REVIEW URRENT C OPINION
Evidence-based treatment of voice and speech disorders in Parkinson disease Leslie A. Mahler a, Lorraine O. Ramig b,c,d, and Cynthia Fox d
Purpose of review Voice and speech impairments are present in nearly 90% of people with Parkinson disease and negatively impact communication and quality of life. This review addresses the efficacy of Lee Silverman Voice Treatment (LSVT) LOUD to improve vocal loudness (as measured by vocal sound pressure level vocSPL) and functional communication in people with Parkinson disease. The underlying physiologic mechanisms of Parkinson disease associated with voice and speech changes and the strength of the current treatment evidence are discussed with recommendations for best clinical practice. Recent findings Two randomized control trials demonstrated that participants who received LSVT LOUD were significantly better on the primary outcome variable of improved vocSPL posttreatment than alternative and no treatment groups. Treatment effects were maintained for up to 2 years. In addition, improvements have been demonstrated in associated outcome variables, including speech rate, monotone, voice quality, speech intelligibility, vocal fold adduction, swallowing, facial expression and neural activation. Advances in technology-supported treatment delivery are enhancing treatment accessibility. Summary Data support the efficacy of LSVT LOUD to increase vocal loudness and functional communication in people with Parkinson disease. Timely intervention is essential for maximizing quality of life for people with Parkinson disease. Keywords LSVT LOUD, neuroplasticity, Parkinson disease, treatment
INTRODUCTION The presence of voice and speech disorders associated with dysarthria in people with Parkinson disease has the potential to decrease functional communication. Even mild dysarthria can influence listener perceptions of speech and a speaker may encounter negative attitudes or discrimination when dysarthria is present. This review includes research demonstrating that Lee Silverman Voice Treatment (LSVT LOUD), a well defined behavioural treatment, can improve vocal loudness and functional communication in people with Parkinson disease and that the benefits of treatment can last for up to 2 years even in the presence of a degenerative neurological disorder. LSVT LOUD methods and research data are presented with an emphasis on how the treatment addresses the underlying physiology of voice and speech changes associated with Parkinson disease and how the treatment incorporates key practice parameters, which drive neural change that may be important for long-term
carryover and generalization to functional communication. Data on the primary outcome variable of increased loudness [as measured by vocal sound pressure level (vocSPL)] and improvements in associated outcome variables following LSVT LOUD are reviewed and recommendations for best clinical practice in treating the voice and speech disorders of people with Parkinson disease are made.
a
Department of Communicative Disorders, University of Rhode Island, Kingston, Rhode Island, bUniversity of Colorado, Boulder, Colorado, c Columbia University, New York, New York and dNational Center for Voice and Speech, Denver, Colorado, USA Correspondence to Leslie A. Mahler, Department of Communicative Disorders, University of Rhode Island, Kingston, RI 02881, USA. Tel: +1 401 874 2490; fax: +1 401 874 4404; e-mail: Leslie_mahler@ uri.edu Curr Opin Otolaryngol Head Neck Surg 2015, 23:209–215 DOI:10.1097/MOO.0000000000000151
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Voice and speech treatment
KEY POINTS Nearly 90% of people diagnosed with Parkinson disease have voice and speech disorders that negatively impact quality of life. LSVT LOUD was specifically designed to address the physiology of voice and speech disorders resulting from Parkinson disease. The studies reviewed in this article provide evidence for long-lasting effects of treatment and improvements in associated voice and speech characteristics. Extensive data at physiological, aerodynamic, acoustic and perceptual levels support the lack of any vocal hyperfunction (e.g. pressed, strained voice) accompanying increased vocal loudness in Parkinson disease. Early referral for treatment and a laryngeal examination prior to treatment are recommended for best clinical practice.
INCIDENCE OF VOICE AND SPEECH DISORDERS IN PEOPLE WITH PARKINSON DISEASE Idiopathic Parkinson disease is one of the most common diagnoses of degenerative neurological disorders [1] affecting 1–2% of individuals over the age of 60 years and those in their 40s and 50s with young onset Parkinson disease [2]. Nearly 90% of people with Parkinson disease will develop voice and speech disorders during the course of the disease [3], which can have a negative impact on functional communication contributing to decreased quality of life [4]. Voice and speech disorders associated with Parkinson disease are most commonly characterized by one or a combination of the following perceptual characteristics; reduced vocal loudness [5]; a breathy or hoarse/harsh voice quality [6]; imprecise consonants and distorted vowels [7]; and reduced voice pitch inflections or monotone voice [8] collectively called hypokinetic dysarthria [9].
ORIGIN OF VOICE AND SPEECH DISORDERS IN PEOPLE WITH PARKINSON DISEASE Recently, there has been a proliferation of published research on the neural bases of Parkinson disease, which has led to an increased understanding of the underlying neurophysiology associated with changes in voice and speech. It is well established that the symptoms of Parkinson disease are associated with alterations in basal ganglia circuitry due to a decrease in dopamine in the substantia nigra pars 210
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compacta [10]. Understanding of the potential underlying neurophysiology of dopamine loss may only be a partial explanation for voice and speech disorders in PD. Recent research suggests that nondopaminergic neurons are also involved in the development of Parkinson disease [11] and that these changes begin before the onset of Parkinson disease symptoms [12]. Physiologic abnormalities associated with voice and speech changes in people with Parkinson disease include reduced vocal fold adduction and asymmetrical patterns of vocal fold vibration [13,14]; reduced neural drive to laryngeal muscles [15]; poor reciprocal suppression of laryngeal and respiratory muscles [16]; and a reduction in respiratory muscle activation patterns [17] all of which contribute to the perceptual feature of significantly decreased loudness. Motor symptoms of rigidity, weakness, bradykinesia and hypokinesia result from dopamine deficiency [18,19] but do not completely account for the voice and speech abnormalities associated with Parkinson disease. Sensory deficits, deficits in internal monitoring of amplitude and maintenance of amplitude of movements across the speech production mechanism may also be significant factors that contribute to decreased loudness, imprecise articulation and monotone [20–23]. One study compared functional MRI (fMRI) in 20 people with Parkinson disease who did not demonstrate overt voice and speech disorders. They were studied in the ON and OFF medication conditions and compared with 20 age-matched controls [24 ]. This study identified decreased striato-prefrontal connectivity and diminished monitoring of external auditory feedback that could interfere with affective modulation of speech prosody (i.e. vocal loudness and intonation) and may contribute to the voice and speech disorders associated with Parkinson disease. For a detailed review of the origins of voice and speech disorders in Parkinson disease, see Sapir, 2014 [25 ]. &
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LSVT LOUD ADDRESSES THE PHYSIOLOGY OF VOICE AND SPEECH DISORDERS IN PEOPLE WITH PARKINSON DISEASE Pharmacological and neurosurgical approaches for managing limb motor symptoms of Parkinson disease have not demonstrated a consistent or significant impact on voice and speech disorders [26–29]. Therefore, a behavioural treatment for people with Parkinson disease is needed to effectively improve voice and speech disorders. LSVT LOUD targets the cardinal perceptual feature of reduced vocal loudness in people with Parkinson Volume 23 Number 3 June 2015
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Evidence-based treatment of voice and speech disorders in Parkinson disease Mahler et al.
disease, which limits audibility and thus functional communication. Multiple physiologic mechanisms may contribute to reduced vocal loudness including reduced amplitude of movement (hypokinesia), slowness of movement (bradykinesia) and reduced ability to maintain movement amplitude [16]. A breathy or harsh/hoarse voice quality and reduced pitch range may be present due to reduced amplitude of vocal fold movement and incomplete adduction of the vocal folds during phonation [14,15]. Thus, increasing vocal loudness in treatment to levels more closely approximating normal loudness while simultaneously addressing sensory deficits, targets the proposed physiologic mechanisms underlying bradykinesia and hypokinesia: inadequate muscle activation [16,17].
