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Journal of Huntington’s Disease 3 (2014) 189–195 DOI 10.3233/JHD-140102 IOS Press
Research Report
A Pilot Study of Virtual Visits in Huntington Disease1 Michael T. Bulla , Kristin Darwinb , Vinayak Venkataramanc , Joseph Wagnera , Christopher A. Beckd , E. Ray Dorseya and Kevin M. Biglana,∗ a Department
of Neurology, University of Rochester, Rochester, NY, USA Hopkins University School of Medicine, University of Rochester, Rochester, NY, USA c Duke University School of Medicine University of Rochester, Rochester, NY, USA d Department of Biostatistics and Computational Biology, University of Rochester, Rochester, NY, USA b Johns
Abstract. Background: Virtual visits through web-based video conferencing can increase access to specialty care for individuals with Huntington disease (HD) and facilitate research participation. Objective: To determine the feasibility of conducting virtual visits directly into the homes of individuals with HD, to assess the reliability of conducting remote versus in-person motor assessments, and to determine the test-retest reliability of conducting motor assessments remotely. Methods: Individuals with mild to moderate HD underwent baseline in-person clinic assessments and completed a HD care survey. Participants were randomized to receive three virtual visits from one of two physicians over four months that included a modified Unified Huntington’s Disease Rating Scale motor examination (excluding rigidity and balance assessments) via webbased video conferencing. Intraclass coefficients (ICC) were calculated to determine the level of agreement between remote and in-person assessments. Participants also completed a survey on their interest in telemedicine. Results: Thirteen individuals underwent baseline assessments, eleven (85%) participants completed at least one virtual visit, and 27 (82%) of 33 total virtual visits were completed. Remote motor scores demonstrated good reliability (ICC = 0.78; n = 11) compared to in-person motor scores. Test-retest reliability of motor scores conducted remotely was excellent (ICC = 0.90; n = 11). Participants expressed moderate future interest in using virtual visits to participate in research and to receive care. Conclusion: In this pilot study, virtual visits into the home were feasible and reliable for conducting motor assessments in HD. Larger scale studies need to confirm and generalize these findings to a broader population of participants. Keywords: Telehealth, Huntington disease, feasibility, reliability, remote assessment
INTRODUCTION Access to specialty care and participation in research for individuals with Huntington disease (HD) may be limited by distance, disability, and the distribution of 1 This study was approved by the University of Rochester Research Subjects Review Board and conducted in compliance with guidelines on human experimentation of the University of Rochester. ∗ Correspondence to: Kevin M. Biglan, M.D., M.P.H., University of Rochester, Rochester, NY 14620, USA. Tel.: +1 585 275 2427; Fax: +1 585 276 1870; E-mail: Kevin [email protected].
specialists [1]. Care from a specialist is associated with improved process measures and health outcomes for many chronic conditions, [2, 3] including other movement disorders [4, 5]. In addition, participation in HD research studies generally requires the ability to access a specialized center. Increasingly, telemedicine or “care at a distance” [6] uses telecommunications technology to increase access to care. Studies have increasingly shown that telemedicine is feasible in a wide range of settings, [7] is comparable to traditional in-person care, [8] generates good
ISSN 1879-6397/14/$27.50 © 2014 – IOS Press and the authors. All rights reserved
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levels of patient satisfaction, [9] demonstrates benefits in home care and in chronic disease, [10, 11] and is cost-effective [12]. Previous studies have shown telemedicine, through the use of simple technology akin to Skype, to be effective in neurology and psychiatry [13, 14]. In Parkinson disease, for example, studies have demonstrated the feasibility and potential value of telemedicine for providing care in the home [15]. In addition, several studies have demonstrated the feasibility and reliability of conducting the Unified Parkinson’s Disease Rating Scale remotely [6, 16, 17]. However, such studies have not been conducted in Huntington disease. We, therefore, conducted a pilot study to determine the feasibility of conducting virtual visits using web-based video conferencing into the homes of individuals with HD and to determine the feasibility and reliability of conducting remote motor assessments. MATERIALS AND METHODS Study design We conducted a pilot study to evaluate the feasibility of conducting virtual visits directly into the homes of individuals with HD through web-based video conferencing. After providing written informed consent, participants in the study underwent a baseline in-person assessment at either the 2012 Annual Huntington Disease Society of America (HDSA) Convention in Las Vegas, NV or at the University of Rochester Medical Center. The baseline assessments included a review of their demographic information, medical and HD history, their medications, and clinical assessments including the motor component of the Unified Huntington’s Disease Rating Scale (UHDRS), [18] Total Functional Capacity, and cognition as measured by the Montreal Cognitive Assessment (MoCA) [19, 20]. The baseline clinical assessments were all conducted by a single investigator (K.M.B.). All participants also completed a survey of their current HD care and of general computer and internet usage. Following the baseline assessments, individuals were randomized using a random number generator to receive three virtual visits over the following four months (months 1, 3, and 4) via web-based video conferencing by a movement disorders specialist at the University of Rochester Medical Center (K.M.B.) or at Johns Hopkins (E.R.D.). All participants were emailed a link to Health Insurance Portability and Accountability Act-compliant, web-based video conferencing software from Vidyo (Hackensack, NJ) and hosted by IDSolutions (Indianapolis, IN). Participants
were also provided a web-camera (Microsoft LifeCam HD-3000) if they did not already own one. Research coordinators (M.T.B., K.D., or V.V.) helped participants, family members or friends, or caretakers with the installation and connection process of the webcamera and video conferencing software by telephone. The virtual visits were scheduled around the availability of the participants. Each virtual visit included assessments of motor function using a modification to the motor UHDRS (“modified motor UHDRS”) that excluded assessments of rigidity and balance that cannot be performed remotely. In addition, participants completed a survey of their satisfaction with various aspects of the virtual visit at month three and a remote cognitive assessment using the MoCA at month four. The surveys were sent via email and returned by email, fax or mail. The MoCA was conducted remotely, with the portion of the form filled out by the participant sent via email. Upon completion, a screenshot of the form was taken and a copy of the form was sent to the research team by email, fax or mail [21, 22]. The institutional review boards at the University of Rochester Medical Center and Johns Hopkins approved the research protocol and consent forms. This study was funded by Lundbeck, LLC. which had no role in the design, analysis or reporting of this study. Participants Participants were men and women at least 18 years of age with a clinical diagnosis of Huntington disease, a total functional capacity score of at least 7 and a MoCA score of at least 20. Individuals also had to have access to a computer with internet access. Exclusion criteria included any neurological, medical or psychiatric condition, in the investigator’s judgment, that would preclude the individual from participating in a telemedicine visit. Outcomes The primary study outcome was feasibility, as measured by the proportion of telemedicine visits completed as scheduled. The secondary outcome was the reliability of conducting the modified motor UHDRS remotely and the test-retest reliability of its assessment remotely. Supplemental outcomes included responses to the baseline care and subsequent virtual visit survey. Survey responses were scored on a 10-point Likert Scale ranging from very uncomfortable (1) to very comfortable (10).
