Translingual Neurostimulation in Treatment of Children with

Cerebral Palsy in the Late Residual Stage. Case Study

T. S. Ignatova

, V. E. Kolbin

, A. M. Sarana

1,2

, S. G. Scherbak

1,2

, Yu. P. Danilov

N. N. Semibratov

, A. V. Sokolov

, G. E. Trufanov

, A. V. Ryzhkov

and A. Yu. Efimtsev

State Health Institution «City Hospital №40», Neurology and Rehabilitation Department, Saint-Petersburg, Russia

State University, Faculty of Medicine, Saint-Petersburg, Russia

TCNL, Kinesiology Department, Wisconsin, U.S.A.

National Medical Research Centre n.a. V.A.Almazov, SIL MRI, Saint-Petersburg, Russia

Keywords: Translingual Neurostimulation, Portable Neurostimulator, Rehabilitation, Cerebral Palsy, fMRI,

Neuroimaging.

Abstract: Management of cerebral palsy is an actual problem of modern medicine. A new direction of

neurorehabilitation, intensively discussed in modern science and practice, includes various types of electrical

stimulation. Constant stimulation of the nervous system is one of the most popular ways to activate neural

networks in order to activate the brain and initiate neuroplasticity processes. Participants in the experiment

were children with cerebral palsy, spastic diplegia form at the age of 6 to 19 (n=6) (mean age - 17,9 ± 5,6

years). All subjects underwent standard treatment, including massage, therapeutic gymnastics simulators,

robotic mechanotherapy, etc., which lasted 20-25 minutes with neurostimulation of the brain (using a PoNS

device). All subjects underwent a functional brain imaging (MRI) before and after neurostimulation course.

Results indicate positive dynamics in all subjects: most of them learned walking without aids, decreased

muscle tonus and improvement in balance, coordination function were noted. Neurostimulation with the PonS

device combined with curative gymnastics (focused exercises), improves the efficiency of motor functions

and development of motor skills. Functional MRI with an active paradigm, with proper and high-quality

performance, is an auxiliary method of objective control of treatment effectiveness.

1 INTRODUCTION

In subjects with cerebral palsy, there are obvious

violations of equilibrium, position of motion,

retention of the pose in space. Each function of the

human body is based on well-organized complex

neural networks, including numerous interconnected

structures (cortex, nuclei, neural clusters) located in

different levels of brain and spinal cord. Collaboration

and synchronization of human performance in

behavioral, cognitive and autonomic functions. This

close integration is especially important in complex

sensory and motor functions, such as vision, hearing,

balance, gait, speech.

Neurorehabilitation of children with cerebral

palsy is multicomponent and includes physiotherapy,

special massage therapy, treatment, special limb

treatment with different stitches, the use of fixing

devices for walking, special, facilitating the motor

activity of the child, and costumes. In modern

medicine, the problems of rehabilitation of children

with cerebral palsy are given particular attention. A

new direction of neurorehabilitation, intensively

discussed in modern science and practice, is the use of

various types of electrostimulation, as well as their

use in or in combination with existing procedures. The

most common among them - electrical stimulation of

muscles and nerves, as well as the spinal cord.

Electrical stimulation was used to treat spastic Erb-

Duchenne paralysis in 1871. Since that time treating

patients with spasticity by electrostimulation of

muscles and nerve structures used in psychotherapy,

the subcutaneous, epidural location of the electrodes,

as well as peroneal implantation (Svozil et al. 2015,

Kublanov et al. 2008). Despite the positive results

achieved by the integrated treatment approach, the

problem of rehabilitation of children with cerebral

palsy in the late residual stage with persistent

stereotypes remains unresolved. The problems of

restoring muscle control and complex sensorimotor

integration (balance, coordination of movement,

retention of the body in space) have so far not been

332

Ignatova, T., Kolbin, V., Scherbak, S., Sarana, A., Sokolov, A., Trufanov, G., Semibratov, N., Ryzhkov, A., Eﬁmtsev, A. and Danilov, Y.

Translingual Neurostimulation in Treatment of Children with Cerebral Palsy in the Late Residual Stage. Case Study.