FUNDAMENTALS OF LSVT LOUD Development of LSVT LOUD began in the late 1980s at a time when no behavioural speech treatments had been identified in the literature to ameliorate voice and speech disorders for people with Parkinson disease. Initial data reported in 1988 by Ramig et al. [30] established the LSVT LOUD treatment protocol. LSVT LOUD consists of exercises to train increased amplitude of motor output (vocal loudness as measured by increased vocSPL) as well as training individuals to monitor vocal loudness and relearn a new internal cue for scaling amplitude of motor output. Training increased vocal loudness (i.e. speak louder) serves as a single motor organizing theme that coordinates effort across multiple speech production subsystems that may be impaired [31]. LSVT LOUD is unique in its intensive and high effort mode of treatment delivery. This mode is consistent with theories of motor learning and principles that drive activity-dependent neuroplasticity, both of which may be important for longterm carryover and generalization of improved vocal loudness to functional communication outside of the treatment room [32]. In addition, LSVT LOUD is unique because it addresses sensory kinesthetic and internal cueing deficits that can be barriers to carryover and generalization of treatment effects for people with Parkinson disease. People with Parkinson disease relearn a new internal cue for the amount of effort they need to produce for normal loudness and are recalibrated to their sensory perception of this loudness to recognize it is within normal limits [31,33]. The LSVT LOUD protocol has been described in detail in previously published research [34,35]. In brief, it consists of 16 individual 1-h sessions administered four times a week for 4 weeks and daily homework and carryover activities every day of
the month and is continued daily throughout life. The treatment incorporates materials that are salient to individual patients. This means that although the exercises and mode of delivery are consistent in all LSVT LOUD treatment sessions, speech materials are customized to address individual goals for improved vocal loudness in each patient’s daily functional communication.
RESEARCH OUTCOMES ON LSVT LOUD The initial randomized control trial (RCT) compared two treatments designed to improve loudness: one treatment focused on voice and the other treatment focused on increasing respiratory support for speech. Those treatments were matched for intensity of treatment within the treatment session (i.e. number of task repetitions, driving of motor effort, reinforcement from speech clinicians) and in treatment dosage including homework and carryover assignments. Evaluations were conducted immediately before treatment, immediately following treatment and at 6, 12 and 24 months after the completion of treatment. The primary outcome variable in this study was vocSPL. The data showed that intensive voice treatment (LSVT LOUD) was more effective in improving vocSPL than the respiratory treatment [36 ]. In another RCT, LSVT LOUD was compared with an untreated group of people with Parkinson disease and an age and sex-matched healthy control group. The groups were evaluated immediately before treatment, immediately after treatment and 6 months after treatment completion. The outcomes of this study also demonstrated statistically significant improvements in vocSPL for people who received LSVT LOUD compared with an untreated group. Across both of these studies, effect sizes were larger than 0.80 for the primary outcome variable of vocSPL indicating that the change was also clinically significant [37 ]. Importantly, increased vocal loudness was documented posttreatment with no evidence of increasing supraglottic hyperfunction, and in one study, a decrease in pretreatment supraglottic false fold and anterior posterior hyperfunction was documented post treatment [38]. Findings from a group study by Smith et al. [14] using laryngostroboscopic assessment documented improved glottal closure with no change in supraglottal tension following LSVT LOUD. These findings suggest that phonatory effort, which is targeted in LSVT LOUD, results in healthy vocal fold adduction without laryngeal hyperfunction. In contrast, de Swart et al. [39] claimed adverse effects of increasing loudness in Parkinson disease. These researchers asked patients to ‘speak
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loud/shout’ during one 30-min stimulability session and concluded that pressed voice and excessive strain accompanied LSVT LOUD. The use of LSVT LOUD or ‘LSVT’ in the de Swart et al. article is incorrect. Pressed voice and strain are never the behaviours elicited or trained during the 16 sessions of LSVT LOUD. Increased loudness is what the person with Parkinson disease may feel when increasing vocal effort; healthy, normal loudness is what
the speech clinician elicits and shapes in terms of actual vocal output. Extensive data at physiological, aerodynamic, acoustic and perceptual levels [6,14, 38,40,41] support the lack of any strain or hyperfunction accompanying increased vocal loudness in Parkinson disease. Data demonstrating associated treatment outcomes have been reported across a range of variables at perceptual, acoustic, aerodynamic, physiologic
Table 1. Summary of research outcome data following LSVT LOUD Primary Outcome Variable Acoustic: Improved vocSPL
•
Associated Outcome Variables Additional acoustic variables: Improved rate; slower
•
[33]Ramig, Countryman, Thompson, Horii, 1995
Improved F0 variation (Pitch); Reduced monotone
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[36]Ramig, Sapir, Countryman, et al, 2001; [43]Ramig, Bonitati, Lemke, Horii, 1994; [42]Whitehill, Kwan, Lee , Chow, 2011
Improved articulation; Larger vowel space area
•
[7]Sapir, Spielman, Ramig, Story, Fox, 2007
Improved formant centralization ratio
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[44]Sapir, Ramig, Spielman, Fox, 2010
Improved articulatory acoustics Perceptual: Listener ratings Improvement in voice quality Less harsh and breathy
•
[45]Dromey, Ramig, Johnson, 1995
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[6]Baumgartner, Sapir, Ramig, 2001
Better voice quality
•
[46]Sapir, Ramig, Hoyt, et al, 2002
Improved speech intelligibility
•
[47]Cannito, Suiter, Beverly, et al, 2012
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[33]Ramig, Countryman, Thompson, Horii, 1995
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[48]Smith, Ramig, Dromey, et al, 1995; [38]Countryman, Hicks, Ramig, Smith, 1997
Electroglottography
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[49]Ramig, Dromey, 1996
More efficient swallow
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[50]El-Sharkawi, Ramig, Logemann, et al., 2002
Improvement in maximum capacity tongue function Aerodynamic: Improved subglottic air pressure
•
[51]Ward, Theodoros, Murdoch, Silburn, 2000
•
[49]Ramig, Dromey, 1996
Family and self ratings Improved ratings of communication characteristics Physiologic: Improved vocal fold adduction with no increase in laryngeal tension Direct visualization
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Data documenting treatment outcomes following LSVT LOUD [33]Ramig, Countryman, Thompson, Horii, 1995; [35]Ramig, Countryman, O’Brien, et al, 1996; [36]Ramig, Sapir, Countryman, et al, 2001; [37]Ramig, Sapir, Fox, Countryman, 2001; [42]Whitehill, Kwan, Lee , Chow, 2011. Data documenting treatment outcomes following LSVT LOUD
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Evidence-based treatment of voice and speech disorders in Parkinson disease Mahler et al. Table 1. (Continued) and MFDR Improved respiratory kinematics Facial expression: Improved frequency and variation of expressions Neural: Activation pattern changes in cortex and subcortical areas and rightsided shift in activation Treatment Accessibility
•
[52]Huber, Stathopoulos, Ramig, Lancaster, 2003
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[53]Spielman, Borod, Ramig, 2003; [54]Dumer, Oster, et al., 2014
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[55]Liotti, Ramig, Vogel, et al., 2003; [56]Narayana, Fox, Zhang, et al., 2010
Data supporting alternate modes of treatment
Primary outcome variable Increased vocSPL: Telepractice LSVT eLOUD™
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[57]Theodoros, Constantinescu, Russell, et al, 2006; [58]Constantinescu G, Theodoros D, Russell T, et al, 2011; [59}Howell, Tripoliti, Pring, 2009; [60]Theodoros, Ramig, 2011
LSVT Companion Software
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[61]Halpern, Ramig, Matos, et al., 2012
Alternate treatment dosage LSVT-X
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[62]Spielman, Ramig, Mahler, Halpern, Petska, 2007
and neural levels accompanying training increased vocSPL and are summarized in Table 1 [7,33,35, 36 ,37 ,42–53,54 ,55–57]. In addition, research studies examining treatment outcomes for people with atypical Parkinsonism [63,64] and those who have received deep brain stimulation [65,66] show a positive impact of LSVT LOUD on voice and speech variables, although these patients may be more challenging to treat [65].
clinician and seven using the LSVT Companion at home on their own. Outcome data demonstrated that all 16 participants had comparable gains in vocSPL immediately posttreatment and at 6 months follow-up as compared with data of people with Parkinson disease who received all 16 sessions in person.
