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The Relationship Between Regulation Disorders of Sensory Processing (RDSP) and White Matter Abnormalities

Authors:
  • Institute of Ortophonology, Rome, Italy
  • Istituto di ortofonologia Roma
  • Istituto di Ortofonologia

Abstract and Figures

Background: The Regulatory-Sensory Processing Disorder (RSPD), defined on the diagnostic manual DC: 0-3 R as "the child's difficulties to regulate his own behaviour, his physiological, sensory, attention, motor or affective processes and difficulties in organizing a state of calm, of alert or a positive affective state", is a neurodevelopmental disorder of the early infancy for which it has not yet been determined a clear etiopathology. Clinical practice has led us to hypothesize an association between "minor" injuries of the white matter, in particular periventricular hyperyntensities (PVHs) and RSPD. Method: We conducted a prospective clinical and neuroradiological study of 20 children aged between 2 and 4 years; the sample was divided into children with RDSP (N = 10) and healthy children (N = 10). Findings: The MRI revealed the presence of a low-grade non-cystic diffuse hyperintensity of the periventricular white matter in 8 of the 10 children with RSPD. In none of the healthy children these anomalies were found. Conclusion: The findings of our study revealed that the RSPD may be associated with periventricular hyperyntensities (PVHs), so demonstrating a probable neurobiological origin of the disorder. An interesting fact is that in the children with RSPD we analyzed, we found a posterior-anterior gradient in the localization of the altered PVWM areas, in two cases with involvement of the deep WM of temporal and parietal areas, and this seems to be in accordance with the estimated dysfunction of the associative posterior and limbic areas, with an inevitable impact on the child's emotional sphere too. However, further studies will be necessary to define the specificity of those neuroimaging findings that can be used as RSPD markers.
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The Relaonship Between Regulaon Disorders of Sensory Processing (RDSP)
and White Maer Abnormalies
Vanadia E1, Di Renzo M1, Trapolino D2, Racinaro L1 and Rea M1
1Instute of Ortofonologia (IdO), Via Tagliamento Salaria, Rome, Italy
2Child Neuropsychiatry School, University of Palermo, Piazza Nicola Leoa, 4, 90127 Palermo, Italy
Corresponding author: Elena Vanadia, Instute of Ortofonologia (IdO), Via Tagliamento Salaria 30, 00198, Rome, Italy, Tel: 0039-068542038;
Fax: 0039-3281421235; E-mail: e.vanadia@ortofonologia.it
Received: Apr 30, 2016; Accepted: May 30, 2016; Published: June 02, 2016
Abstract
Background: The Regulatory-Sensory Processing Disorder
(RSPD), dened on the diagnosc manual DC: 0-3 R as
"the child's dicules to regulate his own behaviour, his
physiological, sensory, aenon, motor or aecve
processes and dicules in organizing a state of calm, of
alert or a posive aecve state", is a
neurodevelopmental disorder of the early infancy for
which it has not yet been determined a clear eo-
pathology. Clinical pracce has led us to hypothesize an
associaon between "minor" injuries of the white maer,
in parcular periventricular hyperyntensies (PVHs) and
RSPD.
Method: We conducted a prospecve clinical and
neuroradiological study of 20 children aged between 2
and 4 years; the sample was divided into children with
RDSP (N = 10) and healthy children (N = 10).
Findings: The MRI revealed the presence of a low-grade
non-cysc diuse hyperintensity of the periventricular
white maer in 8 of the 10 children with RSPD. In none of
the healthy children these anomalies were found.
Conclusion: The ndings of our study revealed that the
RSPD may be associated with periventricular
hyperyntensies (PVHs), so demonstrang a probable
neurobiological origin of the disorder. An interesng fact
is that in the children with RSPD we analyzed, we found a
posterior-anterior gradient in the localizaon of the
altered PVWM areas, in two cases with involvement of
the deep WM of temporal and parietal areas, and this
seems to be in accordance with the esmated dysfuncon
of the associave posterior and limbic areas, with an
inevitable impact on the child's emoonal sphere too.
However, further studies will be necessary to dene the
specicity of those neuroimaging ndings that can be
used as RSPD markers.
