MEDICAL TREATMENT AND SPEECH THERAPY FOR SPASMODIC DYSPHONIA: A LITERATURE REVIEW

(1) Speech Pathologist; PhD Professor at the Department of Speech Pathology, UNESP, Marilia, São Paulo, Brazil; PhD in Education from UNESP. (2) Speech Pathologist; PhD Professor at the Department of Speech Pathology, UNESP, Marilia, São Paulo, Brazil; PhD in Communication Sciences and Disorders from the University of Florida (3) Speech Pathologist; B.A. in Speech Pathology from UNESP Marília, São Paulo, Brazil. (4) Speech Pathologist; PhD Professor at the Department of Speech Pathology, UNESP, Marilia, São Paulo, Brazil; PhD in Education from UNESP. (5) Medical Doctor; Assistant Professor at the Department of Speech Pathology, UNESP, Marilia, São Paulo, Brazil; Master’s Degree.


INTRODUCTION
First described by Traube in 1871, spasmodic dysphonia (SD) was classified as a spastic form of nervous hoarseness.This disorder was discussed in literature as two types of spastic dysphonia: adductor spasmodic dysphonia (AdSD) and abductor spasmodic dysphonia (AbSD) 1 .Adductor spasmodic dysphonia is characterized by the strained-strangled voice, with interruption in word production or difficulty in starting 2 .Abductor spasmodic dysphonia was described as maintaining normal vocal quality followed by moments of breathy or whispery voice 1 .The term spastic dysphonia was ABSTRACT Spasmodic dysphonia (SD) is a voice disorder characterized by a strained-strangled voice, with sound breaks and has implications in one's communication.The purpose of this study is to present a bibliographic review of the speech therapy and medical treatment suggested for SD from 2006 to 2010.The speech therapy and medical treatments described are: botulinum toxin injection, myectomy, neurectomy, denervation and reinnervation selective laryngeal adductor, thyroplasty, radiofrequency thyroarytenoid myothermy, injection of lidocaine, homeopathy and speech therapy.The use of botulinum toxin injection showed results that indicated the satisfaction of the patients who were treated, although some of the articles presented the frequent need of reapplication of the toxin as a disadvantage.The surgical procedures were considered long-lasting and indicated to patients who didn't want to get botulinum toxin injections.The studies, however, presented a restricted contingency of patients, and the outcomes in many studies were based in the patient's own judgment on his/her voice quality.The treatments using lidocaine and homeopathy had positive results in relation to the voice quality of the patients and were suggested as an option for those who wouldn't like to undergo surgical treatment or have botulinum toxin injection.The few studies which discourse on voice therapy presented good results in association with botulinum toxin injection, showing the shortage of information in this field.
A study on the literature review pointed out the need of developing researches to help us understand the neurological functioning in spasmodic dysphonia.Future study involving speech therapy in the treatment of ED is still necessary.KEYWORDS: Dysphonia; Spastic Dysphonia; Dystonia recently published articles is essential in determining the best approach in the treatment and prognosis of this devastating communication disorder.Thus, this study aims to review literature on medical and speech pathology treatment for SD from the period between 2006 and 2010.

METHOD
The study was conducted by searching national and international journal articles available in the databases Lilacs, Medline, and Scielo, which after careful analysis were incorporated into the study.
The criteria used for inclusion were articles that presented data for SD treatment (medical and/ or speech therapy) reported within the last five years, i.e. between 2006 and 2010.Articles on SD that included investigation with animals were disregarded.
In the search for articles, terms related to the theme "spasmodic dysphonia" were chosen and found in the Health Sciences Descriptors (HSD).These terms were used in isolation and cross reference as shown in Figure 1.The filters "year of publication" and "word" were used in each search.And with the database Medline it was possible to use the filter "subject description", which allowed the search for the terms "voice", "focal dystonia" and "spastic dysphonia".subsequently discussed in literature and considered inadequate because it is not an alteration related to injury in the pyramidal or extrapyramidal tract 3 .Currently SD is classified as a focal laryngeal dystonia with a neurological etiology 2 .
The standard treatment for SD, according to that presented in literature, is the injection of type A botulinum toxin (BT-A) 4,5 which can be combined with speech therapy 3 .Speech therapy as the only proposed treatment is considered ineffective mainly due to the fact that emotional states influence the control of speech production in patients diagnosed with SD 7 .However, studies show that following BT injection, speech therapy may help in prolonging its effectiveness, providing longer intervals between applications [8][9][10] .
Several other forms of treatment (medical and/ or surgical) for SD were presented in the literature, however, none showed lasting effectiveness.Despite the various proposals presented for surgical treatment, literature indicates that some patients with SD go to speech therapy in an attempt to minimize communication disorders when other treatments are not effective 7,11 .
The search for treatment options for SD is caused by the need to find a better quality of life for the patients.Given the difficulty in obtaining conclusive information from literature about treatments available for SD, an updated search in