MAKING LSVT LOUD MORE ACCESSIBLE
Today, LSVT LOUD is the most researched treatment for people with Parkinson disease and has been shown to be an effective behavioural treatment of voice and speech disorders associated with Parkinson disease. Speech clinicians must complete a training and certification course to administer the LSVT LOUD protocol. This training is required to maintain treatment fidelity of LSVT LOUD, thus allowing people with Parkinson disease to expect treatment outcomes similar to those documented in the research. As of publication of this article, there are over 14 000 clinicians certified in LSVT LOUD in 62 countries. Voice and speech symptoms have been reported to occur very early in the diagnosis of Parkinson disease and models of the physiology suggest that changes in the nervous system underlying voice and speech disorders begin long before the onset of symptoms [10–12,24 ]. Therefore, it is recommended that
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More recent research has been conducted to evaluate the incorporation of technology to make treatment more accessible to people with Parkinson disease. Several studies have examined the feasibility of telerehabilitation with LSVT LOUD and found that treatment outcomes were similar to administration of treatment wherein all sessions were conducted in person [57–60]. These studies support the feasibility and effectiveness of online administration of LSVT LOUD for people with Parkinson disease after an initial evaluation in person to determine candidacy for treatment. Another study was recently completed assessing the feasibility and effectiveness of an assistive technology system called the LSVT Companion, a U.S. Food and Drug Administration (FDA)-approved medical device [61 ]. People with Parkinson disease received nine LSVT LOUD sessions in person with a speech &
RECOMMENDATIONS FOR BEST CLINICAL PRACTICE
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people with Parkinson disease receive LSVT LOUD soon after diagnosis to obtain the maximum benefit from the treatment for the greatest possible length of time. It is further recommended that each patient receives a laryngeal evaluation by an otolaryngologist prior to initiation of treatment as part of best practice to confirm a diagnosis of voice changes secondary to Parkinson disease and eliminate the possibility of vocal fold disease other than that associated with Parkinson disease such as gastroesophageal reflux disease or unilateral vocal fold paralysis.
CONCLUSION The cumulative evidence from treatment efficacy studies of LSVT LOUD provides support for implementation of behavioural treatment of voice and speech deficits resulting from Parkinson disease. The studies reviewed in this article provide evidence for long-lasting effects of treatment following LSVT LOUD and the data demonstrate distributed effects of treatment to acoustic, perceptual, physiologic, aerodynamic, facial expression and neural control variables. The ability to communicate is essential to quality of life. Our future research will continue to work to improve treatment outcomes and understand the basic mechanisms accompanying treatment-related change. Acknowledgements The authors thank the patients who continue to inspire and challenge us and The National Center for Voice and Speech research team and the University of Colorado, Boulder. Financial support and sponsorship This research was funded by the National Institutes of Health (NIH) grants R01 DC1150, R21 RFA-NS-02006, R21 DC006078 and R21 NS043711 from the National Institutes of Deafness and Other Communication Disorders (NIDCD). Conflicts of interest Leslie A. Mahler receives lecture honoraria and travel reimbursement from LSVT Global, Inc. Lorraine O. Ramig and Cynthia M. Fox receive lecture honoraria and have ownership interest in LSVT Global, Inc. They have disclosed any conflict of interest and their conflict of interest management plan has been approved by their respective Offices of Conflict of Interest and Commitment at the University of Colorado, Boulder and the University of Rhode Island. The authors have no other relevant affiliations or financial involvement with any 214
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organization or entity with a financial interest in or financial conflict with the subject matter or materials discussed in the manuscript apart from those disclosed.
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Evidence-based treatment of voice and speech disorders in Parkinson disease Mahler et al. 27. Goberman A, Coelho C, Robb M. Phonatory characteristics of Parkinsonian speech before and after morning medication: the ON and OFF states. J Commun Disord 2002; 35:217–239. 28. Pinto S, Ozsancak C, Tripoliti E, et al. Treatments for dysarthria in Parkinson’s disease. Lancet Neurol 2004; 3:547–556. 29. Pinto S, Gentil M, Krack P, et al. Changes induced by levodopa and subthalamic nucleus stimulation on Parkinsonian speech. Mov Disord 2005; 20:1507–1515. 30. Ramig LO, Mead C, Scherer R, et al. Voice therapy and Parkinson’s disease: a longitudinal study of efficacy. Presented at: The Clinical Dysarthria Conference. San Diego, CA 1988; 4–7; February. 31. Fox C, Morrison C, Ramig L, Sapir S. Current perspectives on the Lee Silverman Voice Treatment (LSVT). Am J Speech Lang Path 2002; 11:111–123. 32. Maas E, Robin DA, Hula SNA, et al. Principles of motor learning in treatment of motor speech disorders. J Speech-Lang Path 2008; 17:277–298. 33. Ramig LO, Countryman S, Thompson L, Horii Y. A comparison of two forms of intensive speech treatment for Parkinson disease. J Speech Hear Res 1995; 38:1232–1251. 34. Fox C, Ebersbach G, Ramig L, Sapir S. LSVT LOUD and LSVT BIG: behavioral treatment programs for speech and body movement in Parkinson disease. Parkinson’s Dis 2012; 2012:391946. 35. Ramig LO, Countryman S, O’Brien C, et al. Intensive speech treatment for patients with Parkinson disease: short and long-term comparison of two techniques. Neurology 1996; 47:1496–1504. 36. Ramig L, Sapir S, Countryman S, et al. Intensive voice treatment (LSVT1) for && individuals with Parkinson disease: a two-year follow-up. J Neurol Neurosurg Psychiatry 2001; 71:493–498. This study provides data from a RCT comparing treatment outcomes for LSVT LOUD and respiratory treatment. 37. Ramig L, Sapir S, Fox C, Countryman S. Changes in vocal intensity following && intensive voice treatment (LSVT1) in individuals with Parkinson disease: a comparison with untreated patients and age-matched controls. Mov Disord 2001; 16:79–83. This study provides data from a RCT comparing treatment outcomes for LSVT LOUD, untreated individuals with Parkinson disease and a healthy control group. 38. Countryman S, Hicks J, Ramig LO, Smith M. Supraglottal hyperadduction in an individual with Parkinson disease: a clinical treatment note. Am J Speech Lang Path 1997; 6:74–84. 39. de Swart BJM, Willemse SC, Maassen BAM, Horstink MWIM. Improvement of voicing in patients with Parkinson’s disease by speech therapy. Neurology 2003; 60:498–500. 40. Tjaden K, Wilding GE. Rate and loudness manipulations in dysarthria: acoustic and perceptual findings. J Speech Lang Hearing Res 2004; 47:766–783. 41. Huber J, Darling M. Effect of Parkinson’s disease on the production of structured and unstructured speaking tasks: respiratory physiologic and linguistic considerations. J Speech Lang Hearing Res 2011; 54:33–46. 42. Whitehill TL, Kwan L, Lee FP, Chow MM. Effect of LSVT on lexical tone in speakers with Parkinson’s disease. Parkinson’s Dis 2011; 2011:897494. 43. Ramig L, Bonitati CM, Lemke JH, Horii Y. Voice treatment for patients with Parkinson disease: development of an approach and preliminary efficacy data. J Med Speech Lang Path 1994; 2:191–209. 44. Sapir S, Ramig LO, Spielman JL, Fox C. Formant centralization ratio: a proposal for a new acoustic measure of dysarthric speech. J Speech Lang Hear Res 2010; 53:114–125. 45. Dromey C, Ramig LO, Johnson A. Phonatory and articulatory changes associated with increased vocal intensity in Parkinson disease: a case study. J Speech Hear Res 1995; 38:751–764. 46. Sapir S, Ramig L, Hoyt P, et al. Speech vocSPL and quality 12 months after intensive voice treatment (LSVT) for Parkinson’s disease: a comparison with an alternative speech treatment. Folia Phoniatrica 2002; 54:296–303.
