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| CASE REPORT |
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| Year : 2017 | Volume
: 7
| Issue : 1 | Page : 14-17 |
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Botulinum toxin application in the facial muscles for the treatment of stuttering
Abraham Abramovitz, Carlos Goyeneche, Andrés Cánchica, Tamsin Burgues, Eduardo Sucupira
Clinica Essendi, Rio de Janeiro, Brazil
| Date of Web Publication | 14-May-2018 |
Correspondence Address:
Dr. Eduardo Sucupira
Clinica Essendi, Centro Empresarial Mario Henrique Simonsen, Av. Das Americas 3434, BL 05, Conj 419, Barra Da Tijuca, Rio De Janeiro
Brazil
 Source of Support: None, Conflict of Interest: None  | Check |
DOI: 10.4103/jlv.JLV_6_17
 Abstract | | |
Stuttering is a psychosomatic condition with a high degree of social dysfunction characterized by involuntary interruptions in the flow of speech. The etiology is considered to be multifactorial. Speech therapy is the first-line treatment, and therapeutic alternatives have been proposed with different pharmacological agents. In this case report, we describe the use of botulinum toxin type A (BTX-A) in the facial muscles for treating a patient with developmental stuttering which showed a significant temporary improvement after undergoing the procedure. This is a single-subject study. In the current medical literature, there are a few descriptions of using BTX-A injections in the intrinsic laryngeal muscles with favorable results, but no reports of its use in facial muscles for the treatment of stuttering. Therefore, this report may be important to highlight BTX-A as a safe and effective option when managing to stutter. Hopefully, it will assist in the search for effective treatment and form part of the multi-disciplinary approach in the treatment of stuttering, helping these patients to reduce their social dysfunction and improve their speech and their lives.
Keywords: Botulinum toxin type A, stammering, stuttering
How to cite this article:
Abramovitz A, Goyeneche C, Cánchica A, Burgues T, Sucupira E. Botulinum toxin application in the facial muscles for the treatment of stuttering. J Laryngol Voice 2017;7:14-7 |
| Introduction | |  |
Stuttering is a speech disorder characterized by involuntary interruptions of speech which impede the ability to communicate effectively. It can be characterized by the frequent occurrence of one or more of the following signs: repetition of sounds, syllables or whole words, prolongation of sounds, the interjection of breaks in the production of words or interruption between words.[1]
We describe the case of an 18-year-old male patient and our treatment of his stuttering using botulinum toxin type A (BTX-A).
| Stuttering: Types and Treatments | | |
Two types of stuttering are described in the literature: acquired neurological and developmental stuttering. Acquired neurological stuttering is less common and is associated with diseases or organic neurological conditions. Developmental stuttering, with or without an associated psychiatric disorder, is the more common type, and its onset is during childhood. Its resolution can be spontaneous or with treatments like speech therapy. In some cases, the use of alternative medical therapies such as antipsychotics, benzodiazepines, selective serotonin reuptake inhibitors, and tricyclic antidepressants may assist, but with controversial results.[2]
Despite several theories proposed, the causes of stuttering are still poorly understood. Multifactorial etiologies with high emphasis on genetics and without definitive conclusions are presented as simple models.[3] However, studies using electromyographic measurements (EMG) show the muscles involved in phonation, and both intralaryngeal and extralaryngeal muscles reveal hyperactivity and increased tension in patients who stutter.[4]
For intralaryngeal musculature, it is further proposed that there is an alternative dissociation between abductor and adductor muscle fibers, which causes a blockage in the air flow and consequent interruptions, breaks, temporary inhibition, and inability to start phonation.[5] This could increase brain feedback and exacerbate hyperstimulation of the responsible motor neurons.[6]
For extralaryngeal musculature, studies have been conducted measuring EMG in the masseter muscle and orbicularis oris [7] which reveal that patients who stutter show significantly higher EMG levels when starting speech and during the speech, when compared to patients without stutter.
| Botulinum Toxin Treatment | | |
BTX-A for injection is a neurotoxin that has been used as a therapeutic agent since the 1970s and was approved by the FDA for cosmetic use in the 1980s.
Injected BTX-A enters the nerve terminal by endocytosis and interacts with intracellular proteins (SNARE proteins) inhibiting vesicular release of acetylcholine-mediated Ca 2+. Inhibition of this causes temporary chemical denervation of the neuromuscular junction blocking acetylcholine release with consequent mediated striated muscle paralysis. This effect is dose-dependent and in some cases may extend to neighboring muscle groups. Its initial effect appears after 3–7 days, reaching its maximum effect at approximately 2 weeks, and full recovery of muscle function after 3–6 months. This is probably due to axonal regeneration in the fiber terminal.
