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Annals of Physical and Rehabilitation Medicine 63 (2020) 81–84

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Letter to the editor

Could non-invasive brain stimulation help treat this patient. Indeed, previous studies of TBI suggested that tDCS
dysarthria? A single-case study combined with active behavioral training can facilitate cortical
reorganization and consolidation of learning in specific neural
networks [10], as in stroke patients [7]. Moreover, previous
A R T I C L E I N F O literature on post-stroke aphasia demonstrated an improvement
with tDCS in oral production for some patients [5], which provides
Keywords: a possible rationale for applying tDCS in dysarthria.
Language To this purpose, we compared 2 tDCS protocols based on the
Dysarthria
literature, in which the right cerebellum [6] or left IFG [4,5] was
Speech rehabilitation
Brain stimulation stimulated by using cathodal or anodal polarities, respectively,
coupled with speech therapy in one patient. Possible changes in
brain integrity after tDCS were assessed by using diffusion tensor
imaging (DTI), before and after the rehabilitation procedure.
At age 19, our right-handed, 21-year-old, native French-
Dear Editor, speaking patient experienced TBI. He spent 1 month in a coma,
with an initial score of 5 on the Glasgow Coma Scale. The first
Dysarthria is a motor speech disorder that can occur following cerebral MRI, performed at 2 weeks after the TBI, showed diffuse
brain damage. It is characterized by impairments in producing the axonal injury in both hemispheres, predominantly in the right
movements needed to articulate words [1] but does not affect temporal lobe, corpus callosum, bilateral basal ganglia, bilateral
other language processing domains such as writing and compre- internal capsule, and both peduncles. At 3 years after TBI, the
hension. patient remained severely impaired, with right hemiparesis,
Several rehabilitation approaches have been developed to cerebellar syndrome, and pseudo-bulbar palsy and persistent
improve dysarthria; they involve intensive exercise or alternative/ severe chronic dysarthria (see Supplementary Table for the
augmentative communication devices but have shown no strong neuropsychological evaluation). Naming, repetition, and reading
beneficial effects [2]. Recent studies in neuro-modulation have were impaired, as was automatic language, with no further
effectively used non-invasive brain stimulation to treat post-stroke impairments in written and oral language and no signs of aphasic
aphasia [3, for a review]. Anodal transcranial direct current agraphia or oral facial apraxia. The lesional substrate of the
stimulation (tDCS) of the left inferior frontal gyrus (IFG) has been articulatory impairments was attributed to a bi-genicular lesion
found to mitigate language articulation impairments in aphasia (Fig. 1).
[4,5]. Moreover, recent findings from healthy individuals showed The rehabilitation procedure lasted 8 weeks and is described in
that cathodal tDCS over the right cerebellum improved perfor- Fig. 2. It was approved by the local ethics committee, and the study
mance in a sequenced articulation task [6], which suggests followed the ethical standards of the 1964 Declaration of Helsinki.
promising perspectives for treating motor speech disorders. Language evaluation included tests for facial-apraxia, automat-
To the best of our knowledge, tDCS has never been used in ic language, repetition, naming, and reading. The tasks were
the context of dysarthria. However, the aforementioned selected from the Protocole d’examen linguistique de l’aphasie
studies suggest that modulating brain areas involved in speech Montréal-Toulouse [11], a language assessment battery for
articulation and motor control, such as the IFG and cerebellum, evaluating language impairments in French-speaking adults, with
could contribute to dysarthria recovery. Moreover, tDCS the exception of the repetition task, which was chosen from the
has been found to induce long-lasting alterations of cortical Batterie d’évaluation clinique de la dysarthrie [12]. The score for
excitability, by mimicking long-term potentiation and depression automatic language, repetition, naming, and reading was calculat-
[7] and by modulating gamma-aminobutyric acid and glutamate ed as a combination of words (1 point if the word was produced
concentrations [8]. Because both the IFG and the cerebellum are with no articulatory errors) and voice (1 point if the word was
part of a complex network implicated in speech articulation [9], produced aloud/not whispered). The maximum possible score was
we hypothesized that inducing long-lasting alterations of cortical 30 for automatic language, 66 for repetition, 31 for naming, and
excitability in these regions could be effective in dysarthria 33 for reading.
recovery. MRI with DTI sequences was performed before and after the
Here, we describe a single-case study of language rehabilitation rehabilitation procedure by using a Philips 3 T MRI Ingenia system
in a young man with severe chronic dysarthria after severe (Philips Medical Systems, Best, The Netherlands; parameters: b-
traumatic brain injury (TBI), despite management by classical value 800 s/mm2; 32 directions; voxel size 2 mm isotropic; TR/TE
speech therapy twice a week. We assessed whether tDCS coupled 8495/100 ms; EPI factor 55). T2 FLAIR and T1 SWI sequences were
with active training could help attenuate the speech disorder in added for anatomical reference and axonal damage assessment.