Keywords: Regulaon disorders of sensory processing;
Periventricular hyperyntensies; Neurodevelopmental
disorder; Child neurological dysfuncons
Introducon
In developmental neuroscience, the most recent ndings
conrm the wide plascity of the nervous system in the early
years of life, both in the case of typical development and in the
presence of genec abnormalies or lesions. The plascity is
greater during the so-called "crical periods" in which the
environment has a beer chance to play a posive inuence,
but also potenally negave ("maladapve plascity") on the
development of the nervous system [1]. As regards the scope
of clinical research and healthcare organizaon, these
discoveries make it increasingly important the weight of early
diagnosis and mely treatment of developmental disabilies,
starng from psychomotor and sensory disorders ll to those
mental and relaonal.
Research in the eld of child mental health has led to an
important transformaon in the knowledge of early
psychopathological disorders.
At the same me, great aenon has been paid to the so-
called “minor” disorders of perceptual-motor development
such as the Developmental Coordinaon Disorder (DCD) and
the disorders of “new denion” such as the Decit in
Aenon, Motor control and Percepon (DAMP) and the
Regulaon Disorders of Sensory Processing (RDSP),
aributable in most, if not in all cases, to a dysfuncon in the
processing and/or integraon of sensory-percepve
informaon [2]. This aspect refers to the concepts of
"connecvity" and "system", to the extent that it does not
require a macroscopic lesion to the onset of the disorders, but
it is quite conceivable a malfuncon of circuits and bundles of
intra- and interhemispheric bers that underlie them [3].
In 2002, Hadders-Algra’s team published an interesng work
on minor neurological dysfuncons asserng the certain
existence of a correlaon between these and adverse perinatal
factors, the descripon of which, however, remained
unspecic [4].
The current hypothesis is that a minimal brain damage
origins the minimum brain dysfuncon that determines the
minor neurological dysfuncon. The quesons that remain
unresolved are: what is meant by minimal brain damage
and/or dysfuncon? How much the individual vulnerability,
presumably genec, it is crucial in the presentaon of a clinical
Case Report
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framework rather than another for the same period of onset
and topography of the lesion? Moreover, how much the
environment aects through epigenec mechanisms?
Currently the majority of neuroradiological and clinical
neuropsychiatric studies are aimed at idenfying
intensiometric and morphological features which allow to
associate specic neuroimaging characteriscs to certain
developmental disorders, with the ulmate goal of increasing
the predictability of injuries and the ancipate both diagnosis
and therapy [5-7]. At the same me great aenon is paid to
the research of the so-called "suscepbility genes" that are
typical of certain diseases [8]. In the context of
neurodevelopmental disorders lies a clinical framework whose
diagnosis is steadily increasing, that is the Regulaon Disorders
[9].
As part of neurodevelopmental disorders, there is another
disorder of early childhood, the Regulatory-Sensory Processing
Disorder (RSPD) dened on the diagnosc manual DC: 0-3R as
"the child's dicules to regulate his own behaviour, his
physiological, sensory, aenonal, motor or aecve
processes and dicules in organizing a state of calm, of alert
or a posive aecve state". For the diagnosis must be present
sensory processing and motor dicules and a specic paern
of behaviour, characteriscs that allow the denion of the
"type" hypersensive, hyposensive (with the relave
subtypes) or impulsive/disorganized [10].
At the base of the RSPD denion, there are the works of Dr.
Anne Jean Ayres, an occupaonal therapist with a PhD in
developmental psychology, who had always worked at the
Brain Research Instute at the University of California in Los
Angeles. In the course of her research at this instuon, she
coined the term "sensory integraon dysfuncon" to describe
what she considered the basis of learning disabilies of which
she was dealing. These works precisely inuenced the sort of
the publishing of the diagnosc manual DC: 0-3 and then of its
review DC: 03R in which the regulaon disorders clearly refer
to the abnormal processing and sensory integraon. Another
sphere of inuence concerns the presence and nature of
sensory percepon disorders in ausm spectrum as well as on
the development of dierent methods of treatment of
children "with special needs" [11].
Clinical pracce has led us to hypothesize an associaon
between "minor" injuries of the white maer (WM) and
neurodevelopmental disorders with early onset, which will be
the object of the research detailed below.
The main goal of our work was to demonstrate the
associaon between low grade non-cysc diuse lesions of the
periventricular white maer (PVWM) presumably aributable
to a prenatal onset and specic neuropsychiatric paerns with
onset in early childhood.
