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Dysarthria in individuals with
Parkinson’s disease: a protocol for
a binational, cross-sectional,
case-controlled study in French and
European Portuguese (FraLusoPark)
Serge Pinto,
1,2
Rita Cardoso,
3,4
Jasmin Sadat,
1,2
Isabel Guimarães,
4,5
Céline Mercier,
1,6
Helena Santos,
3
Cyril Atkinson-Clement,
1,2
Joana Carvalho,
3
Pauline Welby,
1,2
Pedro Oliveira,
4,7
Mariapaola D’Imperio,
1,2
Sónia Frota,
7
Alban Letanneux,
1
Marina Vigario,
7
Marisa Cruz,
7
Isabel Pavão Martins,
8
François Viallet,
1,2,6
Joaquim J Ferreira
3,4
To cite: Pinto S, Cardoso R,
Sadat J, et al. Dysarthria in
individuals with Parkinson’s
disease: a protocol for
a binational, cross-sectional,
case-controlled study in
French and European
Portuguese (FraLusoPark).
BMJ Open 2016;6:e012885.
doi:10.1136/bmjopen-2016-
012885
▸Prepublication history for
this paper is available online.
To view these files please
visit the journal online
(http://dx.doi.org/10.1136/
bmjopen-2016-012885).
Received 30 May 2016
Revised 21 September 2016
Accepted 21 October 2016
For numbered affiliations see
end of article.
Correspondence to
Dr Serge Pinto;
serge.pinto@lpl-aix.fr
ABSTRACT
Introduction: Individuals with Parkinson’s disease
(PD) have to deal with several aspects of voice and
speech decline and thus alteration of communication
ability during the course of the disease. Among these
communication impairments, 3 major challenges
include: (1) dysarthria, consisting of orofacial motor
dysfunction and dysprosody, which is linked to the
neurodegenerative processes; (2) effects of the
pharmacological treatment, which vary according to the
disease stage; and (3) particular speech modifications
that may be language-specific, that is, dependent on
the language spoken by the patients. The main
objective of the FraLusoPark project is to provide a
thorough evaluation of changes in PD speech as a
result of pharmacological treatment and disease
duration in 2 different languages (French vs European
Portuguese).
Methods and analysis: Individuals with PD are
enrolled in the study in France (N=60) and Portugal
(N=60). Their global motor disability and orofacial
motor functions is assessed with specific clinical rating
scales, without (OFF) and with (ON) pharmacological
treatment. 2 groups of 60 healthy age-matched
volunteers provide the reference for between-group
comparisons. Along with the clinical examinations,
several speech tasks are recorded to obtain acoustic
and perceptual measures. Patient-reported outcome
measures are used to assess the psychosocial impact
of dysarthria on quality of life.
Ethics and dissemination: The study has been
approved by the local responsible committees on
human experimentation and is conducted in
accordance with the ethical standards. A valuable large-
scale database of speech recordings and metadata
from patients with PD in France and Portugal will be
constructed. Results will be disseminated in several
articles in peer-reviewed journals and in conference
presentations. Recommendations on how to assess
speech and voice disorders in individuals with PD to
monitor the progression and management of
symptoms will be provided.
Trial registration number: NCT02753192,
Pre-results.
INTRODUCTION
Dysarthria in Parkinson’s disease (PD)
Dysarthria denotes a motor speech disorder
resulting from a lesion of the peripheral or
central nervous system.
1–3
Dysarthria and
the psychosocial aspects of communication
impairments are particularly disabling for
individuals with PD. During the progression
of the disease, between 70% and 79% of
individuals with PD mention that speech
45
and functional communication are
impaired,
67
contributing to social isolation
8
and degradation of social interactions.
9
These speech and communication disorders
Strengths and limitations of this study
▪A multicentre (binational), cross-sectional, case-
controlled study.
▪A cross-linguistic, multiparametric and holistic
study of speech in Parkinson’s disease (PD).
▪An interdisciplinary approach bringing together
data analyses from the speech sciences and
neurosciences.
▪A clinically reasonable number of individuals
with PD.