M.T. Bull et al. / Virtual Visits in HD
Statistical analysis In this pilot study, we pre-specified the threshold for feasibility as completing at least 80% of virtual visits as scheduled. In addition, secondary feasibility outcomes included the feasibility of completing the modified motor section of the UHDRS (excluding rigidity and retropulsion) using the same feasibility threshold. The reliability of conducting the modified motor UHDRS remotely was assessed by calculating the intra-class correlation coefficients (ICC) to determine the level of agreement between the baseline in-person and remote assessment conducted one month later. A similar analysis was conducted to assess the test-retest reliability of conducting motor assessments remotely one month apart. In addition, weighted Cohens kappa coefficients were calculated to evaluate agreement between individual motor items. Responses to survey items were analyzed descriptively. RESULTS Study participants Thirteen participants from 10 states (see Fig. 1) completed baseline in-person assessments at the HDSA Convention (n = 11) or at the University of Rochester (n = 2). Randomization resulted in unequal randomization with 10 participants randomized to receive telemedicine care with KMB. Participants generally had mild to moderate HD with baseline motor and cognitive impairment and on average, lived over 200
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miles away from their HD care provider whom they saw approximately three times annually (Table 1). Individuals had a high comfort level with technology: 69% were comfortable using a computer and 100% used the internet and send/receive email at least occasionally. Feasibility Eleven (85%) of the thirteen participants completed at least one virtual visit. One individual was lost to follow-up prior to any virtual visits despite numerous attempts to reach them by phone and email. Another individual withdrew from the study prior to the first virtual visit. Both were randomized to receive telemedicine care from KMB. For the remaining eleven participants, 27 (82%) of the 33 virtual visits were completed as scheduled, and the main reasons for noncompletion were because the patient did not remember they had a visit scheduled or because of technical complications, including poor connection causing lag in audio and video, no audio or video at all, dropped connection after starting visit and not being able to connect to the internet in the participants home and contributed to the time establishing a connection. Twenty-nine of the 33 modified motor examinations were scored completely and nearly all (99%) of all individual motor items were scored. The 4 modified motor examinations that were not scored completely were because the rater felt they had an insufficient view to score a particular item or that the picture and/or connection quality prevented them from being able to accurately score a particular item. On average the virtual visits lasted
Fig. 1. Geographic distribution of study participants.
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M.T. Bull et al. / Virtual Visits in HD Table 1 Baseline characteristics All (n = 13) Demographics Age (years) Gender (% female) Ethnicity (% Caucasian) Years form symptom onset Pursued genetic testing (% yes) Modified total motor UHDRS score∗ Total Functional Capacity Montreal cognitive assessment Clinical Care Type of HD provider (% neurologist)# Distance form HD provider (miles) Lost time form work due to clinic visit for HD-Participant (% yes) Lost time form work due to clinic visit for HD-Spouse/Caregiver (% yes) Time spent per HD visit (minutes)∧ Frequency of visit (per year) Technology Use Own a desktop Own a laptop Own a smartphone Own an e-book reader Own a tablet Use the Internent Check e-mail Use Skype or similar services Use You Tube or similar services Use Social Media/Networking Mean comfort level using computers [scale 1(very uncomfortable)-10(very comfortable)]
56.5 ± 16.6 53.8 92.3 9.0 ± 9.2 (n = 12) 84.6 29.9 ± 12.5 11.0 ± 1.8 24.0 ± 3.4 92.3 227 ± 299 23.1 38.5 289 ± 297 2.9 ± 2.9 69.2 76.9 69.2 15.4 15.4 100.0 100.0 30.8 61.5 76.9 7.54 ± 2.70
UHDRS = Unified Huntington disease Rating Scale. ∗ Total motor score excluding assessment for rigidity and balance (“pull” test). # One participant indicated they receive care for their HD from their primary care physician. ∧ Accounted for the time traveling to and from appointment, time waiting for physician, and time spent with physician.
Fig. 2. Time saved via Telemedicine.
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Table 2 Reliability of the mUHDRS motor exam assessed in-person versus remotely Score (Mean ± SD) In-person assessment Remote assessment (Baseline) (Visit 1) Total sample (n = 11) Modified total motor score Oculomotor Dystonia Chorea Total bradykinesia Dysarthria Tongue Finger taps Rapid alternating movement Luria Bradykinesia Gait Tandem walking (n = 10)
31.00 ± 12.62 7.55 ± 3.53 2.55 ± 3.11 9.0 ± 5.15 12.72 ± 5.62 0.55 ± 0.52 1.09 ± 0.70 4.45 ± 1.21 2.64 ± 1.50 0.73 ± 0.90 0.64 ± 0.81 0.73 ± 0.79 0.90 ± 1.29
Measures of reproducibility Intra-class correlation Weighted Cohen’s kappa
34.64 ± 11.18 10.55 ± 4.08 1.18 ± 1.25 11.18 ± 4.14 12.18 ± 4.85 0.55 ± 0.69 0.91 ± 0.94 3.73 ± 0.79 2.55 ± 1.13 1.36 ± 0.92 1.09 ± 0.94 0.82 ± 0.60 0.70 ± 1.06
0.78 0.41 0.31 0.75 0.79 – – – – – – – –
– – – – – 0.69 0.40 0.11 0.16 −0.016 0.34 0.37 0.45
UHDRS = Unified Huntington disease Rating Scale; SD = standard deviation; HD, Huntington disease. In-person UHDRS were measured at baseline. Remote modified UHDRS were conducted via web-based video conferencing at 1, 3 and 4 months for HD patients.
