Background: Orthostatic tremor is an uncommon disorder manifest by high frequency, low amplitude leg tremor upon weight bearing. Treatment with oral tremor agents is inconsistent and usually not satisfactory. Methods: We implanted bilateral ventralis intermedius nuclei deep brain stimulators into an 82-year-old male with refractory orthostatic tremor. Results: The patient had a marked subjective and objective improvement in leg and arm tremor, mainly manifested by an improved ability to stand. Discussion: Bilateral thalamic deep brain stimulation may be considered in refractory cases of orthostatic tremor

Ondo, William G.

Yaltho, Toby C.

Tremor and Other Hyperkinetic Movements

English

PubMed

Columbia University Academic Commons

Case ReportThalamic Deep Brain Stimulation for Orthostatic TremorToby C. Yaltho1& William G. Ondo1*1 Parkinson’s Disease Center and Movement Disorders Clinic, Department of Neurology, Baylor College of Medicine, Houston, Texas, United States of AmericaAbstractBackground: Orthostatic tremor is an uncommon disorder manifest by high frequency, low amplitude leg tremor upon weight bearing. Treatment with oraltremor agents is inconsistent and usually not satisfactory.Methods: We implanted bilateral ventralis intermedius nuclei deep brain stimulators into an 82-year-old male with refractory orthostatic tremor.Results: The patient had a marked subjective and objective improvement in leg and arm tremor, mainly manifested by an improved ability to stand.Discussion: Bilateral thalamic deep brain stimulation may be considered in refractory cases of orthostatic tremor.Keywords: Orthostatic tremor; deep brain stimulation; ventral intermediate nucleus of the thalamusCitation: Yaltho TC, Ondo WG. Thalamic deep brain stimulation for orthostatic tremor. Tremor Other Hyperkinet Mov 2011;1: http://www.tremorjournal.org/article/view/26* To whom correspondence should be addressed. E-mail: wondo@bcm.tmc.eduEditor: Elan D. Louis, Columbia University, United States of AmericaReceived: April 20, 2011 Accepted: June 1, 2011 Published: August 10, 2011Copyright: ’ 2011 Yaltho and Ondo. This is an open-access article distributed under the terms of the Creative Commons Attribution–Noncommerical–No Derivatives License, whichpermits the user to copy, distribute, and transmit the work provided that the original author and source are credited; that no commercial use is made of the work; and that the work is notaltered or transformed.Funding: This research received no specific grant from any funding agency in the public, commercial, or not-for-profit sectors.Competing Interests: TCY is a member of the speaker’s bureau for Teva Neuroscience and a consultant for Merz Pharmaceuticals. WGO is a member of the speaker’s bureau forGlaxoSmithKline, Teva Neuroscience, Boehringer Ingelheim, Allergan, Ipsen, Merz Pharmaceuticals, and Lundbeck.IntroductionOrthostatic tremor (OT) was described in 1984 by Heilman astremor of the legs and trunk when standing, accompanied byunsteadiness or fear of falling, that is relieved by walking or leaningon nearby objects.1 A characteristic electrophysiological finding of a13–18 Hz leg tremor is observed while standing. Treatment responsewith oral medications is often unsatisfactory. We report a patient withmedication-refractory OT treated successfully with bilateral ventralisintermedius nucleus (Vim) thalamic deep brain stimulation (DBS)surgery.Case ReportThe patient is a 78-year-old, right-handed Caucasian male whopresented with a 5-year history of unsteadiness occurring within a fewminutes upon standing, later associated with fine tremor of both legs,left worse than right. He would have to sit down or lean against a walldue to concern for falling. The symptoms resolved with walking orsitting. There was no improvement with alcohol. The patient reporteddifficulty taking a shower and he avoided social functions. His medicalhistory included hyperlipidemia, hypertension, myocardial infarctionwith placement of three cardiac stents, laminectomy of cervical spinelevels 3–5, and benign prostatic hypertrophy. His brother had a headtremor of unknown duration.Initial physical examination demonstrated a fine but visible legtremor with a latency of approximately 1 minute after standing. Hewas able to stand uncomfortably but independently for a maximum of2 minutes. Interestingly, he had a mild postural and kinetic flexion-extension hand tremor that was worse when standing and improvedwith sitting or walking. There was no cogwheel rigidity or evidence oforthostatic hypotension. Surface electromyography (EMG) of thegastrocnemius and vastus lateralis demonstrated a 14 Hz regulartremor consistent with the diagnosis of OT. Magnetic resonanceimaging (MRI) of the brain was unremarkable, and MRI of thecervical spine showed postoperative changes from his prior laminect-omy. Medications subsequently tried for his OT included clonazepam(3 mg/day), alprazolam (1.5 mg/day), gabapentin (3600 mg/day),topiramate (200 mg/day), levodopa (600 mg/day), pregabalin (225mg/day), phenobarbital (90 mg/day), and zonisamide (100 mg/day).His symptoms progressed, and at 82 years of age he was only able tostand independently for 30–45 seconds.Since the patient was completely refractory to medications, heunderwent a bilateral Vim thalamic DBS implantation (Soletra,Freely available onlineTremor and Other Hyperkinetic Movementshttp://www.tremorjournal.orgThe Center for Digital Research and