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976267

review-article2020
CRE0010.1177/0269215520976267Clinical RehabilitationMuñoz-Vigueras et al.

CLINICAL
Systematic Review REHABILITATION

Clinical Rehabilitation

Speech and language therapy 1­–17


© The Author(s) 2020
Article reuse guidelines:
treatment on hypokinetic sagepub.com/journals-permissions
DOI: 10.1177/0269215520976267
https://doi.org/10.1177/0269215520976267

dysarthria in Parkinson journals.sagepub.com/home/cre

disease: Systematic review


and meta-analysis

Natalia Muñoz-Vigueras , Esther Prados-Román ,


Marie Carmen Valenza , Maria Granados-Santiago,
Irene Cabrera-Martos , Janet Rodríguez-Torres
and Irene Torres-Sánchez

Abstract
Objective: To assess the effect of speech and language therapy (SLT) on Hypokinetic dysarthria (HD)
in Parkinson’s disease.
Design: Systematic review and meta-analysis of randomized controlled trials.
Methods: We performed a literature search of randomized controlled trials using PubMed, Web of
Science, Science Direct and Cochrane database (last search October 2020). Quality assessment and risk
of bias were assessed using the Downs and Black scale and the Cochrane tool. The data were pooled and
a meta-analysis was completed for sound pressure levels, perceptual intelligibility and inflection of voice
fundamental frequency.
Results: We selected 15 high to moderate quality studies, which included 619 patients with Parkinson’s
disease. After pooling the data, 7 studies, which compared different speech language therapies to no
treatment, control groups and 3 of their variables, (sound pressure level, semitone standard deviation and
perceptual intelligibility) were included in the analysis.
Results showed significant differences in favor of SLT for sound pressure level sustained phonation
tasks (standard mean difference = 1.79; 95% confidence interval = 0.86, 2.72; p ⩽ 0.0001). Significant
results were also observed for sound pressure level and semitone standard deviation in reading tasks
(standard mean difference = 1.32; 95% confidence interval = 1.03, 1.61; p ⩽ 0.0001). Additionally, sound
pressure levels in monologue tasks showed similar results when SLT was compared to other treatments
(standard mean difference = 0.87; 95% confidence interval = 0.46, 1.28; p ⩽ 0.0001).

Department of Physiotherapy, Faculty of Health Sciences, Corresponding author:


University of Granada, Granada, Spain Marie Carmen Valenza, Department of Physiotherapy,
Faculty of Health Sciences, University of Granada, Av. De la
Ilustración, 60, Granada, 18016, Spain.
Email: cvalenza@ugr.es
2 Clinical Rehabilitation 00(0)

Conclusion: This meta-analysis suggests a beneficial effect of SLT for reducing Hypokinetic Dysarthria
in Parkinson’s disease, improving perceptual intelligibility, sound pressure level and semitone standard
deviation.

Keywords
Parkinson’s disease, language disorders, Hypokinetic Dysarthria, speech language therapy, Lee Silverman
Voice Treatment

Received: 14 January 2020; accepted: 4 November 2020

Introduction
More than 89% of individuals with Parkinson’s dis- evidence was mainly due to the small number of
ease suffer from speaking and swallowing issues examined patients, methodological flaws, and the
during the course of their illness.1 Among those possibility of publication bias. Therefore, the study
impairments, a distinctive perceptual motor speech fails to analyze specifically designed programs for
disorder associated with basal ganglia control cir- Hypokinetic Dysarthria. The effect should be rele-
cuit pathology, called Hypokinetic Dysarthria, is vant for everyday life and lead to long-term
the most frequent one.2 This may appear in all of the improvement,12 but the reported effectiveness,
respiratory, phonatory, resonatory, articulatory and components and duration of the published speech
prosody levels of speech, but its characteristics are and language therapy studies are scarce. Thus, the
most evident in voice, articulation and prosody.3 main objective in this study is to systematically
This specific speech disorder4 is characterized review the results of specific speech and language
by reduced loudness,5 imprecise consonants, vowel therapy programs for Hypokinetic Dysarthria.
centralization, and rate changes6 accompanied by Additionally, a metanalysis will be performed in
involuntary facial movements.7 order to analyze the results found among the differ-
The Hypokinetic Dysarthria deleterious effects ent programs.
on patient’s communication and social participa-
tion may lead to social isolation, reducing the qual-
Methods
ity of life.8
Speech and language therapy in Hypokinetic This review has been written in accordance with the
Dysarthria aims to improve the intelligibility of Preferred Reporting Items for Systematic Reviews
speech with behavioral treatment techniques and and Meta-Analyses (PRISMA) Statement.13 The
instrumental aids.9 Behavioral techniques and review has been registered at the International
instrumental aids pursue the modification of behav- Prospective Register of Systematic Review (PROS
ior, influencing one or several speech motor func- PERO) with registration number: CRD4201911
tions, as well as linguistic or communicative 9605.
behavior, with the possible inclusion of specific An electronic search was conducted using three
devices and/or feedback instruments.10 electronic databases: PudMed, Web of Science,
Speech and language therapy has already been Science Direct and Cochrane database of rand-
seen to be an integral component of treatment in omized controlled trials. The terms used can be
Parkinson’s disease. However, the most recent seen in the online appendix. Relevant publications
review in 201511 concluded that there was insuffi- were included from inception until 9 October 2020.
cient evidence to conclusively support or refute the To define the research question, the PICOS14
efficacy of any form of speech and language ther- (Participants, Interventions, Comparisons, Outcome
apy to treat Hypokinetic Dysarthria. The lack of and Study design) model was applied. The inclusion
Muñoz-Vigueras et al. 3

