DSM-5 Application for Sensory Processing Disorder Appendix A (part 1)

Research (PDF Available) · December 2015with 5,754 Reads
Cite this publication
Abstract
DSM-5 Proposal for inclusion of Sensory Processing Disorder
Appendix A Part 1
3
Appendix A:
Evidence For Sensory Processing Disorder
INTRODUCTION
In 1999, the KID Foundation formed the Sensory Processing Disorder (SPD) Scientific Work
Group (SWG), a multidisciplinary collaboration of leading scientists from university-based
research institutions, to stimulate cross-disciplinary research into sensory processing and sensory
processing impairments, supported by the Wallace Research Foundation and the NIH. Areas
included in past and ongoing research include: neuro-physiological reactions to sensory stimuli
in children and adults; sensation processing at the neural level, sensory-related behavior,
attention and emotion regulation, animal models of neuropathology, genetic studies, and studies
related to clinical issues such as the utility, sensitivity/specificity, and discriminate validity of the
diagnosis of SPD.
The SPD Scientific Work Group is submitting this proposal to include Sensory Processing
Disorder in the Diagnostic and Statistical Manual V. This appendix to the application includes a
summary of empirical data providing evidence about the existence of the SPD syndrome, based
primarily on studies conducted by SWG members. The information herein includes: published,
submitted, and in process work. Ongoing research is occurring in all areas cited in this Appendix.
Members and affiliations of the SPD SWG are:
Margaret Bauman MD, Harvard University Medical School, Boston, MA
Margaret Briggs-Gowen PhD, Yale University, CO
Alice Carter PhD, University of Massachusetts, Boston, MA
Patricia Davies PhD, CO State University, Fort Collins, CO
Winnifred Dunn PhD, University of Kansas Medical Center, KS
John Foxe PhD, U of Dublin, Ireland; and Nathan Kline Institute, NYC, NY
William Gavin PhD, Colorado State University, Ft. Collins, CO
Hill Goldsmith PhD, University of Wisconsin – Madison, WI
Moya Kinnealey PhD, Temple University, Philadelphia, PA
Michael Kisley PhD, University of Colorado in Colorado Springs, CO
Jane Koomar Ph.D, Spiral Foundation, Watertown, MA
Ed Levin PhD, Duke University, Chapel Hill, NC
Jeffrey Lewine, PhD, University of Kansas Medical Center
Sophie Molholm PhD, Nathan Kline Institute NYC, NY
Teresa May-Benson ScD, Spiral Foundation, Watertown, MA
Shula Parush PhD, Tel Aviv University, Jerusalem, Israel
David Pauls PhD, Harvard University Medical School, Boston, MA
Mary Schneider PhD, University of Wisconsin – Madison, SI
Roseann Schaaf PhD, Thomas Jefferson University, Philadelphia, PA
Sinclair Smith ScD, Drexel University and Temple University, PA
Barry Stein PhD, Wake Forest College, IO
Appendix A Part 1
4
The KID Foundation Research Institute: Lucy Jane Miller PhD, Roianne Ahn PhD,
Barbara Brett-Green PhD, Jennifer Brout PsyD, and Sarah Schoen, PhD, Denver,
CO.
Primary support for studies cited in this Appendix came from the Wallace Research
Foundation and from the National Institutes of Health.
Pennington’s Model of Syndrome Validation
This Appendix is organized by Pennington’s model of syndrome validation (Pennington, 1991;
Pennington, 2002) which suggests that empirical data, when provided in five areas, increases the
likelihood that a new syndrome is valid. The five domains are: neuropathology, signs and
symptoms, developmental trajectory, etiology, and treatment effectiveness (see Figure 1 below).
“If a syndrome is valid, it will satisfy tests of both convergent and discriminant validity across
[these] levels of analysis” (Pennington, 1991, p. 24). Thus, if a condition is homogeneous across
these five domains, and can discriminate the condition from other disorders across these five
domains, likely it is a syndrome.
Figure 1. Pennington’s Model of Syndrome Validation
Syndrome Validation
Pathogenesis
Etiology 1
Dx1
Based on model by Pennington, 1991, p. 25
Signs & Symptoms
Dev. Course
2
1
2
1
2
1
2
1
2
Treatment
Dx2
© KID Foundation
Adapted from Pennington, 1991, p 25
Pennington (2002) suggests that most disorders are defined first behaviorally with a set of signs
and symptoms that comprise a phenotype of the disorder. He further suggests that while genetic
and neuropathology studies can not progress without designated behavioral phenotypes, brain
and genetic studies can force revision in phenotype descriptions, thus refining the syndrome
definitions and subtypes. Also needed to verify a syndrome is evidence that the condition is
universal, e.g., all individuals with the condition exhibit similar patterns, and evidence that the
condition is specific, e.g., individuals without this disorder do not exhibit these signs.
Scientific Workgroup Research on SPD
Empirical data on sensory processing and SPD were gathered in each area of Pennington’s
model, by the SPD scientific workgroup 1995 – 2007. The SPD studies reported here are
incorporated within current, ongoing programs of research. Research on SPD is in its early
Appendix A Part 1
5
stages. Empirical data derived from multiple ongoing programs of research are summarized in
the following pages. The following report highlights the consistent results that have emerged so
far:
First, individuals with SPD have a different and less organized pattern of responding to
sensory stimulation in both the autonomic nervous system and central nervous system.
Psychophysiologic data (e.g. electrodermal activity and EEG/event-related potential
studies), suggest that individuals with SPD exhibit atypical and/or unique patterns of
physiological functioning during and after sensory stimulation (e.g., differences from
typical controls and individuals with other disorders on autonomic nervous system
arousal and state, and on cortical function during the classic stages milliseconds after
stimulation, e.g., P50, P100, N100, and P200). In addition, cortical multisensory
integration is much less organized in those with SPD, than it is in controls.
Second, data suggest that clinical groups such as ADHD, Autistic Spectrum Disorder and
other DSM-IV-TR conditions can be discriminated from SPD using physiological
outcomes, standardized performance assessments, and parent/teacher-report measures.
Third, familial/twin studies suggest a genetic component to the etiology of SPD.
Fourth, animal studies suggest that dopamine-related correlates of SPD exist and specific
pharmacological agents can impact one suggested mechanism of the disorder, sensory
gating, and also affect dopamine-related correlates of SPD exist.
Fifth, human studies suggest that a specific sensory-based occupational therapy approach
may be effective in ameliorating features of the disorder.
Sixth, the clinical utility of adding SPD to the DSM-V-TR, and the ability diagnose SPD
accurately were studied. A new performance assessment the Sensory Over and Under-
Responsivity Scale has an 88% overall hit rate in identifying SPD, with a 1.3% false
positive rate.
Appendix A Part 2
26
RESPONSE TO QUESTION 1: DESCRIPTION OF NEW DISORDER
Summary
100 Character Summary – Sensory Processing Disorder: persistent atypical over-responsivity
or under-responsivity to neutral sensation
Sensory Processing Disorder is characterized by persistent atypical over- or under-responsivity
to neutral sensations. The existence of a unique syndrome is supported by data in the five
syndrome validation areas outlined by Pennington (1991, 2002): neuropathology, signs and
symptoms, developmental trajectory, etiology, and treatment effectiveness (Pennington, 2002).
Neuropathology data (from human and primate studies) have found that some autonomic and
central nervous system functions of individuals with SPD are abnormal compared to typical
controls. Signs and symptoms data demonstrate that individuals with SPD can be discriminated
from typically developing individuals in areas of attention, emotion, and sensory processing on
both behavioral and physiological measures resulting in problems in daily life functioning.
Developmental trajectory studies in animal and human models have demonstrated that
behavioral signs and symptoms, along with sensory gating (one likely underlying mechanism of
SPD), improve with maturity. Etiology findings based on twin studies and animal models have
linked SPD to genetic factors, prenatal and birth risk factors, environmental exposures, and
developmental and health factors. Finally, treatment effectiveness data demonstrate that
sensory-based interventions for SPD result in better outcomes compared to no-treatment and
active-placebo controlled treatments.
Definition/Description of Sensory Processing Disorder
The essential behavioral feature of Sensory Processing Disorder (SPD) is the presence of
persistent atypical response patterns to neutral, everyday, “non-noxious” sensory stimuli. When
the responses to sensation of individuals with SPD are compared to the responses of typically
developing individuals at a similar age and stage of development, significant impairments are
observed in SPD that are associated with problems in social, academic, and/or occupational
functioning. In SPD, a response to sensory stimulation occurs; however, behavioral responses are
poorly regulated e.g., the duration of behavioral response, the intensity of the responses, and/or
the type of responses made to neutral levels of sensory input, are abnormal, resulting in poorly
graded responses relative to the sensory input, and hence, non-adaptive behaviors are observed.
The range of normal responses to sensory input is quite large. A response pattern is only labeled
disordered” when an individual’s ability to detect, regulate, interpret, or organize responses to
sensory input is so impaired that abnormal attention, emotion, cognition or motor responses are
observed. Abnormalities in an individual can occur in one or more of the following sensory
domains: auditory, visual, tactile, olfactory, gustatory, vestibular (relation to gravity seen in
response to changes in position or movement), or proprioception (sensation from muscles and
joints). Because the disorder is related to sensory stimulation in the environment, patterns of
responsivity may vary throughout the day and from day to day, resulting in behavioral or
emotional symptoms that may appear inconsistent across contexts (ICDL, 2005; Zero To Three,
2005; Miller, 2007c).
Appendix A Part 2
27
Two atypical patterns (subtypes) are observed in SPD:
Sensory Over-Responsivity, where individuals exhibit severe and persistent aggression,
withdrawal, or fear (“fight or flight” reactions) to specific sensory stimuli, typically
perceived as neutral, non-noxious, and harmless by others.
Sensory Under-Responsivity, where individuals exhibit reactions that are slower and less
intense and less precise than is typical (e.g., unaware of basic sensory input easily
recognized by most people such as pain, hearing their name called).
Empirical Evidence
Neuropathology
Measures of Peripheral Autonomic Nervous System Function in SPD
In Children.
Investigation into the neuropathology of SPD began with studies of the sympathetic nervous
system because individuals with SPD exhibit dramatic “fight or flight” reactions to levels of
sensory stimuli perceived as non-aversive or “neutral” by typically developing individuals.
Sympathetic nervous system function may be examined using electrodermal activity (EDA).
EDA measures changes in the electrical conductance of the skin associated with sympathetic
nervous system activation of eccrine sweat glands (Boucsein, 1992), and is considered a
sensitive index of sensation, attention, emotion and cognition, related to sympathetic arousal
(Critchley, 2002). Consequently, the Sensory Challenge Protocol was created using EDA
outcome measures to establish a reliable laboratory paradigm for examining children’s reactions
to sensory stimuli by recording EDA resulting from serial introduction of sensory stimuli in five
sensory domains during a “space trip” (Miller, McIntosh et al., 1999). Figure 2 displays the EDA
responses of a typically developing child during the Sensory Challenge Protocol. Figure 3 shows
two examples of EDA from a Sensory Over-Responsive child, with larger amplitudes, more
frequent responses, and no habituation. Figure 4 displays the EDA responses of a Sensory
Under-Responsive child.