47. Cannito MP, Suiter DM, Beverly D, et al. Sentence intelligibility before and after voice treatment in speakers with idiopathic Parkinson’s disease. J Voice 2012; 26:214–219. 48. Smith M, Ramig LO, Dromey C, et al. Intensive voice treatment in Parkinson disease: laryngostroboscopic findings. J Voice 1995; 9:453–459. 49. Ramig L, Dromey C. Aerodynamic mechanisms underlying treatment-related changes in vocal intensity in patients with Parkinson disease. J Speech Hear Res 1996; 39:798–807. 50. El-Sharkawi A, Ramig LO, Logemann JA, et al. Swallowing and voice effects of Lee Silverman Voice Treatment (LSVT): a pilot study. J Neurol Neurosurg Psychiatry 2002; 71:31–36. 51. Ward EC, Theodoros DG, Murdoch BE, Silburn P. Changes in maximum capacity tongue function following the Lee Silverman Voice Treatment Program. J Med Speech Lang Path 2000; 8:331–335. 52. Huber JE, Stathopolous ET, Ramig LO, Lancaster SL. Respiratory function and variability in individuals with Parkinson disease: pre and post-Lee Silverman Voice Treatment. J Med Speech Lang Path 2003; 11:185–201. 53. Spielman J, Borod J, Ramig L. Effects of intensive voice treatment (LSVT) on facial expressiveness in Parkinson’s disease: preliminary data. Cog Behav Neuro 2003; 16:177–188. 54. Dumer AL, Oster H, McCabe D, et al. Effects of the Lee Silverman Voice & Treatment on facial expressivity in Parkinson’s disease. J Int Neuropsych Soc 2014; 20:302–312. This article provides evidence for associated changes in facial movement following LSVT LOUD. 55. Liotti M, Ramig LO, Vogel D, et al. Hypophonia in Parkinson’s disease. Neural correlates of voice treatment revealed by PET. Neurology 2003; 60:432–440. 56. Narayana S, Fox PT, Zhang W, et al. Neural correlates of efficacy of voice therapy in Parkinson’s disease identified by performance-correlation analysis. Hum Brain Map 2010; 31:222–236. 57. Theodoros D, Constantinescu G, Russell TG, et al. Treating the speech disorder in Parkinson’s disease online. J Telemed Telecare 2006; 12 (Suppl 3): 88–91. 58. Constantinescu G, Theodoros D, Russell T, et al. Treating disordered speech and voice in Parkinson’s disease online: a randomized controlled noninferiority trial. Intl J Lang Comm Disord 2011; 46:1–16. 59. Howell S, Tripoliti E, Pring T. Delivering the Lee Silverman Voice Treatment (LSVT) by web camera: a feasibility study. Int J Lang Comm Disord 2009; 44:287–300. 60. Theodoros E, Ramig O. Telepractice supported delivery of LSVT LOUD. Sem Speech Lang Path 2011; 21:107–119. 61. Halpern E, Ramig O, Matos E, et al. Innovative technology for the assisted & delivery of intensive voice treatment (LSVT LOUD) for Parkinson disease. Am J Sp-Lang Path 2012; 21:354–367. This study provides evidence for the integration of technology in the delivery of LSVT LOUD with treatment outcomes similar to treatment in person thus increasing accessibility of treatment to patients with Parkinson disease. 62. Spielman J, Ramig L, Mahler L, et al. Effects of LSVT extended (LSVT-X) on voice and speech in Parkinson disease. Am J Speech Lang Path 2007; 16:95–107. 63. Countryman S, Ramig LO. Effects of intensive voice therapy on voice deficits associated with bilateral thalamotomy in Parkinson’s disease: a case study. J Med Speech Lang Path 1993; 1:233–250. 64. Countryman S, Ramig LO, Pawlas A. Speech and voice deficits in Parkinsonian plus syndromes: can they be treated? J Med Speech Lang Path 1994; 2:211–225. 65. Spielman J, Mahler L, Halpern A, et al. Intensive voice treatment (LSVT LOUD) for Parkinson’s disease following deep brain stimulation of the subthalamic nucleus. J Comm Dis 2011; 44:688–700. 66. Tripoliti E, Zrinzo L, Martinez-Torres I, et al. Effects of subthalamic stimulation on speech of consecutive patients with Parkinson disease. Neurology 2011; 76:80–86.
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Evidence-based treatment of voice and speech disorders in Parkinson disease Leslie A. Mahler a, Lorraine O. Ramig b,c,d, and Cynthia Fox d
Purpose of review Voice and speech impairments are present in nearly 90% of people with Parkinson disease and negatively impact communication and quality of life. This review addresses the efficacy of Lee Silverman Voice Treatment (LSVT) LOUD to improve vocal loudness (as measured by vocal sound pressure level vocSPL) and functional communication in people with Parkinson disease. The underlying physiologic mechanisms of Parkinson disease associated with voice and speech changes and the strength of the current treatment evidence are discussed with recommendations for best clinical practice. Recent findings Two randomized control trials demonstrated that participants who received LSVT LOUD were significantly better on the primary outcome variable of improved vocSPL posttreatment than alternative and no treatment groups. Treatment effects were maintained for up to 2 years. In addition, improvements have been demonstrated in associated outcome variables, including speech rate, monotone, voice quality, speech intelligibility, vocal fold adduction, swallowing, facial expression and neural activation. Advances in technology-supported treatment delivery are enhancing treatment accessibility. Summary Data support the efficacy of LSVT LOUD to increase vocal loudness and functional communication in people with Parkinson disease. Timely intervention is essential for maximizing quality of life for people with Parkinson disease. Keywords LSVT LOUD, neuroplasticity, Parkinson disease, treatment
INTRODUCTION The presence of voice and speech disorders associated with dysarthria in people with Parkinson disease has the potential to decrease functional communication. Even mild dysarthria can influence listener perceptions of speech and a speaker may encounter negative attitudes or discrimination when dysarthria is present. This review includes research demonstrating that Lee Silverman Voice Treatment (LSVT LOUD), a well defined behavioural treatment, can improve vocal loudness and functional communication in people with Parkinson disease and that the benefits of treatment can last for up to 2 years even in the presence of a degenerative neurological disorder. LSVT LOUD methods and research data are presented with an emphasis on how the treatment addresses the underlying physiology of voice and speech changes associated with Parkinson disease and how the treatment incorporates key practice parameters, which drive neural change that may be important for long-term
carryover and generalization to functional communication. Data on the primary outcome variable of increased loudness [as measured by vocal sound pressure level (vocSPL)] and improvements in associated outcome variables following LSVT LOUD are reviewed and recommendations for best clinical practice in treating the voice and speech disorders of people with Parkinson disease are made.
a
Department of Communicative Disorders, University of Rhode Island, Kingston, Rhode Island, bUniversity of Colorado, Boulder, Colorado, c Columbia University, New York, New York and dNational Center for Voice and Speech, Denver, Colorado, USA Correspondence to Leslie A. Mahler, Department of Communicative Disorders, University of Rhode Island, Kingston, RI 02881, USA. Tel: +1 401 874 2490; fax: +1 401 874 4404; e-mail: Leslie_mahler@ uri.edu Curr Opin Otolaryngol Head Neck Surg 2015, 23:209–215 DOI:10.1097/MOO.0000000000000151
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Voice and speech treatment
KEY POINTS Nearly 90% of people diagnosed with Parkinson disease have voice and speech disorders that negatively impact quality of life. LSVT LOUD was specifically designed to address the physiology of voice and speech disorders resulting from Parkinson disease. The studies reviewed in this article provide evidence for long-lasting effects of treatment and improvements in associated voice and speech characteristics. Extensive data at physiological, aerodynamic, acoustic and perceptual levels support the lack of any vocal hyperfunction (e.g. pressed, strained voice) accompanying increased vocal loudness in Parkinson disease. Early referral for treatment and a laryngeal examination prior to treatment are recommended for best clinical practice.