criteria were: (1) patients with Parkinson’s disease; March 2011). Because of the clinical heterogene-
(2) speech and language therapy interventions ity of the included studies, meta-analysis was lim-
focused on dysarthria; (3) the speech and language ited. The I2 statistic was utilized to determine the
therapy intervention had to be compared to a con- degree of heterogeneity, where the percentages
trol intervention or no-treatment; (4) Motor speech quantified the magnitude of heterogeneity:
control was included in the outcomes; (5) only ran- 25% = low, 50% = medium, and 75% = high hetero-
domized clinical trials were included. Full texts in geneity. Using this scale, if I2 was 50%, a random
English were included. effects model was used. All the included outcomes
After the records were obtained from the differ- were of continuous data, Sound Pressure Level,
ent databases, duplicates were removed. Then, two Semitones Standard Deviation and perceptual
reviewers (N.M. and I.T.) performed independent intelligibility and the mean difference with 95%
evaluations of the titles and abstracts of all obtained confidence interval (CI) was used in the analysis.
papers and further reviewed the studies to ensure Forest plots were generated to illustrate the overall
eligibility. All disagreement or differences in crite- effect of interventions.
ria where resolved by a third reviewer (E.P).
When the articles were selected, we extracted
Results
the data and performed a quality assessment. The
methodological quality of the included studies was The search results and the final included studies are
assessed using the Downs and Black quality assess- shown in Figure 1.
ment method.15 It consists of 27 elements with five A total of 619 participants with Parkinson’s dis-
subscales (study quality, external validity, study ease and clinical diagnosis of Hypokinetic
bias, confounding and selection bias, and study Dysarthria were recruited. Study design, clinical
power). A study is considered excellent when it characteristics of patients and main outcomes, for-
reaches a score between 26 and 28 points, good mal measures and quality scores are detailed in
between 20 and 25, fair between 15 and 19, and Supplementary Table 1.
poor when it is less or equal to 14. This scale has The severity of the disease in the majority of the
been ranked as one of the six highest quality assess- cases was mild to moderate as measured by the
ment scales suitable for use in systematic reviews, Hoehn-Yahr scale20 and the range of time since
due to its high validity and reliability.16,17 diagnosis was from 4.77 ± 3.021 to 12.5 ± 7.422
The risk of bias was assessed using the Cochrane years.
Risk of Bias Tool for Randomized Controlled The most frequent outcomes were loudness
Trials method.18 It consists of seven elements with measured by sound pressure level22–29 and pitch,
six subscales (selection bias, performance bias, measured by semitone standard deviation.25,27
detection bias, attrition bias, reporting bias and The quality assessment using the Downs and
other bias). It is considered that a study is of high Black scale shows high quality for 722,27,29–33 of the
quality when there is low risk for each domain. Fair included studies and fair for 821,24–29,34,35 of the
quality19 when one criterion does not meet (i.e. studies. However, when Cochrane Risk of bias
high risk of bias for one domain) or two criteria are Assessment was applied, the risk of bias shown is
unclear, and there is no known important limitation generally low.
that could invalidate the results. Poor quality, when Table 1 shows intervention characteristics and
one criterion does not meet or two criteria are main conclusions obtained for each included study.
unclear, and there are important limitations that Among the included studies, five studies23,24,28,33,34
could invalidate the results; and when two or more compared speech and language therapy to no treat-
criteria are listed as high or unclear risk of bias. ment; and five studies21,25–27,31 compared speech and
When possible, study results were pooled and a language therapy to control treatment (Respiratory
meta-analysis was undertaken using Review effort treatment). Additionally, four studies29,30,32,35
Manager Software (RevMan version 5.1, updated included three groups, (Two different groups of
4 Clinical Rehabilitation 00(0)

Identification Records identified through database searching:


Pub Med = 94; Web of Science = 538;
ScienceDirect= 118; Cochrane data base=16

Studies
identified
from
other
sources Total of articles
=1
(n = 766)
Screening

Duplicated articles
(n = 168)

Total of elegible articles

(n = 598)

Excluded articles (n = 434),


reason for exclusion:
Elegibility

Title and abstract not related to


the topic
Full text articles analyzed
(n = 164)

Excluded articles (n=150), reason for


exclusion:

Review: n=59; Quasiexperimental:


n=71; Case report: n=10; Others non
Included articles
RCT: n= 10
(n = 14)
Included

Excluded articles from meta-analysis


Studies included in
(n=8), reason for exclusion:
qualitative analysis:
Lack of agreement in variables used to
(n= 15)
evaluate outcomes

Studies included in
quantitative analysis: (n=7)

Figure 1. Flowchart.
Muñoz-Vigueras et al. 5

Table 1.  Characteristics of interventions.