Appendix A Part 2
28
Figure 2. Example of Electrodermal Response in a Typically Developing Child
©KID Foundation
From Miller, et al. 1999
Figure 3. Examples of Electrodermal Responses in Two Children with Sensory Over-
Responsivity
©KID Foundation
From Miller et al 1999
Figure 4. Example of Electrodermal Responses in a Child with Sensory Under-
Responsivity
From Miller et al 1999
Using the Sensory Challenge Protocol, McIntosh and colleagues (McIntosh, Miller et al., 1999)
demonstrated that children with SPD respond at higher amplitudes and habituate more slowly to
stimuli than typically developing children (see Figure 5 below). They also found a significant
association between children’s physiologic responses and their functional behavior scores based
on parent report.
Appendix A Part 2
29
Figure 5A. Discriminating Children with SPD from Typically Developing Children
on Electrodermal Reactivity
Discriminating Children with Sensory Modulation
Dysfunction and Typically Developing Children (n=25)
0
0.02
0.04
0.06
0.08
0.1
0.12
0.14
1 2 3 4 5 6 7 8
Trial s
Log Magnitude of Peak (micromhos)
SMD
Typical
Group
McIntosh, Miller et al., 1999
© KID Foundation
From McIntosh 1999a
Because regulation of an individual’s reactivity involves a balance of activity occurring within
both the sympathetic and parasympathetic divisions of the autonomic nervous system, Schaaf
and colleagues studied the Sensory Challenge Protocol using parasympathetic nervous system
function outcomes in children with SPD. Their pilot study, using the parasympathetic measure
vagal tone (Porges, 1985), demonstrated that children with SPD have poorer parasympathetic
regulation than controls (p < .05) (Schaaf, 2001).
In Adults.
Smith and colleagues, (Smith, 2004) have been using autonomic nervous system measures to
study adults with sensory-over-responsivity. Sixteen adults (8 SPD) were studied. Sympathetic
nervous system response was assessed by continuously monitoring electrodermal activity as well
as heart rate and peripheral blood flow. After 15 minutes of acclimation, 25 sensory stimuli were
administered (5 each in 5 sensory systems: olfactory, auditory, visual, tactile, and vestibular).
Smith et al. (2004), report that the SPD group demonstrated a significantly larger skin
conductance response following tactile stimuli (68±28%) than the control group (20±5.6%; p =
0.04). There were no differences in heart rate between groups or in the initial peripheral blood
flow response. Smith et al., suggest that because the sensory over-responsive group had a
relatively large initial skin conductance response to tactile stimuli without a relatively large
peripheral blood flow response, sensory over-responsive persons may have an attenuated
adrenergic sympathetic activation of the peripheral vasculature and/or an elevated cholinergic
sympathetic activation of eccrine glands. In another study, Sensory Over-Responsive adults who
score below –2 two standard deviations on the sensory sensitivity portion of the Adult Sensory
Appendix A Part 2
30
Profile when presented with 108 dB white noise via headphones demonstrated an elevated initial
skin conductance (Brown, 2002).
Measures of Central Nervous System Function in SPD
Autonomic nervous system measures such as electrodermal activity, vagal tone, heart rate and
peripheral blood flow have the advantage of measuring an individual’s interaction with the
environment (Stern, Ray et al., 2001), however we can only infer from these measures what
might be occurring in the central nervous system.
Thus, with evidence of peripheral nervous system dysfunction, further investigation into the
neuropathology of SPD naturally evolved into studies of central nervous system function. One
measure being utilized is electroencephalography (EEG), which complements peripheral nervous
system measures by providing a direct measure of cortical activity. Often event-related potentials
(ERPs) are extracted from EEG to examine responses specifically related to sensory stimulation,
Sensory Gating
In Children. Davies and Gavin (Davies, 2007) hypothesized that children with SPD
would demonstrate different sensory gating and organization of sensory information than
controls. Using a paradigm similar to that used previously for assessment of responsivity in
ADHD (Olincy, Ross et al., 2000), autism (Kemner, 2002) and traumatic brain injury
(Arciniegas, Adler et al., 1999), Davies and Gavin used real-time measures of brain activation
during the processing of sensory stimuli (clicks) which were time-locked to the occurrence of
each sensory stimuli (i.e., P50, N100 and P200). They measured both amplitude (in microvolts)
and latency (in milliseconds) of responses to sensory stimulation to examine brain processing of
auditory sensory stimuli in 28 children with SPD and 25 typically developing children, group
matched on age, 5-12 years, and gender (Davies & Gavin, 2007).
First, the paradigm presented tones at different frequencies and intensities known to
effect the threshold at which sensory information is detected e.g., N100 and P200. Second, the
paradigm presented clicks at 500ms intervals. It has been established that typically developing
individuals have a smaller positive ERP response to the second of the two clicks at about 50 ms
post-stimulation (P50), representing suppression of the second response, frequently labeled
sensory gating.
Results for the first paradigm illustrated that children with SPD, unlike controls, did not
have an increased response to increased intensity of stimulation. In addition, results for the
second paradigm indicated that children with SPD had less auditory sensory gating than controls
(p = .04), suggesting that children with SPD have more difficulty filtering out or “gating”
irrelevant sensory information. This may explain in part the behavioral signs and symptoms such
as distractibility, impulsiveness, disorganization and emotional ability seen in SPD. In addition,
sensory gating improved with age for typically developing children, but not for children with
SPD.
Appendix A Part 2
31
Examining group difference scores revealed that some children in the SPD group
demonstrated over-responsivity compared to controls, while others demonstrated under-
responsivity relative to controls. Children with SPD could be divided into Sensory Over
Responsive and Sensory Under Responsive based on their sensory gating scores relative to
typical children.
In Adults. Concurrently, Kisley and colleagues have been examining the relation of
behavioral over-responsivity after auditory stimulation to the underlying neural mechanisms of
auditory processing in adults. Using an EEG/ERP paradigm (including sensory gating and
mismatched negativity) a significant correlation was found between sensory gating and sensory
over-responding in a healthy adult sample (Kisley, Noecker et al., 2004). Responses at P100
were correlated with modulation of stimuli, whereas response at N100 was correlated with
filtering out background sounds. These results suggest that individuals with SPD over-process
stimuli of low salience rather than over-responding to all stimuli in their environment. Typically,
as stimulus “relevance” goes down, so too does the brain’s automatic response to that stimulus.
But individuals that endorse higher rates of sensory over-responding, as quantified by the Adult
Sensory Profile, exhibit similar amplitude brain responses to nearly all stimuli, regardless of their
“relevance.” Kisley et al, suggest that this finding implies that the brain is automatically
processing all stimuli to the same extent, rather than appropriately increasing or decreasing its
response according to higher or lower stimulus relevance. Thus, the brain of adults with SPD is
not over-responding to all stimuli, but only to those stimuli that should be effectively filtered, or
“gated” out, indicating that adults on the SPD spectrum, as well as children with SPD, have
atypical patterns of sensory gating.
Multi-Sensory Integration.
Multi-sensory integration refers to the ability of the brain to integrate information from multiple
sensory systems. In the natural environment, a wide variety of sensory stimuli occur in various
positions in time and space. While the input from the senses is initially separate, sensory input
ultimately converges in the brain so that individual elements in the external world can be
integrated. This integrated representation of the environment creates the foundation for
determining the nature and significance of events, and creating a meaningful response.
One methodology commonly used to evaluate multisensory interactions is comparing neural
responses to two unisensory stimuli, with a third neural response to a simultaneous presentation
of the same two stimuli. Multisensory integration is suggested when responses to a simultaneous
presentation of two stimuli is greater than the sum of the two unisensory responses (Laurienti,
2005). Using this approach, studies in animals (Stein and Meredith, 1993) and humans (Calvert,
2004), provide evidence of multisensory integration in various brain regions including the
midbrain, thalamus and cortex. In addition, psychophysical studies have shown that the
integration of multisensory input profoundly influences behavior (Stein and Meredith, 1993).
Numerous studies focused on specifying the neural basis for the behavioral enhancement that has
been observed during multi-sensory integration are currently underway, setting the stage for
improved understanding of the neural mechanisms underlying multisensory integration.
Appendix A Part 2
32
In Children. Brett-Green and colleagues have adapted an animal model ERP paradigm
(Stein and Meredith, 1993) to measure multisensory auditory-somatosensory integration in
typically developing children. Brett-Green is testing children with SPD, examining the
hypothesis that multisensory integration is atypical in children with SPD. The first phase of the
research involved testing a multisensory event-related potential (ERP) paradigm in a typical
cohort. Demonstrated was that the paradigm can measure multisensory integration in typically
developing children (Brett-Green, submitted). ERPs recorded from electrode site Cz in one
typically developing 10-year-old-boy (TYP) are shown in Figure 6 on the left side.
The figure on the left shows three types of sensory stimuli administered in the multisensory
paradigm: 1) unisensory auditory stimuli, 2) unisensory somatosensory stimuli and 3)
multisensory auditory and somatosensory stimuli administered concurrently. The waveforms
reflect the sensory information processing stages that occur mainly in the cortex, which have a
characteristic sequence of amplitude peaks (P100, N100, P200). Notable is that the ERPs have
clear amplitude peaks. In addition, the multisensory ERP denoting simultaneous auditory and
somatosensory stimulation (green line) exceeds the amplitude of either of the unisensory
responses (red = auditory; black = somatosensory). This indicates the response to multisensory
stimulation is stronger that the sum of the two unisensory responses.
The second phase involved using the paradigm to assess multisensory integration in children
with SPD (n = 9 to date). Preliminary multisensory ERP data demonstrate individual differences
between typically developing children (TYP) and children with SPD. For example, see the data
from one 11-year-old boy with SPD in Figure 6 right side. In the SPD case, neither the
unisensory nor the multisensory responses show clear amplitude peaks. In addition, the responses
to multisensory stimulation (green line) are not larger than either of the unisensory responses.
Appendix A Part 2
33
Figure 6. Comparison of One Typically Developing Child to One Child with SPD on
Multisensory Integration Paradigm
Group Differences between SPD and Typically Developing Children. Thirteen typically
developing children (TYP) were compared to nine children with SPD (sensory over-
responsivity) and several differences were found. In typical children, the classic P100, N100 and
P200 ERP components are evident. (See left side of Figure 7 on page 15.) This figure shows the
grand average multisensory ERP waveform recorded with simultaneous auditory- and-
somatosensory stimulation in typically developing children at electrode site Cz. The classic
P100, N100 and P200 ERP components are evident. Topographical maps (at right side of Figure
7, on page 15) show the voltage distribution across the scalp during multisensory stimulation.