Within the growing field of noncosmetic use of BTX-A, positive results in different areas of medicine have been described, including in neurology, dermatology, urology, gynecology, and otolaryngology. For example, its use has been reported in the treatment of cervical dystonia, hemifacial spasm, multifocal spasticity, chronic musculoskeletal pain, spasmodic dysphonia, and bruxism.
All such reports put forward treatment protocols for the different pathologies; some with specific FDA approvals for each type of disorder and others without it, but with favorable results in empirical clinical practice and within controlled studies.
In the current medical literature, however, there are a few descriptions of BTX-A injections into the intrinsic laryngeal muscles, but they show favorable results and clear improvement in patient groups who stutter.
One of the most cited studies using BTX-A as a treatment for stuttering was published by MF Brin et al., in which 1.25 U was injected into each vocal cord in a sample of 14 patients with chronic developmental stuttering persisting into adulthood. The study concludes that “local injections of BTX-A can effectively reduce the pharyngeal blockage and improve fluency and general function in patients with disabling stutter.”[8]
Another favorable result was published by C. Ludlow, in a case report on a sample of 7 patients who received BTX-A injections to the left thyroarytenoid muscle until a reduction of the function was achieved which required between 10 and 78 U. It was concluded that there was a significant reduction in the disruption of speech following the application.[6]
| Case Study Subject and Method | | |
Our subject was an 18-year-old male patient, a native of Lima, Peru with the diagnosis of developmental stuttering. He had no prior medical management from any specialty, and he had no other significant or relevant medical or family history. He reported having a stutter since he was 9-year-old which significantly hindered his social interaction. When he spoke, there was an obvious contraction in the facial muscles, predominantly in the lower third of the face, only before his speech intention.
The treatment given was several applications of BTX-A, as described below and summarized in [Table 1]. | Table 1: Total number of units of Botulinum toxin type A used in each application in the muscle treated
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| Treatments and Results | | |
Based on the etiological theory that stuttering involves overactivity in muscles involved in facial expression that relate to phonation, the application protocol was designed to use BTX-A to partially paralyze those muscles. We sought to start conservatively and to adjust the dose progressively according to the results:
Application 1
- The first application was made in the upper portion of the orbicularis oris muscle, with a total of 6 U BTX-A distributed equally at six equidistant points. Then an application was made into the masseter muscle with a total of 20 U each side at 2 points. Finally, 12 U was applied to each side of the platysmal bands at 4 points
- One week after the application, the patient showed an obvious improvement in his symptoms, noting that speech disruptions and blocks were significantly reduced. The quality of his speech became less disabling, and his fluency improved
- A gradual mild recurrence of symptoms was noted within 4 months, and 6 months after the application of BTX-A, a subjective assessment was performed which showed that the severity of the stuttering was similar to that of the patient before starting the treatment.
Application 2
The second application used increased doses and more injection points as follows:
- 8 U BTX-A in the orbicularis oris muscle at 8 points, both in its upper portion and a lower portion, 80 U in the masseter muscle on each side at 2 points, and 20 U in platysmal bands on each side at 4 points
- Similarly to what was seen with the first application, the patient presented a significant improvement in his symptoms, greater even than after the first application, according to the assessments of both the patient and the authors.
Application 3
- The third application was made 4 months after the second treatment and consisted of an injection of 60 U BTX-A in each masseter muscle at 2 points and 10 U in platysmal bands on each side at 2 points (no injection into the orbicularis oris muscle)
- Two weeks after this application, a booster dose was given of 10 U in the upper portion of the orbicularis oris at 2 points and 30 U in each masseter muscle at a single point
- There were also favorable results with significant improvements in each of the applications with a recurrence of symptoms between 4 and 7 months.
Application 4
- After 7 months, an adjusted version of the third application was made: 90 U BTX-A into each masseter muscle at 2 points and 30 U in platysmal bands on each side at 3 points (No injection into the orbicularis oris muscle)
- The subject's speech was tested but asking him to read the same text on each consultation (allowing 2 weeks for the BTX-A to fully take effect). The researchers then assessed him on this. The participant was asked if his stuttering had improved subjectively and he stated he was delighted with the results.
A description of the total number of units of BTX-A used in each application is presented in [Table 1].