Authors Interventions (n) Duration of intervention Main conclusions


(year)
Program duration (weeks);
Session duration (minutes);
Frequency (days per week)
Robertson Standard-SLT (Breathing; 2; 210–240; 5 Standard SLT group improved significantly
and phonation; resonance; all measured variables after intervention and
Thomson34 articulation; prosody; remains at follow up.
facial muscles): (n = 12) Between groups analysis post-intervention
No treatment: (n = 6) – showed significant differences in favor of
Standard SLT group that remains at
follow up.
Johnson and Standard-SLT (breathing; 4; 60; 2–3 Standard SLT group improved all measured
Pring24 phonation, resonance; variables significantly after intervention and
articulation; prosody): remains at follow up.
(n = 6) Between groups analysis post-intervention
No treatment: (n = 6) – showed significant differences in favor of
Standard SLT group that remains at
follow up.
Ramig et al.25 LSVT: (n = 26) 4; 50; 4 Both groups improved in all measured
Respiratory treatment: 4; 50; 4 variables during intervention.
(n = 18) LSVT group improved significantly in SPL
subscales, F0, impact profile and family and
self-perceived ratings. RET group improved
significantly in SPL subscales and self-ratings.
After intervention, LSVT group showed
higher results in all measured outcomes
being significantly better in F0 rainbow and
SIP, when compared to RET group.
Ramig and LSVT: (n = 26) 4; 60; 4 LSVT group improved significantly all
Dromey26 Respiratory treatment: 4; 60; 4 measures during treatment.
(n = 19) RET group showed no significant differences
after treatment.
Between group comparisons showed
significant differences in all variables
measured in favor of LSVT group.
Ramig et al.27 LSVT: (n = 21) 4; 60; 4 LSVT group improved significantly all
Respiratory 4; 60; 4 variables after intervention and are
treatment:(n = 12) maintained at follow up.
RET group improved significantly SPL
and STSD rainbow subscores that are
not maintained at follow up. Differences
between groups were found in favor of
LSVT group and maintained at follow up.
Ramig et al.28 LSVT: (n = 14) 4; 60; 4 LSVT group improved significantly in SPL
No treatment:(n = 15) – subscales. There were no significantly
differences between post-intervention and
at follow up in LSVT group.
Between groups analysis showed significant
differences in favor of LSVT group after
intervention and at follow up.
(Continued)
6 Clinical Rehabilitation 00(0)

Table 1. (Continued)

Authors Interventions (n) Duration of intervention Main conclusions


(year)
Program duration (weeks);
Session duration (minutes);
Frequency (days per week)
Baumgartner LSVT: (n = 13) 4; 60; 4 LSVT improved significantly all variables
et al.31 Respiratory treatment 4; 60; 4 after intervention.
(n = 7) RET group showed no differences after
intervention.
LSVT group showed significant differences
in all variables measured compared to RET
group after intervention and follow up.
Sapir et al.21 LSVT: (n = 22) 4; 60; 4 LSVT improved significantly all variables
Respiratory treatment 4; 50; 4 after intervention.
(n = 13) RET group showed no differences after
intervention.
LSVT group showed statistically significant
differences in all variables measured
compared to RET group after intervention
and follow up.
Sapir et al.23 LSVT: (n = 14) 4; 50–60; 4 LSVT group showed significant changes in
No treatment: (n = 15) – SPL, F2u, F2i/F2u and perception of quality
of vowel after intervention.
Between groups analysis post-intervention
showed significant differences in favor of
LSVT group.
Dumer LSVT: (n = 16) 4; 60; 4 LSVT and LSVT artic group improved all
et al.35 ARTIC: (n = 12) 4; 60; 4 variables significantly after intervention.
No treatment: (n = 17) – Comparisons between groups after
intervention showed significant differences
in favor of LSVT groups compared to
untreated group.
LSVT group showed significantly higher
values in the majority of variables after
intervention when compared to LSVT artic
group, with similar results in happiness sub
score.
Sackley LSVT: (n = 30) 4; 50–60; 4 Both intervention groups showed significant
et al.32 Standard-SLT: 6–8; 45; 1 improvements in all variables.
(Breathing; phonation; LSVT group and Standard-SLT group
resonance; articulation; showed significant differences in all variables
prosody): (n = 30) measured after intervention in favor of
No treatment: (n = 29) – LSVT group.
At follow up those differences remains.
Ramig et al.29 LSVT: (n = 22) 4; 60; 4 LSVT group showed improvements in all
LSVT ARTIC: (n = 20) 4; 60; 4 variables after intervention.
No treatment: (n = 22) – LSVT artic group showed no differences
after intervention. Between groups
comparisons showed significant differences
between groups in favor of LSVT group, that
last at follow up.
(Continued)
Muñoz-Vigueras et al. 7