In contrast, data obtained from children with SPD (see left side of Figure 8, on page 15)
shows similar data obtained from children with SPD. Comparison of the multisensory response
between typically developing children and children with SPD reveals several notable differences:
1. The amplitudes the classic ERP components are small for SPD, especially the P100
and N100.
2. The SPD group shows less decrement in response amplitude over time.
3. Topographical maps show the range of potentials (measured in µV) across the scalp is
smaller for the SPD group. (See Table 1 below for values in µV at P100 and N100 for
SPD vs. TYP.)
Table 1. Values of P100 and N100 in µV for SPD and TYP
The extent of the areas activated during multisensory stimulation are smaller in children with
SPD compared to typically developing children. In addition, the responses of children with SPD
begin earlier, continue longer, and appear to be less in frontal areas than the responses of
P100 (100ms)
N100 (160ms)
TYP
4.1
3.7
SPD
3.4
3.2
Appendix A Part 2
34
typically developing children. The tendency for longer response time in SPD is consistent with
the autonomic nervous system findings from McIntosh et al. (1999, described above) that
children with SPD have sympathetic nervous system responses (electrodermal activity) that
habituate more slowly after sensory stimulation compared to the responses of typically
developing children.
In addition, the brain areas activated during multisensory stimulation are clearly larger in
typically developing children.
Appendix A Part 2
35
Figure 7. Multisensory integration in a group of typically developing children
Figure 8. Multisensory integration in a group of children with SPD
Appendix A Part 2
36
Developmental Trajectory
Animal Model Research
Stein and colleagues (Stein, in process) are using a number of animal models to determine when
multisensory integration begins in development. Current results indicate that multisensory
integrative capabilities of the superior colliculus do not develop if the superior colliculus is
anaesthetized and thus inactive during the phase of early life when multisensory capabilities are
first being formed. Deficits have been demonstrated specifically in the association cortex in
animals whose superior colliculus is kept inactive. Stein’s study suggests that anomalies in SPD
may reflect problems with the development of the cortico-collicular axis, providing hypotheses
into the etiology of the symptoms of SPD.
Physiological Evidence
Analyses of developmental trends showed that sensory gating improves with maturity in
typically developing children ages 5-12, but does not improve with age in children with SPD
(Davies, 2007). This suggests that gating abilities in children with SPD do not change as a
function of either biologically driven maturity (e.g., physical growth) or the accumulation of
experiences across time (e.g., learning).
Clinical Evidence
In addition, the signs and symptoms of SPD differ by age. Although individuals may be
diagnosed at any age, symptoms of Sensory Processing Disorder are generally present (as
reflected in developmental histories) early in development and include poor or irregular patterns
in sleeping, eating, eliminating, and self-calming. Infants and toddler over-responsive symptoms
are often attributed to “colic,” “fussiness,” or “difficult temperament,” and if oral-sensitivity is
present the infant is at high risk for severe feeding difficulties. Infant and toddler under-
responsive symptoms include significantly less curiosity and exploration of new objects, and
places. Sensory under-responsivity is usually undiagnosed in infancy/toddlerhood but
retrospectively these children may be described as “easy babies,” “good sleepers,” and
“remarkably undemanding.”
Pre-school or kindergarten teachers are often among the first professionals to identify children
who have sensory problems because school environments require adaptation and accommodation
to sensory events that continually challenge children with SPD. These children display over- or
under-responsive behaviors to classroom noises (e.g., fans, bells, instrumental music); tactile
experiences (e.g., art projects, hands-on exploration); tastes (e.g., snack time); lights (e.g.,
fluorescent lights, blinking lights); unexpected touch (e.g., bumped by children while waiting in
line or playing at recess); and motion (e.g., playground equipment).
Children with SPD also often encounter increased academic difficulties in increasing grades
(e.g., 1st-3rd grade) due to more complex multi-sensory requirements associated with reading,
writing, memorization, and timed tests.
Appendix A Part 2
37
Adolescents and adults tend to display fewer overt signs of SPD as they employ various
compensatory strategies for managing and processing sensory information. However, inability to
maintain consistent levels of energy, mood, and attention across sensory environments persist.
Sensory over-responsivity in particular poses risk factors for intimate relationships and self-
regulation, while sensory under-responsivity tends to be a risk factor for processing speed of
academic information and occupational functioning.
Etiology
Twin Studies
Genetic influences on tactile and auditory over-responsiveness is being studied by Goldsmith and
colleagues (Goldsmith, 2006) using twin methodology. By comparing monozygotic (MZ) to
dizygotic (DZ) twins, variations due to underlying genetics versus environmental factors can be
estimated. Higher concordance rates in MZ twins indicate that status is partly heritable.
Probandwise concordance rates were calculated separately for MZ and DZ twin groups. Using a
population-based sample of 1394 toddler aged twins, the incidence of Sensory Over-
Responsivity (SOR) was widely distributed, with an accumulation of cases in the extreme range.
SOR was relatively distinct from other common dimensions of childhood behavioral dysfunction
although children with SOR were at increased risk for developing internalizing problems,
dysregulation and maladaptive problems. Goldsmith and colleagues (2006) found that in both
auditory and tactile responsivity, MZ twins were more similar than DZ twins, leading to the
inference that SOR has some genetic influence. Concordance rates were MZ (.72) and DZ (.53)
for Auditory SOR and MZ (.82) and DZ (.27) for tactile SOR. From this initial study of familial
aggregation it appears that SOR has moderate genetic influences with tactile overresponsivity
demonstrating somewhat greater heritability.
Retrospective Study
A preliminary study addressing the prevalence of pre-natal, birth, and early childhood health and
development problems in children with SPD was conducted using a retrospective record review
on all children with SPD ages 3-14 years, who were evaluated and treated at a large private OT
clinic near Boston 1996 to 2006 (n=1000), excluding adopted children and children with medical
diagnoses (May-Benson, 2006). A variety of possible risk factors exceeding base rates in the
population were identified in families and/or children treated for SPD including:
Pre-natal and./ or birth factors. 25% pregnancy complications; 42% labor/delivery
complications; 34% assisted deliveries e.g., vacuum, suction and forceps; 13% pre-term < 37
weeks; 5 % umbilical cord insults;
Developmental and health factors. 49% skipped the “terrible two’s;” 37 % brief / absent
crawling, 32% sleep or feeding problems; 62% chronic ear infections; 27% serious injuries or
illness; 25% jaundiced at birth, 20% colic as infants.
Animal Model Studies
Appendix A Part 2
38
Although controlled human studies exploring the relation of SPD to other known etiologic risk
factors have not yet been completed, Schneider and colleagues (Schneider, 2006) are using
animal model research paradigms to explore whether genetic factors might make the brain more
vulnerable to SPD, with the advantage over human research of systematically controlling for
environment and random assignment to groups. In Schneider’s studies, the sensory processing
abilities of Rhesus monkey offspring exposed to risk factors of moderate levels of 1) alcohol in-
utero, 2) prenatal stress, and 3) postnatal lead exposure were compared to the sensory processing
abilities of control monkeys. Significant differences were found in all risk conditions, with the
exposed monkeys showing more likelihood of producing SPD offspring.
Pre-alcohol exposed.
This group was primate mothers who voluntarily consumed .6g/kg of alcohol (~ two drinks) each
day during pregnancy and control monkeys (e.g., mothers received sucrose as control for
alcohol). The control monkeys demonstrated a relatively large initial withdrawal response to
sensory stimuli, followed by a decrease in response across trials (i.e., a typical habituation
pattern characterized by gradually decreasing responsiveness to tactile stimuli over repeated
trials). Monkeys whose mothers had prenatal alcohol exposure (n = 38) were discriminated by
their higher initial magnitudes of tactile withdrawal response that remained high over trials.
Schneider cites literature that demonstrates concordance with humans neonates exposed to
prenatal alcohol who have reduced habituation to auditory and visual stimuli (Barron, 1992),
reduced orienting to olfactory stimuli (Hunt, 2004), and various central nervous system deficits
(Livy, 2003).
Pre-stress.
This group was primate mothers who experienced a 10-minute removal from home cage and
transport to dark room where three random loud noise blasts were administered 5 times a week
over a 10-minute period, (90-145 days from gestation). Offspring demonstrated slightly lower
initial reactions to the feather (tactile stimulus), followed by slightly increased magnitudes of
withdrawal from repeated trials (e.g., this was the only group to show sensitization with
amplitude of responsiveness increasing over trials without habituation).
Lead Exposed.
Monkeys whose mothers were exposed to lead had abnormally strong patterns of withdrawal to
touch stimuli with increasing abnormal reactivity after the first few trials.
Conclusions.
Several conclusions can be drawn from these animal studies. First, the pattern of habituation
/sensitization observed to repeated tactile stimulation differs as a function of prenatal
experiences. Controls with no stress, alcohol, or lead exposure demonstrate increased magnitude
of response to the initial tactile stimulation followed by habitation, like human controls. In
Appendix A Part 2
39
contrast, monkeys exposed to prenatal stress showed behavioral sensitization, and those exposed
to prenatal alcohol showed a higher magnitude of Sensory Over-Responsivity. This suggests that
both alcohol and stress affect Sensory Over-Responsivity and behavioral regulation. Schneider et
al., notes that neuromodulation is likely sensitive to prenatal perturbations, which may cause
cascading effects later in development (Schneider, 2007). Furthermore, results suggest that
reduced regulation to sensory stimuli appears to result in delayed motor abilities, learning
deficits and other adverse developmental outcomes in primates.
Treatment Effectiveness
The gold standard for outcome studies is randomized controlled trials (Bury and Mead, 1998)
comparing the targeted intervention to either an active Alternate Placebo, and/or to a passive
placebo, No Treatment (e.g., a wait-list condition). Criteria for rigorous randomized trials are
well established (Boruch, 1997; Bury and Mead, 1998) and mandate inclusion of four primary
criteria: 1) an objectively defined sample that is homogeneous with regard to the impairment
studied (Bulpitt, 1983); 2) a “manualized intervention” where treatment is detailed in a manual
that others can obtain to replicate the procedures (Boruch, 1997) with a method to monitor
adherence to the specified delivery of treatment (Ottenbacher, 1991); 3) outcomes that are
meaningful, appropriate and sensitive to hypothesized changes (Fuhrer, 1997); and 4)
methodology that is rigorous, e.g., a) random allocation to experimental and control treatment
groups, b) blinded outcome evaluators, and c) adequate power to evaluate the significance of
effects (Jadad, 1998).
Previous Studies of Sensory-Based Occupational Therapy
A review of existing treatment effectiveness studies for SPD found that none of the previously
published research studies evaluating treatment outcomes met all four rigorous criteria for a
randomized trial (and often did not meet even one criterion) (Miller, 2003). Thus as of 2003, the
only accurate conclusion that could be proffered was that no rigorous evidence exists supporting
or denying the effectiveness of this treatment.