▪No analysis of phonetic alterations and so far,
not a longitudinal study: unable to address
individuals’speech deterioration with time.
Pinto S, et al.BMJ Open 2016;6:e012885. doi:10.1136/bmjopen-2016-012885 1
Open Access Protocol
worsen along with the aggravation of other non-motor
symptoms such as self-perception, depression
10
and cog-
nitive impairment.
11
In addition to the alteration of
speech intelligibility, signally a motor-driven speech
deficit, it is also important to consider the importance
of cognitive impairment on everyday communication in
individuals with PD.
12
Dysarthria can appear at any stage
of PD, and usually worsens as the disease progresses,
13
which suggests that it is also linked to the evolution of
the pathological processes and non-dopaminergic brain
circuits.
14–16
The main deficits of PD speech are: loss of
intensity (hypophonia), monotony of pitch and loud-
ness, reduced stress, inappropriate silences, short rushes
of speech, variable rate, imprecise consonant articula-
tion and dysphonia (harsh and breathy voice).
1217
Previous studies have shown that treatments in PD
have variable effects on these voice and speech
symptoms.
18 19
Although behavioural treatments mainly focus on two
key indices of PD speech, pitch and intensity, dysprosody
has been understudied. Yet prosody deficits represent an
acoustic hallmark of dysarthria. First, perceptual and
acoustic investigations of PD speech have reported
alterations on fundamental frequency (F0), as part of
speakers’reduced phonatory capacity, and thus the
reduction of the frequency range is an indicator of dys-
arthria in PD.
20
In particular, individuals with PD show a
loss of the upper part of the frequency range.
21
Degradation of prosody has been found to impact
speech intelligibility and communication (eg, see
ref. 22). Second, the temporal organisation of speech in
PD has been addressed in reading tasks.
23–25
In French,
for example, speech rate tends to be slower in PD,
which in turn seems to be correlated with longer pause
times. Average durations of pauses are found to be
longer in individuals with PD than in healthy indivi-
duals, while the average duration of sound sequences
are similar.
24 25
Studying dysprosody is thus important
for differential diagnosis, identifying severity and the
need and focus of treatment.
20
Remaining challenges to assess dysarthria in PD: the
rationale of the FraLusoPark study
Individuals with PD have to cope with several issues that
contribute to voice and speech decline and thus to the
alteration of communication ability during the course of
the disease. Among these communication impairments,
three major challenges include: (1) dysarthria, consist-
ing of orofacial motor dysfunction and dysprosody,
which is linked to the neurodegenerative processes; (2)
the effects of the pharmacological treatment, which vary
according to the disease stage and (3) the particular
speech modifications that may be language-specific, that
is, dependent on the language spoken by the patients.
The main objective of the FraLusoPark project is to
provide an extensive evaluation of dysarthric speech in
PD as a result of pharmacological treatment and disease
duration, using acoustic parameters (voice and
prosody), perceptual markers (intelligibility) and
patient-reported outcome measures (PROMs; psycho-
social impact on quality of life) in speakers of two differ-
ent languages (French and European Portuguese).
Based on a large-scale binational collaboration, the
interdisciplinary FraLusoPark project aims to address
these issues by providing important insights in the
domains of neurodegenerative disorders, speech
sciences, neuropsychology, clinical research and patient
rehabilitation.
Medication effects along disease progression
Early studies assessing the effect of the levodopa
(L-dopa) on PD speech found favourable results,
arguing for a beneficial effect, as for limb impair-
ments.
26–29
However, the long-term use of L-dopa is asso-
ciated with motor complications which occur in up to
80% of patients.
30 31
This may be the reason why the fol-
lowing studies reported no improvement
32 33
and/or
detrimental effects of L-dopa on speech.
34 35
More
recent studies face similar problems: beneficial effects of
L-dopa can be observed in advanced patients with
PD,
36 37
whereas a lack of improvement is reported for
speech parameters in early stage patients with PD.
38
It is
also commonly accepted that in the later stages of PD,
non-motor symptoms (dementia, psychosis, depression
and apathy) are a major source of disability together
with axial symptoms (eg, alteration of gait, balance,
posture, speech).