Table 1. (Continued)

Authors Interventions (n) Duration of intervention Main conclusions


(year)
Program duration (weeks);
Session duration (minutes);
Frequency (days per week)
Saffarian LSVT: (n = 13) 4; 60; 4 LVST group showed significantly differences
et al.33 No treatment: (n = 10) – in the perception of loudness and quality of
voice after intervention and follow up.
Significant differences were found between
groups after intervention and at follow up in
all measured variables in favor of LSVT.
Tamplin Parkinsong (vocal 52; 180; NR Both intervention groups showed significant
et al.22 warm-ups, exercises improvements in all variables.
to develop respiratory Parkinsong weekly group were significantly
strength and control, louder than the other 3 groups.
vocal loudness, Significant differences were found after
articulation, pitch Parkinsong weekly intervention.
control, communication At 12 months follow up those differences
confidence activities) decreased but remained significantly higher
Weekly sessions: (n = 20) than baseline.
Control (dancing, 52; 180; NR
painting, tai chi or peer
support groups) Weekly
sessions: (n = 27)
Parkinsong (vocal 52; 180; 1
warm-ups, exercises
to develop respiratory
strength and control,
vocal loudness,
articulation, pitch
control, communication
confidence activities)
Monthly sessions:
(n = 15)
Control (dancing, 52; 180; 1
painting, tai chi or peer
support groups) Weekly
Monthly sessions:
(n = 13)
Levy et al.30 Voice treatment (focus 4; 60; 4 Voice and articulation treatment group
on loudness; respiratory- increased significantly TA after intervention.
laryngeal systems): Between groups, the increases in the voice
(n = 19) group were significantly greater than those
Articulation treatment 4; 60; 4 for both the articulation group and the no
(focus on enunciation; treatment group.
orofacial–articulatory Differences in TA changes between the
system): (n = 19) articulation group and the no treatment
No treatment: (n = 19) – group were not significant.

SLT: Speech and language therapy; CG: Control group; PD: Parkinson’s disease; EI: Experimental Intervention; CI: Control
Intervention; LSVT: Lee Silverman Voice Treatment; ARTIC: Articulation; RET: Respiratory effort treatment; F2u: Second formant
vowel u; F2i: Second formant vowel i; NR: No reported.
8 Clinical Rehabilitation 00(0)

speech and language therapies and a non-treatment analyzed to obtain concrete results on the charac-
group). teristics of the treatment. To acquire the sound
The study of Tamplin et al.22 compared four pressure level, we used three subtasks: the sus-
groups, two of them with the same SLT interven- tained phonation, the reading and the monologue
tion with different frequencies. One group received as shown in Figures 2–5. The results on the stand-
treatment once a week and the other one received ard deviation of semitones were obtained by
treatment once per month. There were also two using the reading and monologue subtasks and
respective control groups. can be seen in Figures 6 and 7.
Feedback during speech and language therapy For the sustained phonation subtask, the pooled
interventions was used in eight21,24–27,30,31 of the mean difference (MD) showed significant overall
studies. effect of speech and language therapy: standard
The most commonly applied method was Lee speech and language therapy, Lee Silverman Voice
Silverman’s Voice Treatment in different modali- Treatment Loudness, Lee Silverman Voice Treatment
ties (Loudness and Articulation). All the studies Articulation, ParkinSong Weekly and ParkinSong
carried out the intervention using an individual Monthly; when compared to no intervention
approach, except the one by Robertson and (MD = 1.11; 95% CI = -0.30, 2.52; p = 0.0003), or to a
Thomson that used a group dynamic.32 comparison group: Respiratory Treatment, Control
All the results obtained were in favor of experi- treatment Weekly and Control treatment Monthly
mental interventions. When speech and language (MD = 2,24; 95% CI = 0.97; 3.51; p ⩽ 0.0001).
therapy was compared to non-treatment, signifi- Heterogeneity was high (I2 = 91.28%; I2 = 90.01%)
cant improvements were found. These improve- respectively. Compared to no intervention with the
ments were maintained at the follow-up in all comparison group: (MD = 1.79; 95% CI = 0.86; 2.72;
measured outcomes.24,34 p ⩽ 0.0001). Heterogeneity was high (I2 = 90.54%).
In the case of the studies comparing speech and When the reading subtask was analyzed, the
language therapy with a control group, speech and effect of speech and language therapy was further
language therapy groups showed better results in examined by pooling the data. The mean difference
all outcomes that were maintained at follow up. (MD) shows significant overall effect of speech and
This positive effect lead to a general improvement language therapy: standard speech and language
in communication and dysarthria. therapy, Lee Silverman Voice Treatment Loudness
Especially for loudness and pitch variables, and Lee Silverman Voice Treatment Articulation;
when speech and language therapy treatment was when compared to no intervention (MD = 0.99;
compared with the control group, the results were 95% CI = 0.42, 1.56; p < 0.0001) and comparison
in favor of speech and language therapy after treat- group: Respiratory Treatment (MD  = 
1.62; 95%
ment and at follow up. CI = 1.15, 2.09; p <  0.0001) Heterogeneity was
Detailed data about included aspects of dysar- (I2 = 52.72%; I2 = 0%) respectively. Compared to
thria in the SLT interventions of each included no intervention with comparison group: (MD = 1.32;
study (slurred speech, impaired muscle tone, lack of 95% CI = 1.03; 1.61; p ⩽ 0.0001). Heterogeneity
coordination and hypophonia) were included as was low (I2 = 0%).
supplementary material (see supplementary data When the Monologue subtask was analyzed, the
Table 1). effect of speech and language therapy was further
examined by pooling the data.
For the monologue subtask the mean difference
Results obtained in meta-analysis
(MD) shows significant overall effect of speech and
Results obtained in loudness and pitch characteris- language therapy: standard speech and language
tics have been analyzed across different subtasks therapy, Lee Silverman Voice Treatment Loudness,
(sustained phonation, reading and monologue). Lee Silverman Voice Treatment Articulation and
The measurements of the sound pressure level ParkinSong Weekly and ParkinSong Monthly;
and the standard deviation of semitones were when compared to no intervention (MD = 1.07; 95%
Muñoz-Vigueras et al.