INCIDENCE OF VOICE AND SPEECH DISORDERS IN PEOPLE WITH PARKINSON DISEASE Idiopathic Parkinson disease is one of the most common diagnoses of degenerative neurological disorders [1] affecting 1–2% of individuals over the age of 60 years and those in their 40s and 50s with young onset Parkinson disease [2]. Nearly 90% of people with Parkinson disease will develop voice and speech disorders during the course of the disease [3], which can have a negative impact on functional communication contributing to decreased quality of life [4]. Voice and speech disorders associated with Parkinson disease are most commonly characterized by one or a combination of the following perceptual characteristics; reduced vocal loudness [5]; a breathy or hoarse/harsh voice quality [6]; imprecise consonants and distorted vowels [7]; and reduced voice pitch inflections or monotone voice [8] collectively called hypokinetic dysarthria [9].
ORIGIN OF VOICE AND SPEECH DISORDERS IN PEOPLE WITH PARKINSON DISEASE Recently, there has been a proliferation of published research on the neural bases of Parkinson disease, which has led to an increased understanding of the underlying neurophysiology associated with changes in voice and speech. It is well established that the symptoms of Parkinson disease are associated with alterations in basal ganglia circuitry due to a decrease in dopamine in the substantia nigra pars 210
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compacta [10]. Understanding of the potential underlying neurophysiology of dopamine loss may only be a partial explanation for voice and speech disorders in PD. Recent research suggests that nondopaminergic neurons are also involved in the development of Parkinson disease [11] and that these changes begin before the onset of Parkinson disease symptoms [12]. Physiologic abnormalities associated with voice and speech changes in people with Parkinson disease include reduced vocal fold adduction and asymmetrical patterns of vocal fold vibration [13,14]; reduced neural drive to laryngeal muscles [15]; poor reciprocal suppression of laryngeal and respiratory muscles [16]; and a reduction in respiratory muscle activation patterns [17] all of which contribute to the perceptual feature of significantly decreased loudness. Motor symptoms of rigidity, weakness, bradykinesia and hypokinesia result from dopamine deficiency [18,19] but do not completely account for the voice and speech abnormalities associated with Parkinson disease. Sensory deficits, deficits in internal monitoring of amplitude and maintenance of amplitude of movements across the speech production mechanism may also be significant factors that contribute to decreased loudness, imprecise articulation and monotone [20–23]. One study compared functional MRI (fMRI) in 20 people with Parkinson disease who did not demonstrate overt voice and speech disorders. They were studied in the ON and OFF medication conditions and compared with 20 age-matched controls [24 ]. This study identified decreased striato-prefrontal connectivity and diminished monitoring of external auditory feedback that could interfere with affective modulation of speech prosody (i.e. vocal loudness and intonation) and may contribute to the voice and speech disorders associated with Parkinson disease. For a detailed review of the origins of voice and speech disorders in Parkinson disease, see Sapir, 2014 [25 ]. &
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LSVT LOUD ADDRESSES THE PHYSIOLOGY OF VOICE AND SPEECH DISORDERS IN PEOPLE WITH PARKINSON DISEASE Pharmacological and neurosurgical approaches for managing limb motor symptoms of Parkinson disease have not demonstrated a consistent or significant impact on voice and speech disorders [26–29]. Therefore, a behavioural treatment for people with Parkinson disease is needed to effectively improve voice and speech disorders. LSVT LOUD targets the cardinal perceptual feature of reduced vocal loudness in people with Parkinson Volume 23 Number 3 June 2015
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Evidence-based treatment of voice and speech disorders in Parkinson disease Mahler et al.
disease, which limits audibility and thus functional communication. Multiple physiologic mechanisms may contribute to reduced vocal loudness including reduced amplitude of movement (hypokinesia), slowness of movement (bradykinesia) and reduced ability to maintain movement amplitude [16]. A breathy or harsh/hoarse voice quality and reduced pitch range may be present due to reduced amplitude of vocal fold movement and incomplete adduction of the vocal folds during phonation [14,15]. Thus, increasing vocal loudness in treatment to levels more closely approximating normal loudness while simultaneously addressing sensory deficits, targets the proposed physiologic mechanisms underlying bradykinesia and hypokinesia: inadequate muscle activation [16,17].
FUNDAMENTALS OF LSVT LOUD Development of LSVT LOUD began in the late 1980s at a time when no behavioural speech treatments had been identified in the literature to ameliorate voice and speech disorders for people with Parkinson disease. Initial data reported in 1988 by Ramig et al. [30] established the LSVT LOUD treatment protocol. LSVT LOUD consists of exercises to train increased amplitude of motor output (vocal loudness as measured by increased vocSPL) as well as training individuals to monitor vocal loudness and relearn a new internal cue for scaling amplitude of motor output. Training increased vocal loudness (i.e. speak louder) serves as a single motor organizing theme that coordinates effort across multiple speech production subsystems that may be impaired [31]. LSVT LOUD is unique in its intensive and high effort mode of treatment delivery. This mode is consistent with theories of motor learning and principles that drive activity-dependent neuroplasticity, both of which may be important for longterm carryover and generalization of improved vocal loudness to functional communication outside of the treatment room [32]. In addition, LSVT LOUD is unique because it addresses sensory kinesthetic and internal cueing deficits that can be barriers to carryover and generalization of treatment effects for people with Parkinson disease. People with Parkinson disease relearn a new internal cue for the amount of effort they need to produce for normal loudness and are recalibrated to their sensory perception of this loudness to recognize it is within normal limits [31,33]. The LSVT LOUD protocol has been described in detail in previously published research [34,35]. In brief, it consists of 16 individual 1-h sessions administered four times a week for 4 weeks and daily homework and carryover activities every day of
the month and is continued daily throughout life. The treatment incorporates materials that are salient to individual patients. This means that although the exercises and mode of delivery are consistent in all LSVT LOUD treatment sessions, speech materials are customized to address individual goals for improved vocal loudness in each patient’s daily functional communication.
