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November 15, 2016; 87 (20) Article

Objective, computerized video-based rating of blepharospasm severity

David A. Peterson, Gwen C. Littlewort, Marian S. Bartlett, Antonella Macerollo, Joel S. Perlmutter, H.A. Jinnah, Mark Hallett, Terrence J. Sejnowski
First published October 21, 2016, DOI: https://doi.org/10.1212/WNL.0000000000003336
David A. Peterson
From the Computational Neurobiology Laboratory (D.A.P., T.J.S.) and Howard Hughes Medical Institute (T.J.S.), Salk Institute for Biological Studies; Institute for Neural Computation (D.A.P., G.C.L., M.S.B., T.J.S.), Kavli Institute for Brain and Mind (D.A.P., T.J.S.), Machine Perception Laboratory (G.C.L., M.S.B.), and Division of Biological Sciences (T.J.S.), University of California, San Diego, La Jolla; Sobell Department of Motor Neuroscience and Movement Disorders (A.M.), National Hospital of Neurology and Neurosurgery, Institute of Neurology, University College London, UK; Departments of Neurology, Radiology, and Anatomy and Neurobiology, and Programs in Physical Therapy and Occupational Therapy (J.S.P.), Washington University School of Medicine, St. Louis, MO; Departments of Neurology, Human Genetics, and Pediatrics (H.A.J.), Emory University, Atlanta, GA; and Human Motor Control Section (M.H.), National Institute of Neurological Disorders and Stroke, NIH, Bethesda, MD.
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Gwen C. Littlewort
From the Computational Neurobiology Laboratory (D.A.P., T.J.S.) and Howard Hughes Medical Institute (T.J.S.), Salk Institute for Biological Studies; Institute for Neural Computation (D.A.P., G.C.L., M.S.B., T.J.S.), Kavli Institute for Brain and Mind (D.A.P., T.J.S.), Machine Perception Laboratory (G.C.L., M.S.B.), and Division of Biological Sciences (T.J.S.), University of California, San Diego, La Jolla; Sobell Department of Motor Neuroscience and Movement Disorders (A.M.), National Hospital of Neurology and Neurosurgery, Institute of Neurology, University College London, UK; Departments of Neurology, Radiology, and Anatomy and Neurobiology, and Programs in Physical Therapy and Occupational Therapy (J.S.P.), Washington University School of Medicine, St. Louis, MO; Departments of Neurology, Human Genetics, and Pediatrics (H.A.J.), Emory University, Atlanta, GA; and Human Motor Control Section (M.H.), National Institute of Neurological Disorders and Stroke, NIH, Bethesda, MD.
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Marian S. Bartlett
From the Computational Neurobiology Laboratory (D.A.P., T.J.S.) and Howard Hughes Medical Institute (T.J.S.), Salk Institute for Biological Studies; Institute for Neural Computation (D.A.P., G.C.L., M.S.B., T.J.S.), Kavli Institute for Brain and Mind (D.A.P., T.J.S.), Machine Perception Laboratory (G.C.L., M.S.B.), and Division of Biological Sciences (T.J.S.), University of California, San Diego, La Jolla; Sobell Department of Motor Neuroscience and Movement Disorders (A.M.), National Hospital of Neurology and Neurosurgery, Institute of Neurology, University College London, UK; Departments of Neurology, Radiology, and Anatomy and Neurobiology, and Programs in Physical Therapy and Occupational Therapy (J.S.P.), Washington University School of Medicine, St. Louis, MO; Departments of Neurology, Human Genetics, and Pediatrics (H.A.J.), Emory University, Atlanta, GA; and Human Motor Control Section (M.H.), National Institute of Neurological Disorders and Stroke, NIH, Bethesda, MD.
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Antonella Macerollo
From the Computational Neurobiology Laboratory (D.A.P., T.J.S.) and Howard Hughes Medical Institute (T.J.S.), Salk Institute for Biological Studies; Institute for Neural Computation (D.A.P., G.C.L., M.S.B., T.J.S.), Kavli Institute for Brain and Mind (D.A.P., T.J.S.), Machine Perception Laboratory (G.C.L., M.S.B.), and Division of Biological Sciences (T.J.S.), University of California, San Diego, La Jolla; Sobell Department of Motor Neuroscience and Movement Disorders (A.M.), National Hospital of Neurology and Neurosurgery, Institute of Neurology, University College London, UK; Departments of Neurology, Radiology, and Anatomy and Neurobiology, and Programs in Physical Therapy and Occupational Therapy (J.S.P.), Washington University School of Medicine, St. Louis, MO; Departments of Neurology, Human Genetics, and Pediatrics (H.A.J.), Emory University, Atlanta, GA; and Human Motor Control Section (M.H.), National Institute of Neurological Disorders and Stroke, NIH, Bethesda, MD.
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Joel S. Perlmutter
From the Computational Neurobiology Laboratory (D.A.P., T.J.S.) and Howard Hughes Medical Institute (T.J.S.), Salk Institute for Biological Studies; Institute for Neural Computation (D.A.P., G.C.L., M.S.B., T.J.S.), Kavli Institute for Brain and Mind (D.A.P., T.J.S.), Machine Perception Laboratory (G.C.L., M.S.B.), and Division of Biological Sciences (T.J.S.), University of California, San Diego, La Jolla; Sobell Department of Motor Neuroscience and Movement Disorders (A.M.), National Hospital of Neurology and Neurosurgery, Institute of Neurology, University College London, UK; Departments of Neurology, Radiology, and Anatomy and Neurobiology, and Programs in Physical Therapy and Occupational Therapy (J.S.P.), Washington University School of Medicine, St. Louis, MO; Departments of Neurology, Human Genetics, and Pediatrics (H.A.J.), Emory University, Atlanta, GA; and Human Motor Control Section (M.H.), National Institute of Neurological Disorders and Stroke, NIH, Bethesda, MD.
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H.A. Jinnah
From the Computational Neurobiology Laboratory (D.A.P., T.J.S.) and Howard Hughes Medical Institute (T.J.S.), Salk Institute for Biological Studies; Institute for Neural Computation (D.A.P., G.C.L., M.S.B., T.J.S.), Kavli Institute for Brain and Mind (D.A.P., T.J.S.), Machine Perception Laboratory (G.C.L., M.S.B.), and Division of Biological Sciences (T.J.S.), University of California, San Diego, La Jolla; Sobell Department of Motor Neuroscience and Movement Disorders (A.M.), National Hospital of Neurology and Neurosurgery, Institute of Neurology, University College London, UK; Departments of Neurology, Radiology, and Anatomy and Neurobiology, and Programs in Physical Therapy and Occupational Therapy (J.S.P.), Washington University School of Medicine, St. Louis, MO; Departments of Neurology, Human Genetics, and Pediatrics (H.A.J.), Emory University, Atlanta, GA; and Human Motor Control Section (M.H.), National Institute of Neurological Disorders and Stroke, NIH, Bethesda, MD.
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Mark Hallett
From the Computational Neurobiology Laboratory (D.A.P., T.J.S.) and Howard Hughes Medical Institute (T.J.S.), Salk Institute for Biological Studies; Institute for Neural Computation (D.A.P., G.C.L., M.S.B., T.J.S.), Kavli Institute for Brain and Mind (D.A.P., T.J.S.), Machine Perception Laboratory (G.C.L., M.S.B.), and Division of Biological Sciences (T.J.S.), University of California, San Diego, La Jolla; Sobell Department of Motor Neuroscience and Movement Disorders (A.M.), National Hospital of Neurology and Neurosurgery, Institute of Neurology, University College London, UK; Departments of Neurology, Radiology, and Anatomy and Neurobiology, and Programs in Physical Therapy and Occupational Therapy (J.S.P.), Washington University School of Medicine, St. Louis, MO; Departments of Neurology, Human Genetics, and Pediatrics (H.A.J.), Emory University, Atlanta, GA; and Human Motor Control Section (M.H.), National Institute of Neurological Disorders and Stroke, NIH, Bethesda, MD.
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Terrence J. Sejnowski
From the Computational Neurobiology Laboratory (D.A.P., T.J.S.) and Howard Hughes Medical Institute (T.J.S.), Salk Institute for Biological Studies; Institute for Neural Computation (D.A.P., G.C.L., M.S.B., T.J.S.), Kavli Institute for Brain and Mind (D.A.P., T.J.S.), Machine Perception Laboratory (G.C.L., M.S.B.), and Division of Biological Sciences (T.J.S.), University of California, San Diego, La Jolla; Sobell Department of Motor Neuroscience and Movement Disorders (A.M.), National Hospital of Neurology and Neurosurgery, Institute of Neurology, University College London, UK; Departments of Neurology, Radiology, and Anatomy and Neurobiology, and Programs in Physical Therapy and Occupational Therapy (J.S.P.), Washington University School of Medicine, St. Louis, MO; Departments of Neurology, Human Genetics, and Pediatrics (H.A.J.), Emory University, Atlanta, GA; and Human Motor Control Section (M.H.), National Institute of Neurological Disorders and Stroke, NIH, Bethesda, MD.
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Citation
Objective, computerized video-based rating of blepharospasm severity
David A. Peterson, Gwen C. Littlewort, Marian S. Bartlett, Antonella Macerollo, Joel S. Perlmutter, H.A. Jinnah, Mark Hallett, Terrence J. Sejnowski
Neurology Nov 2016, 87 (20) 2146-2153; DOI: 10.1212/WNL.0000000000003336