The hypothesis is that these abnormalies of the PVWM can
determine an alteraon in brain connecvity systems with a
pathogenic role in certain neurodevelopmental disorders with
onset in the rst years of life, parcularly in RDSP. In favour of
this hypothesis, there are many scienc works that, through
the use of neuroimaging techniques, such as the high-
resoluon MRI and tractography, show the existence of a
dense network of intracerebral bers and alteraons of traits
or bundles of bers corresponding to convenonal MRI
pictures as diuse hyperintensity of the PVWM in FLAIR
sequences (Fluid Aenuated Inversion Recovery). FLAIR is a
pulse sequence used in MRI that allow to visualize the brain in
T2 weighted images and to suppress cerebrospinal uid eects
on the images, so it is used to bring out the periventricular
hyperintense lesions.
It also seems interesng to speculate that early insults,
presumably hypoxic / ischemic, borne by the subplate can
determine not so much macroscopical alteraons but rather
funconal, aecng mulple neural circuits that in the
subplate neurons nd an important relay point between
thalamic, brainstem and subcorcal aerent bers and mainly
directed to the cortex eerent bers.
Methods
Parcipants
We conducted a prospecve clinical and neuroradiological
study of 20 children (15 males and 5 females) aged between 2
and 4 years (mean = 25.5; SD = 6.5). The sample was divided
into children with RDSP (N = 10) and healthy children (N = 10).
All children were born between the 37th and 40th gestaonal
week, with a birth weight appropriate for gestaonal age.
None of the children had a documented perinatal distress,
with an Apgar index ranging from 8 to 10 at the rst and the
h minute.
Procedures
The diagnosis of RDSP was formulated according to the DC:
0-3R, by excluding specic visual or hearing pathologies in
accordance with the inclusion criteria; the clinical evaluaon
was integrated with neurophysiological tests: the visual and
acousc Evoked Potenals (EP) of the sperimental group of
children resulted aected in most cases by a modest increase
of latencies, while in only 1 child epilepform EEG
abnormalies were present. In healthy children no
neurophysiological anomaly was found.
All the MRI analyses were performed using the RM Philips
Full 1.5 T equipment; the images were acquired in SE technical
(spin-echo) and FAST-SE with mulplanar axial, sagial and
coronal weighngs in T1 and T2, with and without infusion of
paramagnec contrast.
Results
As shown in Table 1, in 8 of the 10 children with RDSP the
MRI revealed the presence of a low grade non-cysc diuse
hyperintensity of the periventricular white maer typically
noceable in FLAIR sequences, not associated with
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morphological and structural abnormalies of the lateral
ventricles (LV); this value was signicantly higher than that
found in the group of healthy children (p<0.001). In 5 of the 8
children the PVHs showed a posterior-anterior grading, with
involvement of the deep temporo-parietal area in 2 of them; in
6 of the 8 children with posive MRI, the PVHs was associated
with hypoplasia of the corpus callosum. In none of the healthy
children these anomalies were found. In Table 1 the
informaon of both groups are shown.
Table 1 MRI normal and pathologic ndings of the sample, with the specic typology of pathologic ndings.
RDSP children Healthy children
MRI Normal Findings N = 2 N = 10
MRI Pathologic Findings N = 8 N = 0
Typology of MRI Pathologic Findings
Patient 1
PVH (> post.); CCH
Patient 2 PVH (> ant.); CCH
Patient 3 PVH (> temporo-parietal)
Patient 4 PVH (ant.)
Patient 5 PVH; CCH
Patient 6 PVH (> temporo-parietal); CCH
Patient 7 PVH (> post.); CCH
Patient 8 PVH (> post.); CCH
RDSP: Regulation Disorders of the Sensory Processing; PVH: Periventricular Hyperintensity; CCH: Corpus Callosum Hypoplasia.
Figures 1 to 4, show some of the ndings highlighted in 3
children of the RDSP group and in 1 of the control group
children (Figure 4).
Figure 1 Paent 1: Hyperintense aspect of the PVWM and in parcular of the back secons. Hypoplasc CC.
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Figure 2 Paent 2: Mild hyperintensity of the PVWM adjacent to the frontal horns of LV. Modestly hypoplasc CC.
Figure 3 Paent 3: Hyperintensity of the PVWM and parcularly deep temporo-parietal. Mild hypoplasia of the posterior
poron of the CC.
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Figure 4 Paent 9: Normal MRI aspect.