RESEARCH OUTCOMES ON LSVT LOUD The initial randomized control trial (RCT) compared two treatments designed to improve loudness: one treatment focused on voice and the other treatment focused on increasing respiratory support for speech. Those treatments were matched for intensity of treatment within the treatment session (i.e. number of task repetitions, driving of motor effort, reinforcement from speech clinicians) and in treatment dosage including homework and carryover assignments. Evaluations were conducted immediately before treatment, immediately following treatment and at 6, 12 and 24 months after the completion of treatment. The primary outcome variable in this study was vocSPL. The data showed that intensive voice treatment (LSVT LOUD) was more effective in improving vocSPL than the respiratory treatment [36 ]. In another RCT, LSVT LOUD was compared with an untreated group of people with Parkinson disease and an age and sex-matched healthy control group. The groups were evaluated immediately before treatment, immediately after treatment and 6 months after treatment completion. The outcomes of this study also demonstrated statistically significant improvements in vocSPL for people who received LSVT LOUD compared with an untreated group. Across both of these studies, effect sizes were larger than 0.80 for the primary outcome variable of vocSPL indicating that the change was also clinically significant [37 ]. Importantly, increased vocal loudness was documented posttreatment with no evidence of increasing supraglottic hyperfunction, and in one study, a decrease in pretreatment supraglottic false fold and anterior posterior hyperfunction was documented post treatment [38]. Findings from a group study by Smith et al. [14] using laryngostroboscopic assessment documented improved glottal closure with no change in supraglottal tension following LSVT LOUD. These findings suggest that phonatory effort, which is targeted in LSVT LOUD, results in healthy vocal fold adduction without laryngeal hyperfunction. In contrast, de Swart et al. [39] claimed adverse effects of increasing loudness in Parkinson disease. These researchers asked patients to ‘speak
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loud/shout’ during one 30-min stimulability session and concluded that pressed voice and excessive strain accompanied LSVT LOUD. The use of LSVT LOUD or ‘LSVT’ in the de Swart et al. article is incorrect. Pressed voice and strain are never the behaviours elicited or trained during the 16 sessions of LSVT LOUD. Increased loudness is what the person with Parkinson disease may feel when increasing vocal effort; healthy, normal loudness is what
the speech clinician elicits and shapes in terms of actual vocal output. Extensive data at physiological, aerodynamic, acoustic and perceptual levels [6,14, 38,40,41] support the lack of any strain or hyperfunction accompanying increased vocal loudness in Parkinson disease. Data demonstrating associated treatment outcomes have been reported across a range of variables at perceptual, acoustic, aerodynamic, physiologic
Table 1. Summary of research outcome data following LSVT LOUD Primary Outcome Variable Acoustic: Improved vocSPL
•
Associated Outcome Variables Additional acoustic variables: Improved rate; slower
•
[33]Ramig, Countryman, Thompson, Horii, 1995
Improved F0 variation (Pitch); Reduced monotone
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[36]Ramig, Sapir, Countryman, et al, 2001; [43]Ramig, Bonitati, Lemke, Horii, 1994; [42]Whitehill, Kwan, Lee , Chow, 2011
Improved articulation; Larger vowel space area
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[7]Sapir, Spielman, Ramig, Story, Fox, 2007
Improved formant centralization ratio
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[44]Sapir, Ramig, Spielman, Fox, 2010
Improved articulatory acoustics Perceptual: Listener ratings Improvement in voice quality Less harsh and breathy
•
[45]Dromey, Ramig, Johnson, 1995
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[6]Baumgartner, Sapir, Ramig, 2001
Better voice quality
•
[46]Sapir, Ramig, Hoyt, et al, 2002
Improved speech intelligibility
•
[47]Cannito, Suiter, Beverly, et al, 2012
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[33]Ramig, Countryman, Thompson, Horii, 1995
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[48]Smith, Ramig, Dromey, et al, 1995; [38]Countryman, Hicks, Ramig, Smith, 1997
Electroglottography
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[49]Ramig, Dromey, 1996
More efficient swallow
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[50]El-Sharkawi, Ramig, Logemann, et al., 2002
Improvement in maximum capacity tongue function Aerodynamic: Improved subglottic air pressure
•
[51]Ward, Theodoros, Murdoch, Silburn, 2000
•
[49]Ramig, Dromey, 1996
Family and self ratings Improved ratings of communication characteristics Physiologic: Improved vocal fold adduction with no increase in laryngeal tension Direct visualization
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Data documenting treatment outcomes following LSVT LOUD [33]Ramig, Countryman, Thompson, Horii, 1995; [35]Ramig, Countryman, O’Brien, et al, 1996; [36]Ramig, Sapir, Countryman, et al, 2001; [37]Ramig, Sapir, Fox, Countryman, 2001; [42]Whitehill, Kwan, Lee , Chow, 2011. Data documenting treatment outcomes following LSVT LOUD
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Evidence-based treatment of voice and speech disorders in Parkinson disease Mahler et al. Table 1. (Continued) and MFDR Improved respiratory kinematics Facial expression: Improved frequency and variation of expressions Neural: Activation pattern changes in cortex and subcortical areas and rightsided shift in activation Treatment Accessibility
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[52]Huber, Stathopoulos, Ramig, Lancaster, 2003
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[53]Spielman, Borod, Ramig, 2003; [54]Dumer, Oster, et al., 2014
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[55]Liotti, Ramig, Vogel, et al., 2003; [56]Narayana, Fox, Zhang, et al., 2010
Data supporting alternate modes of treatment
Primary outcome variable Increased vocSPL: Telepractice LSVT eLOUD™
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[57]Theodoros, Constantinescu, Russell, et al, 2006; [58]Constantinescu G, Theodoros D, Russell T, et al, 2011; [59}Howell, Tripoliti, Pring, 2009; [60]Theodoros, Ramig, 2011
LSVT Companion Software
•
[61]Halpern, Ramig, Matos, et al., 2012
Alternate treatment dosage LSVT-X
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[62]Spielman, Ramig, Mahler, Halpern, Petska, 2007
and neural levels accompanying training increased vocSPL and are summarized in Table 1 [7,33,35, 36 ,37 ,42–53,54 ,55–57]. In addition, research studies examining treatment outcomes for people with atypical Parkinsonism [63,64] and those who have received deep brain stimulation [65,66] show a positive impact of LSVT LOUD on voice and speech variables, although these patients may be more challenging to treat [65].
clinician and seven using the LSVT Companion at home on their own. Outcome data demonstrated that all 16 participants had comparable gains in vocSPL immediately posttreatment and at 6 months follow-up as compared with data of people with Parkinson disease who received all 16 sessions in person.
MAKING LSVT LOUD MORE ACCESSIBLE
Today, LSVT LOUD is the most researched treatment for people with Parkinson disease and has been shown to be an effective behavioural treatment of voice and speech disorders associated with Parkinson disease. Speech clinicians must complete a training and certification course to administer the LSVT LOUD protocol. This training is required to maintain treatment fidelity of LSVT LOUD, thus allowing people with Parkinson disease to expect treatment outcomes similar to those documented in the research. As of publication of this article, there are over 14 000 clinicians certified in LSVT LOUD in 62 countries. Voice and speech symptoms have been reported to occur very early in the diagnosis of Parkinson disease and models of the physiology suggest that changes in the nervous system underlying voice and speech disorders begin long before the onset of symptoms [10–12,24 ]. Therefore, it is recommended that
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More recent research has been conducted to evaluate the incorporation of technology to make treatment more accessible to people with Parkinson disease. Several studies have examined the feasibility of telerehabilitation with LSVT LOUD and found that treatment outcomes were similar to administration of treatment wherein all sessions were conducted in person [57–60]. These studies support the feasibility and effectiveness of online administration of LSVT LOUD for people with Parkinson disease after an initial evaluation in person to determine candidacy for treatment. Another study was recently completed assessing the feasibility and effectiveness of an assistive technology system called the LSVT Companion, a U.S. Food and Drug Administration (FDA)-approved medical device [61 ]. People with Parkinson disease received nine LSVT LOUD sessions in person with a speech &
RECOMMENDATIONS FOR BEST CLINICAL PRACTICE
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people with Parkinson disease receive LSVT LOUD soon after diagnosis to obtain the maximum benefit from the treatment for the greatest possible length of time. It is further recommended that each patient receives a laryngeal evaluation by an otolaryngologist prior to initiation of treatment as part of best practice to confirm a diagnosis of voice changes secondary to Parkinson disease and eliminate the possibility of vocal fold disease other than that associated with Parkinson disease such as gastroesophageal reflux disease or unilateral vocal fold paralysis.