Study or Treatment group Comparison group Weight Mean difference dB Mean difference dB
subgroup Mean SD Total Mean SD Total % IV, Random, 95% CI IV, Random, 95% CI
Speech language therapy (Lee Silverman Voice Treatment Loudness) vs No treatment
Ramig 2001 82.4 3.9 14 70.5 4.4 15 25.66 2.78 [1.76, 3.80]
Ramig 2018 84.3 10.6 22 76.1 8.3 22 38.94 0.85 [0.23, 1.46]
Speech language therapy (Lee Silverman Voice Treatment Articulation) vs No treatment
Ramig 2018 75.2 7.9 20 76.1 8.3 20 35,40 -0.11[-0.71, 0.50]
Subtotal (95%CI) 56 57 100 1.11[-0.30, 2.52]
Heterogeneity:Tau2=1.3990,Chi2= 11.47, df= 2 (P= <0.0001 ), I2= 91.28%
Test for overall effect: Z= 3.5837 (P= 0.0003)
Speech language therapy (Lee Silverman Voice Treatment Loudness) vs Comparison group
Ramig 1995 81.2 4.6 26 68.0 4.3 19 26.47 2.87 [2.03, 3.71]
Ramig 1996 81.7 3.1 10 65.2 4.2 7 10 4.37 [2.61, 6.13]
Ramig 2001 82.36 3.9 21 68.7 4.8 12 19.41 3.14 [2.10, 4.18]
Speech language therapy (Parkinsong) vs Comparison group
Tamplin 2020 W 90,08 10,1 20 80,8 9,4 15 20.58 0.92 [0.22, 1.62]
Tamplin 2020 M 80,56 11,6 27 74,5 10,1 13 23.59 0.53 [-0.14, 1.21]
Subtotal (95%CI) 104 66 100 2.24 [0.97, 3.51]
Heterogeneity:Tau2=1.8124,Chi2= 10.01, df= 4 (P= <0.0001 ), I2= 90.01%
Test for overall effect: Z= 8.5421 (P= <0.0001)
Heterogeneity:Tau2=1.5610,Chi2= 10.57, df= 7 (P= <0.0001 ), I2= 90.54%
Test for overall effect: Z= 8.6518 (P= <0.0001)

Figure 2.  Meta-analysis; sustained phonation; sound pressure level.


9
10

Study or Treatment group Comparison group Weight Mean difference dB Mean difference dB
subgroup Mean SD Total Mean SD Total % IV, Random, 95% CI IV, Random, 95% CI
Standard speech language therapy vs No treatment
Johnson 1990 76.5 6.3 6 70.1 2.4 6 9.68 1.24 [0.00, 2.47]
Speech language therapy (Lee Silverman Voice Treatment Loudness) vs No treatment
Ramig 2001 77.9 4.2 14 70.5 4.4 14 22.58 1.67 [0.82, 2.52]
Ramig 2018 76.8 6.2 22 71.3 4.8 22 35.48 0.97 [0.35, 1.60]
Speech language therapy (Lee Silverman Voice Treatment Articulation) vs No treatment
Ramig 2018 73.2 5.1 20 71.3 4.8 20 32.26 0.38 [-0.23, 0.99]
Subtotal (95%CI) 62 62 100 0.99 [042, 1.56]
Heterogeneity:Tau2=0.1713,Chi2= 2.11, df= 3 (P= 0.0960), I2= 52.72%
Test for overall effect: Z= 4.8273 (P= <.0001)
Speech language therapy (Lee Silverman Voice Treatment Loudness) vs Comparison group
Ramig 1995 74.33 4.83 26 68.16 3.33 19 47.37 1.42 [0.76, 2.08]
Ramig 1996 74.2 3.5 10 66.9 3.9 7 17.89 1.89 [0.73, 3.05]
Ramig 2001 75.31 4.22 21 68.03 3.36 12 34.74 1.80 [0.97, 2.64]
Subtotal (95%CI) 57 38 100 1.62 [1.15, 2.09]
Heterogeneity:Tau2=0, Chi2= 1, df= 2 (P=0.6880), I2= 0.00%
Test for overall effect: Z=6.7347 (P= <.0001)
Heterogeneity:Tau2=0, Chi2= 0.89, df= 6 (P=0.4994), I2= 0.00%
Test for overall effect: Z=8.8469 (P= <.0001)

Figure 3.  Meta-analysis; reading; sound pressure level.


