision [Supporting], Writing—review & editing [Supporting])
Ethics Approval
All participants provided written consent per the Johns Hopkins School of Medicine Institutional Review Board (IRB).
Funding
This study was supported by a grant from the National Institutes of Health (R01HD097619-03). A.B. received a consulting fee or honorarium from McGraw Hill Publishers and support for travel to meetings from the Society for Neuroscience.
Disclosures
The authors completed the ICMJE Form for Disclosure of Potential Conflicts of Interest and reported no conflicts of interest.
This article was adapted from Rachel Reoli’s dissertation with the University of Maryland, Baltimore that was posted on ProQuest (https://www.proquest.com/dissertations-theses/telehealth-care-how-reliable-are-our-physical/docview/2777471263/se-2?accountid=41004).
References
- 1. NINDS Ataxias and Cerebellar or Spinocerebellar Degeneration Information Page. What is ataxia and cerebellar or spinocerebellar degeneration? 2016. Accessed January 5, 2024. https://www.ninds.nih.gov/health-information/disorders/ataxia-and-cerebellar-or-spinocerebellar-degeneration.
- 2. Gonzalez-Usigli HA. Cerebellar Disorders. Merck Manual Professional Edition. Merck & Co Inc. Rahway, NJ, USA. 2022. Accessed January 5, 2024. http://www.merckmanuals.com/professional/neurologic-disorders/movement-and-cerebellar-disorders/cerebellar-disorders.
- 3. Morton S, Keller J, Bastian A. Cerebellar Dysfunction. In Lazaro RT, Reina-Guerra SG, Quiben M, eds. Umphred's Neurological Rehabilitation. Maryland Heights, MO: Mosby; 2020:560–579. [Google Scholar]
- 4. Ruano L, Melo C, Silva M, Coutinho P. The global epidemiology of hereditary ataxia and spastic paraplegia: a systematic review of prevalence studies. Neuroepidemiology. 2014;42:174–183. [DOI] [PubMed] [Google Scholar]
- 5. Farghaly W, El-Tallawy H, et al. Population based study of acquired cerebellar ataxia in Al-Kharga district, New Valley, Egypt. Neuropsychiatr Dis Treat. 2011;7:183–187. [DOI] [PMC free article] [PubMed] [Google Scholar]
- 6. Bastian A, Martin A, Keating J, Thach W. Cerebellar ataxia: abnormal control of interaction torques across multiple joints. J Neurophysiol. 1996;76:492–509. [DOI] [PubMed] [Google Scholar]
- 7. Holmes G. The cerebellum of man. Brain. 1939;LXII:1–30. [Google Scholar]
- 8. Shumway-Cook A, Wollacott M. Motor Control: Translating Research into Clinical Practice. 4th ed. Philadelphia, PA: Lippincott Williams and Wilkins; 2012: 71–126. [Google Scholar]
- 9. Gillen G. Improving activities of daily living performance in an adult with ataxia. Am J Occup Phys Ther. 2000;54:89–96. [DOI] [PubMed] [Google Scholar]
- 10. Matsushima A, Yoshida K, Genno H, et al. Clinical assessment of standing and gait in ataxic patients using a triaxial accelerometer. Cerebellum Ataxias. 2015;2:1–7. [DOI] [PMC free article] [PubMed] [Google Scholar]
- 11. Thach T, Bastian A. Role of cerebellum in the control and adaptation of gait in health and disease. Prog Brain Res. 2003;143:347–360. [DOI] [PubMed] [Google Scholar]
- 12. Earhart G, Bastian A. Selection and coordination of human locomotor forms following cerebellar damage. Am Physiol Soc. 2001;759–769. [DOI] [PubMed] [Google Scholar]
- 13. Morton S, Bastian A. Cerebellar control of balance and locomotion. Neuroscientist. 2004;10:247–259. [DOI] [PubMed] [Google Scholar]
- 14. Morton S, Bastian A. Relative contributions of balance and voluntary leg-coordination deficits to cerebellar gait ataxia. J Neurophysiol. 2003;89:1844–1856. [DOI] [PubMed] [Google Scholar]
- 15. Boyd R, Stevens J. Falls, and fear of falling: burden, beliefs, and behaviors. Age Ageing. 2009;38:423–428. [DOI] [PubMed] [Google Scholar]
- 16. Hallford D, Nicholson G, Sanders K, McCabe M. The association between anxiety and falls: a meta-analysis. J Gerontol B Psychol Sci Soc Sci. 2017;72:729–741. [DOI] [PubMed] [Google Scholar]