In spite of relatively universal agreement about the lack of well-controlled outcome studies,
significant controversy exists regarding the interpretation of the findings of over 80 published
articles from the field regarding the effectiveness of occupational therapy for SPD, based
primarily on belief systems, rather than empirical data. Relevant publications include two meta-
analyses (Ottenbacher, 1982; Vargas and Camilli, 1999) and four research syntheses (Schaffer,
1984; Arendt, MacLean et al., 1988; Polatajko, Kaplan et al., 1992; Hoehn and Baumeister,
1994). One meta-analysis suggests that the intervention approach does have a positive effect, but
the article is 25 years old (Ottenbacher, 1982). The four review articles conclude that previous
studies are not rigorous enough to make valid conclusions, while at the same time they conclude
that OT is not effective with SPD. The other meta-analysis suggests that the treatment approach
has no positive effect (Vargas and Camilli, 1999), but significant methodological flaws occurred
in this paper including: 1) studies had extremely small sample sizes (median sample size = 4.5
for 13 studies), 2) samples were heterogeneous regarding diagnostic groups, 3) such general
descriptions of treatment were provided that replication was impossible and, 4) studies had such
poor power that an effect was unlikely to be detected if present (Type II error).
Appendix A Part 2
40
Pilot Treatment Study.
Miller and colleagues therefore conducted a pilot treatment study (Miller, 2007a) correcting
previous research limitations by utilizing a homogeneous sample, manualized treatment,
outcome measures sensitive to change from treatment, and rigorous methodology. Results of the
pilot treatment study (without control groups) demonstrated significant pre-post changes from
treatment (Miller, 2007a) (See Table 2). The findings provided needed support for implementing
a randomized treatment trial.
Table 2. Results of Pilot Treatment Study evaluating the effect of Occupational Therapy
with Children who have Sensory Processing Disorder
Measure
Pre-Treatment
Mean Scores
Post-Treatment
Mean Scores
Change in
Mean Scores
Effect Size
p value
Leiter-R
Attention
5.88
6.32
0.43
0.29 (0.20)
0.20
Cognitive/Social
76.83
80.32
3.57
0.50 (0.03)
0.03
SSP
Total Score
-3.39
-0.39
3.11
1.62
<0.001
Vineland
Socialization
79.04
89.47
11.95
0.82
0.002
CBCL
Externalizing
60.93
56.95
-4.19
0.54
0.02
Internalizing
61.57
57.48
-3.67
0.43
0.07
GAS
30.37
55.68
25.31
2.16
<0.001
Miller et al., 2007a
Key:
SSP- Short Sensory Profile Vine- Vineland Behavior Scale
GAS- Goal Attainment Scale Leiter- Leiter International Performance
CBCL- Child Behavior Checklist Rating- Revised, Parent Rating
EDR- Electrodermal Reactivity
Pilot Randomized Controlled Trial.
Next, Miller and colleagues (Miller, 2007b) conducted a pilot randomized controlled trial of the
effectiveness of occupational therapy with children who have SPD. They evaluated the
effectiveness of three treatment groups: Occupational Therapy (OT), an active placebo called the
Activity Protocol (a play protocol), and a passive placebo e.g., wait-list condition. Twenty-four
children with SPD were randomly assigned to one of the three treatment conditions. Pre- and
post- measures of behavior, sensory, adaptive function and physiology were administered. The
manualized treatment was administered twice a week for 10 weeks in 50-minute sessions.
Fidelity to the treatment protocol was analyzed using Fidelity Measure (Parham, 2007). The
overall design is depicted in Table 3 below.
Table 3. Design of Randomized Controlled Pilot Study
Appendix A Part 2
41
Design of Randomized Controlled Pilot Study
Number
in
Group
Pre-test
Time1
10 Week
Treatment1
Week 2-11
Post-test
Time2
Group A
7
OT
Group B
10
Alternate Treatment
Group C
7
Week 1
No-Treatment
Week 11
Group differences at time-point two, after the first 10 weeks of treatment have been analyzed and
results demonstrate that the group that received OT, compared to the other two groups, made
significant gains on Goal Attainment Scaling (p <0.001) compared to No Treatment (the passive
control) and Activity Protocol (the active control). The OT group also made significantly greater
gains in Attention compared to No Treatment (p = .03); and compared to Activity Protocol
(trend toward significance) (p = .07). The OT group also made significantly greater gains and on
the Cognitive/Social Composite of Leiter-R (p = .02) compared to Activity Protocol. For both
the Short Sensory Profile (SSP) total test score, and the CBCL Internalizing composite score,
change scores were greater, but not significant, in the hypothesized direction for the OT group.
These findings are displayed graphically in Figure 9. Effect sizes were: Leiter-P Attention and
Cognitive/Social scores, (0.29), SSP total (.08), Vineland Socialization (.14), CBCL
Externalizing (.10) and Internalizing (.07), and GAS (1.62).
Figure 9. Findings of Randomized Controlled Pilot Study
Appendix A Part 2
42
-5
0
5
10
OT Group AP Group NT Group
a b c d e f g
a b c d e f g
a b c d e f g
a = Attention e = Externalizing
b = Cognitive/Social f = Internalizing
c = Short Sensory Profile (SSP) g = Goal Attainment Scaling (GAS)
d = Socialization per 10 units
Physiologically, even with a very small sample, the OT group showed greater reduction in
amplitudes of EDR compared to the AP and NT group. Though not significant, a trend was
observed for the OT group to improve in the hypothesized direction (reduced hyper-reactivity),
although the trend was not significant due likely to the small sample size. Inspite of the small
sample, this randomized trial of treatment suggests that OT may be effective in ameliorating
some signs and symptoms of children with SPD.
These preliminary findings suggest the need for completion of a larger randomized controlled
trial of the effectiveness of Occupational Therapy with a sensory-based approach, so that a more
definitive conclusion can be offered with reasonable assurance that results are not attributable to
chance and that external and internal sources of invalidity have been fully controlled.
Pilot Multiple Case Studies.
Psychophysiological measures show promise as treatment outcome measures in a multiple case
study of children with SPD before and after treatment (Miller, in process) using the multisensory
event-related potential (ERP) paradigm. ERP data is collected on children with SPD before and
after 20 occupational therapy treatment sessions to determine if there is a measurable change in
multisensory integration. In multiple case studies (n = 5), meaningful changes in ERP data have
been found, suggesting the multisensory event-related potential (ERP) paradigm may be useful
as a measure of treatment effectiveness. Data from one subject pre-and post-treatment is
provided below to illustrate preliminary results for this pilot study (see Figure 10 below).
Figure 10. Sample of changes in ERP data after OT intervention in one child
Appendix A Part 2
43
The child before treatment (on the left) has no clear peaks in response to auditory or
somatosensory stimuli, however the post treatment data appear to indicate improvement. ERP
amplitude peaks are clearer, and the amplitude of the multisensory ERP after treatment clearly
exceeds the amplitude of either unisensory response. Although these results are preliminary, they
suggest that: (1) the multisensory ERP paradigm may be a useful measure of treatment
effectiveness, and (2) sensory-based occupational therapy may produce measurable changes in
multisensory integration at the neurological level.
Deep Pressure Treatment.
Deep pressure and proprioceptive stimulation is one of the primary treatment methods used by
occupational therapists to reduce heightened sensitivity to tactile stimulation in individuals who
exhibit Sensory Over-Responsivity. Thus Smith and colleagues (Smith, submitted) studied the
physiological outcomes of applying heavy touch and deep pressure. In their paradigm, first
pressure was applied to the torso and thighs using a weighted blanket to two groups of adults,
one with Sensory Over-Responsivity and the other matched controls. Next, a pressurized suit
was used to measure the effect of pressure during the Sensory Challenge Protocol (Miller,
McIntosh et al., 1999). The results, as measured by electrodermal activity, peripheral blood flow,
and heart rate variability, are noted below.
Electrodermal Activity. Smith et al (submitted), found that the Sensory Over-responsive
group had a significant difference in initial skin conductance response (electrodermal reactivity)
to tactile stimuli, however the intervention reduced the response in both groups (p=0.01),
eliminating the initial difference (p = .02). The application of deep tactile pressure to the torso
and thighs appeared to differentially attenuate the sympathetic response to sensory stimuli in the
Sensory Over-Responsive and control groups (p=0.01). There was no difference in skin
conductance response between the groups in the pressure condition (p=0.2).
When tested during vestibular stimulation, (tip back in a chair), for both groups, the skin
conductance response to vestibular stimuli was reduced overall in the pressure condition
(p=0.02). Skin conductance responses to vestibular stimuli were not significantly different
Appendix A Part 2
44
between the Sensory Over-Responsive and control groups in either the no pressure or pressure
conditions. The skin conductance response for the visual, auditory, and olfactory stimuli also
demonstrated no significant differences between sensory over-responsive and control groups, but
showed reduced responses in the pressure condition compared to no pressure (p<0.1).
Peripheral blood flow. To tactile stimuli, the overall peripheral blood flow response was
reduced in the pressure condition compared to the no pressure condition for both groups
(p=0.004). In addition, peripheral blood flow response was reduced in the pressure condition
when compared to the no pressure condition for both groups during vestibular stimulation.
Heart Rate. Normal heart rate variability was observed; however, no discernible changes
were noted in heart rate as a result of any of the sensory stimuli in the study.
Summary. Overall this study suggests that deep pressure may have a calming effect on
the sympathetic nervous system. In addition, results suggest that Sensory Over-Responsive
adults may have a differential response to deep pressure when compared to control adults. Skin
conductance and peripheral blood flow responses to interventions may be useful in further
differentiating effective treatment of SPD.
Pharmacological Treatment
Levin and colleagues (Levin, Petro et al., 2004) are studying neural mechanisms of normal and
impaired sensory modulation, as was well as potential pharmacological therapeutic approaches
using pre-pulse inhibition (PPI) paradigms in a rat model. PPI, easily modeled in experimental
animals, is useful for determining of the neural bases for receptor systems. Studying inhibition of
the startle response (e.g., another measure of sensory gating) has implications for
neurotransmitter interactions and potential future pharmacological therapeutics for SPD.
Normally a warning stimulus reduces startle reactions. Recently, both nicotine effects and the
effect of nicotinic glutamate interactions on pre pulse inhibition (PPI) startle after auditory and
tactile stimulation (after nicotine has been administered, and after dizocilpine is administered)
has been studied (Levin, Petro et al., 2004). They found that nicotine facilitates PPI over various
intensities and inter-stimulus intervals. Notably, low doses of nicotine enhance the sensory
gating deficit. When nicotine and dizocilpine are both administered, there is a decreasing effect
on PPI as the dose of dizocilpine increases (dizocilpine acting as a blockade on the nicotinic
response). However, when clozapine is added to nicotine and dizocilpine, the % PPI increases
significantly in relation to the dose of clozopine administered.