39
Thus, both clinicians and researchers
have to dissociate various intermingled effects. For
example, when individuals with PD respond to L-dopa at
an early stage of the disease, in time they are likely to
experience speech decline that may be the result of the
degeneration of non-dopaminergic structures and/or
adverse effects of L-dopa (ie, dyskinesia). Despite the
large number of recent studies that have focused on the
effect of L-dopa on speech in PD,
36–44
the question of
disease evaluation still remains a matter of debate.
Language specificities of prosody
One missing component in the description of PD
speech deficits is language-specific aspects, in particular
of dysprosody. Prosodic information, including inton-
ation, tempo, stress and rhythm, serves many functions
for the listener and speaker: it helps to segment the con-
tinuous flow of spoken language into words, groups
these words into phrases for interpretation, and indi-
cates the relative importance and function of the inter-
preted meanings.
45–48
Each language has its own
prosodic structure. For example, although they are sister
languages, French and European Portuguese (both
Romance languages) differ prosodically in a number of
important ways. European Portuguese has contrastive
lexical stress: each content word (noun, adjective, verb,
etc) has one syllable that is particularly salient or
stressed, and changing the position of the lexical stress
can change the meaning of a word.
47 49–51
Stressed sylla-
bles may be accompanied by a pitch accent, realised as a
2Pinto S, et al.BMJ Open 2016;6:e012885. doi:10.1136/bmjopen-2016-012885
Open Access
modulation in F0 (eg, a rise or a fall) and aligned in
language-specific ways with the syllable. In contrast,
French has a fixed stress, characterised by a systematic
F0 rise on the last syllable in a word.
52 53
In French,
stress is not a property of the word, but of a larger unit
called the accentual phrase that can include one or
more content words and any preceding function words
(articles, prepositions, etc), often realised with F0 rises
at its right and left edges. French listeners use these F0
rises as cues to word segmentation, finding the begin-
ning and ends of words in the speech stream, and to
lexical access, retrieving words from the mental
lexicon.
48 54
Such differences across languages make the compari-
son of prosodic deficits in individuals with PD particu-
larly interesting. Very few studies of PD dysprosody have
looked beyond global measures to examine the extent to
which linguistically important, language-specific patterns
are affected.
55 56
Therefore, studying speech in indivi-
duals with PD whose languages include different pros-
odic modulations is essential in determining the role of
prosody in patients’speech intelligibility and quality of
life. Does a Portuguese patient experience different
communication impairments compared with a French
patient? If this is the case, could this difference be
related, for example, to the fact that in Portuguese stress
is distinctive and varies in position? Finally, how do these
differences relate to disease duration and pharmaco-
logical treatment? Our project presents a novel
approach to these questions and is characterised by the
collection of cross-linguistic data in a single cohort.
METHODS AND ANALYSIS
FraLusoPark is a binational (data collection is performed
in two countries: France and Portugal), cross-sectional
(data are collected once for each participant) and case-
controlled (both individuals with PD and control subjects
are recruited) study, carried out in two different lan-
guages (French and European Portuguese).
Aims and hypotheses
The main objectives of our project are to evaluate modu-
lations in voice/speech acoustics parameters (acoustics
and prosody), perceptual markers (intelligibility) and
PROMs (psychosocial impact of dysarthria in PD) across
two different languages (French and European
Portuguese).
Our three a priori hypotheses are the following: (1)
global acoustic features are altered similarly in French
and Portuguese individuals with PD; (2) language-
specific prosodic patterns are altered differently in
French and Portuguese individuals with PD and (3) the
impact of speech disorders on intelligibility and quality
of life depends on the cultural and linguistic environ-
ment. In addition, the FraLusoPark project will allow for
a better understanding of the progression of speech
symptoms and their response to pharmacological
treatment, which is important for pathophysiological
aspects and clinical management.
Participants
Two groups of 60 healthy volunteers (one in France
and one in Portugal) are age-matched and sex-matched
with the individuals with PD to provide control refer-
ences for the obtained performance measures.