DOI: 10.5220/0006732403320337

In Proceedings of the 11th International Joint Conference on Biomedical Engineering Systems and Technologies (BIOSTEC 2018) - Volume 4: BIOSIGNALS, pages 332-337

ISBN: 978-989-758-279-0

given great attention. Artificial stimulation of the

nervous system is one of the most popular ways to

activate neural networks in order to activate the brain

and initiate neuroplasticity processes (Petrenko et al.

2017).

2 MATERIALS AND METHODS

2.1 Participants

This study involved 6 children with cerebrally palsy,

form of spastic diplegia. Patients with intact intellect,

no seizures in anamnesis. All children obtained

standard treatment, including massage, medical

gymnastics with simulators, robotic mechanotherapy,

hydrotherapy, and 10 daily sessions of physical

therapy, which lasted for 20-25 minutes and

neurostimulation of the brain (using the PonS device).

Patients underwent functional MRI of the brain before

the start of and at the end of the course of treatment

using neurostimulation. The patients were aged 8 to

14 years. Patients were evaluated by standard scales

GMFSC Scale (gross motor skills), FMS (functional

motor scale), Berg balance scale, the Ashworth scale

(spasticity). 5 patients with a GMFCS level of

development 3 field of the treatment received positive

dynamics in FMS.

2.2 Neurostimulation

Along with the existing modern high-performance

methods of use of peripheral nerve stimulation

(Kublanov et al. 2017), an innovative method was

developed at the University of the state of Wisconsin,

USA, in the laboratory, which was headed by

renowned scientist Paul Bach-y-Rita, one of the

founders of the modern concept of neuroplasticity. An

instrument for electrotactile stimulation of human

skin, in the most densely innervated tactile region -

the tongue, was designed in the laboratory of tactile

communication and neurorehabilitation (TCNL)

(Danilov et al. 2008). Electrotactile stimulation of

language is, at the moment, the most effective and

safe stimulation of the Central nervous system.

Language is the most thin portion relative to the other

surfaces of the skin, full of different types of

mechano-, thermo - and taste buds, with the addition

of free nerve endings. This area has the highest

density of mechanoreceptors per unit area and has a

minimum two-point discrimination threshold: 0.5-1

mm for mechanical stimulation and 0.25-0.5 mm for

electrical stimulation (Danilov et al. 2006, Danilov et

al. 2007). Two major cranial nerves (branch of

trigeminal, 20-22 000 nerve fibers and the facial

nerve, 3-6000 nerve fibres) from the anterior surface

of the tongue provide the transmission of nerve

impulses directly to the structure of the brain stem,

activating the complex nuclei of the trigeminal nerve

(mesencephalic, sensory and spinal cord-the large

core of the trunk) and, at the same time, neighboring

nucleus of solitary tract on the branch of the facial

nerve is stimulated. The cochlear nuclei are being

activated as well, medulla and upper sections of the

cervical spine (C2 and C3). The reticular formation of

the brain stem, a complex of vestibular nuclei and the

ventral part of the cerebellum enters the area of

secondary activation (Barbara et al. 2009). As is

known, the area of the brain stem has a large cluster

of neuronal nuclei (N=86), some of them engaged in

Autonomous regulation (circulation, respiration), and

other sensorimotor integration. One should not

exclude possible secondary activation of several

common systems of neurochemical regulation of

activity of brain nuclei located in the brainstem –

noradrenergic, dopamine-mediated error, and

acetylcholinergic by dopamine and serotonin. From

the same region the trigeminal-spinal path, regulating

the activity of spinal motoneurons, goes down

solitary-three spinal and Vestibulo-spinal directly

involved in the regulation of the activity of the lower

limbs and walk (Michelle et al. 2009). Intense

rhythmic stimulation of active neurons leads to a

corresponding activation of synaptic contacts and

axon, including the whole complex of pre - and

postsynaptic neurochemical mechanisms. Such

phenomena as long potentiate or depression of neural

networks may underlie the effects observed when

using electrotactile stimulation of the tongue.

Potentiate long-term (Long-termpotentiation, LTP)

and long-term depression (Longterminhibition, LTI),

this enhancement/inhibition of synaptic transmission

between two neurons that persist long after exposure

to the synaptic pathway. LTP is involved in the

mechanisms of synaptic plasticity provides the

nervous system of a living organism the ability to

adapt to changing environmental conditions. Most

theorists of neurophysiology believe that long-term

potentiate together with long-term depression

underlie the cellular mechanisms of learning and

memory (Danilov et al. 2008).