https://doi.org/10.1016/j.rehab.2019.06.011
1877-0657/ C 2019 Elsevier Masson SAS. All rights reserved.
82 Letter to the editor / Annals of Physical and Rehabilitation Medicine 63 (2020) 81–84

Fig. 1. A, B. Diffuse axonal injury (black spots) on susceptibility weighted MR images with bilateral lesions at the medial part of the cerebral peduncles and in both striatum. D,
E. Coronal reconstructions of the pyramidal tract based on 3-D FLAIR sequences and merged with diffusion tensor tractography. C, F. Track-density imaging shows a slight
lower density of fibers on the right pyramidal tract. G, H. Corpus callosum axonal injury on diffusion tensor tractography and track-density imaging. I. 3-D SAG FLAIR sequence
demonstrates corpus callosum defect. J. Tractography of the pyramidal tracts (yellow and blue) and corpus callosum (orange) with a large defect on the corpus of the CC
(orange arrow). K. Coronal reconstruction of the track density map with no significant change between the 2 MRI scans at 2 weeks after traumatic brain injury and at 3 years.

Fig. 2. The 21-year-old male patient participated in an 8-week rehabilitative treatment combining speech therapy and functional brain stimulation (transcranial direct
current stimulation [tDCS]) targeting the right cerebellum (R. Cb-tDCS) or the left inferior frontal gyrus (L. IFG-tDCS). Language was evaluated before the rehabilitative
protocol (T0, T1), at the end of each stimulation protocol (T2, T3 and T4) and 15 days after the end of the whole rehabilitative procedure (T5).

The post-processing tractography steps were performed with a Under both conditions, the patient received concurrent speech
Philips extended workspace (Portal from Philips, Medical Imaging, therapy on a daily basis for 2 weeks, excluding weekends (5 days/
Best, The Netherlands) with Mrtrix3 package software (J.-D. week). Thus, the patient received 10 cerebellar tDCS sessions and
Tournier, Brain Research Institute, Melbourne, Australia). Regions 10 IFG-tDCS sessions. During each rehabilitation session, the
of interest were placed to include the corpus callosum at midline patient and speech therapist were seated face to face and the
and both the precentral gyri and the cerebral peduncles. A track patient was asked to repeat simple and more complex sounds in a
density map was reconstructed on the basis of a full-brain progressive manner starting from syllables, to disyllabic words,
probabilistic tracking of 1 million fibers with an interpolated and finally short sentences. Two stimuli lists, of 12 words
resolution of 0.3 mm [13]. organized by sounds, were alternated on a daily basis. Each list
tDCS involved use of a battery-driven stimulator (NeuroConn, contained from 3 to 8 words of 1 to 2 syllables; 18 sentences with
GmbH, Germany) with a pair of surface-soaked electrodes. up to 8 syllables were also used (e.g., ‘‘Il a lu le livre de Louis’’). The
Cerebellar stimulation consisted of 20 min of 2 mA direct current speech therapy sessions lasted about 30 min each day and were
with the cathode electrode (5  5 cm) centered on the cerebellum combined with tDCS during the first 20 min. The stimulus lists
(3 cm lateral to the inion on the right cerebellum) [6] and the anode used remained constant despite the change in stimulation
(5  5 cm) placed on the right deltoid muscle (Cb-tDCS). IFG protocols. The speech and language rehabilitation program
stimulation (IFG-tDCS) consisted of 20 min of 1 mA direct current performed during this study was more intensive (5 sessions per
with the anodic electrode (5  7 cm) centered over the left IFG (BA week) than the speech and language therapy the patient
44/45, F5 of the 10–20 system for electroencephalography) and the underwent before inclusion in this study (2 sessions per week),
cathode centered on the contralateral supraorbital region (FP2) [4,5]. which on top of repetition, also comprised spontaneous speech,
Letter to the editor / Annals of Physical and Rehabilitation Medicine 63 (2020) 81–84 83

Fig. 3. Results from the language evaluations during the 8-week rehabilitative period in (from left to right and top to bottom) automatic language, reading, repetition, and
naming. The gray lines reflect the patient’s performance in producing words with no articulatory errors and the black lines the ability to produce the sounds aloud.