Clinical Rehabilitation 00(0)
Muñoz-Vigueras et al.

Study or subgroup Treatment group Comparison group Weight Mean difference dB Mean difference dB
Mean SD Total Mean SD Total % IV, Random, 95% CI IV, Random, 95% CI
Standard Speech language therapy vs No treatment
Johnson 1990 77.5 8.8 6 69.5 3.8 6 9.45 1.09 [-0.12, 2.30]
Speech language therapy (Lee Silverman Voice Treatment Loudness) vs No treatment
Ramig 2001 74.5 4 14 64.9 3.9 15 22.83 2.36 [1.41, 3.31]
Ramig 2018 74.9 6.2 22 70.2 4.7 22 34.64 0.84 [0.22, 1.46]
Lee Silverman Voice Treatment Articulation vs No treatment
Ramig 2018 71,4 5.6 20 70.2 4.7 22 33.07 0.23 [-0.38, 0.84]
Subtotal (95%CI) 62 65 100 1.07 [0.22, 1.93]
Heterogeneity: Tau2=0.5812, Chi2= 4.64, df= 3 (P= 0.0030), I2= 78.46%
Test for overall effect: Z= 4.5452 (P= <.0001)
Speech language therapy (Lee Silverman Voice Treatment Loudness) vs Comparison group
Ramig 1995 69.17 4.1 29 66.2 2.1 13 21.76 0.85 [0.08, 1.61]
Ramig 1996 70 3.5 17 67.1 2.9 7 12.43 0.84 [-0.17, 1.85]
Ramig 2001 69.4 3.4 18 65.8 2.7 6 12.43 1.07 [0.03, 2.11]
Speech language therapy (Parkinsong) vs Comparison group
Tamplin 2020 W 68,9 5,5 35 62.9 5.7 15 25.90 1.04 [0.32, 1.75]
Tamplin 2020 M 62,2 8,3 40 61.4 9.0 13 27.46 0.10 [0.03, 2.11]
Subtotal (95%CI) 139 54 100 0.90 [-0.56, 0.76]
Heterogeneity: Tau2=0.0303, Chi2=1.18, df= 4 (P= 0.3182), I2= 15.10%
Test for overall effect: Z= 3.8716 (P= 0.0001)
Heterogeneity: Tau2=0.2204, Chi2=2.38, df= 8 (P= 0.0147), I2= 57.96%
Test for overall effect: Z= 5.9379 (P= 0.0147)

Figure 4.  Meta-analysis; monologue; sound pressure level.


11
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Study or subgroup Treatment group Comparison group Weight Mean difference dB Mean difference dB
Mean SD Total Mean SD Total % IV, Random, 95% CI IV, Random, 95% CI
Speech language therapy (Lee Silverman Voice Treatment Loudness) vs No treatment
Ramig 2001 74.4 4.3 14 70.7 4.1 15 25.22 0.86 [0.10, 1.62]
Ramig 2018 74.8 5.9 22 69.8 4.9 22 38.26 0.91 [0.28, 1.53]
Speech language therapy (Lee Silverman Voice Treatment Articulation) vs No treatment
Ramig 2018 71.8 6.3 20 69.8 4.9 22 36.52 0.35 [-0.26, 0.96]
Subtotal (95%CI) 56 59 100 0.68 [0.30, 1.06]
Heterogeneity: Tau2=0, Chi2= 1.00, df= 2 (P= 0.3988 ), I2= 0.00%
Test for overall effect: Z= 3.5316 (P= 0.0004)

Figure 5.  Meta-analysis; picture description; sound pressure level.

Study or subgroup Treatment group Comparison group Weight Mean difference dB Mean difference dB
Mean SD Total Mean SD Total % IV, Random, 95% CI IV, Random, 95% CI
Speech language therapy (Lee Silverman Voice Treatment Loudness) vs Comparison group
Ramig 1995 2.38 0.67 26 2.09 0.34 17 57.33 0.50 [-0.12, 1.12]
Ramig 2001 2.48 0.71 20 2.17 0.36 12 42.66 0.50 [-0.23, 1.22]
Subtotal (95%CI) 46 29 100 0.50 [0.03, 0.97]
Heterogeneity: Tau2=0, Chi2=1.00, df= 2 (P= 0.9915), I2= 0.00%
Test for overall effect: Z= 2.0855 (P= 0.0370)

Figure 6.  Meta-analysis; reading; semitone standard deviation.