Myectomy
The treatment with myectomy has been described since the 1990s.The surgical procedure is performed with an incision on the lateral surface of the vocal fold which exposes the TA muscle, followed by resection.This surgical treatment is irreversible and, in consequence, has the advantage of maintaining the long-term positive effect.Studies have shown that the TA muscle does not regenerate after resection 12 .
Myectomy of the TA and LCA muscle may be performed with local anesthesia and intravenous sedation so that the voice and function of the vocal folds may be evaluated during the procedure 13 .

Neurectomy of the thyroarytenoid branch of the inferior laryngeal nerve associated with the partial myectomy of the TA muscle
Upon completion of the partial myectomy of the TA muscle with CO 2 laser, there is the sectioning by electrocoagulation of the thyroarytenoid branch of the recurrent laryngeal nerve (RLN), located between the internal perichondrium of the thyroid cartilage and the fascia of the TA and LCA muscles.The procedure described by the authors is performed in both vocal folds 14 .

Selective laryngeal adductor denervation and reinnervation
Selective adductor denervation interrupts the abnormal neural signals to the TA and LCA muscles.The adductor branch of the RLN is divided at its insertion in the TA and LCA muscles and the proximal stump is exteriorized from the larynx to prevent the regeneration of axons in this anatomical structure.The ansa cervicalis of the cervical nerve is then anastomosed to the distal stump of the TA to maintain muscle tone and volume and also prevent the regeneration of axons in the RLN endplates of the TA and LCA muscles 15 .

Thyroplasty
Type II thryoplasty, as described in the consulted literature, is carried out with local anesthesia followed by a midline incision of the thyroid cartilage holding the underlying tissue intact.During speech, the edges of the cartilage are separated from 2 to 6 mm, average 4 mm, to verify any voice change 16,17 .A variety of material has been described for securing the edges of cartilage, such as pieces of silicon or cartilage, and titanium plates and bridges above and below the anterior commissure [16][17][18] .

LITERATURE REVIEW
A total of 3833 articles were found in the survey of the databases Scielo, Lilacs, and Medline.Each article was presented in one or more databases.
From the analysis of the abstracts of these articles, we excluded those that had previously been selected in another database, those that did not meet the inclusion criteria, and also those from journals that did not provide the full article.Thus, we included 30 articles related to the study.These articles reported on treatment for SD by means of medical and speech pathological procedures with: (a) 11 (37%) articles on BT injection; (b) 10 (33%) on surgical procedures, such as myectomy (2, 7%), neurectomy (2, 7%), selective laryngeal adductor denervation and reinnervation (1, 3%), thyroplasty (4, 13%), and thyroarytenoid myothermy (1, 3%); (c) 2 (7%) on other medical treatments, such as homeopathy and lidocaine injection; and (d) 1 (3%) speech therapy.In addition to these, there were 6 (20%) literature review articles on the treatment of SD.
The information on the proposed treatments for SD, as shown in the literature, is summarized below.
Botulinum toxin is a protein produced by Clostridium botulinum bacteria with a potent neurotoxic action that blocks the release of acetylcholine from nerve endings at the neuromuscular joint.BT has been a treatment option for SD since the 1980s.The injection of BT into the intrinsic muscles of the larynx results in a temporary paresis or paralysis of the injected muscle.The toxin may be injected into one or both vocal folds simultaneously.
Several injection techniques may be used for BT, some of which are performed with the support of percutaneous electromyography or nasalaryngoscopy.The injection of BT is usually carried out in the thyroarytenoid muscle (TA) 4 , however there are also reports of injection in other laryngeal muscles, such as the lateral cricoarytenoid muscle (LCA).
One of the drawbacks of BT is the fact that the effect is temporary and it is necessary to reapply every three to six months.There is also the possibility of the body developing antibodies against BT which reduces its effectiveness.The following complications have been reported regarding the use of BT: transient dysphonia, glottic incompetence with an extremely hoarse voice, dysphagia, and asthenia.