CONCLUSION The cumulative evidence from treatment efficacy studies of LSVT LOUD provides support for implementation of behavioural treatment of voice and speech deficits resulting from Parkinson disease. The studies reviewed in this article provide evidence for long-lasting effects of treatment following LSVT LOUD and the data demonstrate distributed effects of treatment to acoustic, perceptual, physiologic, aerodynamic, facial expression and neural control variables. The ability to communicate is essential to quality of life. Our future research will continue to work to improve treatment outcomes and understand the basic mechanisms accompanying treatment-related change. Acknowledgements The authors thank the patients who continue to inspire and challenge us and The National Center for Voice and Speech research team and the University of Colorado, Boulder. Financial support and sponsorship This research was funded by the National Institutes of Health (NIH) grants R01 DC1150, R21 RFA-NS-02006, R21 DC006078 and R21 NS043711 from the National Institutes of Deafness and Other Communication Disorders (NIDCD). Conflicts of interest Leslie A. Mahler receives lecture honoraria and travel reimbursement from LSVT Global, Inc. Lorraine O. Ramig and Cynthia M. Fox receive lecture honoraria and have ownership interest in LSVT Global, Inc. They have disclosed any conflict of interest and their conflict of interest management plan has been approved by their respective Offices of Conflict of Interest and Commitment at the University of Colorado, Boulder and the University of Rhode Island. The authors have no other relevant affiliations or financial involvement with any 214
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organization or entity with a financial interest in or financial conflict with the subject matter or materials discussed in the manuscript apart from those disclosed.
REFERENCES AND RECOMMENDED READING Papers of particular interest, published within the annual period of review, have been highlighted as: & of special interest && of outstanding interest 1. Schapira AH. Neurobiology and treatment of Parkinson’s disease. Trends Pharmacol Sci 2009; 30:41–47. 2. deLau LM, Breteler MM. Epidemiology of Parkinson’s disease. Lancet Neurol 2006; 5:525–535. 3. Ho A, Iansek R, Marigliani C, et al. Speech impairment in a large sample of people with Parkinson’s disease. Behav Neurol 1998; 11:131–137. 4. Miller N, Noble E, Jones D, Burn D. Life with communication changes in Parkinson’s disease. Age Ageing 2006; 35:235–239. 5. Ho A, Iansek R, Bradshaw JL. Motor instability in Parkinsonian speech intensity. Neuropsychiatry Neuropsychol Behav Neurol 2001; 14:109– 116. 6. Baumgartner C, Sapir S, Ramig LO. Voice quality changes following phonatory-respiratory effort treatment (LSVT1) versus respiratory effort treatment for people with Parkinson disease. J Voice 2001; 14:105–114. 7. Sapir S, Spielman J, Ramig LO, et al. Effects of intensive voice treatment (the Lee Silverman Voice Treatment [LSVT]) on vowel articulation in dysarthric individuals with idiopathic Parkinson disease: acoustic and perceptual findings. J Speech Lang Hear Res 2007; 50:899–912. 8. Forrest K, Weismer G, Turner G. Kinematic, acoustic and perceptual analysis of connected speech produced by Parkinsonian and normal geriatric adults. J Acoust Soc Am 1989; 85:2608–2622. 9. Duffy J. Motor speech disorders; substrates, differential diagnosis and management. (3rd ed. pp. 2013; 165–189. 10. Damier P, Hirsch EC, Agid Y, Graybiel AM. The substantia nigra of the human brain II. Patterns of loss of dopamine-containing neurons in Parkinson’s disease. Brain 1999; 122:1437–1448. 11. Ahlskog J. Beating a dead horse: dopamine and Parkinson disease. Neurology 2007; 69:1701–1711. 12. Braak H, Ghebremedhin E, Rub U, et al. Stages in the development of Parkinson’s disease-related pathology. Cell Tissue Res 2004; 318:121– 134. 13. Perez K, Ramig LO, Smith M, Dromey C. The Parkinson larynx: tremor and videostroboscopic findings. J Voice 1996; 10:354–361. 14. Smith M, Ramig LO, Dromey C, et al. Intensive voice treatment in Parkinson’s disease: laryngostroboscopic findings. J Voice 1995; 9:453–459. 15. Baker K, Ramig LO, Luschei E, Smith M. Thyroarytenoid muscle activity associated with hypophonia in Parkinson disease and aging. Neurology 1998; 51:1592–1598. 16. Vincken W, Gauthier SG, Dollfuss RE, et al. Involvement of upper-airway muscles in extrapyramidal disorders, a cause of airflow limitation. N Engl J Med 1984; 7:438–442. 17. Solomon N, Hixon TJ. Speech breathing in Parkinson’s disease. J Speech Hear Res 1993; 36:294–310. 18. Bartels AL, Leenders KI. Parkinson’s disease: the syndrome, the pathogenesis and pathophysiology. Cortex 2009; 45:915–921. 19. Berardelli A, Rothwell JC, Thompson PD, Hallett M. Pathophysiology of bradykinesia in Parkinson’s disease. Brain 2001; 124:2131–2146. 20. Sapir S, Ramig LO, Fox CM. Intensive voice treatment in Parkinson’s disease: Lee Silverman Voice Treatment. Expert Rev 2011; 11:815–830. 21. Desmurget M, Grafton ST, Vindras P, et al. The basal ganglia network mediates the planning of movement amplitude. Exp Brain Res 2003; 150:197–209. 22. Schneider S, Diamond SG, Markham DH. Deficits in orofacial sensorimotor function in Parkinson’s disease. Ann Neurol 1986; 19:275–282. 23. Gallena S, Smith PJ, Zeffro T, Ludlow CL. Effects of levodopa on laryngeal muscle activity for voice onset and offset in Parkinson disease. J Speech Hear Res 2001; 44:1284–1299. 24. Arnold C, Gehrig J, Gispert S, et al. Pathomechanisms and compensatory & efforts related to Parkinsonian speech. Neuroimage Clin 2014; 4:82–97. This article identifies a pathological interplay between the limbic and sensorimotor striatum that could interfere with sensory feedback and modulation of speech routines. 25. Sapir S. Multiple factors are involved in the dysarthria associated with && Parkinson’s disease: a review with the implications for clinical practice and research. J Speech Lang Hearing Res 2014; 57:1330–1343. This work describes multiple factors that cause speech and voice disorders in Parkinson’s disease and provides a rationale for the LSVT LOUD protocol. 26. DeLetter M, Santens P, VanBorsel J. The effects of levodopa on word intelligibility in Parkinson’s disease. J Commun Disord 2005; 38:187–196.