With tactile startle, a dose effect of clozopine on the intensity of response is also noted. As the
amount of clozopine increases, the effect of the dizocilpine on reducing the %PPI decreases. In
other words, clozopine increases sensory gating when gating has been pharmacologically
reduced (using dizocilpine.). Levin et al, (2005), suggest that the added blockade of DA D2, D4
or 5-HT2 receptors with clozopine added may effectively reverse the impairment caused by the
blockade of NMDA.
Appendix A Part 2
45
Clozopine is often used in the treatment of schizophrenia to reduce the risk of suicide. Off label,
it is used to decrease mania, tardive dyskinesia, insomnia, obsessive-compulsive disorder, and
may have other uses. Use in humans with SPD has not been investigated yet in a controlled
study. Study of combination treatments affecting nicotinic receptors and receptor systems acted
upon by clozapine may be a fruitful avenue for study. This is particularly interesting in light of
the findings by Schneider et al (in press) for primates with SPD-like symptoms. Schneider found
the sensory over-responsive primates had reduced habituation to repeated stimuli that was
associated with increased D2 receptor binding of radiotracer suggesting up-regulation (super
sensitivity) of striatal D2 receptors.
Conclusion
Sensory Processing Disorder (SPD) is hypothesized to explain the abnormal behavioral
responses observed after sensory stimuli is experienced. SPD occurs along a spectrum from mild
to severe and affects both children and adults, causing either over-responsivity or under-
responsivity symptoms. In the proceeding section we have highlighted the significant differences
between individuals with SPD and typically developing individuals in a variety of areas needed
for syndrome validation based on Pennington’s model (1999, 2002). These areas include
underlying brain processes, signs and symptoms, developmental trajectory, etiology, and
treatment effectiveness. Collectively the empirical findings above suggest that the diagnostic
category of SPD may be valid. Future research in all areas above is ongoing and will advance the
knowledge base related to this disorder significantly over the next few years.
RESPONSE TO QUESTION 2:
THE NEW DISORDER DESCRIBES A CONDITION THAT IS
NOT ADEQUATELY COVERED BY THE EXISTING DSM-IV-TR CATEGORIES.
Summary
The key difference between Sensory Processing Disorder (SPD) and other DSM-IV-TR
diagnoses is that SPD is the only diagnosis that has sensory processing impairment as a primary
and essential feature. In addition, SPD is the only diagnosis where behavioral disruptions can be
directly tied to responses to sensory stimuli. No other disorder has sensory impairments as a
core feature, although some do include references to sensory functioning (e.g., autism,
schizophrenia). Discriminant validity studies of SPD have found significant differences between
SPD and existing DSM-IV conditions such as ADHD, Autistic Spectrum Disorder, and Fragile X
Syndrome. Both physiological and behavioral data highlight that SPD is the only condition that
has sensory processing impairment as a universal and specific feature. In addition, cases of
“pure SPD” have been identified in which subjects did not meet criteria for other DSM-IV
conditions, but did meet criteria for SPD.
Empirical Data Comparing SPD to Other Conditions
Work by some members of the SPD Scientific Work Group has focused on discriminant validity
of SPD compared to certain DSM-IV conditions, specifically Attention Deficit Disorder with and
Appendix A Part 2
46
without hyperactivity (ADHD) and Autistic Spectrum Disorder (ASD). Data related to these
studies is summarized below.
In addition, Pauls and colleagues are currently conducting a longitudinal family study of Sensory
Over-Responsiveness in individuals with Attention Deficit Hyperactivity Disorder (N=200),
Obsessive Compulsive Disorder (N=200) or Gilles de la Tourette (N=200) Syndrome and their
first-degree relatives. It will be the first systematic study designed to obtain discriminant validity
data in a prospective sample of children with psychiatric disorders and their families. In addition,
DNA is being collected from all families participating in this study. Since data is being collected
from all family members, examining the familial patterns and testing specific genetic hypotheses
regarding the transmission of Sensory Over-Responsivity within families is planned. Future
studies can include candidate gene studies to provide more quantitative discriminant data
comparing SPD to those disorders (Pauls, in process).
These studies will build on results obtained from other discriminant studies by members of the
SPD Scientific Work Group outlined below.
Attention Deficit Disorder (ADHD)
The following summarizes physiological and behavioral differences that have been found
between children with ADHD and SPD both physiologically and behaviorally. In general:
the group with SPD does not habituate to repeated sensory stimulation, while those with
ADHD do;
Individuals with SPD have more sensory aversion and withdrawal behaviors compared to
individuals with ADHD;
Individuals with SPD can inhibit responses on a computerized rapid response task but
those with ADHD can not;
Behaviorally, more sensory aversion and withdrawal responses are noted in SPD.
Individuals with SPD report problems with sustained attention, impulsivity and activity
as being less salient than sensory aversiveness, while the opposite is reported for those
with ADHD.
Sensory Responsivity
Introduction
Mangeot et al (Mangeot, Miller et al., 2001) compared 26 children with ADHD to 30 typically
developing children and found behaviorally, that ADHD is characterized behaviorally by
inappropriate impulsivity, inattention, and hyperactivity; however, these behaviors are not seen
specifically in response to sensory stimuli, but rather appear fairly universal across settings.
When the sensory functioning of children with ADHD is compared to typically
developing children, the group with ADHD displayed significantly greater abnormalities
in sensory responsivity on both physiological and parent report measures (Mangeot,
Miller et al., 2001; Dunn and Bennett, 2002).
Appendix A Part 2
47
Physiologically, children with ADHD demonstrate significant differences from typically
developing children in response to sensory stimuli, particularly on the first stimuli of a set
of repeated stimuli.
However, of note is that children with ADHD display significant variation in response to sensory
stimuli (Mangeot, Miller et al., 2001).
In order to begin examining if ADHD and SPD were the same syndrome (e.g., all children
exhibiting symptoms of one disorder would be expected to exhibit symptoms of the other
disorder), Miller and colleagues studied in a national sample of 2410 children (from national
standardization of Leiter International Performance Scale – Revised; (Roid and Miller, 1997),
who were stratified by age, gender, ethnicity and socio-economic status. Ahn et al., (Ahn, Miller
et al., 2004) found that 181 children (approximately 7.5 % of total sample) had significant
symptoms of either atypical sensory responsivity or attention problems.
If ADHD and SPD were the same syndrome, all children exhibiting symptoms of one disorder
would be expected to exhibit symptoms of the other disorder. In fact only 74 children exhibited
symptoms of both disorders (~ 3% of total sample or 40% of impaired sample). Fifty-seven
children exhibited only ADHD symptoms (2.4% of total sample or 31.5% of impaired sample)
and 50 children exhibited only symptoms of disordered sensory processing (~ 2% of total sample
and 27.5% of impaired sample) (see Table 4 below). The preliminary evidence from this study
supports existence of two separate syndromes, ADHD and SPD, although a high co-morbidity
rate (40%) appeared to exist.
Table 4. Percent of General Population with Symptoms of Attentional vs. Sensory
Disorders in a National Stratified Sample
Percent of National Stratified Sample with
Symptoms of Sensory and/or Attention
Impairments
© KID Foundation
ADHD Symptoms
No
Yes
Total
No
2229
57
2286
Sensory
Symptoms
Yes
50
74
124
Total
2279
131
2410
Ahn, Miller, et al., 2004
As a follow up, Miller and colleagues (Miller, Reisman et al., 2001) examined physiological
reactivity in children with SPD (n = 32) compared to children with ADHD (n = 40) and controls
(n = 46). The results demonstrated significant physiological differences between groups of
Appendix A Part 2
48
children with ADHD and SPD, particularly in the habituation variable. Children with ADHD did
habituate to stimuli similarly to typically developing controls. In contrast, children with SPD did
not habituate to sensory stimuli (Figure 11).
Figure 11. Discrimination of children with SPD and ADHD from Typically Developing
Children during Sensory Challenge Protocol.
Comparison of Children with ADHD and SMD
0
0.01
0.02
0.03
0.04
0.05
0.06
0.07
12345 678
Trials
Log Amplititude of Main Peak (micromhos)
Typical
ADHD
SMD
© KID Foundation Miller et al., 2001
From Miller et al., 2002
Behaviorally, the ADHD and SPD groups were compared on parent-rated behavior scales (i.e.,
Child Behavior checklist, Leiter International Performance Scale – Revised, and Short Sensory
Profile). The characteristics (in Table 6) were found to have differences greater than .5 standard
deviations between groups. The ADHD group had more difficulties with Attention, Impulsivity,
Activity level, and Auditory Filtering, as well poor Social Abilities characterized by Aggression
and poor Adaptation. By contrast, the SPD group had more difficulties with Tactile sensitivity,
Taste/Smell sensitivity, Low Energy, and Withdrawal. The two groups were rated
approximately the same on Anxiety, Social problems, Somatic complaints and Thought
problems.
Table 5. Discriminating Behavioral Characteristics of ADHD vs. SPD
Groups
ADHD
more
impaired
Auditory
Filtering
Attention
Problems
Activity
Level
Impulsivity
Adaptation
Aggression
Social
Abililties
ADHD
-3.2
-2.3
-1.8
-1.9
-2.1
-1.8
-1.6
SPD
-2.6
-1.8
-1.1
-1.2
-1.7
-1.2
0.8
SPD
more
impaired
Tactile
Sensitivity
Taste &
Smell
Sensitivity
Low Energy
/ Weak
Withdrawn
ADHD
-2
0.8
-2
0.5
SPD
-2.6
-1.1
-3
-1
Appendix A Part 2
49
Miller et al., 2002
Finally, (Ognibene, McIntosh et al., 2004) conducted a pilot study comparing typically
developing children (n = 25) to children with ADHD (n = 20), SPD (n = 11), or both ADHD and
SPD (n = 12). Significant differences between groups was found, after controlling for problem
behaviors, IQ, age, gender, and anxiety, on habituation and response inhibition. Although SPD
and ADHD shared some behavioral features, children with SPD had a sensory habituation deficit
(did not habituate to repeated sensory stimuli), and children with ADHD did not demonstrate that
impairment (i.e., they did habituate to repeated stimuli). In contrast, children with ADHD
demonstrated a significant response inhibition deficit on a computerized continuous performance
task, whereas children with SPD did not have the same inhibition problem. These data suggest an
apparent double dissociation between ADHD and SPD. Ognibene concluded based on this pilot
data that habituation and inhibition may prove useful in differentiating these disorders (see Table
6).