At the moment, the device for electrotactile

stimulation is called PoNS (Portable

Neurostimulator), and its use for stimulation of the

brain in children with cerebral palsy is a new direction

in neurorehabilitation. The matrix, in which are the

electrodes of irregular shape; optimized to stimulate

the most sensitive areas of language (Fig.1A). The

Translingual Neurostimulation in Treatment of Children with Cerebral Palsy in the Late Residual Stage. Case Study

333

matrix itself includes 143 electrodes divided into nine

16 - electrode sectors (Fig.1B). Within each sector,

only one electrode is active at a given time, the rest

are grounded. Stimulation through one electrode

occur simultaneously in nine sectors. The electrodes

are alternated with a frequency of 50 Hz. The

incentive is a triplet of rectangular pulses of

microsecond duration.

Figure 1: The PoNS device (Portable Neurostimulator).

Appearance (A) and close-up of the matrix (B).

Regular stimulation of the PonS device,

activating an extensive region of the brain, increases

the efficiency of existing neural networks, increases

the likelihood of the formation of new synaptic

contacts (synaptogenesis), enhances the innate ability

of the brain to improve motor functions. The purpose

of a successful rehabilitation when such stimulation

to restore motor function or to teach new motor skills

is done by combining specialized exercises with a

wide activation of the brain with the PoNS device.

The studies were conducted in patients with

peripheral and Central vestibular disorders (Lomo

2003, Badke et al. 2011, Amanda and Raza 2014,

Kublanov 2008), multiple sclerosis (Kublanov et al.

2010), stroke and ischemic damage (Danilov et al.

2015), head injury and spinal cord injury

Figure 2: The PoNS device (Portable Neurostimulator).

New design.

(Wildenberg 2011, Wildenberg et al. 2013). High

effectiveness of peripheral neurostimulation in

combination with a specialized physical therapy to

restore motor control of the body, balance, walking,

speech, eye movements, different aspects of

sensorimotor integration was shown. Additional

studies, using functional MRI, unambiguously

confirmed the presence of a powerful activation of

the brain stem and ventral parts of the cerebellum

upon stimulation of the tongue, and also the long-

term effects of after-effect, the persistence of pockets

of activity in the brain of subjects for hours or even

days after the last stimulation (Bach-y-Rita 2003,

Joseph et al. 2011). Additional data analysis showed

that while the activation of subcortical structures of

the brain are changed and the coefficients of coupling

between areas of the cerebral cortex involved in the

integrative processes of training (Petrenko et al.

2017).

2.3 fMRI Data Acquisition and

Postprocessing

To investigate the organization of functions, to

localize activated cortical areas and to assess the

therapeutic effect we used functional magnetic

resonance imaging. Conventional T1- and T2-

weighted images in three orthogonal planes were

obtained also. fMRI was performed on 3.0 T MR-

scanner with BOLD (Blood Oxygenation Level

Dependent) technique. Functional MR images were

acquired using echo-planar imaging (EPI) with

repetition time (TR) = 3000 ms, echo time (TE) = 50

ms, flip angle = 90º, field of view (FOV) = 230 mm

NENT 2018 - Special Session on Neuro-electrostimulation in Neurorehabilitation Tasks

334

and matrix size 128*128. The research was carried out

using active movement functional paradigms for each

extremity (both feet and hands) and active count

paradigm for patients who could perform these tasks

and depending on clinical status, and passive

movement or sensory paradigms was performed for

those, who could not. Besides, a “virtual walking”

paradigm was performed to some patients: they had to

imagine they walk. Each activation paradigm lasted

5 minutes with five epochs of movement, 30 seconds

each. Taking into consideration patients’

hyperactivity, scanning protocol (number of

performed paradigms) could be reduced in some cases

to focus on most weak extremity (hand and leg). To

obtain high resolution images of whole brain for

Talairach coregistration and reslicing along different

planes, we used 3D MPRAGE (Magnetization

Prepared Rapid Acquisition Gradient Echo) – T1-

sequence with the following parameters: repetition

time (TR) = 2000 ms, echo time (TE) = 4,38 ms, flip

angle = 10º, field of view (FOV) = 250 mm, 160 slices

and matrix size 256*256. Processing of neuroimaging

data with the identification of activation sites in each

patient and evaluation of the results were carried out

using the software package SPM12 (Welcome

Department of Imaging Neuroscience, University

College, London, UK) software package running

under MATLAB R2011b (The Mathworks, Sherborn,

MA, USA) programming. Template space was

defined by standard EPI template data in SPM (MNI

coordinates - Montreal neurologic Institute, McGill

University, Montreal, Canada).