37 minutes from “log-on to log-off” time, compared to the participants’ usual physician encounters, which lasted on average 289 minutes “roundtrip.” Moreover, the majority of time associated with a virtual visit was in direct physician interaction, whereas the majority of time associated with usual encounters was in travel (Fig. 2). Finally, as scheduling telemedicine visits was more flexible, none of the participants or their caregivers had to miss work for the virtual visits. Reliability of motor assessments The modified total motor scores for in-person visits demonstrated good reliability (ICC of 0.78; n = 11) compared to the remote assessments (Table 2). The test-retest reliability of modified total motor score conducted via telemedicine between the final two visits (Table 3) was excellent (ICC of 0.90; n = 11). Survey of participants All 11 participants who had at least one virtual visit completed a survey on their experience and interest in future virtual visits. Eight of the eleven participants rated their level of comfort in enrolling in a telemedicine program as an 8 or higher, with six of the eight rating their comfort level a 10 (very comfortable). Likewise, eight participants expressed high levels of interest in participating in research visits via telemedicine as opposed to research visits conducted in-person, scoring an 8 or higher, with six of the eight rating their interest level as a 10. Seven participants indicated high levels of interest in enrolling
in a telemedicine program to receive care for their HD as an alternative method to in-person clinic visits, giving a rating of 8 or higher, with five of the seven rating their interest level a 10. Overall, the average score out of 10 for comfort level in a telemedicine program for HD care was 7.9, for interest in enrolling in a telemedicine program for HD care, 7.4, and interest in participating in HD research virtually,7.3. The majority (n = 7) of participants also indicated that they would be willing to pay some amount of money above insurance coverage to receive their HD care via telemedicine. DISCUSSION In this pilot study, conducting virtual visits using simple web-based video conferencing directly into the homes of individuals with HD was feasible. In addition, the reliability of motor assessments conducted remotely compared to in-person was good and the test-retest reliability of remote motor assessments was excellent. Given the small sample size of the population and the selective participation of individuals with comfort in the use of technology, the results of this study require replication and confirmation in a larger, more representative sample of individuals with HD. This study in HD confirms that most elements of the standard motor examination can be performed and assessed remotely as is this case with Parkinson disease [6]. Total chorea (ICC = 0.75) and bradykinesia (ICC = 0.79) when scored remotely demonstrated the best reliability compared to in-person. However, the motor examination in HD relies more heavily on
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M.T. Bull et al. / Virtual Visits in HD Table 3 Test re-test reliablility of the mUHDRS motor exam Score (Mean ± SD) Remote assessment (Visit 2) Remote assessment (Visit 3)
Total sample (n = 11) mTMS total Oculomotor Dystonia (n = 10) Chorea Total bradykinesia Dysarthria Tongue Finger Taps RAM Luria B Bradykinesia (n = 9) Gait (n = 10) Tandern walking
35.91 ± 12.51 11.55 ± 5.47 1.00 ± 1.49 00.09 ± 3.99 13.18 ± 4.58 0.72 ± 0.47 0.82 ± 0.75 4.27 ± 0.79 2.91 ± 1.30 1.27 ± 0.90 0.78 ± 1.09 0.60 ± 0.70 1.18 ± 1.17
36.27 ± 13.45 9.64 ± 4.54 1.40 ± 1.90 11.27 ± 4.54 15.18 ± 6.29 0.91 ± 0.54 1.00 ± 1.00 4.36 ± 1.43 2.45 ± 0.93 1.64 ± 1.36 1.18 ± 1.00 0.90 ± 0.74 1.45 ± 1.13
Measures of reproducibility Intra-class correlation Weighted Cohen’s kappa 0.90 0.77 0.39 0.67 0.86 – – – – – – – –
– – – – – 0.59 0.40 0.48 0.43 0.48 0.37 0.61 0.63
mUHDRS, Modified Unified Huntington disease Rating Scale; SD, Standard Deviation; ICC, Intra-class Correlation; mTMS, Modified Total Motor Score; HD, Huntington disease Disease; RAM, Rapid Alternating Movement; B Bradykinesia, Body Bradykinesia. In-person UHDRS were measured at baseline. Remotem UHDRS were conducted via web-besed video conferencing at 1, 3 and 4 months for HD patients.
visualizing the entire body, which is harder to perform in a video call that typically visualizes the upper torso and head, to assess more subtle features (e.g., chorea in the toes) and measures that require higher resolution (e.g., saccadic eye movements), bandwidth speeds (512 Kbps minimum for Audio and HD Video) that are not universally available as well as a space conducive to perform the tasks necessary to evaluate all elements of the examination. These features were subjectively harder to assess and generally had more modest level agreements (occulomotor, ICC = 0.41; dystonia, ICC = 0.31) than other elements of the examination. Despite these limitations, this study adds HD to the list of conditions that stand to benefit from advances in internet technologies. As indicated in this study, individuals with HD can live hundreds of miles from their HD specialist and research centers. As such, care and research participation can be limited by distance and its requisite travel and time costs, and as the disease progresses, disability. In Parkinson disease where distances and disability may not be as great as in HD, over 40% of individuals with the condition do not see a neurologist, and those that do not have worse health outcomes [4]. One can imagine that similar disparities are present in HD although this remains to be demonstrated. In addition to care, a recent study in Alzheimer disease suggested that home visits would enable greater participation in clinical trials [23]. By using technology and without the requirement for any health provider in the home, virtual visits could reduce the burden to participate in observational and interventional
research studies. Moreover, telemedicine assessments in conjunction with remote measures of function and potentially quantitative motor assessments could be used in clinical trials to reduce participant burden, and more accurately reflect the effects of treatment. Hopefully, this pilot study will represent the first step to using increasingly available and inexpensive technology to increase access to care and research for individuals with HD. ACKNOWLEDGMENTS Funding for this study was provided through a grant from Lundbeck, LLC. Accommodations and space for the research conducted at the 2012 HDSA annual convention in Las Vegas, NV, was provided by the Huntington Disease Society of America. CONFLICT OF INTEREST AND FINANCIAL DISCLOSURE Drs. Biglan and Dorsey have filed a patent related to telemedicine and neurological disorders and are consultants to Lundbeck. Dr. Dorsey is a member of the Medical Advisory Board for Grand Rounds, an online second opinion service, and an unpaid adviser to SBR Health and Vidyo. STUDY FUNDING Funding for this study was provided through a grant from Lundbeck, LLC.