Appendix A Part 2
50
Table 6. Comparison of ADHD and SPD on Sensory Habituation and Response
Inhibition
Groups
Habituates to
repeated sensory stimuli
Inhibits on a computerized
continuous performance task
ADHD Group
Yes
No
SPD Group
No
Yes
From Ognibene et al, (2004)
Autistic Spectrum Disorder (ASD)
Although significant literature exists suggesting both Sensory Over-Responsivity and Sensory
Under-Responsivity are present in children with autism by age 2.5 (Rogers, Hepburn, & Wehner,
2003), sensory dysfunction is not a core deficit in autism. Kientz and Dunn (1997) and VerMaas
Lee (1999) found significant Sensory Over-Responsivity in children with autism compared to
controls. Dunn and colleagues (2002) also report significant Sensory Over-Responsivity in
children with Asperger’s Syndrome compared to controls.
In a physiologic study of the responses of children with Autism Spectrum Disorder to sensory
stimulation, Miller and colleagues evaluated 30 children with ASD using the Sensory Challenge
Protocol (Miller et al, 1999) with electrodermal activity as an outcome. Findings suggest that
children with ASD (ages 5-12) fall into two groups. One group demonstrates higher levels of
tonic electrodermal activity (e.g. general arousal state) maintained over sensory challenges (e.g.
phasic reactions or “trait”). The other group demonstrates lowered tonic electrodermal activity
(e.g. general arousal state is decreased) and they also display lowered reactions to sensory
challenges (e.g. phasic reactions or “trait”) (Schoen et al, submitted). The data for these two
ASD groups are displayed below in Figure 12.
Figure 12. Groups in ASD based on Electrodermal Activity during Sensory Stimulation
Grouping of Skin Conductance Level in
Autistic Spectrum Disorder
© KID Foundation
Appendix A Part 2
51
Although most individuals with ASD do exhibit sensory symptoms, most individuals with SPD
do not exhibit autistic signs and symptoms. The three core features of ASD, impaired interaction,
impaired communication, and restricted repetitive, stereotyped patterns of behavior are not
present in SPD unless the individual has co-morbid SPD and ASD. Studies evaluating theory of
mind, repetitive movements and socialization/communication in SPD compared to ASD are
initiated but have not yet been completed (Brout, in process).
Fragile X Syndrome
Fragile X syndrome is another clinical condition in which individuals of all ages exhibit quite
significant Sensory Over-Responsivity (Miller, McIntosh et al., 1999). Physiologically, the group
with the most severe sensory reactivity was observed in boys with Fragile X Syndrome who
demonstrate significantly increased amplitude of electrodermal activity and poor habituation to
sensory stimuli compared to controls (Miller, Reisman et al., 2001). These children differ from
SPD, however, in cognitive level and genetic etiology.
“Pure” SPD Case Studies
A pilot study targeting 30 subjects is underway to ascertain the existence of individuals with
SPD who do not meet criteria for other DSM-IV-TR conditions (Miller, in process). Subjects
receive a battery of tests designed specifically to identify conditions such as ADHD, autism,
OCD, and anxiety disorders, and are further evaluated by a developmental pediatrician and a
psychiatrist to rule out psychiatric and medical conditions. To date, ten individuals have met the
criteria for SPD only. This demonstrates that there are individuals who meet criteria of SPD and
who do not meet criteria for other diagnoses listed in the DSM-IV-TR. The study will continue
until 30 individuals have been identified to who meet the study criteria (Miller, in process).
Conclusion
The discriminant validity of SPD and several existing DSM conditions has been evaluated on
both physiologic and behavioral levels. Discriminant studies using both physiological and
behavioral measures have documented significant differences between SPD and ADHD. A
possible double dissociation has been suggested with SPD having poor sensory habituation but
adequate response inhibition, and ADHD exhibiting the inverse with poor response inhibition
and adequate sensory habituation. Differences between Autistic Spectrum Disorder and SPD
include socialization, communication and repetitive movements in the former but not the latter;
and electrodermal activity higher and lower than typically developing individuals. Individuals
with Fragile X syndrome have a genetic disorder in which severe Sensory Over-Responsivity is
found; however, individuals with SPD, unlike those with Fragile X syndrome, have much higher
intelligence (often above average) and no communication disabilities, and SPD is unlikely a
single gene disorder. In addition, multiple case studies suggest that a group of individuals have
significant sensory processing impairments in the absence of other diagnosed conditions. Further
studies are needed to cross-validate findings above and to evaluate differences from other key
disordered groups, including Generalized Anxiety Disorder, Obsessive Compulsive Disorder and
Gilles de la Tourette Syndrome. These findings suggest strongly that the new disorder describes
a condition that is not adequately covered by the existing DSM-IV-TR categories.
Appendix A Part 2
52
RESPONSE TO QUESTION 3:
ADDING THE DISORDER WILL IMPROVE CLINICAL UTILITY
Summary
Studies of prevalence have found that 5% of children in the general population exhibit
signs and symptoms of SPD severe enough to qualify for a diagnosis. Pilot consumer
surveys of clinical populations of children suggest that parents support inclusion of a
diagnosis of SPD in the DSM, primarily due to experiences of positive outcomes of
sensory-based treatment for their children’s sensory symptoms, after having no responses
to treatments for other diagnoses (e.g., ADHD, anxiety disorders, etc.). Surveys and focus
groups of physicians have found that a majority of physicians are aware of SPD and
support the inclusion of SPD in the revised DSM. Both consumers and physicians
anticipate that the inclusion of SPD will lead to a better risk/benefit ratio for treatment of
individuals with symptoms of SPD.
Prevalence Studies
For individuals with diagnosed developmental disabilities the rate of co-morbid SMD is
estimated to be 40% - 80% (Baranek, Chin et al., 2002), depending on the specific
developmental diagnosis. The prevalence of SMD in children in a general population is
estimated in a recent survey study (Ahn, Miller et al., 2004). Parents of incoming
kindergartners from a public school district were surveyed using the Short Sensory
Profile, a parent-report screening tool assessing functional correlates of SMD (McIntosh,
Miller et al., 1999). Of the 703 children surveyed, a conservative prevalence estimate
suggested that 5.3% of the sample met criteria for SPD. In a subsequent study of 440
children who were gifted and talented (e.g. IQs +2 SD above mean), an even higher
prevalence of SPD was demonstrated, 36%. See Table 8 below. (Miller, in process)
Table 8. Prevalence of SPD in Gifted and Talented Children
SMD
270
64.0
64.0
64.0
152
36.0
36.0
100.0
422
100.0
100.0
No SMD
Yes SMD
Total
Valid
Frequency
Percent
Valid Percent
Cumulative
Percent
SMD (No SS/A)
317
75.1
75.1
75.1
105
24.9
24.9
100.0
422
100.0
100.0
No SMD
Yes SMD
Total
Valid
Frequency
Percent
Valid Percent
Cumulative
Percent
Prevalence of SMD in Special
Populations: Gifted and Talented
© KID Foundation
Appendix A Part 2
53
Carter and Briggs-Cowen are examining the prevalence of Sensory Over-Responsivity in a prospective study of a longitudinal
birth cohort of children in the Greater New Haven area who have been followed from either 1- or 2-years of age and assessed
with the Sensory Over-Responsivity Scale in Second Grade. Data about early emerging social-emotional problems and
competencies, including a measure of sensory sensitivities at ages 1, 2, and 3 years of age and psychiatric diagnostic status in
Kindergarten and Second Grade is available in the longitudinal study. By 2008, this project will provide data on the prevalence
and correlates of Sensory Over-Responsivity in a representative community sample. It is also examining associations between
infant/toddler and concurrent social-emotional and behavior problem correlates of Sensory Over-Responsivity in school-aged
children.
Clinical Utility
Physician Responses
Two relevant studies have occurred examining clinical utility to date. The first was a
series of focus groups led by Edward Goldson MD, Professor of Pediatrics at the
University of CO Health Sciences Center. The second was a survey of 2400 physicians
(pediatricians, neurologists, and pediatric psychiatrists) in Massachusetts (5% response
rate). These studies show that most physicians are aware of the diagnosis of SPD and of
those aware, a majority support inclusion in the revised DSM (64%). A small cadre of
physicians are strongly opposed to inclusion (5%) and strongly advise parents not to seek
treatment for SPD (10%). A group of physicians are either unaware of the diagnosis or
are asking for addition information before deciding whether SPD should be included in
the DSM (~30%). Details of these studies appear below.
Focus Groups with Pediatricians
Two pilot focus groups were conducted by Edward Goldson, MD, Professor of Pediatrics
at the University of Colorado Health Sciences Center. Dr. Goldson and colleagues
interviewed eight experienced Colorado pediatricians in two small groups to explore the
perceptions of clinical providers about the clinical utility of adding SPD as a separate
disorder to the DSM (Goldson, in process). These focus groups are being expanded to
other medical and psychological disciplines in other cities nationwide in the next few
years. The purpose of the focus groups was determine what pediatricians understand
about SPD, whether they see children with these symptoms in their practices, and what
advantages or /disadvantages they perceive by adding SPD to the DSM.
In general, the pediatricians were aware of SPD but requested clarification about the
definition of subtypes. The pediatricians acknowledged that they were seeing children in
their practices with the defined signs and symptoms of SPD, who tended to have
diagnoses that included attention deficit/hyperactivity disorder, mood instability, and/or
coordination disorder. However, most were unclear about how to provide services for
these children. They reported that clinical care for these children is extremely time-
consuming and taxing both for the families and as well as for physicians. Inaccurate
diagnoses frequently result in children going from one specialist to another seeking better
diagnoses and care. In some cases children are referred for developmental evaluations or
are referred directly to occupational therapists where an evaluation for sensory
functioning is provided and OT treatment instituted.
Appendix A Part 2
54
A further concern of the pediatricians was that the lack of an accepted diagnostic code
makes it difficult for the physician to be reimbursed for his/her time, resulting in using a
“pseudo- diagnosis” to access services for these children.
The focus groups provided preliminary evidence that physicians (pediatricians) are
interested in learning more about SPD, how to manage children with this problem, and
how to provide effective services for them. Both focus groups clearly articulated that
inclusion of a new diagnosis would likely increase their own diagnostic accuracy,
enhancing their ability to make appropriate referrals for intervention, improve outcomes
for the children and their families (Goldson, in process).
A Survey of Physicians
A large survey study examining the beliefs and knowledge of physicians related to SPD
was conducted in Massachusetts by mailing an 11-question survey to pediatricians,
neurologists, and pediatric psychiatrists (May-Benson, T., Koomar, J. & Teasdale, A.,
2006). The survey was mailed to 2400 physicians listed with the Massachusetts State
Board of Registration in Medicine. Of the 122 surveys returned (5%), a majority (64%)
supported the inclusion of SPD in the DSM, and a few were strongly opposed to
inclusion (5%). Of the remaining respondents who did not express an opinion, many
reported being unaware of the diagnosis. (See Table 8 below.)
Table 8 Physician Survey Related to the Utility of SPD as a Diagnosis
Question
Results (n = 122)
How familiar are you with SPD?
65% very familiar;
28% somewhat;
7% not familiar
Do you believe SPD is a valid
diagnosis?
