Deep Brain Stimulation, Laryngeal Dystonia, Spasmodic Dysphonia
Conditions
Brief summary
Laryngeal Dystonia (LD), also commonly referred to as spasmodic dysphonia, is a neurological voice disorder characterized by involuntary dystonic contractions of the laryngeal muscles. Current treatments such as botox and voice therapy only provide temporary relief and thus, the investigators are exploring new strategies to provide long-term, sustained improvement. Deep Brain Stimulation (DBS) is a neurosurgical procedure that involves the implantation of electrodes to deliver electrical stimuli to specific brain regions. It is the standard surgical treatment for many other movement disorders such as Parkinson's disease, essential tremor, and primary dystonia. This trial has been designed to test the hypothesis that DBS can improve the vocal dysfunction of LD.
Interventions
DEVICEVIM Thalamic Deep Brain Stimulation ON
DEVICEVIM Thalamic Deep Brain Stimulation OFF
Sponsors
University of British Columbia
Study design
Allocation
RANDOMIZED
Intervention model
CROSSOVER
Primary purpose
TREATMENT
Masking
TRIPLE (Subject, Caregiver, Outcomes Assessor)
Eligibility
Sex/Gender
ALL
Age
18 Years to 75 Years
Healthy volunteers
No
Inclusion criteria
* Clinically diagnosed isolated laryngeal dystonia (adductor spasmodic dysphonia) * Able to give informed consent * Patients who fall into the age range of 18-75 years old * Patients with inadequate medical and BTX management of spasmodic dysphonia
Exclusion criteria
* Dystonia present in other body parts (i.e- eyes, neck, limbs) in addition to larynx * History of laryngeal denervation surgery for spasmodic dysphonia * History of intracranial pathology (such as multiple sclerosis, tumors, or aneurysms) that may account for dystonia or essential tremor. * History or evidence of ongoing psychiatric or neurodegenerative disorders (such as Parkinson's disease, Alzheimer's disease). * Incompetent adults or those unable to communicate.
Design outcomes
Primary
| Measure | Time frame | Description |
|---|---|---|
| Unified Spasmodic Dysphonia Rating Scale (USDRS) | After the first 6 months, the participants completed the USDRS twice (once per crossover). | Assessments of spasmodic dysphonia using a standardized spasmodic dysphonia rating scale were done pre-operatively, blinded DBS ON, blinded DBS OFF, and open DBS ON. Voice recording taken of each patient reading standardized sentences were blinded and evaluated by two speech language pathologists. The scale ranges from 0-7, with lower scores being better and higher scores being worse. |
| Voice-Related Quality of Life | After the first 6 months, the participants completed the V-RQoL twice (once per crossover). | Double-blinded assessments of voice-related quality of life with DBS ON/OFF were conducted using the V-RQoL for Spasmodic Dysphonia. The V-RQoL is a questionnaire that measures a patient's voice related quality of life. The scale ranges from 0-50, with higher scores representing a lower quality of life with respect to voice. |
Secondary
| Measure | Time frame | Description |
|---|---|---|
| Beck's Depression Inventory Scale | All participants completed the BDI-II once pre-operatively and once 1 year post-operatively (after the 6-month open unblinded phase). | Beck's Depression Inventory Scale (BDI-II) was used in this study to evaluate the mood of the patients both pre-operatively and at the end of the one-year study. All 6 patients completed the BDI-II, which is a scale that identifies symptoms and attitudes associated with depression, and evaluates the presence and severity of depression. The BDI-II ranges from 0-63. Scores ranging from 0-10 are considered to be absent or minimal depression, 10-18 indicate mild to moderate depression, 19-29 indicate moderate depression, and 30-63 indicate severe depression. |
| Montreal Cognitive Assessment Scale (MoCA) | All participants completed the MoCA once pre-operatively and once 1 year post-operatively (after the 6-month open unblinded phase). | The MoCA was used to analyze the cognitive ability of the patients. It was first evaluated pre-operatively and again evaluated at the end of the one-year study. The MoCA scale ranges from 0-30 with higher scores indicating less cognitive impairment. Scores ranging from 18-25 indicate mild cognitive impairment, 10-17 indicate moderate cognitive impairment, and scores less than 10 indicate severe cognitive impairment. |
| Voice-Handicap Index | All participants completed the VHI once pre-operatively and once 1 year post-operatively (after the 6-month open unblinded phase). | The Voice-Handicap Index (VHI) is a questionnaire that measures the patients' perceived degree of handicap related to their voice. The VHI ranges from a scale of 0-100, with higher scores reflecting higher perceived degree of voice handicap and lower scores reflecting lower perceived degree of voice handicap. |
Countries
Canada
Participant flow
Participants by arm
| Arm | Count |
|---|---|
| All Study Participants 1. Patients who are currently receiving BTX injections for their voice disorder
2. Who experience fluctuations in their symptom control as a result of their BTX therapy
3. Patients who fall into the age range of 18-75 years old | 6 |
| Total | 6 |
Baseline characteristics
| Characteristic | All Study Participants | — |
|---|---|---|
| Age, Continuous | 65.17 years STANDARD_DEVIATION 8.27 | — |
| Patients who are currently receiving BTX injections for their voice disorder | 6 Participants | — |
| Patients who experience fluctuations in their symptom control as a result of their BTX therapy | 6 Participants | — |
| Race and Ethnicity Not Collected | — | — Participants |
| Region of Enrollment Canada | 6 Participants | — |
| Sex: Female, Male Female | 5 Participants | — |
| Sex: Female, Male Male | 1 Participants | — |
Adverse events
| Event type | EG000 affected / at risk | EG001 affected / at risk | EG002 affected / at risk |
|---|---|---|---|
| deaths Total, all-cause mortality | 0 / 6 | 0 / 6 | 0 / 6 |
| other Total, other adverse events | 0 / 6 | 0 / 6 | 0 / 6 |
| serious Total, serious adverse events | 0 / 6 | 0 / 6 | 0 / 6 |
Outcome results
Primary
Unified Spasmodic Dysphonia Rating Scale (USDRS)
Assessments of spasmodic dysphonia using a standardized spasmodic dysphonia rating scale were done pre-operatively, blinded DBS ON, blinded DBS OFF, and open DBS ON. Voice recording taken of each patient reading standardized sentences were blinded and evaluated by two speech language pathologists. The scale ranges from 0-7, with lower scores being better and higher scores being worse.