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Abstract

Objective: To compare clinical rating scales of blepharospasm severity with involuntary eye closures measured automatically from patient videos with contemporary facial expression software.

Methods: We evaluated video recordings of a standardized clinical examination from 50 patients with blepharospasm in the Dystonia Coalition's Natural History and Biorepository study. Eye closures were measured on a frame-by-frame basis with software known as the Computer Expression Recognition Toolbox (CERT). The proportion of eye closure time was compared with 3 commonly used clinical rating scales: the Burke-Fahn-Marsden Dystonia Rating Scale, Global Dystonia Rating Scale, and Jankovic Rating Scale.

Results: CERT was reliably able to find the face, and its eye closure measure was correlated with all of the clinical severity ratings (Spearman ρ = 0.56, 0.52, and 0.56 for the Burke-Fahn-Marsden Dystonia Rating Scale, Global Dystonia Rating Scale, and Jankovic Rating Scale, respectively, all p < 0.0001).

Conclusions: The results demonstrate that CERT has convergent validity with conventional clinical rating scales and can be used with video recordings to measure blepharospasm symptom severity automatically and objectively. Unlike EMG and kinematics, CERT requires only conventional video recordings and can therefore be more easily adopted for use in the clinic.

Glossary

BFM=
Burke-Fahn-Marsden Dystonia Rating Scale;
CERT=
Computer Expression Recognition Toolbox;
CI=
confidence interval;
GDRS=
Global Dystonia Rating Scale;
ICC=
intraclass correlation;
JRS=
Jankovic Rating Scale;
UCSD=
University of California, San Diego;
WUSM=
Washington University School of Medicine

Footnotes

  • Go to Neurology.org for full disclosures. Funding information and disclosures deemed relevant by the authors, if any, are provided at the end of the article.

  • Supplemental data at Neurology.org

  • Received May 2, 2016.
  • Accepted in final form August 2, 2016.
  • © 2016 American Academy of Neurology
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