64% yes, valid;
26% unsure;
10% not valid
Do you support inclusion of SPD in
the DSM?
66% support;
29% not definite, wanted more information;
5% strongly opposed
Do you encourage treatment for
SPD?
64 % actively encourage;
26 neither encourage nor discourage;
10% actively discourage parents from seeking
services
Particularly notable from this survey was that a large percentage (30%) of physician
respondents wanted more information before deciding whether SPD should be included
in the DSM revision. However, survey results suggest that a majority of physicians
surveryed (64%) support SPD as a separate diagnosis from other conditions such as
ADHD, Anxiety Disorders and Autistic Spectrum Disorder (May Benson, Submitted.).
Appendix A Part 2
55
Consumer Responses
To gather data from a consumer perspective on the clinical utility of SPD, a pilot survey
was administered to 250 participants who attended educational SPD workshops (Ahn, in
progress). More than half of the 124 respondents reported they were at the workshop as a
parent (n = 67). First, families were queried about their experience of effectiveness and
efficiency with regard to their children’s’ diagnosis. Respondents reported having spent
an average of more than $3000, meeting with an average of 5 clinical professionals (e.g.,
physicians, psychologists), and receiving an average of seven different diagnoses (e.g.,
ADHD, anxiety, etc.), before obtaining a referral to an occupational therapist for
evaluation of sensory processing abilities (Ahn, in progress). In addition, a qualitative
review of descriptions of an open-ended question that asked about child diagnoses and
symptoms found that behaviors that precipitated seeking clinical services, included
tantrums, irritability, clinginess, inflexibility, and extreme emotional distress during daily
routines (e.g., crying, aggressiveness, etc. associated with meals, traveling, bathing, etc.).
When asked about effectiveness of previous treatments for these children, respondents
reported mixed outcomes with medication management (e.g., Ritalin) or psychological
treatments (e.g., family counseling), and reported positive outcomes with occupational
therapy. From this pilot a more specific data are being collected through the KID
Foundation website (see www.KIDFoundation.org/survey) and through other
professional sources. Data on this survey will be collected through 2008 when results will
be summarized and published. In addition, more than 6000 consumers have signed a
petition posted on the KID Foundation website endorsing recognition of SPD in the
DSM-V.
RESPONSE TO QUESTION 4:
A LOW RISK OF FALSE POSITIVES
Summary
Several valid and reliable parent and self-report screening measures exist to evaluate
SPD. In addition, three performance measures exist. The newest performance measure,
called the Sensory Over-responsivity and Under-responsivity Scale (SensOUR),
demonstrates a low false positive rate using a cut-point of -2 standard deviations (False
Positive rate: 1.3%; False Negative rate: 10.7%; Overall Hit rate 88%).
Instruments for Screening and Diagnosis
Several parent and self-report screening measures exist to evaluate SPD, including the
Sensory Processing Measure and three versions of the Sensory Profiles. Three
performance assessments have also been developed: (1) The Sensory Integration and
Praxis Scale (SIPT: Ayres, 1989), a diagnostic assessment widely used to evaluate related
aspects of SPD (e.g. sensory discrimination, postural disorders, and praxis); (2) The
Miller Assessment for Preschoolers (MAP: Miller, 1982, 1988) that evaluates related
Appendix A Part 2
56
aspects of SPD, but does not focus on Sensory Over- and Under-Responsivity; and (3)
the Sensory Over-Responsive and Under-Responsive (SensOUR) Scale, which is under
development (Schoen, Miller et al., 2005) These scales and available false positive data is
provided below.
Parent or Self Report Measures
The Sensory Profile(s) (The Psychological Corporation: San Antonio)
The most widely used report measures are Dunn’s series of three scales: (1), the original
Sensory Profile, and the School Companion, ages 3 to 10 (Dunn, 2001; 2006) for
classroom and home; (2) the Adolescent and Adult Sensory Profile, ages 11 and older
(Brown and Dunn, 2002); and (3) the Infant Toddler Sensory Profile ages birth to 3 years
(2002). The Sensory Profiles examine sensory processing patterns in individuals who are
at-risk or have specific disabilities related to sensory processing issues. Responses are
based on self- or caretaker-reports. The scales have been standardized nation-wide
(samples ranging from 500 to 1200). Resulting profiles from the measures highlight the
effects of impaired sensory processing on functional performance in the daily life of an
individual.
The SP measures have good reliability (coefficient Alpha’s ranging from .47-.91 on the
Sensory Profile, from .64 - .78 on the Adolescent/Adult Profile and from .42 to .85 for
the Infant/Toddler Profile). The adolescent/Adult profile correlates with the New York
Longitudinal Scales Adult Temperament questionnaire (between .42 to .46), and the
original Profile correlates with the School Function Assessment (.68 to .72)
demonstrating some concurrent validity.
False positive rates using the Sensory Profile appear to be 19.9% for Factor 1 Sensory
Seeking, 23% for Factor 2 Emotionally Reactive and 11.2% for Factor 5
Inattention/Distractibility.
The Sensory Processing Measure (Western Psychological Services, Los Angeles)
A new scale, the Sensory Processing Measure (SPM: 2006), is modeled on diagnostic
subtypes in the Interdisciplinary Council on Developmental and Learning Disorders
(ICDL, 2006) and the DC: 0-3 (Zero to Three, 2006). The SPM screens for Sensory
Over-Responsivity and Sensory Under-Responsivity specifically with both home
(caretaker) and school (teacher) scales. The SPM was standardized on 1051 children in
grades K through 6 and has excellent reliability (scale internal consistencies range from
.75 to .95; two-week test-retest correlations range from .94 to .98) and validity (evidence
for validity include factor analytic studies, correlational results, and clinical
discrimination studies). False positive rates are 15% using a cutoff T-score of 60 (lower
bound of “Some Problems”) and 2% using a cutoff T-score of 70 (lower bound of
“Definite Dysfunction”.)
Performance Measures
Appendix A Part 2
57
Sensory Integration and Praxis Test (SIPT; (Ayres, 1989)
The Sensory Integration and Praxis (Ayres, 1989) scale has been widely used by
occupational therapists to evaluate SPD based on Ayres’ (1972) constructs. Standardized
nationwide on 1,997 children, the SIPT includes 17 subtests each having separate
reported reliability and validity. SIPT Validity studies are numerous (Carrasco, 1991;
Royeen, 1991; Walker and Burris, 1991; Lai, 1996; Mulligan, 1996; Rinner, 2002;
Royeen and Mu, 2003). One study used 10,000 SIPT protocols to conduct a validity
study using structural equation modeling (Mulligan, 1996). Mulligan found that the SIPT
defines four separate “subtypes” including sensory over-responsivity. The SIPT does not
provide a direct measure of Sensory Over-Responsivity and Sensory Under-responsivity,
the two subtypes we are applying to include in DSM-V, but rather focuses on evaluation
of other presumed aspects of SPD: sensory discrimination, postural disorder, and praxis,
which are other presumed aspects of SPD.
The Miller Assessment for Preschoolers (MAP; 1982, 1988)
The MAP is an assessment for children ages 2 ½ to 5 1/2 that evaluates children’s
neurological maturity and includes several subtests of sensory processing abilities.
Standardized on 1200 children nationwide, the scale has good reliability (.98) and
validity (e.g., discrimination between typically developing and pre-academic delay group:
.75 specificity, .75 sensitivity). A longitudinal predictive validity study, evaluating the
standardization sample four years after original testing, found an 80% correct hit rate,
with false positives < 5% using a cutpoint 2 standard deviations from the mean.
The Sensory Over-Responsive and Under-Responsive Scales (SensOUR)
The Sensory Over-Responsive and Sensory Under-Responsive Scales (SensOUR; in
development since 2004) measures sensory functions in all sensory domains (vision,
audition, touch, olfaction, taste, proprioception and vestibular). The scale includes both a
parent/self report inventory and a performance Assessment where specific standard items
are administered by a trained examiner. Psychometrics of the instrument were established
in three stages of study: 1) Instrument development; 2) Reliability and validity of the
research edition and 3) Cross-validation of findings on the research edition with a second
sample.
For the Over-responsive subtests, data were collected from typically developing
individuals (N=104) and individuals with Sensory Over-Responsivity (N= 113), ages 3 to
55 in Phase 1. All items were reviewed for their internal reliability consistency,
discriminant validity and construct validity and a revised Research Edition was
constructed. Then a new unrelated sample was evaluated. Analyses of the Sensory Over-
Responsivity portion of the SensOUR Assessment revealed moderate to high internal
consistency reliability for the domains (.60 to .89) and the total test (.92). The reliability
estimates for the Over-Responsive Inventory ranged from .65 to .88 for the domains, and
.97 for the total test.
Appendix A Part 2
58
Construct validity of the scales included factor analysis of the Inventory and Assessment
separately. A seven-component factor analytic solution for both the Assessment and the
Inventory provided the most interpretable pattern of loadings with no singleton factors.
Although groups with and without Sensory Over-Responsiveness were factored
separately on the Inventory and Assessment, both had the same factor solution. Factor
loading were .46 - .95 with the greatest number between .6 and .95.
With respect to discriminant validity the typically developing group was compared to the
group with Sensory Over-Responsivity for the Assessment and the Inventory. For both
the total test and domain scores discriminated groups (over-responsive vs. typically
developing) at a meaningful and statistically significant level (significance by domain
ranged from p < .05 to p < .001) (Schoen, in process). False positive rate was 1.3%,
calculated using a cut-point of -2 standard deviations. To be considered “positive” for
SPD both the Inventory and the examiner administered performance Assessment were
rated “positive”. Data on the SensOUR included: (False Positive rate: 1.3%; False
Negative rate: 10.7%; True Positive rate: 25.3%; True Negative Rate: 25.3%; and At Risk
category: 37.3%; and an Overall Hit rate 88%) (Schoen et al., in process).
Appendix A Part 2
59
RESPONSE TO QUESTION 5:
DATA SETS AVAILABLE FOR REANALYSIS
Summary
As scientists have not historically been asking the set of questions needed to identify
Sensory Processing Disorder, no current archived data sets contain sufficient items
needed to meet SPD classification criteria—with the exception of the data sets used by
members of the SPD Scientific Work Group (referred to in this proposal). While it is
possible that other existing data sets (e.g. NIH, MTA study) may contain information
related to sensory-based items, and item analyses might identify those that have
relevance to SPD, it is unlikely that those existing data sets would include sufficient items
needed to meet meaningful criteria for SPD.