Individuals with PD are recruited in France (N=60;
Neurology Department, Centre Hospitalier du Pays
d’Aix, Aix-en-Provence, France) and in Portugal (N=60;
Movement Disorders Unit, Hospital de Santa Maria,
Lisbon, and Campus Neurológico Sénior (CNS), Torres
Vedras, Portugal) and correspond to the UK Parkinson’s
Disease Brain Bank Criteria
57
for the diagnosis of idio-
pathic PD. Individuals with PD and healthy controls are
all native speakers of French or European Portuguese
speakers (French–European Portuguese bilinguals were
excluded) and right-handed (Handedness Edinburgh
test >80%).
58
Inclusion and exclusion criteria of patients
with PD and healthy controls are summarised in table 1.
To assess the effects of L-dopa at various stages of the
disease, we consider three subgroups of patients (N=20
patients each): subgroup 1, early, with a disease duration
between 0 and 3 years and no motor fluctuations; sub-
group 2, medium, with a disease duration between 4
and 9 years, or between 0 and 3 years and experiencing
motor fluctuations; subgroup 3, advanced, with a disease
duration of over 10 years.
Study design
Healthy control participants undergo the same non-
invasive assessments and examinations as individuals
with PD. The only difference for patients is that they are
evaluated twice, in the OFF and ON L-dopa states. This
entails: (1) at least 12 hours after withdrawal of all
anti-Parkinsonian drugs and (2) following at least 1 hour
after the administration of the usual medication. The
full study design is illustrated in figure 1.
Speech recordings
In a quiet room, specialised speech recording equip-
ment (EVA2 system, SQLab, Aix-en-Provence, France;
http://www.sqlab.fr/; Marantz PMD661 MKII recorder,
USA) is used for the speech/voice recordings.
Participants are recorded while performing several
speech production tasks with increasing complexity in a
fixed order: (1) steady vowel /a/phonation (at a com-
fortable pitch and loudness) repeated three times; (2)
maximum phonation time (vowel /a/sustained as long
as possible on one deep breath at a comfortable pitch
and loudness), repeated twice; (3) oral diadochokinesia
(repetition of the pseudoword pataka at a fast rate for
30 s); (4) reading aloud of 10 words and 10 sentences
created by adapting the intelligibility part of V.2 of the
Frenchay Dysarthria Assessment (FDA-2);
59
(5) reading
aloud of a short text (‘The North Wind and the Sun’,
French and European Portuguese adaptations);
50 60
(6)
Pinto S, et al.BMJ Open 2016;6:e012885. doi:10.1136/bmjopen-2016-012885 3
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storytelling speech guided by visual stimuli ( pictures
from the wordless story ‘Frog, Where are you?’,
61
for the
rationale of using this procedure and this book, see
ref. 62); (7) reading aloud of a set of sentences with
specific language-dependent prosodic properties (31
sentences in French, 20 in European Portuguese); and
(8) free conversation for 3 min.
Acoustic measures
The acoustic measures characterise dimensions of aero-
phonatory control.
63
For the steady vowel /a/phonation,
two kinds of measures will be extracted: first, for a
macro analysis, F0 (Hz) and F0 variation (%); and
second, for a micro analysis, perturbation measures such
as jitter factor (%), absolute shimmer (dB) and
harmonics-to-noise ratio (HNR, %). For the maximal
phonation time, the longest duration (in seconds) of
the sustained vowel /a/will be extracted. For the oral
diadochokinesia task, the extracted measures will be the
following: (1) the number of breath groups, that is, the
period during which the pseudoword was repeated in a
single expiration, (2) the ratio between the cumulated
speech duration of the breath groups and the total dur-
ation of the session, that is, the proportion of speech,
(3) the articulatory rate (syllables/second), (4) the
pause-to-sound ratio (%) and (5) the speech proportion
per number of breath groups. As an initial step to inves-
tigate global prosodic aspects of PD speech compared
with the speech of healthy controls, we will extract the
F0 curve of one sentence selected from the short text
(‘The North Wind and the Sun’). This sentence is
selected to be comparable across French and European
Portuguese in terms of semantics and syllable length.