Speech Therapy
Although the articles comment that speech therapy is a possible treatment for SD, there are no reports in these articles on the strategies used in speech therapy sessions.
After a brief exposition of the information on the treatment proposed for SD, the following are the results of the literature reviewed, classified by type of treatment, presented in tables that include: the year of publication, the author(s) involved, the objective(s) of the study, and the results.
Table 1 shows the articles found in the bibliographic survey with the proposed treatment of BT injection.
Table 2 presents the articles that include surgical procedures for the treatment of SD.
Table 3 presents the articles categorized as other medical treatments for SD.
Table 4 presents articles that contemplate speech therapy in the treatment for SD.
Table 5 presents the articles that contemplate studies of literary review in the treatment of SD.
The literature review on the proposed topic showed that in the last five years the medical treatments and speech therapy described in the literature were: BT injection, myectomy, neurectomy, selective laryngeal adductor denervation and reinervation, thyroplasty, radiofrequency thyroarytenoid myothermy, lidocaine injection, homeopathy and speech therapy.

Radiofrequency thyroarytenoid myothermy
This procedure involves the insertion of a bipolar radiofrequency probe at two points of the vocal folds, one in the membranous portion and the other in the anterior-lateral portion of the vocal process, 2 cm from the surface of the vocal fold mucosa.The authors have described an ablation from a depth of 20 to 11 mm to protect the vocal fold mucosa and that it was carried out five times during the procedure 19 .

Other medical treatments
In addition to the surgeries described, treatments with injection of lidocaine, homeopathy, and speech therapy were published.

Injection of lidocaine
The authors described the injection of lidocaine 1% into the RLN in doses that varied from 2.5 to 5 ml.For this a syringe and 27 gauge needle was used to penetrate the neck to the right along the tracheoesophageal groove, below the cricothyroid articulation in the region of the RLN entry point into the larynx 20 .After injection, laryngoscopy was performed on all subjects to confirm vocal fold paralysis on the right.

Homeopathy
The literature reported on the use of Argentum nitricum 21 in the treatment of SD.The dosage of BT in treating laryngeal dystonia may be reduced during treatment without harming the interval between applications and duration of vocal quality effectiveness.

2009
Upile et al 29 Compare the effects of uni-and bilateral BT injection in the TA muscle for the treatment of AdSD.The study was conducted with 31 patients (16 women and 15 men) who had received 5 or more consecutive uni-or bilateral applications of Dysport.Self-evaluation protocols were used for the analysis.
There was no significant difference in the results with low doses of uni-or bilateral BT injection when considering the duration of the toxin action, the self-evaluation vocal score, and the complication rate.However, only in the unilateral treatment was there no report of loss of voice after BT application.The authors recommend the unilateral use of BT for treatment.