Time frame: After the first 6 months, the participants completed the USDRS twice (once per crossover).
Population: One patient requested to have their DBS on after 2 weeks of being in the blinded-DBS OFF after the first crossover.
| Arm | Measure | Value (MEDIAN) | Dispersion |
|---|---|---|---|
| Blinded-DBS ON | Unified Spasmodic Dysphonia Rating Scale (USDRS) | 2.5 units on a scale | Standard Deviation 1.4 |
| Blinded-DBS OFF | Unified Spasmodic Dysphonia Rating Scale (USDRS) | 3.75 units on a scale | Standard Deviation 1 |
p-value: 0.02595% CI: [0.75, 1.75]Wilcoxon (Mann-Whitney)
Primary
Voice-Related Quality of Life
Double-blinded assessments of voice-related quality of life with DBS ON/OFF were conducted using the V-RQoL for Spasmodic Dysphonia. The V-RQoL is a questionnaire that measures a patient's voice related quality of life. The scale ranges from 0-50, with higher scores representing a lower quality of life with respect to voice.
Time frame: After the first 6 months, the participants completed the V-RQoL twice (once per crossover).
| Arm | Measure | Value (MEDIAN) | Dispersion |
|---|---|---|---|
| Blinded-DBS ON | Voice-Related Quality of Life | 74.7 units on a scale | Standard Deviation 18.3 |
| Blinded-DBS OFF | Voice-Related Quality of Life | 19 units on a scale | Standard Deviation 18.2 |
Secondary
Beck's Depression Inventory Scale
Beck's Depression Inventory Scale (BDI-II) was used in this study to evaluate the mood of the patients both pre-operatively and at the end of the one-year study. All 6 patients completed the BDI-II, which is a scale that identifies symptoms and attitudes associated with depression, and evaluates the presence and severity of depression. The BDI-II ranges from 0-63. Scores ranging from 0-10 are considered to be absent or minimal depression, 10-18 indicate mild to moderate depression, 19-29 indicate moderate depression, and 30-63 indicate severe depression.
Time frame: All participants completed the BDI-II once pre-operatively and once 1 year post-operatively (after the 6-month open unblinded phase).
| Arm | Measure | Group | Value (MEAN) | Dispersion |
|---|---|---|---|---|
| Blinded-DBS ON | Beck's Depression Inventory Scale | Pre-Operative BDI-II | 13.33 units on a scale | Standard Deviation 7.2 |
| Blinded-DBS ON | Beck's Depression Inventory Scale | One Year Follow-Up BDI-II | 6.50 units on a scale | Standard Deviation 6.47 |
Secondary
Montreal Cognitive Assessment Scale (MoCA)
The MoCA was used to analyze the cognitive ability of the patients. It was first evaluated pre-operatively and again evaluated at the end of the one-year study. The MoCA scale ranges from 0-30 with higher scores indicating less cognitive impairment. Scores ranging from 18-25 indicate mild cognitive impairment, 10-17 indicate moderate cognitive impairment, and scores less than 10 indicate severe cognitive impairment.
Time frame: All participants completed the MoCA once pre-operatively and once 1 year post-operatively (after the 6-month open unblinded phase).
| Arm | Measure | Group | Value (MEAN) | Dispersion |
|---|---|---|---|---|
| Blinded-DBS ON | Montreal Cognitive Assessment Scale (MoCA) | Pre-Operative MoCA | 24.17 units on a scale | Standard Deviation 3.08 |
| Blinded-DBS ON | Montreal Cognitive Assessment Scale (MoCA) | One Year Follow-Up MoCA | 26.67 units on a scale | Standard Deviation 3.09 |
Secondary
Voice-Handicap Index
The Voice-Handicap Index (VHI) is a questionnaire that measures the patients' perceived degree of handicap related to their voice. The VHI ranges from a scale of 0-100, with higher scores reflecting higher perceived degree of voice handicap and lower scores reflecting lower perceived degree of voice handicap.
Time frame: All participants completed the VHI once pre-operatively and once 1 year post-operatively (after the 6-month open unblinded phase).
| Arm | Measure | Group | Value (MEAN) | Dispersion |
|---|---|---|---|---|
| Blinded-DBS ON | Voice-Handicap Index | Pre-Operative VHI | 84.83 units on a scale | Standard Deviation 15.52 |
| Blinded-DBS ON | Voice-Handicap Index | 1 Year Post-Operative VHI | 38.17 units on a scale | Standard Deviation 13.15 |