This will provide a global phrasal pattern of F0 and
intensity for patients and controls within and across lan-
guages (see below in the discussion for subsequent
studies…). A summary of the acoustic measures that will
be analysed is listed in table 2.
Clinical assessments
The neurological assessment is the Unified Parkinson’s
Disease Rating Scale,
64
using the revised version pro-
vided by the Movement Disorders Society (MDS-UPDRS).
65
The FDA-2 is used to assess the functions of the speech
Figure 1 Overview of the
FraLusoPark study design.
Patient OFF-medication
assessments (clinical history,
speech recordings and clinical
assessments, without medication)
are shown in dark grey.
ON-medication assessments
(speech recordings and clinical
assessments after medication are
effective) are shown in light grey.
Table 1 Inclusion and exclusion criteria
All participants
Inclusion criteria
Age between 35 and 85 years
Good cooperation
Ability to understand the information sheet
Given signed consent
Enrolled in a medical insurance plan
Other stable medical problems not interfering with the proposed study
Idiopathic Parkinson’s disease*
Absence of any neurological, psychiatric or behavioural pathology
†
Exclusion criteria
Illiteracy
French/Portuguese not native language, or bilingual
Participant under tutorship or guardianship, or any other administrative or legal dependence
No cooperation or consent withdrawn
Cognitive deficits, severe depression, dementia, psychosis (including medication-induced) or behavioural, neurological,
medical, psychological disorders that may interfere with evaluations
Non-idiopathic Parkinson’s disease*
Deep brain stimulation*
Severe motor impairment impeding participation in the study*
*Patients with Parkinson’s disease.
†
Control subjects.
4Pinto S, et al.BMJ Open 2016;6:e012885. doi:10.1136/bmjopen-2016-012885
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organs,
59
reflecting the state of the muscular effectors
involved in speech production. The original FDA-2, in
English, includes an evaluation of intelligibility through
10 words and 10 short sentences. Using the same meth-
odology, we developed a set of cross-linguistically
adapted words and sentences in French and European
Portuguese that will be used for the intelligibility assess-
ment in each language. During the OFF medication
state, individuals with PD will be administered the FDA-2
and the motor part ( part 3) of the MDS-UPDRS.
During the ON medication state, these two assessments
are performed together with the non-motor ( part 1.A)
and motor complication ( part 4) part of the
MDS-UPDRS. During the ON medication state, the parti-
cipants’cognitive abilities are evaluated using the
Montreal Cognitive Assessment (MoCA),
66
and the
Clinical Global Impression (CGI) is also reported.
67
For
healthy controls, the assessment is similar to that of the
patients with PD during ON medication (except part 4
of the MDS-UPDRS). A summary of the clinical assess-
ments is given in table 3.
Patient-reported outcome measures
PROMs, such as the Dysarthria Impact Profile (DIP),
68
are used to obtain self-reported information about the
functional impact of an individual’s speech/communica-
tion impairment.
69
Additional self-assessments focus on
the patients’perception of their quality of life (the
39-Item Parkinson’s Disease Questionnaire
(PDQ-39))
70 71
and on how voice/speech impairment
may induce a handicap (Voice Handicap Index, VHI).
72
The French
73
and European Portuguese adapted DIP,
VHI
74 75
and PDQ-39
76–78
are used in our study. The
Patient Global Impression (PGI) scoring
79
and the Beck
Depression Inventory (BDI)
80
are also administered.
The MDS-UPDRS also includes a patient self-assessment
(parts 1.B and 2), which is administered together with
the other questionnaires in the ON condition. For
healthy controls, the assessment is the same as for
individuals with PD. A summary of the PROMs from the
self-administered questionnaires are listed in table 3.
Statistical analyses
The analyses of the data (acoustic, clinical measures and
PROMs) will be performed with linear mixed-effects
models that account for the variability across individuals,
using the latest version of the statistical software R (R
Development Core Team. R: a language and environ-
ment for statistical computing. R Foundation for
Statistical Computing, Vienna, Austria: 2014. ISBN 3--
900051-07-0, http://www.R-project.org/). For each per-
formance measure, the between-group factors group
(patients vs controls), disease duration (early vs medium
vs advanced) and language (French vs European
Portuguese) will be investigated. In addition, we will
explore the effects of the within-patient factor medication
(OFF vs ON) for all measures. Further relevant
participant-related measures such as age, gender and
education level will also be taken into account in the
analyses.
