The Early Development of Stereotypy and Self-Injury

Journal of Intellectual Disability Research
144
doi: 10.1111/j.1365-2788.2004.00632.x
pp
Blackwell Science, LtdOxford, UKJIRJournal of Intellectual Disability Research-Blackwell Publishing Ltd, 2144148Original ArticleEarly
development of stereotypy and self-injuryF. J. Symons et al.
The early development of stereotypy and self-injury:

a review of research methods
F. J. Symons,1 L. A. Sperry,2 P. L. Dropik1 & J. W. Bodfish3
1 University of Minnesota, Minneapolis, MN, USA
2 Division TEACCH, Department of Psychiatry, University of North Carolina at Chapel Hill, NC, USA
3 Human Development Research and Training Institute,Western Carolina Center, University of North Carolina at Chapel Hill, NC,
USA
Abstract
Background The origin and developmental course
of stereotypic and self-injurious behaviour among
individuals with developmental disabilities such as
intellectual disability (ID) or pervasive development
disorders such as autism is not well understood.
Method Twelve studies designed to document the
prevalence, nature, or development of stereotypic
and/or self-injurious behaviour in children under
years of age and identified as at risk for developmental delay or disability were reviewed. Comparisons were made with similar studies with typically
developing children.
Results It appears that the onset of naturally occurring rhythmic motor stereotypies is delayed in young
at-risk children, but that the sequencing may be similar. A very small database, differences in samples,
measures, and designs limited the degree to which
comparisons could be made across studies.
Conclusion Future work is needed based on appropriately designed prospective comparison studies and
uniform quantitative measures to provide an empirical basis for new knowledge about the early developCorrespondence: Frank Symons, PhD, Department of Educational
Psychology, College of Education and Human Development,
Burton Hall Pillsbury Drive SE, University of Minnesota,
Minneapolis, MN , USA (e-mail: symon@umn.edu).
Blackwell Publishing Ltd
ment of one of the most serious behaviour disorders

afflicting children with ID and related problems of
development.
Keywords developmental disability, early
development, self-injurious behavior, stereotypy
Introduction
Normally developing infants engage in a broad class
of repetitive stereotyped behavioural movements such
as toe sucking, foot kicking and body rocking involving a predictable developmental sequence of the
limbs, torso, head, and whole body (Thelen ,
). Stereotypies are most commonly defined as
motor behaviours that are repetitive, topographically
invariant, often rhythmical, and appearing without
obvious purpose (Powell et al. ). In humans, the
progression through rhythmical stereotypies is considered transient and associated with the development of motor skills (Wolff ), neuromuscular
development (Thelen ), and the general maturation of the central nervous system (CNS) (Sprague
& Newell ).
Early theories of child development included
hypotheses about the potential adaptive significance
of repetitive behaviour (Gesell ; Piaget ).
Gesell () observed that young children between

145
F. J. Symons et al. Early development of stereotypy and self-injury
and years of age engage in behaviour characterized as compulsive with strong tendencies toward
repetitive stereotyped ritualized behaviours. Piaget
() suggested that repetition in infancy might
develop from basic reflexive behaviour to goaloriented behaviour that is adaptive for changing
environments. More recently Evans et al. ()
found that compulsive-like, ritualistic, repetitive
behaviours change with age and hypothesized that
repetitive behaviour may have broader implications
related to the study of developmental psychopathology and developmental disorders of childhood onset
such as autism and related disabilities.
The clinical significance of repetitive stereotyped
behaviour has not gone unnoticed. In both humans
and nonhuman primates, aberrant stereotyped
behaviour including self-injury has been associated
with adverse environmental circumstances and early
CNS damage (Lewis et al. ). Unusual stereotyped repetitive movements are considered essential
features of many forms of psychopathology (Stein
et al. ), neurological disorders (Teitelbaum et al.
, ), and are associated with developmental
disabilities including intellectual disability (ID) and
autism (Bodfish et al. ).
Children at risk for and identified with ID and
related developmental disabilities appear to be particularly vulnerable to the development and maintenance of some form of significant stereotypic
behaviour or self-injurious behaviour (SIB) over the
course of their lifetime. Self-injurious behaviour is
among the most disturbing and serious of all behaviours exhibited by children with ID and related developmental disabilities (National Institutes of Health
). Although the developmental course (i.e. onset,
offset, sequence) of early rhythmic behaviours in typical infants is well established (Thelen ), it is less
certain for infants, toddlers, or children at risk for ID
or pervasive developmental disorders. Whether early
patterns of rhythmic motor behaviour are related to
later development of aberrant forms of motor stereotypies and self-injury is not clear. Although it is well
known that severity of intellectual impairment and
the presence of self-injury are inversely related (i.e.
lower intellectual competence, higher likelihood of
SIB; Berkson ), the specific nature of this risk is
unknown as are the underlying biological and behavioural mechanisms associated with the onset of
self-injury. In most cases, the aetiology of SIB is
unknown. Furthermore, it is likely that the original