Discussion
The RDSP is a neurodevelopmental disorder with onset in
early childhood. In literature, there is only one study [12] in
which the associaon between the RDSP and neuroimaging is
invesgated. Owen and colleagues explored the role of white
maer microstructural abnormalies in Sensory Processing
Disorders (SPD) using Diusion Tensor Imaging (DTI), nding a
signicant decrease in Fraconal Anisotropy (FA) and increase
in Mean Diusivity (MD) and Radial Diusivity (RD) in the SPD
cohort compared to controls, primarily involving posterior
white maer including the posterior corpus callosum,
posterior corona radiata and posterior thalamic radiaons.
These ndings were correlated with stascal signicance with
behavioural measures, parcularly with auditory, mulsensory
and inaenon scores.
These data are in line with what we found through MRI
pictures and the hypotheses about anatomical and funconal
correlaons that will be stated below. Compared with Owen’s
work, whose sample consists of children aged 8 to 11 years,
our study focuses on pre-school children, according to the
concepts of early diagnosis and plascity. To have found a
stascal signicance between behavioral paern aributable
to RSPD and neuroimaging data is, in our view, an important
starng point for further studies on the possible
neurobiological or epigenec vulnerabilies of some children
respect to RSPDs but, in terms of development, especially on
the posive eects of a therapy centered on the specic needs
of each child.
Therefore only few data concerning the neuroanatomical
and neurofunconal basis of the disorder are currently
available.
Recent studies related to other neurodevelopmental
pathologies, such as Ausm Spectrum Disorders (ASD) and
intellectual disabilies of genec origin, have shown the
presence of alteraons in the neural connecvity systems
[13,14], as was the case in the epileptologic eld with the new
denion of “system epilepsy” (SystE) [15].
The ndings of our study revealed that the RDSP may be
associated with periventricular hyperyntensies (PVHs), and in
our sample this happens in the 80% of cases. According to the
results of the research in eld of neuroscience and
neuropathology about the relaon between psychiatric
diseases and PVHs, it is hypothesized that PVHs could be due
to one of the three main causes: ependymal loss, diering
degrees of myelinaon in adjacent bre tracts and cerebral
ischaemia with associated demyelinaon [16].
A more accurate analysis of the topography of the WM next
to LV paves the way for anatomical and funconal correlaon
studies in our opinion very interesng:
The lateral poron of the frontal horn of LV is covered by
inferior fronto-occipital fasciculus (IFOF) that ‘can be
considered as a “mul-funcon” bundle, with each
anatomical subcomponent subserving dierent brain
processing The supercial layer and the posterior
component of the deep layer, which connects the occipital
extra-striate, temporo-basal and inferior frontal corces,
might subserve semanc processing. The middle
component of the deep layer could play a role in
mulmodal sensory–motor integraon. Finally, the anterior
component of the deep layer might be involved in
emoonal and behavioural aspects’ [17];
The upper poron of the frontal horn of LV is covered by
the corpus callosum (CC), that is the major neural pathway
that connects homologous corcal areas of the two
cerebral hemispheres [18];
The body of the LV is laterally covered by the bers of the
internal capsule formed by bundles of bers called
“projecon bers” because they start from or arrive to the
cortex, leading informaons from thalamus and basal
ganglia, with the funcon to control the voluntary motor
skills and receive the sensivity of the body, and from the
arcuate fasciculus, which connects the areas of Broca and
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Wernike and transmits auditory-verbal messages aer they
have reached the associave parietal-temporal-occipital
area; frontal-subcorcal cognive and limbic feedback
loops modulate higher cognive funconing [19];
The lower poron of the LV is in relaon to the body of
fornix, which is an interhemispheric commissural formaon
in the shape of a "C" that puts the hippocampus in
communion with the mammillary bodies and then with the
thalamic nuclei and the cingulate gyrus (posterior bers)
and the amygdala (anterior bers); it is involved in
cognive funcons such as memory and complex visual
percepon;
The temporal horn of LV is covered by opcal radiaon in
the depths of the middle temporal gyrus. The auditory
radiaons cross the opcal radiaons to the level of the
roof of the lower horn.
It therefore seems clear the importance of the bundles of
periventricular, associave and commissural bers in sensory-
perceptual processing and integraon, that is the basis for
mulple funcons including learning, visual-spaal, motor
planning, social behaviour and language.