DISCUSSION
The present study will provide a unique, thorough and
reliable assessment of PD voice, speech and prosody dis-
orders and an evaluation of the impact of these aspects
on the quality of life of individuals with PD.
Main and subsequent analyses of the FraLusoPark study
Acoustic and prosodic measurements (table 2), clinical
assessment and PROMs (table 3) are the dependent
variables to be analysed according to the statistical plan.
These findings will be reported in the primary analysis
as the main results of the project. However, the
FraLusoPark investigation protocol will allow us to
conduct additional analyses focusing on specific subdi-
mensions of speech and voice deficits. There are at least
four such subsequent analyses, exploring important
Table 2 Acoustic measures
Speech tasks Function assessed Acoustic measures
Steady vowel /a/ phonation Phonation Mean F0 (Hz)
F0 variation (SD, in Hz)
Shimmer (%)—cycle-to-cycle F0 variation
Jitter (%)—cycle-to-cycle intensity variation
HNR (%)
Maximal phonation time of the vowel /a/ Aero-phonatory control Longest duration (in seconds)
Oral diadochokinesia Supralaryngeal articulatory control Number of breath groups
Proportion of breath groups (%)
Articulatory rate (in syllables/second)
Pause-to-sound ratio (%)
Speech proportion ratio (%)
Reading aloud of text Prosody F0 range (Hz)
Intensity (dB)
F0, fundamental frequency; HNR, harmonics-to-noise ratio.
Pinto S, et al.BMJ Open 2016;6:e012885. doi:10.1136/bmjopen-2016-012885 5
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Table 3 Clinical assessments and patient-reported outcome measures
Description Subsections
Minimum–maximum scores
(worst values in bold)
A. Clinical assessments
Movement Disorders Society—Unified Parkinson’s Disease Rating Scale
(MDS-UPDRS)—Assessment of motor and non-motor features of
Parkinson’s disease
65
Non-motor experience of daily living—motor experience of
daily living—motor examination—motor complications
0–260
Frenchay Dysarthria Assessment, second edition (FDA-2)—Assessment of
speech and voice organs
59
Reflexes—Respiration—Lips—Palate—Larynx—Tongue—
Intelligibility
0–104
Montreal Cognitive Assessment (MoCA)—Global assessment of cognitive
functions
66
Visuospatial—Naming—Memory—Attention—Verbal fluency
—Abstraction—Orientation
0–30
Clinical Global Impression (CGI)—Global impression of the clinician for the
symptom
67
Speech 1–7
B. Patient-reported outcome measures (PROMs)
39-Item Parkinson’s Disease Questionnaire (PDQ-39)—Quality of life in
Parkinson’s disease
71
Mobility—Daily living activities—Emotional well-being—
Stigma—Social support—Cognition—Communication—Body
discomfort
0–156
Voice Handicap Index (VHI)
72
Physical—Functional—Emotional 0–120
Dysarthria Impact Profile (DIP)—Psychosocial impact of speech deficits
68
Effect of dysarthria on me—Accepting my dysarthria—How I
feel others react to my speech—How dysarthria affects my
communication—Dysarthria relative to other worries and
concerns
48–240
Patient Global Impression (PGI)—Global impression of the patient on the
dysfunction
79
Speech 1–7
Beck Depression Inventory (BDI)—Global assessment of the depression
profile
80
0–84
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aspects of PD speech and communication in more
detail.
First, the intelligibility part of the FDA-2
59
requires the
words and sentences produced by the patient to be per-
ceptually rated by the clinician in charge of the assess-
ment. Since these speech productions are recorded
during the FraLusoPark protocol, an evaluation of
speech intelligibility by panels of auditory judges in
France and Portugal will be run. This will allow an add-
itional and unbiased judgement of speech and voice dis-
orders beyond that of the speech therapists and experts
involved in the study. This approach further comple-
ments the global assessment of dysarthric speech in
patients with PD.