ScholarshipColumbia University Libraries/Information Services1Medtronic, Minneapolis, Minnesota), 9 years after the onset of hissymptoms. Pharmacological treatment was discontinued. At 6-monthspost-implantation his stimulator settings were as follows: amplitude 2.6 V,pulse width 90 us, rate 170 Hz for the right active electrode (case cathodal,contacts 0, 1, 3 anodal); amplitude 2.1 V, pulse width 90 us, rate 135 Hzfor the left active electrode (case cathodal, contacts 0 and 3 anodal) afterfour programming sessions. The patient noted marked improvement inboth his OT and hand tremor, and he was able to stand for more than 4minutes unassisted during follow-up evaluations (Video 1).DiscussionOT can negatively impact quality of life,2 and pharmacologictreatment response is often found to be modest and inconsistent.3Surgical intervention has been tried in a small number of patients withsevere OT (Table 1). Spinal cord dorsal column stimulation (SCS),typically used for treatment of medically refractory pain, was beneficial intwo patients with OT,4 suggesting that the spinal cord is associated withthe generation or modulation of the tremor. Two patients with OT havebeen previously reported to benefit from bilateral Vim thalamic DBSimplantation.5,6 However, one patient with unilateral Vim thalamic DBSdid not have sustained improvement.6 Although controlled trials are notavailable, our case supports the consideration of bilateral thalamic DBSimplantation in patients with disabling, medication refractory OT.While the pathogenesis of OT is not known, it is widely believed toinvolve a primary central tremor generator and a cerebello-thalamo-cortico-spinal loop. The unique observation that our patient’s handtremor also improved with gait suggests that tremor in OT canoverflow to other anatomical regions not directly stimulated by anti-gravity-induced muscle contraction and that the lower-frequency handtremor and higher-frequency leg tremor share the same pathophysiol-ogy. Whether the presence of mild arm tremor predicts a good DBSresponse for OT awaits confirmation with additional cases.References1. Heilman KM. Orthostatic tremor. Arch Neurol 1984;41(8):880–881.2. Gerschlager W, Katzenschlager R, Schrag A, et al. Quality of life inpatients with orthostatic tremor. J Neurol 2003;250(2):212–215, http://dx.doi.org/10.1007/s00415-003-0980-9.3. Gerschlager W, Munchau A, Katzenschlager R, et al. Natural history andsyndromic associations of orthostatic tremor: a review of 41 patients. MovDisord 2004;19(7):788–795, http://dx.doi.org/10.1002/mds.20132.4. Krauss JK, Weigel R, Blahak C, et al. Chronic spinal cord stimulation inmedically intractable orthostatic tremor. J Neurol Neurosurg Psychiatry 2006;77(9):1013–1016, http://dx.doi.org/10.1136/jnnp.2005.086132.5. Guridi J, Rodriguez-Oroz MC, Arbizu J, et al. Successful thalamicdeep brain stimulation for orthostatic tremor. Mov Disord 2008;23(13):1808–1811, http://dx.doi.org/10.1002/mds.22001.6. Espay AJ, Duker AP, Chen R, et al. Deep brain stimulation of the ventralintermediate nucleus of the thalamus in medically refractory orthostatic tremor:preliminary observations. Mov Disord 2008;23(16):2357–2362, http://dx.doi.org/10.1002/mds.22271.Video 1. Patient 4 Months Prior to DBS Implantation and 6 MonthsAfter DBS ImplantationSegment 1 of the video shows the patient 4 months prior to DBS implantation. Hehas a 1+ postural tremor of the hands while sitting. Shortly after standing there is avisible tremor of the legs and worsening of his postural hand tremor. The patientappears uncomfortable and has to sit down approximately 45 seconds afterstanding. He has improvement in his OT symptoms with walking. Segment 2 ofthe video shows the patient 6 months post-DBS implantation. There is significantimprovement in his hand tremor while standing and the leg tremor is not visible.The patient is able to stand for several minutes without difficulty.Table 1. Surgical Treatment of Orthostatic TremorReference Patient Disease Duration(Years)Age at Surgery(Years)Surgery Sustained ClinicalImprovementKrauss et al.4 1 10 58 Spinal cord stimulation Yes2 8 72 Spinal cord stimulation YesGuridi et al.5 3 11 55 Bilateral Vim DBS YesEspay et al.6 4 28 73 Bilateral Vim DBS Yes5 30 67 Unilateral Vim DBS NoCurrent patient 6 9 82 Bilateral Vim DBS YesToby C. Yaltho, William G. Ondo Thalamic Deep Brain Stimulation for Orthostatic TremorTremor and Other Hyperkinetic Movementshttp://www.tremorjournal.orgThe Center for Digital Research and ScholarshipColumbia University Libraries/Information Services2

Thalamic Deep Brain Stimulation for Orthostatic Tremor

Background: Orthostatic tremor is an uncommon disorder manifest by high frequency, low amplitude leg tremor upon weight bearing. Treatment with oral tremor agents is inconsistent and usually not satisfactory. Methods: We implanted bilateral ventralis intermedius nuclei deep brain stimulators into an 82-year-old male with refractory orthostatic tremor. Results: The patient had a marked subjective and objective improvement in leg and arm tremor, mainly manifested by an improved ability to stand. Discussion: Bilateral thalamic deep brain stimulation may be considered in refractory cases of orthostatic tremor.</p

Toby C. Yaltho

William G. Ondo

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Chronic spinal cord stimulation in medically intractable orthostatic tremor.

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