The second interesng fact is that in children with RDSP we
analysed, we found a posterior-anterior gradient in the
localizaon of the altered PVWM areas, in two cases with
involvement of the deep WM of temporal and parietal areas,
and this seems to be in accordance with the esmated
dysfuncon of the associave posterior and limbic areas, with
an inevitable impact on the child's emoonal sphere too [20].
Moreover, the fact that the widespread periventricular
hyperintensity was not associated with morphological and
structural abnormalies of the LV gives evidence of a prenatal
origin of the damage, in accordance with the hypothesis that
intrauterine repeated insults in early gestaonal ages can
determine a damage to the WM (being aected the precursors
of oligodendrocytes and potenally the IVZ) of subplate and
neural circuits that depend on it [21].
In parcular, the intensiometric characteriscs (non-cysc
widespread hyperintensity of the PVWM in FLAIR images) lay
for glioc lesions with loss of oligodendrocytes and axonal
damage.
It should also be remembered that the neurons of the
subplate show both glutamatergic and GABAergic acvity and
are involved in the early formaon of funconal circuits not
only at a local level of the subplate itself, but also at a distance
with cortex and thalamus.
Although the subplate is a transional structure, which will
gradually disappear aer the birth, its role is crucial as it forms
a hub between aerent projecons from the brainstem, the
thalamus and subcorcal nuclei and eerent projecons
mainly addressed to the thalamus, the cortex and the spinal
cord; however, most of the thalamic axons not establish
synapses at the level of subplate, remaining "on hold" to enter
the corcal plate around the 24th GW [22].
A very interesng fact is that some thalamocorcal bers
move to the level of corcal plaque in the visual,
somatosensory, auditory and associave areas constung a
connecvity "sensory-driven" framework, while others remain
engaged in the formaon of endogenous circuits at the level of
the subplate. The laer undergoes to a gradual disappearance
around the 36th GW; however, it is present as a band below
the corcal VI layer and represents an important indicator of
the growth of corcocorcal and callosal circuits [23].
It seems plausible to assume that insults to the subplate can
thus determine an alteraon in the formaon of mulple
neural circuits that remain over its disappearance.
Finally also the nding of hypoplasia of the corpus callosum,
and in parcular of the posterior third, is in accordance with
the clinical presentaon of our paents, as in the Witelson
division [24], it would contain the connecng bers between
the upper temporal, parietal and occipital regions closely
related with auditory-verbal and visual-perceptual funcons.
Our hypothesis, which deserves funconal neuroimaging
and ber tracking invesgaons, then is that intensiometric
alteraons of the WM, and in parcular the non-cysc
periventricular leukomalacia should be underpinned by
abnormalies borne by the intra and interhemispheric
connecon bers so determining the formaon of dis-
funconal circuits which above all alter the percepon and
sensory integraon processes. This leads to the inevitable
consequences on motor planning and control, but also and
especially on the behaviour on the relaonship and emoon.
In fact, complex cognive-emoonal behaviours have their
basis in dynamic coalions of networks of brain areas, none of
which should be conceptualized as specically aecve or
cognive. ‘Central to cognive-emoonal interacons are
brain areas with a high degree of connecvity, called hubs,
which are crical for regulang the ow and integraon of
informaon between regions [25].
The future perspecve is to integrate the instrumental
studies with single-voxel spectroscopy evaluaon and with the
applicaon of the diusion ber tracking tensor in order to
idenfy quantave and quality changes of bundles of bers
and of the specic areas aected by the damage in relaon to
the clinical presentaon and as markers that support the
diagnosis [26].
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JOURNAL OF NEUROLOGY AND NEUROSCIENCE
ISSN 2171-6625 Vol.7 No.3:108
2016
© Copyright iMedPub 7
... Związek pomiędzy zaburzeniami sensorycznymi a jakością chodu najprawdopodobniej wynika z zaburzeń schematu ciała i orientacji przestrzennej, o czym świadczą inne badania [21,22]. W badaniach neurofizjologicznych wykazano ponadto związek RDSP z anomaliami funkcji aksonów w korze mózgowej [23]. ...
... The relationship between sensory disturbances and gait quality most likely results from disturbances in the body schema and spatial orientation, as evidenced by other studies [21,22]. Moreover, neurophysiological studies have shown a relationship between RDSP and abnormalities of axonal function in the cerebral cortex [23]. ...
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