conditions giving rise to the behaviour disorder, and
the current factors controlling the same disorder
often appear to be different (Oliver ; Symons &
Thompson ; Kennedy ).
Self-injury also co-occurs in a number of syndromes associated with intellectual impairments and
associated developmental disorders including Lesch
Nyhan syndrome (LNS) (Nyhan & Wong ),
SmithMagenis syndrome (SMS) (Dykens & Smith
), Rett syndrome (Oliver et al. ; Deb ),
de-Lange syndrome (Jackson et al. ), Prader
Willi syndrome (PWS) (Dykens & Kasari ;
Symons et al. ), and fragile X syndrome (FXS)
(Symons et al. ). In LNS, self-injury is reported
as chronic, localized primarily to the fingers and oral
areas (e.g. lips, tongue), and primarily consists of
biting (Anderson & Ernst ). In SMS, self-injury
is episodic and characterized by body squeezing,
removal of fingernails, and inserting objects into
bodily orifices (Deb ; Smith et al. ). In contrast, people with PWS tend to skin pick over wide
areas of their body (Symons et al. ). In FXS,
boys (who are more severely affected) commonly bite
their fingers and the back of their hands are disproportionately targeted as the most prevalent self-injury
body site (Symons et al. ). Overall, given findings to date, the within-syndrome expression of selfinjury appears to vary along a number of dimensions
including severity, form, and body locations. Comparatively little work, however, has been conducted
with respect to onset.
The literature is replete with numerous models
and theories concerning the origin of aberrant
stereotyped and self-injurious behaviour (Lewis &
Baumeister ; Guess & Carr ; Jones et al.
). Many of the models are constructed from
multiple sources of evidence and a variety of theoretically based arguments. Less clear, however, is the
nature of the evidence based directly on studies of
very young children. Most recently, Turner ()
and Berkson & Tupa () reviewed the literature
on repetitive behaviour disorders in autism and developmental disabilities, respectively. Both reviews concluded that most studies have been designed to
document prevalence and there is a need for further
systematic comparative study of the origins and natural history of repetitive behaviour disorders to
inform intervention. Because of this, it is difficult to
Blackwell Publishing Ltd, Journal of Intellectual Disability Research ,

146
reach consensus about the most empirically defensible position regarding the early development of
aberrant repetitive behaviour among children with
developmental disabilities. The purpose of this paper
therefore was to review in detail the specific methods
and procedures used and the outcomes documented
in empirically based reports addressing the early
development of stereotypy and self-injury in children
under years of age. Our overall objective was to
synthesize the procedural knowledge base underlying
current conceptualizations of the early development
of aberrant stereotyped and self-injurious behaviour
to clarify areas of consensus and suggest specific
directions for further research.
Method
met our inclusion criteria and journals were cited