Second, further prosodic analysis will be conducted
on the defined sets of sentences, which manipulate
language-general and language-specific details of
prosody. One particular focus will be the analysis of
tonal alignment in the F0 curve, that is, the temporal
coordination of high and low tones with specific syllables
in the sentences.
81–83
Tonal alignment is likely to be a
relevant factor in the study of PD dysprosody since it
relies on the precise coordination of glottal and supra-
glottal articulatory gestures required to achieve
language-specific temporal patterns for pitch accents
and boundary tones.
Third, taking into consideration patients’personal
feelings with respect to the physical, psychological and
social domains has received increasing interest over the
past decade. Individuals with PD are affected by voice
and speech disorders, which contribute to an impair-
ment of general communication abilities. Individuals
with PD are therefore less likely to participate in conver-
sations or social interactions.
69
Several studies suggest
that a growing discomfort in verbal communication
during the progression of the disease leads to an import-
ant negative impact on social life.
70 84 85
Taken together,
these studies argue for experimental designs that
include different types of speech assessments (clinical,
perceptual, instrumental and psychosocial), as in the
current protocol, and that explore the relationships
between these different measures. Further analyses will
therefore focus on linking the different dimensions of
voice and speech description (eg, acoustic measures,
FDA-2) with the contributions of various participant-
related measures such as intelligibility, cognition and
functional communication.
Fourth, production and prosodic parameters from
the three different speech tasks (ie, short text reading,
orientated picture description and conversation) will
be compared. This will allow us to compare speech
and voicing disorders in increasingly more complex
communication contexts. These comparisons are of
interest since communication abilities in PD are quite
different in the presence of external cueing, such as
during reading, compared with spontaneous speech,
which involves more complex speech planning
strategies.
Finally, the FraLusoPark study might provide us the
opportunity to address the deterioration of the speech
of individuals with PD over time. In a longitudinal
follow-up study, we would recruit a subgroup of patients
from group 1 (disease duration up to 4-year) and group
2 (disease duration between 4 and 10 years). These
patients would be evaluated again at a later point in
time (about 5 years after the original recordings). This
would allow us to describe the precise progression of
speech deficits associated with PD within the same indi-
vidual for French and Portuguese speakers.
ETHICS AND DISSEMINATION
This study has been approved by the local responsible
committees on human experimentation (France:
Comité de Protection des Personnes, Sud Méditerranée
1, project reference number 13-84, approval date 9
January 2014; Portugal: Ethics Committee of the Lisbon
Academic Medical Centre, project reference number
239-14, approval date 1 June 2014). The study is con-
ducted in accordance with the ethical standards of the
Declaration of Helsinki.
86
The patients are included in
the study after providing their written informed consent.
The FraLusoPark trial is registered under the reference
NCT02753192 (26 April 2016) on https://clinicaltrials.
gov/.
Results of the FraLusoPark project will be dissemi-
nated at several research conferences at the national
and international levels and published as articles in
peer-reviewed journals and clinical magazines. The pub-
lication strategy is based on one principal article report-
ing the main results of the project and several
subsequent articles deriving from it, including more
detailed analyses of specific subdimensions of the
speech and voice deficits.
Owing to inter-speaker variability, any generalisation
drawn from speech parameters in the clinical population
requires data from a large number of speakers.
87
The
FraLusoPark project is in line with this idea in that it builds
a large-scale corpus of PD speech recordings and includes a
large set of metadata (clinical examinations, speech
measurements, linguistic features, patient-based indices).
This allows a more accurate description of PD dysarthria,
documenting the evolution of the symptoms and their
response to pharmacological treatment. Both in medical
(eg, http://www.mrc.ac.uk/research/research-policy-ethics/
data-sharing/data-sharing-population-and-patient-studies/)
and linguistic (http://sldr.org/) domains, data sharing is
important to maximise the lifetime value of human
health data. It is our intention to contribute to this prac-
tice by archiving our data for long-term preservation and
making them accessible after the completion of our
analyses.