Injection of BT (n = 11) Year Author(s) Objetive(s) Results
Botulinum Objectively evaluate the type A Botulinum Toxin dosage by means of statistical relationship between the amount of injection, duration of side effects, and the normal voice in AdSD.There were 101 patients (70 women and 31 men) that participated in the study at a private clinic.The doses varied from 0.5 to 15 units of BT.
There was a significant and foreseeable correlation between the duration of side effects and the duration of normal voice quality after BT injection.The smaller the side effects, the lower the durability of normal voice quality.The most severe tendency of side effects was found in higher dosages of BT injections.There was a correlation in the relationship between the perception of vocal impairment reported by the patient and the voice quality and quality of life in the mild to moderate and moderate to severe dysphonia group, and also a weak correlation between patient assessment and the clinician's perceptual judgement with the use of the CAPE-V only in the moderate to severe dysphonia group.Another issue that was studied was the correlation between the patient's quality of life data and the clinical judgement on vocal quality.There was a weak correlation in the moderate to severe dysphonia group.
nalyze the effectiveness of type II Thyroplasty using a titanium plate in AdSD.The review was conducted with a questionnaire in which patients responded on the ease of phonation and vocal quality before and after surgery.Forty-one patients participated in the study.
The answers to the questionnaire showed that 70% of the patients reported having achieved excellent results.Report analysis findings of aerodynamic and acoustic evaluations before and after type II thyroplasty in patients with AdSD.The study was conducted with ten women from 20 to 76 years old who underwent surgery and performed examinations before and six months after surgery.
After surgery, patients did not present voice strangulation and expressed satisfaction.
There was no significant difference among the aerodynamic measurements after surgery, however the acoustic measurements (jitter, shimmer, HNR, SDFO and DVB) improved significantly.The study demonstrated that type II thyroplasty is indicated for patients who were treated with BT and obtained either poor results or none.Surgery is also indicated for patients who desire permanent results.2010 Isshiki; Sanuki 18 After analyzing the dissatisfaction of patients undergoing type II thyroplasty, the researchers presented the possible causes of failure and, in this article, described the changes made in the surgical procedure for treating AdSD.The authors also reflect on the poor acceptance of surgery in AdSD patients.
The authors stated that the review of the unsuccessful cases in the surgical procedure was important for the proposed changes.Study the treatment of AdSD by means of a modification in the Remacle surgical procedure.
The study was conducted with twenty women with spasmodic dysphonia who had already received BT injection with success in the treatment.
The result of treatment with radiofrequency thyroarytenoid myothermy was considered an alternative treatment, however the results were effective for two months after surgery.After six months a reduction in the results was noted in 50% of the patients.
Among the articles found, 37% reported medical treatment with BT injection and results indicated an improvement in the vocal quality analyzed by means of vocal and quality of life self-evaluation protocols.However, the observed results demonstrated the temporary effectiveness of the treatment and the need to reapply the toxin [22][23][24][25][26][27][28][29][30][31][32] The articles that reported on surgical procedures (33%) were based on the premise that surgery would be a long-lasting treatment option for SD, without the need to return to control pathology symptoms.
Among the surgical procedures, myectomy alone or combined with neurectomy was presented as an option in cases in which the patient desires a long-lasting outcome.The results presented were positive in most cases and the scholars stressed the irreversibility of the surgery [12][13][14]33 .
Regarding selective laryngeal adductor denervation and reinnervation surgery, two articles from the same group of researchers were found.While one of these articles addressed a literature review 36 , the other reported on the satisfactory outcome of the surgical procedure from the vocal analysis done by the voice handicap index protocol 15 .
In relation to thyroplasty, the results of this treatment were analyzed in more than one article showing the concern of a surgical team in improving the surgical procedures initially proposed.In general, the researchers discussed the indication and contraindication of this surgery, presented modifications to the surgical technique, and pointed to the patient's own satisfaction in his/her voice quality [16][17][18] .
The use of the Remacle modified radiofrequency thyroarytenoid myothermy was reported on in one study.The result was described as a good alternative to treat SD, though not considered effective in all cases since 50% of the patients had BT injection one year following myothermy 19 .
There was only one single study that used lidocaine injection (lidocaine block of the RLN) Investigate the effects on speech by using lidocaine to block the RLN in treating AdSD.
The study was conducted with twenty-one patients with spasmodic dysphonia.
The results showed that during the blockage of RLN, the patients reported a reduction in the severity of the symptoms and vocal effort.
In the perceptual-auditory evaluation, judges verified that the voices were breathier and less tense.Homeopathy (n=1)

2009
Xue; Schepper; Hao 21 Describe the perceptual and physiological changes in the vocal function of patients treated with classical homeopathy.The study was conducted with a 57 year-old male patiente diagnosed with AdSD.The medicine was offered after analysis of the initial medical interiew (Argentum Nitricum -30C from Borion).Perceptual analysis was performed with the CAPE-V protocol and analysis of some acoustic parameters.
The findings show that after three months of treatment there was a significant reduction in the severity of the strained-strangled voice quality.The number of breaks in the voice was reduced and the patients demonstrated increased control of the speech mechanism, as well as reporting an emotional well-being.This may be a possible treatment for those patients who desire to not use botox.

Year
Examine the Health Locus of Control (HLoC), a protocol for evaluating the patient's locus of control over their health in three groups of patients: 1 -spasmodic dysphonia, 2functional dysphonia, and 3 -a group without dysphonia and without laryngeal dystonia (control group).
The study showed that in spasmodic dysphonia, the relationship between the frequency of injection and the health locus of control may be an interesting focus study.The study demonstrated the effect of vocal therapy on health control or the validity of health control as an indicator for prognosis of treatment.
The authors argued that when the patient has a high value of internal control (Internal LoC), the voice therapy may have a positive outcome because the patient will be able to increase the sense of voice control.The LoC protocol was chosen as a good prognostic indicator for treatment in Speech therapy.However, they suggest that the use of BT before therapy may help the results.
may be a possible treatment for those patients who do not wish to use botox 21 .
Regarding speech pathology treatment, no articles that discussed the effects of vocal technique action in the treatment of SD during the period studied were found.Only one article commented on achieving positive results with speech therapy associated with BT injection in patients with good results in the Health Locus of Control "internal control" index (evaluation protocol of the patient's locus of control in his/her health) 35 .Another literature review article pointed to two studies that discussed speech therapy as a treatment for SD.One of these studies reported that speech therapy improved speech intelligibility, voice functionality, confidence of the patient, when the therapy was combined with BT injection.In the other study, it was concluded in the AdSD in order to investigate the effects on phonation.The results showed that blockage is possible, however the study did not present length of effect.The authors pointed out that this procedure may be used as a differential diagnostic resource for SD 20 .
Homeopathy treatment was found in only one article which aimed to describe the perceptual and physiological changes in the vocal function of patients treated with classic homeopathy.The results found in this study showed that after three months of treatment, a significant reduction in the strained-strangled severity and quality of the voice was observed.The number of voice breaks was reduced and the patients demonstrated greater control of the speech mechanism, as well as a reported emotional well-being.For the authors, this