No Existing Data Sets
All data sets from work of the members of the Sensory Processing Disorder (SPD)
Scientific Work Group (SWG) used to derive data cited in the articles above are available
for reanalysis. They include data from: Margaret Bauman MD, Harvard University
Medical School; Margaret Briggs-Gowen PhD, Yale University; Alice Carter PhD,
University of Massachusetts; Patricia Davies PhD, and William Gavin, CO State
University; Hill Goldsmith PhD, University of Wisconsin Madison; Michael Kisley PhD,
University of Colorado in Colorado Springs; Ed Levin PhD, Duke University, NC; David
Pauls PhD, Harvard University Medical School; Mary Schneider PhD, University of
Wisconsin – Madison; Roseann Schaaf PhD, Thomas Jefferson University; Sinclair
Smith ScD, Drexel University and Temple University; Barry Stein PhD, Wake Forest
College and research from The KID Foundation Research Institute: Lucy Jane Miller
PhD, Roianne Ahn PhD, Barbara Brett-Green PhD, Jennifer Brout PsyD, and Sarah
Schoen, PhD).
In addition, several national databases exist that might shed light on this issue, (e.g., the
NIMH national ADHD Multi-site (MTA) study, and studies related to temperament,
OCD and anxiety). However, scientists have not typically collected data related to
Sensory Processing Disorder in research subjects. Therefore reanalysis would require
item analyses of archived data sets, and are unlikely to contain the necessary raw data
needed for meaningful SPD classifications. Currently no convenient databases exist from
which a set of diagnostic screening questions could be pulled (except the data bases of
the Scientific Work Group referred to in this proposal).
Appendix A Part 2
60
Arciniegas, D., L. Adler, et al. (1999). "Attention and memory dysfunction after
traumatic brain injury: Cholinergic mechanisms, sensory gating, and a hypothesis for
further investigation." Brain Injury 13(1): 1-13.
Arendt, R. E., W. E. MacLean, et al. (1988). "Critique of sensory integration therapy and
its application in mental retardation." American Journal on Mental Retardation 92: 401-
411.
Barron, S., & Riley, E. P. (1992). "The effects of prenatal alcohol exposure on behavioral
and neuroanatomical components of olfaction." Neurotoxicology Teratology 14(4): 291-
297.
Boruch, R. F. (1997). Randomized experiments for planning and evaluation: A practical
guide. Thousand Oaks, CA, SAGE Publications.
Boucsein, W. (1992). Electrodermal activity. New York, Plenum Press.
Brett-Green, B. A., Miller, L. J., Gavin, W. J., Davies, P. L. (submitted). "Multisensory
integration in children: an event-related potential study."
Brown, C., & Dunn, W. (2002). The adult sensory profile. San Antonio, TX.,
Psychological Corporation.
Bulpitt, C. J. (1983). Randomized controlled clinical trials. Hingham, MA, Kluwer
Boston.
Bury, T. J. and J. M. Mead, Eds. (1998). Evidence-based healthcare: A practical guide to
therapists. MA, Butterworth-Heinemann.
Calvert, G. A., & Thesen, T. (2004). "Multisensory integration: methodological
approaches and emerging principles in the human brain." Journal of Physiology Paris
98(1-3): 191-205.
Critchley, H. D. (2002). "Electrodermal responses: What happens in the brain."
Neuroscientist 8(2): 132-142.
Davies, P. L., Gavin, W. J. (2007). "Validating the diagnosis of sensory processing
disorders using EEG technology." The American Journal of Occupational Therapy 61(2).
Fuhrer, M. J. (1997). Assessing medical rehabilitation practices: The promise of
outcomes research. Baltimore, MD, Paul H. Brookes Publishing Co.
Goldsmith, H. H., VanHulle, C.A., Arneson, C.L., Schreiber, J.E., and Gernsbacher,
M.A. (2006). "A population-based twin study of parentally reported tactile and auditory
Appendix A Part 2
61
defensiveness in young children." Journal of Abnormal Child Psychology 34(3): 393-
407.
Hoehn, T. P. and A. A. Baumeister (1994). "A critique of the application of sensory
integration therapy to children with learning disabilities." Journal of Learning Disabilities
27(6): 338-350.
Hunt, P. S., & Phillips, J. S. (2004). "Postnatal binge ethanol exposure affects habituation
of the cardiac orienting response to an olfactory stimulus in preweanling rats."
Alcoholism Clinical and Experimental Research 28(1): 123-130.
ICDL (2005). Diagnostic manual for infancy and early childhood: Mental health,
developmental, regulatory-sensory processing and language disorders and learning
challenges (ICDL-DMIC). Bethesda, MD, Interdisciplinary Council on Developmental
and Learning Disorders (ICDL).
Jadad, A. (1998). Randomized controlled trials: A user's guide. London, BMJ Publishing
Group.
Kemner, C., Oranje, B., Vergaten, M. N., & van Engeland, H. (2002). "Normal P50
gating in children with autism." Journal of Clinical Psychiatry 63(6): 214-217.
Kisley, M. A., T. L. Noecker, et al. (2004). "Comparison of sensory gating to mismatch
negativity and self-reported perceptual phenomena in healthy adults." Psychophysiology
41(4): 604-612.
Laurienti, P. J., Perrault, T. J., et al. (2005). "On the use of superadditivity as a metric for
characterizing multisensory integration in functional neuroimaging studies." Exp. Brain
Res. 166(3-4): 289-297.
Levin, E. D., A. Petro, et al. (2004). "Nicotine and clozapine actions on pre-pulse
inhibition deficits caused by NMDA glutamatergic receptor blockade. Manuscript in
preparation."
Livy, D. J., Miller, E. K., Maier, S. E., & West, J. R. (2003). "Fetal alcohol exposure and
temporal vulnerability: Effects of binge-like alcohol exposure on the developing rat
hippocampus." Neurotoxicology Teratology 25(4): 447-458.
May-Benson, T. A., Koomar, J., Teasdale, A. (2006). Prevalence of Pre-/Post-Natal and
Developmental Factors in 1000 Children with SPD. 124 Watertown, MA 02472, The
Spiral Foundation.
McIntosh, D. N., L. J. Miller, et al. (1999). "Sensory-modulation disruption,
electrodermal responses, and functional behaviors." Developmental Medicine and Child
Neurology 41: 608-615.
Appendix A Part 2
62
Miller, L., J., Coll, J. R., & Schoen, S. A. (2007b). "A randomized controlled pilot study
of the effectiveness of occupational therapy for children with sensory modulation
disorder." The American Journal of Occupational Therapy 61(2): 228-238.
Miller, L., Schoen, S. A., James, K., Schaaf, R. C. (2007a). "Lessons learned: a pilot
study on occupational therapy effectiveness for children with sensory modulation
disorder." The American Journal of Occupational Therapy 61(2): 161-169.
Miller, L. J. (2003). "Empirical evidence related to therapies for sensory processing
impairments." Communique' 31(5): 34-37.
Miller, L. J., Anzalone, M. E., Lane, S. J., Cermak, S. A., Osten, E. T. (2007c). "Concept
evolution in sensory integration: a proposed nosology for diagnosis." The American
Journal of Occupational Therapy 61(2): 135-140.
Miller, L. J., D. N. McIntosh, et al. (1999). "Electrodermal responses to sensory stimuli in
individuals with fragile X syndrome: A preliminary report." American Journal of Medical
Genetics 83(4): 268-279.
Miller, L. J., Schoen, S.A., Brett-Green, B., Ahn, R. R. (in process). "Multiple case study
review of sensory processing disorder."
Olincy, A., R. G. Ross, et al. (2000). "The P50 auditory event-evoked potential in adult
attention-deficit disorder: Comparison with schizophrenia." Biol Psychiatry 47(11): 969-
977.
Ottenbacher, K. (1982). "Sensory integration therapy: Affect or effect." American Journal
of Occupational Therapy 36(9): 571-578.
Ottenbacher, K. (1991). Research in sensory integration: Empirical perceptions and
progress. Sensory integration: Theory and practice. A. G. Fisher, E. A. Murray and A. C.
Bundy. Philadelphia, F.A. Davis Company: 385-399.
Parham, D., Cohn, E. S., Spitzer, S., Koomar, J., Miller, L. J., Burke, J., Brett-Green, B.,
Mailloux, Z., May-Benson, T. A., Smith-Roley, S., Schaaf, R. C., Schoen, S., Summers,
C. A. (2007). "Fidelity in sensory integration intervention research."
Pennington, B. F. (1991). Issues in syndrome validation. Diagnosing learning disorders:
A neuropsychological framework. B. F. Pennington. New York, The Guilford Press: 23-
31.
Pennington, B. F. (2002). The development of psychopathology: Nature and nurture.
New York, Guilford Press.
Appendix A Part 2
63
Polatajko, H. J., B. J. Kaplan, et al. (1992). "Sensory integration treatment for children
with learning disabilities: Its status 20 years later." Occupational Therapy Journal of
Research 12: 323-341.
Porges, S. (1985). Respiratory sinus arrhythmia: An index of vagal tone.
Psychophysiology of cardiovascular control: Models, methods and data. J. F. Orlebeke,
G. Mulder and L.J.P. van Dornen. New York, Plenum: 437-450.
Schaaf, R. C. (2001). Parasympathetic nervous system functions in children with sensory
modulation dysfunction. Bryn Mawr, PA, Bryn Mawr College.
Schaffer, R. (1984). "Sensory integration therapy with learning disabled children: A
critical review." Canadian Journal of Occupational Therapy 51: 73-77.
Schneider, M. L., Moore, C. F., Gajewski, L. L., Larson, J. A., Gay, C.L., Roberts, A.D.,
Converse, A.K., DeJesus, O.T. (2007). "Sensory processing disorder in a nonhuman
primate model: evidence for occupational therapy practice." The American Journal of
Occupational Therapy 61(2).
Schneider, M. L., Moore, C. F., Gajewski, L. L., Larson, J. A., Roberts, A., Converse, A.
et al. (2006). "Sensory processing disorder in a primate model: Evidence from a
longitudinal study of prenatal alcohol and prenatal stress effects." Manuscript submitted
for publication.
Smith, S. A., Kinnealey, M. (submitted). "Effect of deep tactile pressure on the
sympathetic response to sensory stimuli in sensory defensive and non-defensive adults."
Smith, S. A., Kinnealey, M., Barriocanal, J., Witt, D., Im, J., and Kanamalla, U. (2004).
Central Nervous System Metabolism in Sensory-defensive and Non-defensive Adults: A
Proton Magnetic Resonance Spectroscopy Study. Experimental Biology Annual
Conference, Washington, D.C.
Stein, B. (in process). "Multisensory Integration from birth through early months in
anaesthicized cats."
Stein, B. B. and M. A. Meredith (1993). The merging of the senses. Cambridge, MA,
MIT Press.
Stern, R. M., W. J. Ray, et al. (2001). Psychophysiological recording. New York, Oxford
University Press, Inc.
Vargas, S. and G. Camilli (1999). "A meta-analysis of research on sensory integration
treatment." American Journal of Occupational Therapy 53(2): 189-198.
Zero To Three (2005). Diagnostic classification: 0-3-Revised. Arlington, VA, National
Center for Clinical Infant Programs.
Appendix A Part 2
64
This research hasn't been cited in any other publications.