There were no complications or adverse effects during these applications of BTX-A.
| Discussion | | |
The emerging use of BTX-A in various nonesthetic fields of medicine, as listed above, has increased the number of studies reporting favorable results, and this case study supports its use in the treatment of stuttering.
Despite being a treatment of limited durability, BTX-A has several advantages: It is a technique that is easily learnt, and it is a widely available product whose safety and low rate of adverse effects and complications make it an alternative to consider for various pathologies.
The mechanism of action of BTX-A on striated muscle fibers appears to allow the etiology to support an overactive extralaryngeal [7] muscle and therefore to achieve a noticeable improvement. Moreover, injecting into the extralaryngeal facial musculature (shown in EMG studies to be hyperactive and with more tension in stuttering, specifically in the orbicularis oris muscle, masseter, and platysmal bands) is far less invasive when compared to injecting into the larynx, and is a less stressful experience for the patient.
The use of BTX-A in developmental stuttering can offer patients benefits to their emotional health and a positive impact on their quality of life by observing a significant improvement in their symptoms compared to other therapeutic measures, without significant adverse effects. One limitation is that the effect is time-limited, which could lead to some patients not adhering to their treatment.
We may contrast BTX-A treatment and conventional speech therapy by noting that the latter can generate a level of frustration in patients owing to its limited effect on stuttering. We consider BTX-A a good therapeutic alternative, with more immediate results and great benefits for the patient.
Daniel Kahneman distinguishes two modes of thinking: one operating automatically and quickly with little or no effort, and another demanding attention and focuses on complex situations.[9]
Generally speech operates automatically, but we believe that stuttering patients are forced into his second mode because they have so many recommendations from their therapies, prejudices, and fears that their speech becomes too conscious: it makes them reason and be cautious. BTX-A may help patients to drive their speech toward being more automatic, and to make it easy and natural again.
A recent article found that there are a range of abnormalities in both structural architecture and functional organization of the brains of individuals who stutter. However, this article does no pinpoint the facial musculature which is an interesting concept and one that improved the individual in this case.[10]
This is a single-subject study and obviously, there are limitations in terms of being able to generalize to the general population due to this. There are also limitations in terms of how accurately to measure the stutter. However, from the subjective participant's point of view and from the point of view of the researchers the stutter had decreased dramatically. More research is definitely needed to see if this could perhaps be a verified treatment for stutter/stammer in the future.
| Conclusion | | |
The use of BTX-A for the treatment of developmental stuttering should be further investigated for it to be considered a therapeutic alternative with a high level of effectiveness. Its use, in this case, has revealed a significant if temporary, improvement of symptoms in one patient with stuttering.
These findings encourage expanding the use of noncosmetic BTX-A to offer physical and emotional benefits to various groups of patients.
Declaration of patient consent
The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.
Financial support and sponsorship
Nil.
Conflicts of interest
There are no conflicts of interest.
| References | | |
| 1. | Gordon N. Stuttering: Incidence and causes. Dev Med Child Neurol 2002;44:278-81.  [ PUBMED] |
| 2. | Vila-Nova C, Queirós F, Fortaleza T, Lucena R. Tratamento farmacológico da gagueira: Evidências e controvérsias. J Bras Psiquiatr 2006;55:244-8. |
| 3. | Packman A. Theory and therapy in stuttering: A complex relationship. J Fluency Disord 2012;37:225-33. [ PUBMED] |
| 4. | Starkweather CW. The epigenesis of stuttering. J Fluency Disord 2002;27:269-87. [ PUBMED] |
| 5. | Freeman FJ, Ushijima T. Laryngeal muscle activity during stuttering. J Speech Hear Res 1978;21:538-62. [ PUBMED] |
| 6. | Ludlow CL. Treatment of speech and voice disorders with botulinum toxin. JAMA 1990;264:2671-5. [ PUBMED] |
| 7. | Kalotkin M, Manschreck T, O'Brien D. Electromyographic tension levels in stutterers and normal speakers. Percept Mot Skills 1979;49:109-10. |
| 8. | Brin MF, Stewart C, Blitzer A, Diamond B. Laryngeal botulinum toxin injections for disabling stuttering in adults. Neurology 1994;44:2262-6. [ PUBMED] |
| 9. | Kahneman D. Thinking, Fast and Slow. New York: Farrar, Straus and Giroux; 2011. |
| 10. | Etchell AC, Civier O, Ballard KJ, Sowman PF. A systematic literature review of neuroimaging research on developmental stuttering between 1995 and 2016. J Fluency Disord 2018;55:6-45. [ PUBMED] |
[Table 1]
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