Abstract
Background:To date, there are only six published reports of adductor spasmodic dysphonia (SD) responding to awake thalamic deep brain stimulation (DBS).Methods:We retrospectively reviewed cases of Essential Tremor (ET) with SD that were seen in our center from 2012 to 2020. We further identified those that have undergone thalamic DBS, and had a blinded laryngologist rate first the audio voice recordings before and after DBS using the Unified Spasmodic Dysphonia Rating Scale (USDRS), and the video recordings last to rate the related movements and facial grimacing.Results:We identified three cases of adductor SD with ET that had undergone bilateral ventralis intermedius (VIM) DBS under general anesthesia. All patients noted improvement of their limb and voice tremor, as well as their SD post-DBS. Although improvement of tremor was observed even with initial programming in all three, improvement of SD was noted only upon reaching higher amplitudes or wider pulse widths. Blinded voice assessments showed improvement of USDRS scores post-DBS compared to pre-DBS, and with stimulator on compared to stimulator off.Discussion:We report the first three cases of SD responding favorably to bilateral VIM asleep DBS and summarize the nine cases so far of SD who have undergone thalamic DBS.
Highlights
Spasmodic dysphonia (SD) is a primary focal dystonia characterized by loss of control of the vocal muscles during phonation, accompanied by laryngeal muscle spasms [1].Its pathophysiology is thought to be caused by abnormalities of the basal ganglia [2]
In 2009, we reported the first case of adductor spasmodic dysphonia with Essential Tremor (ET) responding to awake deep brain stimulation (DBS) of the ventralis intermedius (VIM) nucleus of the thalamus [8, 9]
B.H. is a 74-year-old right-handed woman presenting with hand tremors since age 68 that were diagnosed to be consistent with ET
Summary
Spasmodic dysphonia (SD) is a primary focal dystonia characterized by loss of control of the vocal muscles during phonation, accompanied by laryngeal muscle spasms [1].Its pathophysiology is thought to be caused by abnormalities of the basal ganglia [2]. The most effective treatment for SD involves onabotulinumtoxinA (BTX) injections into the larynx [4, 5]. BTX therapy requires regular injections every three or so months to ensure continuity of bene- †. Barrow Neurological Institute, Phoenix, Arizona, US ‡. Patients often experience bothersome side effects, including pain from injections, breathiness, dysphagia, and hypophonia [6, 7]. In 2009, we reported the first case of adductor spasmodic dysphonia with Essential Tremor (ET) responding to awake deep brain stimulation (DBS) of the ventralis intermedius (VIM) nucleus of the thalamus [8, 9]. There are only six published reports of adductor spasmodic dysphonia (SD) responding to awake thalamic deep brain stimulation (DBS)
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