reading, and picture description following the principles of the The finding of an effect on speech vocal sound after cerebellar
Promoting Aphasia Communication Effectiveness (PACE) protocol. stimulation is compatible with the anatomical organization of
After the treatment, the patient showed improved intelligibili- neural control of speech [14]. Indeed, the cerebellum is part of a
ty, particularly after Cb-tDCS (Fig. 3), for voice but not words. network involved in speech articulation that includes areas such as
Performance concerning the reading task improved (for voice only) the anterior cingulate, supplementary motor area, and basal
between the 2 pre-tests and after Cb-tDCS but not IFG-tDCS. ganglia and is also connected to the ventral portion of the sensory-
Repetition for voice and words was improved after Cb-tDCS, with motor cortex. This region ultimately projects to the articulatory
an additional improvement after IFG-tDCS for voice only. We muscles via the corticobulbar tract and the oro-facial motor nuclei,
observed no improvement in naming. For all language and which has been shown to somatotopically arrange speech
articulatory evaluations, no long-term improvement was eviden- articulator representations [9]. Cerebellar tDCS may have modu-
ced. lated the activity of this complex network, thereby improving
Because of no significant morphologic modification observed speech production.
on MRI before and after the rehabilitative procedure, only lesion Results from this single-case report should be interpreted
analysis after the rehabilitation procedure is reported. We found with caution. Indeed, a sham phase was not included in the
no significant changes between the 2 MRI scans, whether in experimental design and the 1-week wash-out period did not
targeted or full tractography (Fig. 1). allow for assessing possible long-term improvements after the
The finding of an improvement in intelligibility after Cb-tDCS rehabilitative intervention. The lack of a sham phase limits the
suggests the possibility of using this technique in patients with possibility to exclude language improvements due to the
motor speech disorders. This effect appears specific, particularly increased intensity of the daily intervention. However, the fact
for automatic language, because we observed no improvement that: 1) the improvement was found not only in abilities that
during the 2 pre-tests or after IFG-tDCS. A similar conclusion can be were trained daily and 2) the stimuli used for the daily speech
proposed for reading, although the baseline was not stable in this therapy sessions were not the same as those used during
task. These results agree with the role of the cerebellum in speech language evaluations, allow for partially excluding this issue.
production that can be modulated by tDCS, although articulatory Moreover, the clinical nature of the intervention did not allow for
impairment per se was not improved, as suggested previously in 2 different speech therapists for the therapy sessions and the
healthy individuals [6]. The lack of improvement in abilities not language evaluations, although the speech therapist was blinded
trained for specifically during tDCS seems to suggest that tDCS can to the stimulation protocol used between each language
enhance the effect of speech therapy for only abilities in active assessment. Finally, we noted some variability in the patient’s
training. pre-test scores. However, the pre–post design adopted and the
The mechanisms underlying the observed effect of Cb-tDCS rapid performance impairment 1 week after Cb-tDCS can be
should be discussed with caution. The absence of a long-term effect considered to favour an effect of Cb-tDCS and as a further control
together with no modification of white-matter organization on the for practice effects.
second DTI strongly argues against structural brain reorganization. Well-designed single-case studies allow for exploring the exact
Improvement likely involves more a functional brain-plasticity pathophysiological mechanisms involved in these cases and
process. The cumulative effect of Cb-tDCS and IFG-tDCS on testing new therapeutic approaches. Considering the severity of
repetition likely involves a different network of areas and suggests the speech disorder and the absence of side effects in our patient,
that the mechanism underlying recovery depends on many we believe that repetitive Cb-tDCS could be a possible treatment
variables such as brain lesion topography, delay after onset, the for patients with dysarthria. However, further research is essential
precise nature of the impairment, and treatment type. before any recommendations can be made.
84 Letter to the editor / Annals of Physical and Rehabilitation Medicine 63 (2020) 81–84

Disclosure of interest [10] Villamar MF, Santos Portilla A, Fregni F, Zafonte R. Noninvasive brain stimu-
lation to modulate neuroplasticity in traumatic brain injury. Neuromodulation
2012;15:326–38.
The authors declare that they have no competing interest. [11] Joanette Y, Nespoulous JL, Roch Lecours A. MT 86 - Protocole Montréal-
Toulouse d’examen linguistique de l’aphasie. Ortho; 1998.
[12] Auzou P, Rolland-Monnoury V. Batterie d’Evaluation Clinique de la Dysarthrie
Funding (BECD). Ortho; 2006.
[13] Calamante F, Tournier JD, Jackson GD, Connelly A. Track-density imaging
(TDI): super-resolution white matter imaging using whole-brain track-density
The authors received no specific funding for this work. mapping. Neuroimage 2010;53:1233–43.
[14] Conant D, Bouchard KE, Chang EF. Speech map in the human ventral sensory-
motor cortex. Curr Opin Neurobiol 2014;24:63–7.
Appendix A. Supplementary data
Francesco Panicof,*, Manel Ben-Romdhaneb, Timothee Jacquessonc,d,e,
Supplementary data associated with this article can be found, in Stuart Nashb, Francois Cottonc,d,e, Jacques Luautéa,b,e,**
the online version, at http://doi.dx.org/10.1016/j.rehab.2019.06.011. a
Inserm UMR-S 1028, CNRS UMR 529, ImpAct, Centre de Recherche en
Neurosciences de Lyon, université Lyon-1, 16, avenue Lépine, 69676 Bron,
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b
Service de rééducation neurologique, Hôpital Henry-Gabrielle, Hôpital
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due to stroke and other adult-acquired, non-progressive brain injury. The CREATIS, CNRS UMR 5220, INSERM U1044, Université Lyon 1, INSA Lyon,
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**Co-corresponding author. Inserm UMR-S 1028, CNRS UMR 529,
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