Clinical Rehabilitation 00(0)
Muñoz-Vigueras et al. 13

CI = 0.22, 1.93; p < 0.001) and comparison group:


Respiratory Treatment, Control treatment Weekly
and Control treatment Monthly (MD = 0.90; 95%
CI = –0.56, 0.76; p = 0.0008). Analysis showed a het-
erogeneity (I2 = 78.46%; I2 = 15.10%), respectively.
Compared to no intervention with comparison
group: (MD = 0.87; 95% CI = 0.46, 1.28; p = 0.0147).
Heterogeneity was (I2 = 57.96%).
IV, Random, 95% CI
Mean difference dB

When the picture description task was analyzed,


the mean difference (MD) showed significant over-
all effect of speech and language therapy: standard,
Lee Silverman Voice Treatment Loudness and Lee
Silverman Voice Treatment Articulation) when
compared to no intervention (MD = 0.68; 95%
CI = 0.30, 1.06; p = 0.0004). Analysis showed no
heterogeneity (I2 = 0%).
Results obtained in pitch characteristics have
IV, Random, 95% CI
Mean difference dB

been analyzed across different subtasks (reading


-0.07 [-0.96, 0.81]
0.03 [-0.70, 0.76]

0.07 [-0.40, 0.54]

and monologue). For the reading subtask, semitone


standard deviation scores were analyzed and pre-
sented in speech and language therapy: Lee
Silverman Voice Treatment when compared to
comparison group: Respiratory Treatment, signifi-
Weight

59.18
40.82
100
%
Speech language therapy (Lee Silverman Voice Treatment Loudness) vs Comparison group

cant effects were found in favor of speech and lan-


guage therapy. The mean difference (MD) shows
Total

overall effects when compared to control interven-


13

22
9
Comparison group

tions in semitone standard deviation (MD = 0.50;


Figure 7.  Meta-analysis; monologue; semitone standard deviation.
0.34
0.36

95% CI = 0.03, 0.97; p = 0.0370). Heterogeneity


SD

was (I2 = 0%).
Heterogeneity: Tau2=0, Chi2=1.00, df= 1 (P= 0.8561), I2= 0.00%
Mean

2.09
2.17

For the monologue subtask, semitone standard


deviation scores were analyzed and presented in
Total

Figure 7.
16
11
27
Treatment group

Compared to comparison group: Respiratory


Test for overall effect: Z= -0.0417 (P= 0.9667)
0.58
0.56

Treatment. No significant effects were found in


SD

semitone standard deviation (MD  = 0.07; 95%


Mean

1.97
2.09

CI = –0.40, 0.54; p = 0.9667). Both analyses showed


no heterogeneity (I2 = 0%).
When the Intelligibility (%) perceptual task was
analyzed (presented in Figure 8), the mean differ-
Study or subgroup

Subtotal (95%CI)

ence (MD) showed significant overall effect of


Ramig 1995
Ramig 2001

speech and language therapy: Lee Silverman Voice


Treatment Loudness and ParkinSong Weekly and
ParkinSong Monthly) when compared to compari-
son group: Respiratory Treatment and Control
treatment Weekly and Control treatment Monthly
(MD = 0.22; 95% CI = –0.29, 0.74; p = 0.1454).
Analysis showed heterogeneity (I2 = 48.14%).
14 Clinical Rehabilitation 00(0)

Discussion
Our results support the idea that speech and lan-
guage therapy treatment is effective in the treat-
ment of Hypokinetic Dysarthria in Parkinson’s
disease patients. Nonetheless, our systematic anal-
ysis of the literature, related to components and
design, has to be interpreted with caution due to the
differences in the intensity of therapy, differences
IV, Random, 95% CI

in the application of speech and language therapy,


Mean difference dB

and differences in effect sizes of the included


studies.
Among speech and language therapy interven-
tions, the Lee Silverman Voice Treatment program
was the most used and the most frequent control
treatment was respiratory training. When com-
pared, speech and language therapy showed sig-
nificantly better results in all measured variables
IV, Random, 95% CI
Mean difference dB

-0.05 [-0.64, 0.54]

-0.01 [0.68, 0.66]

0.22 [-0.29, 0.74]

and were better maintained at the different timeline


0.78 [0.10, 1.47]

follow up. Despite this, more research is needed to


determine the concrete effects of SLT in different
phenotypes of the Parkinson’s disease.
Our findings are in line with previous system-
atic reviews performed in the Parkinson’s disease
Weight

59.16

29.16
33.33
Speech language therapy (Lee Silverman Voice Treatment Loudness) vs Comparison group

100
%

population.11,21,36–41 These suggest that the speech


problems are due to hypokinetic dysarthria and
Total

19
Comparison group

15
13

Lee Silverman’s Voice Treatment publications


Heterogeneity: Tau2=0.1012, Chi2= 1.93, df= 2 (P= 0.1454 ), I2= 48.14%

also suggest the same. This paper is relevant and


19.9

12.5
21.3
SD

needed because it is updated and provides high


quality level of evidence. The results of our sys-
Mean

92.9
72.9
72
Speech language therapy (Parkinsong) vs Comparison group

tematic review suggest that speech and language


therapy can have a beneficial effect on aspects of
Total

Figure 8.  Meta-analysis; perceptual intelligibility (%).