M.T. Bull et al. / Virtual Visits in HD
REFERENCES [1]
[2]
[3]
[4]
[5]
[6]
[7]
[8]
[9] [10]
[11]
[12]
Dorsey ER, George BP, Leff B, Willis AW. The coming crisis: Obtaining care for the growing burden of neurodegenerative conditions. Neurology. 2013;80(21):1989-96. Wolfs C, Kessels A, Dirksen CD, Severens JL, Verhey F. Integrated multidisciplinary diagnostic approach for dementia care: Randomized controlled trial. Br J Psychiatry. 2008;192:300-5. Ahmed SN, Mann C, Sinclair DB, Heino A, Iskiw B, Quigley D, Ohinmaa A. Feasibility of epilepsy follow-up care through telemedicine: A pilot study on the patient’s perspective. Epilepsia. 2008;49(4):573-85. Willis AW, Schootman M, Evanoff BA, Perlmutter JS, Racette BA. Neurologist care in Parkinson disease: A utilization, outcomes, and survival study. Neurology. 2011;77:851-7. Willis AW, Schootman M, Tran R, Kung N, Evanoff BA, Perlmutter JS, Racette BA. Neurologist-associated reduction in PD-related hospitalizations and health care expenditures. Neurology. 2012;79:1774-80. Dorsey ER, Deuel LM, Voss TS, Finnigan K, George BP, Eason S, Miller D, Reminick J, Appler A, Polanowicz J, Viti L, Smith S, Joseph A, Biglan KM. Increasing access to specialty care: A pilot, randomized controlled trial of telemedicine for Parkinson disease. Mov Disord. 2010;25:1652-9. Pervez MA, Silva G, Masrur S, Betensky RA, Furie KL, Hidalgo R, Lima F, Rosenthal ES, Rost N, Viswanathan A, Schwamm LH. Remote supervision of IV-tPA for acute ischemic stroke by telemedicine or telephone before transfer to a regional stroke center is feasible and safe. Stroke. 2010;41:e18-e24. Aoki N, Dunn K, Johnson-Throop KA, Turley JP. Outcomes and methods in telemedicine evaluation. Telemed J E Health. 2003;9:393-401. Mair F, Whitten P. Systematic review of studies of patient satisfaction with telemedicine. BMJ. 2000;320:1517-20. Hailey D, Roine R, Ohinmaa A. Systematic review of evidence for the benefits of telemedicine. J Telemed Telecare. 2002;8(Suppl 1):1-30. Hersh WR, Hickam DH, Severance SM, Dana TL, Pyle Krages K, Helfand M. Diagnosis, access and outcomes: Update of a systematic review of telemedicine services. J Telemed Telecare. 2006;12(Suppl 2):S3-31. Rojas SV, Gagnon MP. A systematic review of the key indicators for assessing telehomecare cost-effectiveness. Telemed J E Health. 2008;14:896-904.
[13]
[14]
[15]
[16]
[17]
[18]
[19]
[20]
[21]
[22]
[23]
195
Hubble JP, Pahwa R,Michalek DK, Thomas C, Koller WC. Interactive video conferencing: A means of providing interim care to Parkinson’s disease patients. Mov Disord 1993;8: 380-2. Samii A, Ryan-Dykes P, Tsukuda RA, Zink C, Franks R, Nichol WP. Telemedicine for the delivery of health care in Parkinson’s disease. J Telemed Telecare 2006;12:16-8. Dorsey ER, Venkataraman V, Grana MJ, Bull MT, George BP, Boyd CM, Beck CA, Raja B, Seidmann A, Biglan KM. Randomized controlled clinical trial of “virtual house calls” for Parkinson Disease. JAMA Neurol. 2013;70(5):565-70. Goetz CG, Tilley BC, Shaftman SR, Stebbins GT, Fahn S, Martinez-Martin P, Poewe W, Sampaio C, Stern MB, Dodel R, Dubois B, Holloway R, Jankovic J,Kulisevsky J, Lang AE, Lees A, Leurgans S, LeWitt PA, Nyenhuis D, Olanow CW, Rascol O, Schrag A, Teresi JA, van Hilten JJ, LaPelle N; Movement Disorder Society UPDRS Revision Task Force. Mov Disord. 2008;23(15):2129-70. Cubo E, Gabriel-Gal´an JM, Mart´ınez JS, Alcubilla CR, Yang C, Arconada OF, P´erez NM. Comparison of office-based versus home Web-based clinical assessments for Parkinson’s disease. Mov Disord. 2012;27(2):308-11. Kieburtz K, Penney Jr. JB, Como P, Ranen N, Shoulson I, Huntington Study Group. Unified Huntington’s Disease Rating Scale: Reliability and Consistency. Mov Disord. 1996;11(2):136-42. Gluhm S, Goldstein J, Brown D, Van Liew C, Gilbert PE, Corey-Bloom J. Usefulness of the Montreal Cognitive Assessment (MoCA) in Huntington’s disease. Mov Disord. 2013;28(12):1744-7. Bezdicek O, Majernova V, Novak M, Nikolai T, Ruzicka E, Roth J. Validity of the montreal cognitive assesment in the detection of cognitive dysfunction in huntington’s disease. Appl Neuropsychol Adult. 2013;20(1):33-40. Bull MT, Grana M, Venkataraman V, Dorsey ER, Biglan KM. Conducting the montreal cognitive assessment remotely in parkinson disease [Abstract]. Mov Disord. 2013;28(10): e4-e5. Bull MT, Darwin K, Venkataraman V, Wagner J, Dorsey ER, Biglan KM. Conducting the montreal cognitive assessment remotely in huntington’s disease [Abstract]. Neurotherapeutics. 2014;11:224. Karlawish J, Cary MS, Rubright J, TenHave T. How redesigning AD clinical trials might increase study partners’ willingness to participate. Neurology. 2008;71(23):1883-8.