Furthermore, an important recommendation from the
International Classification of Functioning, Disability
and Health
88
is to improve quality of healthcare and to
encourage clinicians to adopt a more holistic approach
Pinto S, et al.BMJ Open 2016;6:e012885. doi:10.1136/bmjopen-2016-012885 7
Open Access
to the assessment and treatment of patients. Research in
the field of speech sciences needs to incorporate this
vantage point when studying pathological speech. The
FraLusoPark project is in line with this perspective and
will provide important recommendations for speech and
voice assessments in patients with PD. This will be
helpful to health practitioners and clinicians when mon-
itoring the progression of symptoms and their manage-
ment, and will also advance our understanding of
dysarthria in PD within a cross-linguistic and cross-
cultural context.
Author affiliations
1
Aix Marseille Université, Centre National de la Recherche Scientifique
(CNRS), Laboratoire Parole et Langage (LPL), Aix-en-Provence, France
2
Brain and Language Research Institute, Aix-en-Provence, France
3
Campus Neurológico Sénior (CNS), Torres Vedras, Portugal
4
Faculty of Medicine, Instituto de Medicina Molecular (IMM), University of
Lisbon, Lisbon, Portugal
5
Speech Therapy Department, Escola Superior de Saude do Alcoitão,
Alcabideche, Portugal
6
Neurology Department, Centre Hospitalier du Pays d’Aix, Aix-en-Provence,
France
7
Centre of Linguistics, School of Arts and Humanities, University of Lisbon,
Lisbon, Portugal
8
Neurology Department, Faculty of Medicine, Language Research Laboratory,
University of Lisbon, Lisbon, Portugal
Contributors SP and JJF are the principal investigators of the FraLusoPark
study. They designed the study and ensure the good performance of the
study. JJF and FV are the neurologists in charge of patient recruitment and
neurological assessments. RC, JS, HS, CM, JC, FV and SP perform data
acquisition and other clinical examinations. RC, JS, PO and IG are in charge
of the pre-processing and analyses of acoustic measurements. CA-C and AL
are in charge of the analyses of the PROMs and clinical assessments. PW,
PO, MD, MC, SF and MV are the linguistic experts in charge of the prosody
evaluations. IPM is the neurobehaviour, language and cognition expert. SP
wrote the draft of the present article. All coauthors commented and revised it
critically for important intellectual content, and approved the final version to
be published.
Funding This study is supported by a bilateral transnational funding between
France and Portugal: support from the French government, through the
French National Agency for Research (ANR—Agence Nationale de la
Recherche—grant number ANR-13-ISH2-0001-01) and from the Portuguese
government, through the Portuguese National Foundation for Science and
Technology (FCT—Fundação para a Ciência e a Tecnologia—grant number
FCT-ANR/NEU-SCC/0005/2013). CA-C wishes to thank his PhD grant scheme
co-funders: PACA Regional Council and Orthomalin (http://www.orthomalin.
com/).
Competing interests None declared.
Patient consent Obtained.
Ethics approval France: Comité de Protection des Personnes, Sud
Méditerranée 1, project reference number 13-84, approval date 09/01/2014;
Portugal: Ethics Committee of the Lisbon Academic Medical Centre, project
reference number 239-14, approval date 12/06/2014.
Provenance and peer review Not commissioned; externally peer reviewed.
Data sharing statement We addressed this point in the “Ethics and
dissemination”section of the article, as follows: Both in medical (eg, http://
www.mrc.ac.uk/research/research-policy-ethics/data-sharing/data-sharing-
population-and-patient-studies/) and linguistic (http://sldr.org/) domains, data
sharing is important to maximise the lifetime value of human health data. It is
our intention to contribute to this practice by archiving our data for long-term
preservation and making them accessible after the completion of our
analyses.
Open Access This is an Open Access article distributed in accordance with
the Creative Commons Attribution Non Commercial (CC BY-NC 4.0) license,
which permits others to distribute, remix, adapt, build upon this work non-
commercially, and license their derivative works on different terms, provided
the original work is properly cited and the use is non-commercial. See: http://
creativecommons.org/licenses/by-nc/4.0/
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