2006
Chhetri; Berke 36 Present, with literature support, the modifications in selective adductor laryngeal denervation and reinnervation since its proposal.Furthermore, the article presents step-by-step surgical procedure and research results of the impact on vocal quality and patient satisfaction.
The research results of the impact on vocal quality and patient satisfaction showed that the surgery is an alternative therapy in treating SD, and patient satisfaction is high, and most patients managed voice fluency with minimum breathiness.The complications could be minimized by conservative LCA myotomy.Truong; Bhidayasiri 37 Review in the literature about laryngeal muscle hyperactivity syndromes, techniques and types of toxin injections available, as well as doses used.
In the literature, the use of BT is the most often described treatment for AdSD, especially applied in the TA muscle.It also described its application in the LCA and interarytenoid muscles depending on whether it is adductor or abductor SD.The studies presented results of uni or bilateral applied BT.In addition, the authors found the description of intramuscular toxin injection techniques as: percutaneous, transoral, transnasal, and point touch.For laryngeal dystonia three articles were found: one of them, classification B, pointed out that vocal therapy combined with BT is beneficial to the patient and prolonging the interval of Botox injection improved respiratory measurements and the acoustic parameters.The other studies, with classification B, showed conflicting results, one of which suggested that speech therapy improved intelligibility, speech functionality, and patient confidence.The other concluded that speech therapy, psychotherapy, and biofeedback had not positive effect on SD.
and homeopathy.The use of BT injection showed results that indicated the satisfaction of the patients treated, although some of the articles point out the disadvantage of this treatment, i.e. the need to reapply BT after a few months.With regard to surgical procedures, they may be considered longlasting and suitable for patients that do not wish to undergo BT injections.The studies, however, presented contingency of restricted patients and the results were based on the patient's own judgement as to his/her vocal quality.The use of lidocaine and homeopathy showed positive results in relation to the vocal quality of the patient and was suggested as an option for those patients who would not like to submit to surgery or BT injection.The few studies that did contemplate speech therapy showed positive results from this treatment when combined with BT injection.However, it is clear the lack of evidence on the effects of speech therapy in treating SD, although associated with medical treatment.Thus, there is the need for further research involving speech therapy in SD in associated with clinical and/ or surgical treatment.
that speech therapy, as well as pyschotherapy and biofeedback did not bring positive effect in controlling pathological symptoms 40 .
In this survey, there were six literature review articles and half of them attempted to learn about treatment options published in a given period of time, whose scientific methodology could prove the effectiveness of treatments 4,38,40 .The others also tried to obtain the knowledge of proposed treatment of SD by only considering the results of surgery or the use of BT 36,37,39 .
A single literature review identified the need for further studies in the understanding of neurological functioning in SD, for then it would be possible to attempt a treatment without side effects 39 .

Figure 1 -
Figure 1 -List of words and subject descriptors used in the search

Table 1 -Articles on the use of botulinum toxin (BT) injection for the treatment of spasmodic dysphonia
Report the case of a patient with adductor type laryngeal dystonia treated with BT and discuss the advantages and the reports presented in the literature.
breathy voice in the period after BT application and the drop in the vocal quality at the end of a treatment cycle involves a reduction in the patient's quality of life with an average score of 52.8.The authors propose long-term treatment to aid the periods in which the vocal quality is not good.
Examine the correlation between the clinical assessment and that of patients with AdSD on the effects of Botox in the voice quality and quality of life.The responses from selfassessment of voice impairment, EIS (Equal Interval Scale), and the CAPE-V (Consensus Auditory Perceptual Evaluation of Voice) and V-RQOL (Voice-Related Quality of Life) protocols were used.Charts from 199 patients (149 women and 50 men) with ages from 18 to 90 years with AdSD who received two consecutive injections of Botox were reviewed.A retrospective study from 2004 to 2007.

Table 5 -Articles categorized as literature review of spasmodic dysphonia treatments divided by year, author(s), objective(s), and results
Studies with type and toxin dosage control were also found.2006 Watts; Nye; Whurr 4 Determine the efficacy of BT in the treatment of SD through a randomized systematic analysis in the Cochrane database.