Journal of Huntington’s Disease 3 (2014) 189–195 DOI 10.3233/JHD-140102 IOS Press
Research Report
A Pilot Study of Virtual Visits in Huntington Disease1 Michael T. Bulla , Kristin Darwinb , Vinayak Venkataramanc , Joseph Wagnera , Christopher A. Beckd , E. Ray Dorseya and Kevin M. Biglana,∗ a Department
of Neurology, University of Rochester, Rochester, NY, USA Hopkins University School of Medicine, University of Rochester, Rochester, NY, USA c Duke University School of Medicine University of Rochester, Rochester, NY, USA d Department of Biostatistics and Computational Biology, University of Rochester, Rochester, NY, USA b Johns
Abstract. Background: Virtual visits through web-based video conferencing can increase access to specialty care for individuals with Huntington disease (HD) and facilitate research participation. Objective: To determine the feasibility of conducting virtual visits directly into the homes of individuals with HD, to assess the reliability of conducting remote versus in-person motor assessments, and to determine the test-retest reliability of conducting motor assessments remotely. Methods: Individuals with mild to moderate HD underwent baseline in-person clinic assessments and completed a HD care survey. Participants were randomized to receive three virtual visits from one of two physicians over four months that included a modified Unified Huntington’s Disease Rating Scale motor examination (excluding rigidity and balance assessments) via webbased video conferencing. Intraclass coefficients (ICC) were calculated to determine the level of agreement between remote and in-person assessments. Participants also completed a survey on their interest in telemedicine. Results: Thirteen individuals underwent baseline assessments, eleven (85%) participants completed at least one virtual visit, and 27 (82%) of 33 total virtual visits were completed. Remote motor scores demonstrated good reliability (ICC = 0.78; n = 11) compared to in-person motor scores. Test-retest reliability of motor scores conducted remotely was excellent (ICC = 0.90; n = 11). Participants expressed moderate future interest in using virtual visits to participate in research and to receive care. Conclusion: In this pilot study, virtual visits into the home were feasible and reliable for conducting motor assessments in HD. Larger scale studies need to confirm and generalize these findings to a broader population of participants. Keywords: Telehealth, Huntington disease, feasibility, reliability, remote assessment
INTRODUCTION Access to specialty care and participation in research for individuals with Huntington disease (HD) may be limited by distance, disability, and the distribution of 1 This study was approved by the University of Rochester Research Subjects Review Board and conducted in compliance with guidelines on human experimentation of the University of Rochester. ∗ Correspondence to: Kevin M. Biglan, M.D., M.P.H., University of Rochester, Rochester, NY 14620, USA. Tel.: +1 585 275 2427; Fax: +1 585 276 1870; E-mail: Kevin [email protected].
specialists [1]. Care from a specialist is associated with improved process measures and health outcomes for many chronic conditions, [2, 3] including other movement disorders [4, 5]. In addition, participation in HD research studies generally requires the ability to access a specialized center. Increasingly, telemedicine or “care at a distance” [6] uses telecommunications technology to increase access to care. Studies have increasingly shown that telemedicine is feasible in a wide range of settings, [7] is comparable to traditional in-person care, [8] generates good
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levels of patient satisfaction, [9] demonstrates benefits in home care and in chronic disease, [10, 11] and is cost-effective [12]. Previous studies have shown telemedicine, through the use of simple technology akin to Skype, to be effective in neurology and psychiatry [13, 14]. In Parkinson disease, for example, studies have demonstrated the feasibility and potential value of telemedicine for providing care in the home [15]. In addition, several studies have demonstrated the feasibility and reliability of conducting the Unified Parkinson’s Disease Rating Scale remotely [6, 16, 17]. However, such studies have not been conducted in Huntington disease. We, therefore, conducted a pilot study to determine the feasibility of conducting virtual visits using web-based video conferencing into the homes of individuals with HD and to determine the feasibility and reliability of conducting remote motor assessments. MATERIALS AND METHODS Study design We conducted a pilot study to evaluate the feasibility of conducting virtual visits directly into the homes of individuals with HD through web-based video conferencing. After providing written informed consent, participants in the study underwent a baseline in-person assessment at either the 2012 Annual Huntington Disease Society of America (HDSA) Convention in Las Vegas, NV or at the University of Rochester Medical Center. The baseline assessments included a review of their demographic information, medical and HD history, their medications, and clinical assessments including the motor component of the Unified Huntington’s Disease Rating Scale (UHDRS), [18] Total Functional Capacity, and cognition as measured by the Montreal Cognitive Assessment (MoCA) [19, 20]. The baseline clinical assessments were all conducted by a single investigator (K.M.B.). All participants also completed a survey of their current HD care and of general computer and internet usage. Following the baseline assessments, individuals were randomized using a random number generator to receive three virtual visits over the following four months (months 1, 3, and 4) via web-based video conferencing by a movement disorders specialist at the University of Rochester Medical Center (K.M.B.) or at Johns Hopkins (E.R.D.). All participants were emailed a link to Health Insurance Portability and Accountability Act-compliant, web-based video conferencing software from Vidyo (Hackensack, NJ) and hosted by IDSolutions (Indianapolis, IN). Participants
were also provided a web-camera (Microsoft LifeCam HD-3000) if they did not already own one. Research coordinators (M.T.B., K.D., or V.V.) helped participants, family members or friends, or caretakers with the installation and connection process of the webcamera and video conferencing software by telephone. The virtual visits were scheduled around the availability of the participants. Each virtual visit included assessments of motor function using a modification to the motor UHDRS (“modified motor UHDRS”) that excluded assessments of rigidity and balance that cannot be performed remotely. In addition, participants completed a survey of their satisfaction with various aspects of the virtual visit at month three and a remote cognitive assessment using the MoCA at month four. The surveys were sent via email and returned by email, fax or mail. The MoCA was conducted remotely, with the portion of the form filled out by the participant sent via email. Upon completion, a screenshot of the form was taken and a copy of the form was sent to the research team by email, fax or mail [21, 22]. The institutional review boards at the University of Rochester Medical Center and Johns Hopkins approved the research protocol and consent forms. This study was funded by Lundbeck, LLC. which had no role in the design, analysis or reporting of this study. Participants Participants were men and women at least 18 years of age with a clinical diagnosis of Huntington disease, a total functional capacity score of at least 7 and a MoCA score of at least 20. Individuals also had to have access to a computer with internet access. Exclusion criteria included any neurological, medical or psychiatric condition, in the investigator’s judgment, that would preclude the individual from participating in a telemedicine visit. Outcomes The primary study outcome was feasibility, as measured by the proportion of telemedicine visits completed as scheduled. The secondary outcome was the reliability of conducting the modified motor UHDRS remotely and the test-retest reliability of its assessment remotely. Supplemental outcomes included responses to the baseline care and subsequent virtual visit survey. Survey responses were scored on a 10-point Likert Scale ranging from very uncomfortable (1) to very comfortable (10).
