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Research paper
The long-term outcome of orthostatic tremor
  1. Christos Ganos1,2,
  2. Lucie Maugest3,4,
  3. Emmanuelle Apartis4,5,6,
  4. Carmen Gasca-Salas7,
  5. María T Cáceres-Redondo8,
  6. Roberto Erro1,
  7. Irene Navalpotro-Gómez1,9,
  8. Amit Batla1,
  9. Elena Antelmi1,
  10. Bertrand Degos3,6,
  11. Emmanuel Roze3,4,6,
  12. Marie-Laure Welter3,4,6,
  13. Tiago Mestre7,10,
  14. Francisco J Palomar8,
  15. Reina Isayama11,12,
  16. Robert Chen7,11,12,
  17. Carla Cordivari13,
  18. Pablo Mir8,14,
  19. Anthony E Lang7,
  20. Susan H Fox7,
  21. Kailash P Bhatia1,
  22. Marie Vidailhet3,4,6
  1. 1Sobell Department of Motor Neuroscience and Movement Disorders, UCL Institute of Neurology, Queen Square, London, UK
  2. 2Department of Neurology, University Medical Center Hamburg-Eppendorf (UKE), Hamburg, Germany
  3. 3Department of Neurology, AP-HP, Groupe Hospitalier Pitié-Salpêtrière, Paris, France
  4. 4CRICM UPMC/INSERM UMR 1127 CNRS UMR7225 Brain and Spine Institute, Paris, France
  5. 5Neurophysiology Unit, AP-HP, Saint-Antoine Hospital, Paris, France
  6. 6Pierre Marie Curie Paris-6 University, Paris, France
  7. 7Morton and Gloria Shulman Movement Disorders Clinic and the Edmond J. Safra Program in Parkinson's Disease, University Health Network, Toronto, Canada
  8. 8Unidad de Trastornos del Movimiento, Servicio de Neurología y Neurofisiología Clínica, Instituto de Biomedicina de Sevilla, Hospital Universitario Virgen del Rocío/CSIC/Universidad de Sevilla, Seville, Spain
  9. 9Department of Neurology, Hospital del Mar, Barcelona, Spain
  10. 10Parkinson's Disease and Movement Disorder Centre, division of Neurology, department of Medicine, University of Ottawa, The Ottawa Hospital, Ottawa, Canada
  11. 11Department of Medicine, University of Toronto, Toronto, Canada
  12. 12Division of Brain, Imaging and Behavior—Systems Neuroscience, Toronto Western Research Institute, Toronto, Canada
  13. 13Department of Clinical Neurophysiology, National Hospital for Neurology and Neurosurgery, London, UK
  14. 14Centro de Investigación Biomédica en Red sobre Enfermedades Neurodegenerativas (CIBERNED), Spain
  1. Correspondence to Dr Kailash P Bhatia, Clinical Movement Disorders Group, Sobell Department of Motor Neuroscience and Movement Disorders, UCL Institute of Neurology, Queen Square, London WC1N 3BG, UK; k.bhatia{at}ucl.ac.uk

Abstract

Objectives Orthostatic tremor is a rare condition characterised by high-frequency tremor that appears on standing. Although the essential clinical features of orthostatic tremor are well established, little is known about the natural progression of the disorder. We report the long-term outcome based on the largest multicentre cohort of patients with orthostatic tremor.

Methods Clinical information of 68 patients with clinical and electrophysiological diagnosis of orthostatic tremor and a minimum follow-up of 5 years is presented.

Results There was a clear female preponderance (76.5%) with a mean age of onset at 54 years. Median follow-up was 6 years (range 5–25). On diagnosis, 86.8% of patients presented with isolated orthostatic tremor and 13.2% had additional neurological features. At follow-up, seven patients who initially had isolated orthostatic tremor later developed further neurological signs. A total 79.4% of patients reported worsening of orthostatic tremor symptoms. These patients had significantly longer symptom duration than those without reported worsening (median 15.5 vs 10.5 years, respectively; p=0.005). There was no change in orthostatic tremor frequency over time. Structural imaging was largely unremarkable and dopaminergic neuroimaging (DaTSCAN) was normal in 18/19 cases. Pharmacological treatments were disappointing. Two patients were treated surgically and showed improvement.

Conclusions Orthostatic tremor is a progressive disorder with increased disability although tremor frequency is unchanged over time. In most cases, orthostatic tremor represents an isolated syndrome. Drug treatments are unsatisfactory but surgery may hold promise.

  • MOVEMENT DISORDERS
  • TREMOR

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