26

20
27
Treatment group

Hypokinetic Dysarthria like: slurred speech, lack


Test for overall effect: Z= 1.1018 (P= 0.2706)

of coordination and hypophonia. However, we


16.7

17.4
SD

8.6

found no effect on impaired muscle tone. This


Mean

71.1

92.7
87.9

could be due to the insufficient data to analyze the


pooled effect.
When analyzing the results obtained comparing
Study or subgroup

speech and language therapy to no treatment, other


Subtotal (95%CI)
Tamplin 2020 W

reviews found similar results with less available


Tamplin 2020 M

evidence.11 However, when the analysis was per-


Ramig 1995

formed taking into account the main symptoms of


Hypokinetic Dysarthria in Parkinsonians, the evi-
dence showed that speech and language therapy pro-
grams fail to include the work of facial muscles in
the designs. Previous studies42,43 have analyzed the
importance of the assessment of orofacial muscle
Muñoz-Vigueras et al. 15

tone due to their interest in differential diagnosis of The Dumer et al.35 study compares two inter-
the dysarthrias. The relevance of evaluating facial ventions, Lee Silverman Voice Treatment Loudness
muscle tone in patients with Hypokinetic Dysarthria vs Lee Silverman Voice Treatment Articulation.
in Parkinson’s,42,43 highlights the clinical relevance However, they do not measure tone and volume of
of standardized procedures and normative data. Our voice, making it difficult to compare with other
analysis of the SLT interventions showed that this articles.11
aspect is usually not included as a measure or a com- This study has some limitations; firstly, the major-
ponent in the proposed programs. Those limitations ity of interventional groups used the Lee Silverman
yield inconclusive results regarding facial muscles Method as a treatment, being scarce in the descrip-
tone and activity after speech and language therapy tion of the dosage, intensity and individualization of
treatment. the treatment. Different modalities of application
In addition to the positive effects of speech and were carried out, with the number of sessions rang-
language therapy in Hypokinetic Dysarthria after ing from 8 to 16, with a training frequency ranging
intervention, our review found that the reported from 1 to 5 sessions per week and a treatment time
changes continue at follow up in the majority of ranging from 45 to 240 minutes per session.
reviewed articles.21,24,28,29,32,34 The follow up time Secondly, some heterogeneity in the outcome
period ranged between 1 month,24 3, 6, 12 months,32 measures within the included studies was observed.
1–7 months29 and 2 years.27 One of the included studies applied tests such as
What has been found regarding Hypokinetic estimated subglottal pressure, maximum flow dec-
Dysarthria in Parkinson’s disease affirms the need lination rate, open quotient and electroglotto-
for monitoring for signs and symptoms of dysar- graphic signal pulse with adduction to measure
thria in order to propose interventions that use variables of the upper airway.25 These instruments
breathing, phonation, resonance, articulation, pros- were not used in any of the other articles reviewed.
ody and facial muscles to improve dysarthria More studies that assess these variables may be
symptoms in Parkinson’s disease. needed to measure the effect of different types of
When speech and language therapy interven- speech and language therapy on more specific
tions were compared with other treatments, the aspects that are affected by the Hypokinetic
results were in favor of the speech and language Dysarthria of Parkinson’s disease.
therapy. However, in some cases such as the mono- Thirdly, there are significant differences in the
logue subtask, it was difficult to draw a significant results obtained in our meta-analysis that affect, in
conclusion. This could be due to the high cognitive a relevant manner, our conclusions. The heteroge-
demand of the monologue subtask which may have neity of outcomes has impacted the number of
affected the results but had not been measured. included studies in the analysis performed for each
The studies included in our review combine dif- independent variable.
ferent speech and language therapy programs with- This meta-analysis has also some clinical impli-
out a conceptual framework of the speech and cations for rehabilitation practice. There is a num-
language therapy discipline, highlighting the impor- ber of functions attributed to speech and language
tance of developing a definition of speech and therapy in rehabilitation of Hypokinetic Dysarthria
language therapy interventions. Some studies23,32,34 in Parkinson’s disease. As well as improving
included in the review use the “Standard speech and patient’s outcomes on standardized measures,
language therapy intervention”, which is not recog- speech and language therapy is also thought to be
nized as a concrete and structured intervention. The an essential part of multidisciplinary therapeutic
Lee Silverman Voice Treatment defines a structured approach for enhancing global patient functional-
and replicable intervention that permits a scientific ity. Speech and language therapy has demon-
recognition of the evidence associated with speech strated specific results in speech parameters
and language therapy intervention in Parkinson’s (sound pressure level, semitone standard devia-
disease. tion and perceptual intelligibility). The therapy is
16 Clinical Rehabilitation 00(0)

usually administered in a weekly format, in an Supplemental material


individualized approach and can have mid-term Supplemental material for this article is available online.
lasting effects (3–6  months after intervention).
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