- 17. Sartor-Glittenberg C, Brickner L. A multidimensional physical therapy program for individuals with cerebellar ataxia secondary to traumatic brain injury: a case series. Physiother Theor Pract. 2014;30:138–148. [DOI] [PubMed] [Google Scholar]
- 18. Schroeder T. Rehabilitation of a U.S. army soldier diagnosed with cerebellar atrophy with ataxic gait. Mil Med. 2013;178:e879–e883. [DOI] [PubMed] [Google Scholar]
- 19. Bastian A, Keller J. A home balance exercise program improved walking in people with cerebellar ataxia. Neurorehabil Neural Repair. 2014;28:770–778. [DOI] [PMC free article] [PubMed] [Google Scholar]
- 20. Gill-Body K, Popat R, Parker S, Krebs. Rehabilitation of balance in two patients with cerebellar dysfunction. Phys Ther. 1997;77:534–552. [DOI] [PubMed] [Google Scholar]
- 21. Cakrt O, Vyhnalek M, Slaby K, et al. Balance rehabilitation therapy by tongue electrotactile biofeedback in patients with degenerative cerebellar disease. Neurorehabilitation. 2012;31:429–434. [DOI] [PubMed] [Google Scholar]
- 22. Calvo S, Fernandez G, Verdejol C, Perez R, Atutxa F, Torres UJ. Improvements in quality of life in individuals with Friedreich’s ataxia after participation in a 5-year program of physical activity: an observational study pre-posttest design, and two years follow up. Int J Neurorehabilitation Eng. 2014;1:1–8. [Google Scholar]
- 23. Burciu R, Fritsche N, Granert O, et al. Brain changes associated with postural training in patients with cerebellar degeneration: a voxel-based morphometry study. J Neurosci. 2013;33:4594–4604. [DOI] [PMC free article] [PubMed] [Google Scholar]
- 24. Gibson-Horn C. Balance-based torso-weighting in a patient with ataxia and multiple sclerosis: a case report. JNPT. 2008;32:139–146. [DOI] [PubMed] [Google Scholar]
- 25. Widener G, Allen D, Gibson-Horn C. Balance-based torso-weighting may enhance balance in persons with multiple sclerosis: preliminary evidence. Arch Phys Med Rehabil. 2009b;90:602–609. [DOI] [PubMed] [Google Scholar]
- 26. Widener G, Allen D, Gibson-Horn C. Randomized clinical trial of balance-based torso weighting for improving upright mobility in people with multiple sclerosis. Neurorehabil Neuro Repair. 2009;23:784–791. [DOI] [PubMed] [Google Scholar]
- 27. Vaz D, Schettino R, de Castro T, Teixeira V, Furtado S, Figueiredo E. Treadmill training for ataxic patients: a single subject experimental design. Clin Rehabil. 2008;22:234–241. [DOI] [PubMed] [Google Scholar]
- 28. Freund J, Stetts D. Use of trunk stabilization and locomotor training in an adult with cerebellar ataxia: a single system design. Physiother Theory Pract. 2010;26:447–458. [DOI] [PubMed] [Google Scholar]
- 29. Milne S, Corben L, Georgiou-Karistianis G, Delatycki M, Yiu E. Rehabilitation for individuals with genetic degenerative ataxia: a systematic review. Neurorehabil Neural Repair. 2017;31:609–622. [DOI] [PubMed] [Google Scholar]
- 30. Fonteyn E, Heeren A, Engels J, Den Boer J, van de Warrenburg B, Weerdesteyn V. Gait adaptability training improves obstacle avoidance and dynamic stability in patients with cerebellar degeneration. Gait Posture. 2014;40:247–251. [DOI] [PubMed] [Google Scholar]
- 31. World Health Organization . How to use the ICF: a practical manual for using the International Classification of Functioning, Disability and Health (ICF): Exposure Draft for Comment. Geneva: WHO; 2013. [Google Scholar]
- 32. Block V, Pitsch E, Tahir P, Cree B, Allen D, Gelfand J. Remote physical activity monitoring in neurological disease: a systematic review. PLoS One. 2016;1–41. [DOI] [PMC free article] [PubMed] [Google Scholar]
- 33. Fillyaw M, Badger G, Bradley W, et al. Quantitative measures of neurological function in chronic neuromuscular diseases and ataxia. J Neurol Sci. 1989;92:17–36. [DOI] [PubMed] [Google Scholar]