3 RESULTS

The first patient before the treatment was able to walk

using multisupporting canes within the room, and for

longer distances used walkers (500 meters or more),

after a course of treatment learned to walk using one

cane single-bearing alternator within the room and in

school, for longer distances uses a multisupporting

cane. The second patient before treatment used

multisupporting canes to walk across the room and on

the street, could not stand alone without support; the

end of the course of treatment learned to walk

independently on a level surface (within the room),

the patient can stand on his own without support, in

school and on the street uses one single-bearing

alternator cane. The third patient before the start of

treatment used to walk the Walker across the room, at

school, and for longer distances used stroller. Upon

completion of the course of treatment the patient has

mastered multisupporting cane across the room, uses

a Walker in school and can go to the playground, for

longer distances use stroller active type. A fourth

patient before treatment went using two single-

bearing alternator canes within the space and outside,

independently and without reliance might stand for a

few seconds at the end of the course of treatment

learned to walk independently on level surfaces,

standing safely alone on the street uses one single-

bearing alternator cane. Patient N5 before treatment

used multisupporting canes for walking, at the end of

treatment mastered walking within the premise of

relying on one single-bearing alternator cane, for

longer distances uses a multisupporting cane. Patients

N6 with the level of development GMFSC 4, before

the treatment could move around within the premises

using a walker, at school and on the street used active

type stroller, at the end of the treatment the patient

learned to walk with the use of multisupporting canes

within the premises and in school, for longer distances

confidently uses a Walker. Also all patients noted

decreased muscle tone and improve balance,

coordination functions. Improvement of balance,

assessed on a scale of Berg, ranged from two to seven

units (average 4.5) and as a percentage of the original

state improvement was observed from 12 to 70 %

(average 31%). (Fig. 3)

Figure 3: Dynamical differences in patients by Berg balance

scale (black – before, grey - after).

According to obtained fMRI data for each subject

individually, we detected activations of

corresponding areas in response to presentation of

movement stimuli, active and passive, located in the

hemispheres, before and after neurostimulation.

First patient showed increased activation in right

hand motor area, and activation in foot motor area in

response to “virtual walking” paradigm after

neurostiumulation (before it was weaker and appeared

in “wrong place”) (Fig.4). Patients N4 and N6 showed

significant changes in activation patterns for foot and

hand motor areas. All other patients also have had

Translingual Neurostimulation in Treatment of Children with Cerebral Palsy in the Late Residual Stage. Case Study

335

more expressed activations in response to different

stimuli, primarily in foot motor areas and in response

to “virtual walking”.

A B

C D

E F

Figure 4: Activation areas in patient N1. A, B – right hand

fingers movement, С, D – count paradigm active response,

E, F – right leg movement activation, before and after

neurostimulation respectively.

4 CONCLUSIONS

Given the limitations and the minimum intensity of

the workouts, the main objective of the study was

limited by the formation of new motor skills. The

patient had to form a new motor skill in 10 sessions,

A B

Figure 5: Activation areas in patient N6. Illustration of

activation pattern in projection of left leg motor area before

(A) and after (B) neurostimulation.

A B

Figure 6: Activation areas in patient N4. Illustration of

activation pattern in projection of right hand motor area

before (A) and after (B) neurostimulation.

to strengthen it and to confidently use in a daily life.

Based on these considerations, it is understandable

why overall motor control (scale FМS) statistically

significantly improved. Since the task was to develop

the skills of motor control, these neural networks

improved their functional activity as a result of the

neurostimulation. This technique is non-invasive,

innovative in the field of neurostimulation, safe and

easy to use. Daily 20 minute stimulation of the tongue

within two weeks increases the innate ability of the

brain to improve motor function, promotes the

formation of new motor skills. Neurostimulation with

the use of PoNS device, combined with therapeutic

exercises (targeted sessions) allows to improve the

efficiency of recovery of motor function and motor

skills development. Functional MRI active

paradigms, with proper and high-quality

implementation is an auxiliary method of the

objective control of efficiency of treatment.

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336

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