Gianina Toller1, Kamalini Ranasinghe1, Yann Cobigo1, Adam Staffaroni1, Brian Appleby1, Danielle Brushaber1, Giovanni Coppola1, Bradford Dickerson1, Kimiko Domoto-Reilly1, Julie Fields1, Jamie Fong1, Leah Forsberg1, Nupur Ghoshal1, Neill Graff-Radford1, Murray Grossman1, Hilary Heuer1, Gink-Yuek Hsiung1, Edward Huey1, David Irwin1, Kejal Kantarci1, Daniel Kaufer1, Diana Kerwin1, David Knopman1, John Kornak1, Joel Kramer1, Irene Litvan1, Ian Mackenzie1, Mario Mendez1, Bruce Miller1, Rosa Rademakers1, Eliana Ramos1, Katya Rascovsky1, Erik Roberson1, Jeremy Syrjanen1, Carmela Tartaglia1, Sandra Weintraub1, Brad Boeve1, Adam Boxer1, Howard Rosen1, Katherine Rankin2. 1. From the Department of Neurology (G.T., K.R., Y.C., A.S., J. Fong, H.H., J.K., J.K., B.M., A.B., H.R., K.R.), Memory and Aging Center, University of California, San Francisco; Case Western Reserve University (B.A.), Cleveland, OH; Mayo Clinic (D.B., J. Fields, L.F., N.G.-R., K.K., D. Knopman, J.S., B.B.), Rochester, MN; University of California (G.C., M.M., E.R.), Los Angeles; Department of Neurology (B.D.), Massachusetts General Hospital, Harvard Medical School, Boston; University of Washington (K.D.-R.), Seattle; Washington University (N.G.), St. Louis, MO; University of Pennsylvania (M.G., D.I., K.R.), Philadelphia; University of British Columbia (G.-Y.H., I.M.), Vancouver, Canada; Columbia University (E.H.), New York, NY; University of North Carolina (D. Kaufer), Chapel Hill; University of Texas Southwestern (D. Kerwin), Dallas; Department of Neuroscience (I.L.), Parkinson and Other Movement Disorder Center, University of California, San Diego; Mayo Clinic (R.R.), Jacksonville, FL; University of Alabama at Birmingham (E.R.); University of Toronto (C.T.), Ontario, Canada; and Mesulam Center for Cognitive Neurology and Alzheimer's Disease (S.W.), Northwestern University, Chicago, IL. 2. From the Department of Neurology (G.T., K.R., Y.C., A.S., J. Fong, H.H., J.K., J.K., B.M., A.B., H.R., K.R.), Memory and Aging Center, University of California, San Francisco; Case Western Reserve University (B.A.), Cleveland, OH; Mayo Clinic (D.B., J. Fields, L.F., N.G.-R., K.K., D. Knopman, J.S., B.B.), Rochester, MN; University of California (G.C., M.M., E.R.), Los Angeles; Department of Neurology (B.D.), Massachusetts General Hospital, Harvard Medical School, Boston; University of Washington (K.D.-R.), Seattle; Washington University (N.G.), St. Louis, MO; University of Pennsylvania (M.G., D.I., K.R.), Philadelphia; University of British Columbia (G.-Y.H., I.M.), Vancouver, Canada; Columbia University (E.H.), New York, NY; University of North Carolina (D. Kaufer), Chapel Hill; University of Texas Southwestern (D. Kerwin), Dallas; Department of Neuroscience (I.L.), Parkinson and Other Movement Disorder Center, University of California, San Diego; Mayo Clinic (R.R.), Jacksonville, FL; University of Alabama at Birmingham (E.R.); University of Toronto (C.T.), Ontario, Canada; and Mesulam Center for Cognitive Neurology and Alzheimer's Disease (S.W.), Northwestern University, Chicago, IL. kate.rankin@ucsf.edu.
Abstract
OBJECTIVE: To investigate whether the Revised Self-Monitoring Scale (RSMS), an informant measure of socioemotional sensitivity, is a potential clinical endpoint for treatment trials for patients with behavioral variant frontotemporal dementia (bvFTD). METHODS: We investigated whether RSMS informant ratings reflected disease severity in 475 participants (71 bvFTD mutation+, 154 bvFTD mutation-, 12 behavioral mild cognitive impairment [MCI] mutation+, 98 asymptomatic mutation+, 140 asymptomatic mutation-). In a subset of 62 patients (20 bvFTD mutation+, 35 bvFTD mutation-, 7 MCI mutation+) who had at least 2 time points of T1-weighted images available on the same 3T scanner, we examined longitudinal changes in RSMS score over time and its correspondence to progressive gray matter atrophy. RESULTS: RSMS score showed a similar pattern in mutation carriers and noncarriers, with significant drops at each stage of progression from asymptomatic to very mild, mild, moderate, and severe disease (F 4,48 = 140.10, p < 0.001) and a significant slope of decline over time in patients with bvFTD (p = 0.004, 95% confidence interval [CI] -1.90 to -0.23). More rapid declines on the RSMS corresponded to faster gray matter atrophy predominantly in the salience network (SN), and RSMS score progression best predicted thalamic volume in very mild and mild disease stages of bvFTD. Higher RSMS score predicted more caregiver burden (p < 0.001, 95% CI -0.30 to -0.11). CONCLUSIONS: The RSMS is sensitive to progression of both socioemotional symptoms and SN atrophy in patients with bvFTD and corresponds directly to caregiver burden. The RSMS may be useful in both neurologic practice and clinical trials aiming to treat behavioral symptoms of patients with bvFTD.
OBJECTIVE: To investigate whether the Revised Self-Monitoring Scale (RSMS), an informant measure of socioemotional sensitivity, is a potential clinical endpoint for treatment trials for patients with behavioral variant frontotemporal dementia (bvFTD). METHODS: We investigated whether RSMS informant ratings reflected disease severity in 475 participants (71 bvFTD mutation+, 154 bvFTD mutation-, 12 behavioral mild cognitive impairment [MCI] mutation+, 98 asymptomatic mutation+, 140 asymptomatic mutation-). In a subset of 62 patients (20 bvFTD mutation+, 35 bvFTD mutation-, 7 MCI mutation+) who had at least 2 time points of T1-weighted images available on the same 3T scanner, we examined longitudinal changes in RSMS score over time and its correspondence to progressive gray matter atrophy. RESULTS: RSMS score showed a similar pattern in mutation carriers and noncarriers, with significant drops at each stage of progression from asymptomatic to very mild, mild, moderate, and severe disease (F 4,48 = 140.10, p < 0.001) and a significant slope of decline over time in patients with bvFTD (p = 0.004, 95% confidence interval [CI] -1.90 to -0.23). More rapid declines on the RSMS corresponded to faster gray matter atrophy predominantly in the salience network (SN), and RSMS score progression best predicted thalamic volume in very mild and mild disease stages of bvFTD. Higher RSMS score predicted more caregiver burden (p < 0.001, 95% CI -0.30 to -0.11). CONCLUSIONS: The RSMS is sensitive to progression of both socioemotional symptoms and SN atrophy in patients with bvFTD and corresponds directly to caregiver burden. The RSMS may be useful in both neurologic practice and clinical trials aiming to treat behavioral symptoms of patients with bvFTD.
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