M.T. Bull et al. / Virtual Visits in HD
Statistical analysis In this pilot study, we pre-specified the threshold for feasibility as completing at least 80% of virtual visits as scheduled. In addition, secondary feasibility outcomes included the feasibility of completing the modified motor section of the UHDRS (excluding rigidity and retropulsion) using the same feasibility threshold. The reliability of conducting the modified motor UHDRS remotely was assessed by calculating the intra-class correlation coefficients (ICC) to determine the level of agreement between the baseline in-person and remote assessment conducted one month later. A similar analysis was conducted to assess the test-retest reliability of conducting motor assessments remotely one month apart. In addition, weighted Cohens kappa coefficients were calculated to evaluate agreement between individual motor items. Responses to survey items were analyzed descriptively. RESULTS Study participants Thirteen participants from 10 states (see Fig. 1) completed baseline in-person assessments at the HDSA Convention (n = 11) or at the University of Rochester (n = 2). Randomization resulted in unequal randomization with 10 participants randomized to receive telemedicine care with KMB. Participants generally had mild to moderate HD with baseline motor and cognitive impairment and on average, lived over 200
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miles away from their HD care provider whom they saw approximately three times annually (Table 1). Individuals had a high comfort level with technology: 69% were comfortable using a computer and 100% used the internet and send/receive email at least occasionally. Feasibility Eleven (85%) of the thirteen participants completed at least one virtual visit. One individual was lost to follow-up prior to any virtual visits despite numerous attempts to reach them by phone and email. Another individual withdrew from the study prior to the first virtual visit. Both were randomized to receive telemedicine care from KMB. For the remaining eleven participants, 27 (82%) of the 33 virtual visits were completed as scheduled, and the main reasons for noncompletion were because the patient did not remember they had a visit scheduled or because of technical complications, including poor connection causing lag in audio and video, no audio or video at all, dropped connection after starting visit and not being able to connect to the internet in the participants home and contributed to the time establishing a connection. Twenty-nine of the 33 modified motor examinations were scored completely and nearly all (99%) of all individual motor items were scored. The 4 modified motor examinations that were not scored completely were because the rater felt they had an insufficient view to score a particular item or that the picture and/or connection quality prevented them from being able to accurately score a particular item. On average the virtual visits lasted
Fig. 1. Geographic distribution of study participants.
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M.T. Bull et al. / Virtual Visits in HD Table 1 Baseline characteristics All (n = 13) Demographics Age (years) Gender (% female) Ethnicity (% Caucasian) Years form symptom onset Pursued genetic testing (% yes) Modified total motor UHDRS score∗ Total Functional Capacity Montreal cognitive assessment Clinical Care Type of HD provider (% neurologist)# Distance form HD provider (miles) Lost time form work due to clinic visit for HD-Participant (% yes) Lost time form work due to clinic visit for HD-Spouse/Caregiver (% yes) Time spent per HD visit (minutes)∧ Frequency of visit (per year) Technology Use Own a desktop Own a laptop Own a smartphone Own an e-book reader Own a tablet Use the Internent Check e-mail Use Skype or similar services Use You Tube or similar services Use Social Media/Networking Mean comfort level using computers [scale 1(very uncomfortable)-10(very comfortable)]
56.5 ± 16.6 53.8 92.3 9.0 ± 9.2 (n = 12) 84.6 29.9 ± 12.5 11.0 ± 1.8 24.0 ± 3.4 92.3 227 ± 299 23.1 38.5 289 ± 297 2.9 ± 2.9 69.2 76.9 69.2 15.4 15.4 100.0 100.0 30.8 61.5 76.9 7.54 ± 2.70
UHDRS = Unified Huntington disease Rating Scale. ∗ Total motor score excluding assessment for rigidity and balance (“pull” test). # One participant indicated they receive care for their HD from their primary care physician. ∧ Accounted for the time traveling to and from appointment, time waiting for physician, and time spent with physician.
Fig. 2. Time saved via Telemedicine.
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Table 2 Reliability of the mUHDRS motor exam assessed in-person versus remotely Score (Mean ± SD) In-person assessment Remote assessment (Baseline) (Visit 1) Total sample (n = 11) Modified total motor score Oculomotor Dystonia Chorea Total bradykinesia Dysarthria Tongue Finger taps Rapid alternating movement Luria Bradykinesia Gait Tandem walking (n = 10)
31.00 ± 12.62 7.55 ± 3.53 2.55 ± 3.11 9.0 ± 5.15 12.72 ± 5.62 0.55 ± 0.52 1.09 ± 0.70 4.45 ± 1.21 2.64 ± 1.50 0.73 ± 0.90 0.64 ± 0.81 0.73 ± 0.79 0.90 ± 1.29
Measures of reproducibility Intra-class correlation Weighted Cohen’s kappa
34.64 ± 11.18 10.55 ± 4.08 1.18 ± 1.25 11.18 ± 4.14 12.18 ± 4.85 0.55 ± 0.69 0.91 ± 0.94 3.73 ± 0.79 2.55 ± 1.13 1.36 ± 0.92 1.09 ± 0.94 0.82 ± 0.60 0.70 ± 1.06
0.78 0.41 0.31 0.75 0.79 – – – – – – – –
– – – – – 0.69 0.40 0.11 0.16 −0.016 0.34 0.37 0.45
UHDRS = Unified Huntington disease Rating Scale; SD = standard deviation; HD, Huntington disease. In-person UHDRS were measured at baseline. Remote modified UHDRS were conducted via web-based video conferencing at 1, 3 and 4 months for HD patients.
