Anne-Marie Wills1, Alexander Pantelyat2, Alberto Espay3, James Chan4, Irene Litvan5, Tao Xie6, Marian L Dale7, Steven A Gunzler8, Maria Carmela Tartaglia9, Susan H Fox10, Federico Rodriguez-Porcel11, Mansi Sharma12, Anthony E Lang10, Adam L Boxer13, Lawrence I Golbe14. 1. Department of Neurology, Massachusetts General Hospital, Harvard Medical School, Boston, Massachusetts, USA. 2. Department of Neurology, Johns Hopkins University School of Medicine, Baltimore, Maryland, USA. 3. James J. and Joan A. Gardner Family Center for Parkinson's Disease and Movement Disorders, Department of Neurology, University of Cincinnati, Cincinnati, Ohio, USA. 4. Biostatistics Center, Massachusetts General Hospital, Boston, Massachusetts, USA. 5. Parkinson and Other Movement Disorders Center, UC San Diego, Department of Neurosciences, La Jolla, California, USA. 6. Movement Disorder Program, Department of Neurology, University of Chicago Medicine, Chicago, Illinois, USA. 7. Department of Neurology, Oregon Health & Science University, Portland, Oregon, USA. 8. Parkinson's and Movement Disorders Center, Neurological Institute, University Hospitals Cleveland Medical Center and Case Western Reserve University, Cleveland, Ohio, USA. 9. Tanz Centre for Research in Neurodegenerative Diseases, Rossy Progressive Supranuclear Palsy Centre, University of Toronto, Krembil Brain Institute, University Health Network, Toronto, Ontario, Canada. 10. Edmond J. Safra Program in Parkinson's Disease, Rossy Progressive Supranuclear Palsy Centre, Movement Disorders Clinic, Krembil Brain Institute, Toronto Western Hospital, University Health Network and Division of Neurology, University of Toronto, Toronto, Ontario, Canada. 11. Department of Neurology, Medical University of South Carolina, Charleston, South Carolina, USA. 12. Department of Neurology, Massachusetts General Hospital, Boston, Massachusetts, USA. 13. Memory and Aging Center, Department of Neurology, University of California, San Francisco, California, USA. 14. Rutgers Robert Wood Johnson Medical School, New Brunswick, New Jersey, USA.
Abstract
BACKGROUND: The reliability of the Progressive Supranuclear Palsy Rating Scale (PSPRS) using teleneurology has not been assessed. OBJECTIVES: To test whether removing items inadequately assessed by video would impact measurement of PSP severity and progression. METHODS: We performed secondary analyses of two data sets: the phase 2/3 trial of Davunetide in PSP and a large single-center cohort. We examined two modifications of the PSPRS: (1) removing neck rigidity, limb rigidity, and postural stability (25 items; mPSPRS-25) and (2) also removing three ocular motor items and limb dystonia (21 items; mPSPRS-21). Proportional agreement relative to the possible total scores was measured using the intraclass correlation coefficient, compared to the original PSPRS baseline values and change over 6 and 12 months. We examined the ability of both scales to predict survival in the single-center cohort using proportional hazards models. RESULTS: The mPSPRS-25 showed excellent agreement (0.99; P < 0.001) with the original PSPRS at baseline, 0.98 (P < 0.001) agreement in measuring change over 6 months, and 0.98 (P < 0.001) over 12 months. The mPSPRS-21 showed agreement of 0.94 (P < 0.001) with the original PSPRS at baseline, 0.92 (P < 0.001) at 6 months, and 0.95 (P < 0.001) at 12 months. Baseline and 6-month change in both modified scales were highly predictive of survival in the single-center cohort. CONCLUSIONS: Modified versions of the PSPRS which can be administered remotely show excellent agreement with the original scale and predict survival in PSP. The mPSPRS-21 should facilitate clinical care and research in PSP via teleneurology.
BACKGROUND: The reliability of the Progressive Supranuclear Palsy Rating Scale (PSPRS) using teleneurology has not been assessed. OBJECTIVES: To test whether removing items inadequately assessed by video would impact measurement of PSP severity and progression. METHODS: We performed secondary analyses of two data sets: the phase 2/3 trial of Davunetide in PSP and a large single-center cohort. We examined two modifications of the PSPRS: (1) removing neck rigidity, limb rigidity, and postural stability (25 items; mPSPRS-25) and (2) also removing three ocular motor items and limb dystonia (21 items; mPSPRS-21). Proportional agreement relative to the possible total scores was measured using the intraclass correlation coefficient, compared to the original PSPRS baseline values and change over 6 and 12 months. We examined the ability of both scales to predict survival in the single-center cohort using proportional hazards models. RESULTS: The mPSPRS-25 showed excellent agreement (0.99; P < 0.001) with the original PSPRS at baseline, 0.98 (P < 0.001) agreement in measuring change over 6 months, and 0.98 (P < 0.001) over 12 months. The mPSPRS-21 showed agreement of 0.94 (P < 0.001) with the original PSPRS at baseline, 0.92 (P < 0.001) at 6 months, and 0.95 (P < 0.001) at 12 months. Baseline and 6-month change in both modified scales were highly predictive of survival in the single-center cohort. CONCLUSIONS: Modified versions of the PSPRS which can be administered remotely show excellent agreement with the original scale and predict survival in PSP. The mPSPRS-21 should facilitate clinical care and research in PSP via teleneurology.
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