in this review.
Review parameters
In addition to a conventional narrative review for
studies with at-risk children, two sets of review
parameters were used to characterize study methodology and findings. Methodological characteristics
included study group (typically developing, delayed/
at-risk development), age, study design (retrospective, prospective, group comparison, single group,
case, other), and measurement strategy (direct observation, interview, survey/questionnaire, rating scale,
combination, or other). Study findings included
prevalence, onset/development, severity, and topographies of repetitive stereotyped behaviour.
Selection criteria
Studies were included for the review if they met the
following criteria: () study participants ranged in age
from to months, or the overall mean age fell in
this range; and () studies were empirically based
in that some form of repetitive motor movement
(rhythmic motor behaviour, motor stereotypy, selfstimulation, or SIB) was operationally defined and
measured. Studies were excluded for the review if:
() they were treatment-only studies that did not
include some additional aspect of onset, severity, or
development; and/or () they were studies of motor
development per se or developmental milestones only.
Search procedures
The three literature bases searched were MedLine,
PsychLit, and ERIC during the years
(Summer). Search terms included developmental
delay, developmental disability, intellectual disability,
at-risk, medically fragile, infant, toddler, children in
multiple combinations with self-injury, self-injurious
behaviour, stereotypies, stereotyped behaviour, selfstimulation, self-stimulatory behaviour, and rhythmic
motor behaviour. Reliability of the selection criteria
was checked by two authors (F.S., L.S.) independently reviewing each retrieved article along the
dimensions of the inclusions/exclusion criteria outlined above. Agreement rate (number of agreements
divided by number of agreements plus disagreements) was %. Eighteen studies were found that
Results
A total of studies were identified using the search
procedures specified. Twelve studies included data on
children who were at risk for developmental delay or
identified with a developmental disability (see
Table ) and studies included data on typically
developing children (Table ).
Subject characteristics
For each study reviewed, the mean age of the subject
sample and the total sample size studied are listed in
Tables and . In the studies on typically developing
children the range of ages studied was months
and the sample sizes ranged from to . In the
studies on children at risk/delayed the range of ages
studied was months (although one studied
reported studying newborns without specifying exact
age at the time of study Kravitz & Boehm )
and the sample sizes ranged from to . On average, it appears that the study of repetitive behaviours
in at-risk children frequently is begun at a later age
relative to studies of typically developing children.
This most likely relates to a latency period following
birth before atypical conditions are identified.
Design characteristics
In the studies with children at risk for delay or
identified with a disability, five studies used prospec-
34
20
500
12
224
351
18
59
59
59
3
39
Kravitz & Boehm (1971)

Field et al. (1979)
Berkson et al. (1985)
Schwartz et al. (1986)
Campbell et al. (1990)
Short & Simeonsson (1990)
MacLean et al. (1991)
Wehmeyer (1991)
Wehmeyer (1994)
Schneider et al. (1996)
Hall et al. (2001b)
Berkson et al. (2001)
NR
NR
NR
22
54
35.5
19
27.4
27.4
NR
24
21/17*
Mean age
in months
P
GC
GC
GC
SG
GC
P, GC
CS
GC
R
P, SG
P, SG
Design
DO, I
I
S
DO
DO
DO
DO
DO
DO
I
DO, I
DO, I
Measurement
Y
N
Y
N
Y
Y
N
Y
N
N
N
Y
Prevalence
N
N
N
Y
Y
N
Y
Y
N
N
Y
Y
Severity
Y
Y
N
N
N
N
Y
Y
Y
Y
Y
Y
Age of onset
Y
Y
Y
Y
Y
Y
Y
Y
Y
Y
N
Y
Stereotypy
Y
N
N
N
N
Y
N
N
N
Y
Y
Y
Self-injury
P, prospective; GC, group comparison; SG, single group; CS, cross-sectional; R, retrospective; DO, direct observation; I, interviews/ratings; S, survey; NR, not reported; Y, yes; N, no.
* Self-injurious behaviour (SIB) subgroup, months; stereotypy subgroup, months.
Study
Table 1 Methodological characteristics of studies reviewed that included children at risk for or identified with disability
147


148
P, prospective; GC, group comparison; CS, cross-sectional; R, retrospective; DO, direct observation; I, interview; S, survey, Q, questionnaire; Y, yes; N, no.
* Mean age not reported. Figures given are reported age ranges.
Y
Y
N
N
Y
N
N
Y
N
Y
Y
Y
Y
Y
Y
Y
Y
Y
Y
Y
Y
Y
N
N
Y
N
N
Y
N
Y
140
525
20
79
156
12
10
57
10
132
Kravitz & Boehm (1971)
Sallustro et al. (1978)
Field et al. (1979)
Thelen 1979
Werry et al. (1983)
Schwartz et al. (1986)
MacLean et al. (1991)
Troster (1994)
Tan et al. (1997)
Foster (1998)
(012)*
18
NR
(152)*
(359)*
4
5.8
(1060)*
60
55
P
R
GC
P
GC
GC
P, GC
CS
CS
CS
DO, I
S
I
DO
S, Q
DO
DO
S
DO
I
N
Y
N
Y
N
Y
Y
Y
N
N
Y
Y
Y
Y
Y
N
Y
N
Y
N
Self-injury
Prevalence
Measurement
Design
Mean age
in months
n
Study
Table 2 Methodological characteristics of studies reviewed that included typically developing children
Severity
Age of onset
Stereotypy
tive designs (%), six were group comparison