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Evidence-based treatment of voice and speech disorders in Parkinson disease Mahler et al. 27. Goberman A, Coelho C, Robb M. Phonatory characteristics of Parkinsonian speech before and after morning medication: the ON and OFF states. J Commun Disord 2002; 35:217–239. 28. Pinto S, Ozsancak C, Tripoliti E, et al. Treatments for dysarthria in Parkinson’s disease. Lancet Neurol 2004; 3:547–556. 29. Pinto S, Gentil M, Krack P, et al. Changes induced by levodopa and subthalamic nucleus stimulation on Parkinsonian speech. Mov Disord 2005; 20:1507–1515. 30. Ramig LO, Mead C, Scherer R, et al. Voice therapy and Parkinson’s disease: a longitudinal study of efficacy. Presented at: The Clinical Dysarthria Conference. San Diego, CA 1988; 4–7; February. 31. Fox C, Morrison C, Ramig L, Sapir S. Current perspectives on the Lee Silverman Voice Treatment (LSVT). Am J Speech Lang Path 2002; 11:111–123. 32. Maas E, Robin DA, Hula SNA, et al. Principles of motor learning in treatment of motor speech disorders. J Speech-Lang Path 2008; 17:277–298. 33. Ramig LO, Countryman S, Thompson L, Horii Y. A comparison of two forms of intensive speech treatment for Parkinson disease. J Speech Hear Res 1995; 38:1232–1251. 34. Fox C, Ebersbach G, Ramig L, Sapir S. LSVT LOUD and LSVT BIG: behavioral treatment programs for speech and body movement in Parkinson disease. Parkinson’s Dis 2012; 2012:391946. 35. Ramig LO, Countryman S, O’Brien C, et al. Intensive speech treatment for patients with Parkinson disease: short and long-term comparison of two techniques. Neurology 1996; 47:1496–1504. 36. Ramig L, Sapir S, Countryman S, et al. Intensive voice treatment (LSVT1) for && individuals with Parkinson disease: a two-year follow-up. J Neurol Neurosurg Psychiatry 2001; 71:493–498. This study provides data from a RCT comparing treatment outcomes for LSVT LOUD and respiratory treatment. 37. Ramig L, Sapir S, Fox C, Countryman S. Changes in vocal intensity following && intensive voice treatment (LSVT1) in individuals with Parkinson disease: a comparison with untreated patients and age-matched controls. Mov Disord 2001; 16:79–83. This study provides data from a RCT comparing treatment outcomes for LSVT LOUD, untreated individuals with Parkinson disease and a healthy control group. 38. Countryman S, Hicks J, Ramig LO, Smith M. Supraglottal hyperadduction in an individual with Parkinson disease: a clinical treatment note. Am J Speech Lang Path 1997; 6:74–84. 39. de Swart BJM, Willemse SC, Maassen BAM, Horstink MWIM. Improvement of voicing in patients with Parkinson’s disease by speech therapy. Neurology 2003; 60:498–500. 40. Tjaden K, Wilding GE. Rate and loudness manipulations in dysarthria: acoustic and perceptual findings. J Speech Lang Hearing Res 2004; 47:766–783. 41. Huber J, Darling M. Effect of Parkinson’s disease on the production of structured and unstructured speaking tasks: respiratory physiologic and linguistic considerations. J Speech Lang Hearing Res 2011; 54:33–46. 42. Whitehill TL, Kwan L, Lee FP, Chow MM. Effect of LSVT on lexical tone in speakers with Parkinson’s disease. Parkinson’s Dis 2011; 2011:897494. 43. Ramig L, Bonitati CM, Lemke JH, Horii Y. Voice treatment for patients with Parkinson disease: development of an approach and preliminary efficacy data. J Med Speech Lang Path 1994; 2:191–209. 44. Sapir S, Ramig LO, Spielman JL, Fox C. Formant centralization ratio: a proposal for a new acoustic measure of dysarthric speech. J Speech Lang Hear Res 2010; 53:114–125. 45. Dromey C, Ramig LO, Johnson A. Phonatory and articulatory changes associated with increased vocal intensity in Parkinson disease: a case study. J Speech Hear Res 1995; 38:751–764. 46. Sapir S, Ramig L, Hoyt P, et al. Speech vocSPL and quality 12 months after intensive voice treatment (LSVT) for Parkinson’s disease: a comparison with an alternative speech treatment. Folia Phoniatrica 2002; 54:296–303.
47. Cannito MP, Suiter DM, Beverly D, et al. Sentence intelligibility before and after voice treatment in speakers with idiopathic Parkinson’s disease. J Voice 2012; 26:214–219. 48. Smith M, Ramig LO, Dromey C, et al. Intensive voice treatment in Parkinson disease: laryngostroboscopic findings. J Voice 1995; 9:453–459. 49. Ramig L, Dromey C. Aerodynamic mechanisms underlying treatment-related changes in vocal intensity in patients with Parkinson disease. J Speech Hear Res 1996; 39:798–807. 50. El-Sharkawi A, Ramig LO, Logemann JA, et al. Swallowing and voice effects of Lee Silverman Voice Treatment (LSVT): a pilot study. J Neurol Neurosurg Psychiatry 2002; 71:31–36. 51. Ward EC, Theodoros DG, Murdoch BE, Silburn P. Changes in maximum capacity tongue function following the Lee Silverman Voice Treatment Program. J Med Speech Lang Path 2000; 8:331–335. 52. Huber JE, Stathopolous ET, Ramig LO, Lancaster SL. Respiratory function and variability in individuals with Parkinson disease: pre and post-Lee Silverman Voice Treatment. J Med Speech Lang Path 2003; 11:185–201. 53. Spielman J, Borod J, Ramig L. Effects of intensive voice treatment (LSVT) on facial expressiveness in Parkinson’s disease: preliminary data. Cog Behav Neuro 2003; 16:177–188. 54. Dumer AL, Oster H, McCabe D, et al. Effects of the Lee Silverman Voice & Treatment on facial expressivity in Parkinson’s disease. J Int Neuropsych Soc 2014; 20:302–312. This article provides evidence for associated changes in facial movement following LSVT LOUD. 55. Liotti M, Ramig LO, Vogel D, et al. Hypophonia in Parkinson’s disease. Neural correlates of voice treatment revealed by PET. Neurology 2003; 60:432–440. 56. Narayana S, Fox PT, Zhang W, et al. Neural correlates of efficacy of voice therapy in Parkinson’s disease identified by performance-correlation analysis. Hum Brain Map 2010; 31:222–236. 57. Theodoros D, Constantinescu G, Russell TG, et al. Treating the speech disorder in Parkinson’s disease online. J Telemed Telecare 2006; 12 (Suppl 3): 88–91. 58. Constantinescu G, Theodoros D, Russell T, et al. Treating disordered speech and voice in Parkinson’s disease online: a randomized controlled noninferiority trial. Intl J Lang Comm Disord 2011; 46:1–16. 59. Howell S, Tripoliti E, Pring T. Delivering the Lee Silverman Voice Treatment (LSVT) by web camera: a feasibility study. Int J Lang Comm Disord 2009; 44:287–300. 60. Theodoros E, Ramig O. Telepractice supported delivery of LSVT LOUD. Sem Speech Lang Path 2011; 21:107–119. 61. Halpern E, Ramig O, Matos E, et al. Innovative technology for the assisted & delivery of intensive voice treatment (LSVT LOUD) for Parkinson disease. Am J Sp-Lang Path 2012; 21:354–367. This study provides evidence for the integration of technology in the delivery of LSVT LOUD with treatment outcomes similar to treatment in person thus increasing accessibility of treatment to patients with Parkinson disease. 62. Spielman J, Ramig L, Mahler L, et al. Effects of LSVT extended (LSVT-X) on voice and speech in Parkinson disease. Am J Speech Lang Path 2007; 16:95–107. 63. Countryman S, Ramig LO. Effects of intensive voice therapy on voice deficits associated with bilateral thalamotomy in Parkinson’s disease: a case study. J Med Speech Lang Path 1993; 1:233–250. 64. Countryman S, Ramig LO, Pawlas A. Speech and voice deficits in Parkinsonian plus syndromes: can they be treated? J Med Speech Lang Path 1994; 2:211–225. 65. Spielman J, Mahler L, Halpern A, et al. Intensive voice treatment (LSVT LOUD) for Parkinson’s disease following deep brain stimulation of the subthalamic nucleus. J Comm Dis 2011; 44:688–700. 66. Tripoliti E, Zrinzo L, Martinez-Torres I, et al. Effects of subthalamic stimulation on speech of consecutive patients with Parkinson disease. Neurology 2011; 76:80–86.
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