37 minutes from “log-on to log-off” time, compared to the participants’ usual physician encounters, which lasted on average 289 minutes “roundtrip.” Moreover, the majority of time associated with a virtual visit was in direct physician interaction, whereas the majority of time associated with usual encounters was in travel (Fig. 2). Finally, as scheduling telemedicine visits was more flexible, none of the participants or their caregivers had to miss work for the virtual visits. Reliability of motor assessments The modified total motor scores for in-person visits demonstrated good reliability (ICC of 0.78; n = 11) compared to the remote assessments (Table 2). The test-retest reliability of modified total motor score conducted via telemedicine between the final two visits (Table 3) was excellent (ICC of 0.90; n = 11). Survey of participants All 11 participants who had at least one virtual visit completed a survey on their experience and interest in future virtual visits. Eight of the eleven participants rated their level of comfort in enrolling in a telemedicine program as an 8 or higher, with six of the eight rating their comfort level a 10 (very comfortable). Likewise, eight participants expressed high levels of interest in participating in research visits via telemedicine as opposed to research visits conducted in-person, scoring an 8 or higher, with six of the eight rating their interest level as a 10. Seven participants indicated high levels of interest in enrolling
in a telemedicine program to receive care for their HD as an alternative method to in-person clinic visits, giving a rating of 8 or higher, with five of the seven rating their interest level a 10. Overall, the average score out of 10 for comfort level in a telemedicine program for HD care was 7.9, for interest in enrolling in a telemedicine program for HD care, 7.4, and interest in participating in HD research virtually,7.3. The majority (n = 7) of participants also indicated that they would be willing to pay some amount of money above insurance coverage to receive their HD care via telemedicine. DISCUSSION In this pilot study, conducting virtual visits using simple web-based video conferencing directly into the homes of individuals with HD was feasible. In addition, the reliability of motor assessments conducted remotely compared to in-person was good and the test-retest reliability of remote motor assessments was excellent. Given the small sample size of the population and the selective participation of individuals with comfort in the use of technology, the results of this study require replication and confirmation in a larger, more representative sample of individuals with HD. This study in HD confirms that most elements of the standard motor examination can be performed and assessed remotely as is this case with Parkinson disease [6]. Total chorea (ICC = 0.75) and bradykinesia (ICC = 0.79) when scored remotely demonstrated the best reliability compared to in-person. However, the motor examination in HD relies more heavily on
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M.T. Bull et al. / Virtual Visits in HD Table 3 Test re-test reliablility of the mUHDRS motor exam Score (Mean ± SD) Remote assessment (Visit 2) Remote assessment (Visit 3)
Total sample (n = 11) mTMS total Oculomotor Dystonia (n = 10) Chorea Total bradykinesia Dysarthria Tongue Finger Taps RAM Luria B Bradykinesia (n = 9) Gait (n = 10) Tandern walking
35.91 ± 12.51 11.55 ± 5.47 1.00 ± 1.49 00.09 ± 3.99 13.18 ± 4.58 0.72 ± 0.47 0.82 ± 0.75 4.27 ± 0.79 2.91 ± 1.30 1.27 ± 0.90 0.78 ± 1.09 0.60 ± 0.70 1.18 ± 1.17
36.27 ± 13.45 9.64 ± 4.54 1.40 ± 1.90 11.27 ± 4.54 15.18 ± 6.29 0.91 ± 0.54 1.00 ± 1.00 4.36 ± 1.43 2.45 ± 0.93 1.64 ± 1.36 1.18 ± 1.00 0.90 ± 0.74 1.45 ± 1.13
Measures of reproducibility Intra-class correlation Weighted Cohen’s kappa 0.90 0.77 0.39 0.67 0.86 – – – – – – – –
– – – – – 0.59 0.40 0.48 0.43 0.48 0.37 0.61 0.63
mUHDRS, Modified Unified Huntington disease Rating Scale; SD, Standard Deviation; ICC, Intra-class Correlation; mTMS, Modified Total Motor Score; HD, Huntington disease Disease; RAM, Rapid Alternating Movement; B Bradykinesia, Body Bradykinesia. In-person UHDRS were measured at baseline. Remotem UHDRS were conducted via web-besed video conferencing at 1, 3 and 4 months for HD patients.
visualizing the entire body, which is harder to perform in a video call that typically visualizes the upper torso and head, to assess more subtle features (e.g., chorea in the toes) and measures that require higher resolution (e.g., saccadic eye movements), bandwidth speeds (512 Kbps minimum for Audio and HD Video) that are not universally available as well as a space conducive to perform the tasks necessary to evaluate all elements of the examination. These features were subjectively harder to assess and generally had more modest level agreements (occulomotor, ICC = 0.41; dystonia, ICC = 0.31) than other elements of the examination. Despite these limitations, this study adds HD to the list of conditions that stand to benefit from advances in internet technologies. As indicated in this study, individuals with HD can live hundreds of miles from their HD specialist and research centers. As such, care and research participation can be limited by distance and its requisite travel and time costs, and as the disease progresses, disability. In Parkinson disease where distances and disability may not be as great as in HD, over 40% of individuals with the condition do not see a neurologist, and those that do not have worse health outcomes [4]. One can imagine that similar disparities are present in HD although this remains to be demonstrated. In addition to care, a recent study in Alzheimer disease suggested that home visits would enable greater participation in clinical trials [23]. By using technology and without the requirement for any health provider in the home, virtual visits could reduce the burden to participate in observational and interventional
research studies. Moreover, telemedicine assessments in conjunction with remote measures of function and potentially quantitative motor assessments could be used in clinical trials to reduce participant burden, and more accurately reflect the effects of treatment. Hopefully, this pilot study will represent the first step to using increasingly available and inexpensive technology to increase access to care and research for individuals with HD. ACKNOWLEDGMENTS Funding for this study was provided through a grant from Lundbeck, LLC. Accommodations and space for the research conducted at the 2012 HDSA annual convention in Las Vegas, NV, was provided by the Huntington Disease Society of America. CONFLICT OF INTEREST AND FINANCIAL DISCLOSURE Drs. Biglan and Dorsey have filed a patent related to telemedicine and neurological disorders and are consultants to Lundbeck. Dr. Dorsey is a member of the Medical Advisory Board for Grand Rounds, an online second opinion service, and an unpaid adviser to SBR Health and Vidyo. STUDY FUNDING Funding for this study was provided through a grant from Lundbeck, LLC.