(%), two were a single group (%), one was crosssectional (%), and two were retrospective (%).
Percentages do not tally to % because a study
could be classified into more than one design category. In the studies with typically developing children, three studies used prospective designs (%),
three were group comparison (%), three were cross
sectional (%), and one was retrospective (%).
Overall, the most common design was based on
group comparison (% of the studies).
Measurement procedures
A variety of measurement methods were used in the
studies reviewed. Among the studies of children at
risk or delayed, % used direct observation
measures, % used parent/teacher interview/rating
measures, and % (one study) used a survey measurement procedure. Percentages exceed % of
studies as two studies used two measurement strategies. Among the studies of typically developing children % used direct observation measures, %
used parent interview measures, and % used survey measurement procedures. Overall, the most common measurement strategy was based on direct
observation (% of studies).
Prevalence studies
Ten of the studies reviewed produced data on the
prevalence of specific types of repetitive behaviours
(five studies of typically developing children, five
studies of children at risk/delayed). Eight of these
studies also included data on the prevalence of SIB.
Of these prevalence studies, two utilized a prospective
development method and direct observation procedures in whole or in part for quantifying repetitive
behaviour (Kravitz & Boehm ; Berkson et al.
).
Kravitz & Boehm () studied several cohorts
of infants for repetitive and rhythmic behaviours.
Their cohorts included typically developing infants
(n = ), greater than g/perinatal disease
(n = ), low birthweight/perinatal disease (n = ),
and low birthweight/no perinatal disease (n = ).
Kravitz and Boehm included a second sample comprised of typically developing infants, infants
with cerebral palsy and infants with Down syn-

149
drome. These children were followed monthly from

age month to year. During each monthly visit,
mothers were queried regarding the onset of a range
of rhythmical movements. Repetitive behaviours
measured included hand sucking, foot kicking, lip
sucking and biting, body rocking, toe sucking, head
rolling, head banging, and teeth grinding. Direct
observation was used for the onset of hand sucking
in the first study sample and a maternal questionnaire
for prevalence of rhythmic behaviours in the second
study sample. Overall, they found that % engaged
in body rocking, % in head rolling, and % in head
banging. Also reported was a . : male to female
ratio for head banging. Unfortunately, their data were
reported in such a manner as to make it impossible
to examine group differences with regard to
prevalence.
Short & Simeonsson () investigated
infants and children with handicapping conditions
(ID, auditory/speech problems, orthopaedic handicaps, Down syndrome, and multiple handicaps)
(mean = . months). Using the Carolina Record of
Infant Behaviour (CRIB) (Simeonsson ), they
measured repetitive stereotypical behaviours including hand/finger sucking, foot kicking, lip biting, body
rocking, pica, head rolling, body arching, head banging, tooth grinding, hair pulling, breath holding,
ruminating, tongue thrusting, throwing head back,
and hand flapping. Overall, the most frequent repetitive behaviours were hand/finger sucking, foot kicking, body arching, throwing body back, and body
rocking (> % in each category of disability) with
handicapping condition reported to be only weakly
related to stereotyped behaviour.
Campbell et al. () reported on baseline stereotypic movements in children with autism (mean
age = . years). All but one child was rated as having at least mild symptoms related to stereotyped
behaviours (.%). A subset of children was
directly observed and the Timed Stereotypies Rating
Scale (TSRS; Campbell ) was completed. Based
on the TSRS, the most predominant stereotypies
involved the oral facial area, other upper extremities,
and other lower extremities. The specific movements
in the category other, however, were not described.
Wehmeyer (, ) found that % (/) of
a sample of young children at risk for moderate,
severe, and profound retardation were judged by professionals to exhibit pathological stereotypies with a
mean age of onset at . months. All children in this