- 34. Heldner M, Vanbellingen T, Bohlhalter S, Mattle H, Muri R, Kamm C. Coin rotation task: a valid test for manual dexterity in multiple sclerosis. Phys Ther. 2014;94:1644–1651. [DOI] [PubMed] [Google Scholar]
- 35. Matsuo Y, Asai Y, Nomura T, et al. Intralimb incoordination in patients with ataxia. Neuro Report. 2003;14:2057–2059. [DOI] [PubMed] [Google Scholar]
- 36. Saute J, Donis K, Serrano-Munuera C, et al. Ataxia rating scales-psychometric profiles, natural history and their application in clinical trials. Cerebellum. 2012;11:488–504. [DOI] [PubMed] [Google Scholar]
- 37. Schmitz-Hubsch T, Fimmers R, Rakowicz M, et al. Responsiveness of different rating instruments in spinocerebellar ataxia patients. Neurology. 2010;74:678–684. [DOI] [PubMed] [Google Scholar]
- 38. Reoli R, Cherry-Allen K, Therrien A, et al. Can the ARAT be used to measure arm function in people with cerebellar ataxia? Phys Ther. 2021;101:pzza203. [DOI] [PMC free article] [PubMed] [Google Scholar]
- 39. Reoli R, Therrien A, Cherry-Allen K, et al. Is the dynamic gait index a useful outcome to use to measure balance and ambulation in patients with cerebellar ataxia? Gait Posture. 2021;89:200–205. [DOI] [PMC free article] [PubMed] [Google Scholar]
- 40. Schmitz-hubsch T, Du Montcel S, Baliko L, et al. Scale for the assessment and rating of ataxia: development of a new clinical scale. Neurology. 1006;66:1717–1720. [DOI] [PubMed] [Google Scholar]
- 41. Burk K, Malzig U, Wolf S, et al. Comparison of three clinical rating scales in Friedreich ataxia (FRDA). Mov Disord. 2009;24:1779–1784. [DOI] [PubMed] [Google Scholar]
- 42. Weyer A, Able M, Schmitz-hubsch T, et al. Reliability and validity of the scale for the assessment and rating of ataxia: a study in 64 ataxia patients. Mov Disord. 2007;22:1633–1637. [DOI] [PubMed] [Google Scholar]
- 43. Subramony SH. SARA- a new clinical scale for the assessment and rating of ataxia. Nat Clin Pract Neurol. 2007;3:136–137. [DOI] [PubMed] [Google Scholar]
- 44. Kim B, Lim J, Lee S, et al. Usefulness of the scale for assessment and rating of ataxia (SARA) in ataxic stroke patients. Ann Rehabil Med. 2011;35:772–780. [DOI] [PMC free article] [PubMed] [Google Scholar]
- 45. Ferguson S. Is the end of the pandemic the end of telerehabilitation? Phys Ther. 2022;102:1–3. [DOI] [PMC free article] [PubMed] [Google Scholar]
- 46. Dorsey R, Bloem B, Okun M. A new day: the role of telemedicine in reshaping care for persons with movement disorders. Mov Disord. 2020;35:1897–1902. [DOI] [PubMed] [Google Scholar]
- 47. Truter P, Russell T, Fary R. The validity of physical therapy assessment of low back pain via telerehabilitation in a clinical setting. Telemed J E Health. 2014;20:161–167. [DOI] [PubMed] [Google Scholar]
- 48. Havaran M, Bidelspach D. Virtual physical therapy and telerehabilitation. Phys Med Rehabil Clin N Am. 2021;32:419–428. [DOI] [PubMed] [Google Scholar]
- 49. Cohen J, Cohen P, West SG, Aiken LS. Applied Multiple Regression/Correlation Analysis for the Behavioral Sciences. 3rd ed. Mahwah, NJ: Lawrence Erlbaum Associates; 2003. [Google Scholar]
- 50. Koo T, Li M. A guideline of selecting and reporting intraclass correlation coefficients for reliability research. J Chiropract Med. 2016;15:155–163. [DOI] [PMC free article] [PubMed] [Google Scholar]
- 51. Gordon H, Solanki P, Bokhour B, Gopal R. “I’m not feeling lime I’m part of the conversation”: patients’ perspectives on communicating in clinical video telehealth visits. J Gen Intern Med. 2020;35:1751–1758. [DOI] [PMC free article] [PubMed] [Google Scholar]
- 52. Landon K, Portneu T, Perugini J, et al. Perceptions of telehealth vs in-person visits among older adults with advanced kidney disease, care partners, and clinicians. JAMA Netw Open. 2021;4:e2137193. [DOI] [PMC free article] [PubMed] [Google Scholar]