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REFERENCES [1]
[2]
[3]
[4]
[5]
[6]
[7]
[8]
[9] [10]
[11]
[12]
Dorsey ER, George BP, Leff B, Willis AW. The coming crisis: Obtaining care for the growing burden of neurodegenerative conditions. Neurology. 2013;80(21):1989-96. Wolfs C, Kessels A, Dirksen CD, Severens JL, Verhey F. Integrated multidisciplinary diagnostic approach for dementia care: Randomized controlled trial. Br J Psychiatry. 2008;192:300-5. Ahmed SN, Mann C, Sinclair DB, Heino A, Iskiw B, Quigley D, Ohinmaa A. Feasibility of epilepsy follow-up care through telemedicine: A pilot study on the patient’s perspective. Epilepsia. 2008;49(4):573-85. Willis AW, Schootman M, Evanoff BA, Perlmutter JS, Racette BA. Neurologist care in Parkinson disease: A utilization, outcomes, and survival study. Neurology. 2011;77:851-7. Willis AW, Schootman M, Tran R, Kung N, Evanoff BA, Perlmutter JS, Racette BA. Neurologist-associated reduction in PD-related hospitalizations and health care expenditures. Neurology. 2012;79:1774-80. Dorsey ER, Deuel LM, Voss TS, Finnigan K, George BP, Eason S, Miller D, Reminick J, Appler A, Polanowicz J, Viti L, Smith S, Joseph A, Biglan KM. Increasing access to specialty care: A pilot, randomized controlled trial of telemedicine for Parkinson disease. Mov Disord. 2010;25:1652-9. Pervez MA, Silva G, Masrur S, Betensky RA, Furie KL, Hidalgo R, Lima F, Rosenthal ES, Rost N, Viswanathan A, Schwamm LH. Remote supervision of IV-tPA for acute ischemic stroke by telemedicine or telephone before transfer to a regional stroke center is feasible and safe. Stroke. 2010;41:e18-e24. Aoki N, Dunn K, Johnson-Throop KA, Turley JP. Outcomes and methods in telemedicine evaluation. Telemed J E Health. 2003;9:393-401. Mair F, Whitten P. Systematic review of studies of patient satisfaction with telemedicine. BMJ. 2000;320:1517-20. Hailey D, Roine R, Ohinmaa A. Systematic review of evidence for the benefits of telemedicine. J Telemed Telecare. 2002;8(Suppl 1):1-30. Hersh WR, Hickam DH, Severance SM, Dana TL, Pyle Krages K, Helfand M. Diagnosis, access and outcomes: Update of a systematic review of telemedicine services. J Telemed Telecare. 2006;12(Suppl 2):S3-31. Rojas SV, Gagnon MP. A systematic review of the key indicators for assessing telehomecare cost-effectiveness. Telemed J E Health. 2008;14:896-904.
[13]
[14]
[15]
[16]
[17]
[18]
[19]
[20]
[21]
[22]
[23]
195
Hubble JP, Pahwa R,Michalek DK, Thomas C, Koller WC. Interactive video conferencing: A means of providing interim care to Parkinson’s disease patients. Mov Disord 1993;8: 380-2. Samii A, Ryan-Dykes P, Tsukuda RA, Zink C, Franks R, Nichol WP. Telemedicine for the delivery of health care in Parkinson’s disease. J Telemed Telecare 2006;12:16-8. Dorsey ER, Venkataraman V, Grana MJ, Bull MT, George BP, Boyd CM, Beck CA, Raja B, Seidmann A, Biglan KM. Randomized controlled clinical trial of “virtual house calls” for Parkinson Disease. JAMA Neurol. 2013;70(5):565-70. Goetz CG, Tilley BC, Shaftman SR, Stebbins GT, Fahn S, Martinez-Martin P, Poewe W, Sampaio C, Stern MB, Dodel R, Dubois B, Holloway R, Jankovic J,Kulisevsky J, Lang AE, Lees A, Leurgans S, LeWitt PA, Nyenhuis D, Olanow CW, Rascol O, Schrag A, Teresi JA, van Hilten JJ, LaPelle N; Movement Disorder Society UPDRS Revision Task Force. Mov Disord. 2008;23(15):2129-70. Cubo E, Gabriel-Gal´an JM, Mart´ınez JS, Alcubilla CR, Yang C, Arconada OF, P´erez NM. Comparison of office-based versus home Web-based clinical assessments for Parkinson’s disease. Mov Disord. 2012;27(2):308-11. Kieburtz K, Penney Jr. JB, Como P, Ranen N, Shoulson I, Huntington Study Group. Unified Huntington’s Disease Rating Scale: Reliability and Consistency. Mov Disord. 1996;11(2):136-42. Gluhm S, Goldstein J, Brown D, Van Liew C, Gilbert PE, Corey-Bloom J. Usefulness of the Montreal Cognitive Assessment (MoCA) in Huntington’s disease. Mov Disord. 2013;28(12):1744-7. Bezdicek O, Majernova V, Novak M, Nikolai T, Ruzicka E, Roth J. Validity of the montreal cognitive assesment in the detection of cognitive dysfunction in huntington’s disease. Appl Neuropsychol Adult. 2013;20(1):33-40. Bull MT, Grana M, Venkataraman V, Dorsey ER, Biglan KM. Conducting the montreal cognitive assessment remotely in parkinson disease [Abstract]. Mov Disord. 2013;28(10): e4-e5. Bull MT, Darwin K, Venkataraman V, Wagner J, Dorsey ER, Biglan KM. Conducting the montreal cognitive assessment remotely in huntington’s disease [Abstract]. Neurotherapeutics. 2014;11:224. Karlawish J, Cary MS, Rubright J, TenHave T. How redesigning AD clinical trials might increase study partners’ willingness to participate. Neurology. 2008;71(23):1883-8.