sample exhibiting pathological stereotypies as determined by an ad hoc Atypical Rating Scale (see below)
were years of age or older. However, it was not clear
what proportion of the total sample exhibited general
stereotyped behaviour patterns.
Berkson et al. () reported a prevalence of .%
(/) of a sample of infants/toddlers enrolled in
early intervention programmes engaged in some form
of self-injurious and/or proto-injurious behaviour and
% (/) were observed or reported to body rocking. Considering the longitudinal nature of the study,
.% and % could also be interpreted as the incidence of self-injury/proto-injury and body rocking,
respectively, among at-risk children with several caveats specific to the nature of the study (i.e. no random
sampling, fidelity of measures, restricted sample).
Severity studies
Nine of the studies reviewed produced data on the
severity of repetitive behaviours (five studies of typically developing children, five studies of children at
risk/delayed). Severity was typically operationalized
and measured using either a Likert severity rating or,
alternatively, in terms of duration of occurrence. For
example, Schwartz et al. () reported topographical differences between children with and without ID
for duration of hand gazing and body rocking. Children with severe ID spent more time hand gazing and
repetitively moving their hands while hand gazing and
they body rocked with a greater number of repetitions
but with smaller amplitudes than typically developing
children. None of the studies provided any standardized measures of the severity of behaviour in terms of
its intensity or in terms of perceived interference with
other activities or behaviours. Berkson et al. (),
however, did classify early SIB as injurious, protoinjurious, or transient although it is not exactly clear
how the term injurious was operationally defined.
Wehmeyer () constructed an Atypical Rating
Scale for use by teaching staff to judge the extent to
which the childs stereotyped movements interfered
with instruction, required intervention, and were perceived as atypical. Hall et al. (b) used a similar
strategy for three young children with LNS and asked
nursery school staff to rate their level of concern
specific to each childs topography of self-injury. In
addition, Hall et al. (b) documented that for two

150
of their LNS cases, protective equipment was in place

by as early as and months to prevent biting with
one case having already experienced tooth extraction.
Overall, all studies of severity involved measuring
severity of stereotypies, with two studies examining
the severity of self-injury during early development
(Berkson et al. ; Hall et al. b).
Age of onset
Fifteen of the studies reviewed included data on the
age of onset of the repetitive behaviour under study
(seven studies of typically developing children, eight
studies of children at risk/delayed). Three of the ageof-onset studies utilized a prospective, direct observation methodology (Thelen ; MacLean et al.
; Berkson et al. ) and one study that used
direct observation to track self-injury also used parent
interview to document estimated age of onset of SIB
(Hall et al. b). Seven of the age-of-onset studies
included data on self-injury. Comparing typical
and at-risk/delayed children, MacLean et al. ()
found that on average typically developing children
exhibited repetitive motor behaviours between
and months of age, whereas the children with
developmental delay/disability (Down syndrome)
exhibited repetitive motor behaviours between
and months of age. In contrast, Kravitz & Boehm
() reported that self-injury (head banging)
occurred after months of age in over % of children in both the typical and atypical development
groups. Berkson et al. () did not have a comparison group, but did document the apparent sequence
of development of severity of self-injury with the
mean age of onset of injurious behaviours (i.e. tissuedamaging SIB) occurring at months, the mean age
of onset of transient-injurious behaviours occurring
at months, and the mean age of onset of protoinjurious behaviours occurring at months.
Although Berkson et al. () reported on body
rocking, it was difficult to ascertain from the manuscript the age of onset by type of body posture. Based
on this very limited amount of available data: ()
stereotypies may tend to occur prior to self-injury;
and () atypically developing children may exhibit a
developmental lag in the onset of stereotypies but not
self-injury.
In the MacLean et al. () study, three groups
of pre-ambulatory children matched on motor
age typically developing (. months), Down syndrome (. months), and profound motor impairment (. months) were examined. Rhythmic
motor behaviours head, mouth, arm, legs, hands,
feet, and torso were recorded bi-weekly using direct
observation embedded in a longitudinal design over
the course of months. Each group exhibited
rhythmic motor behaviour of the type reported
in previous studies of typically developing preambulatory children (Thelen ). Further longitudinal analyses over the entire observation period
revealed that the rhythmic motor behaviour of children with Down syndrome was more similar to that
exhibited by the nondisabled children than was the
rhythmic motor behaviour of the children with motor
impairment.
Field et al. () compared two groups of highrisk infants with a typically developing group over the
first year of life. Their findings, based on maternal
report, suggested that the onset of rhythmic behaviours were related to gestational age differences. In
other words, apparent initial differences in the onset
of rhythmic behaviour disappeared when a correction
was made for prematurity. Group differences, however, were reported for first-year motor skills (as measured by the Bayley Scales of Infant Development)
(Bayley ) even after correcting for gestational
age, suggesting that gross motor behaviour was
affected negatively by perinatal complications.
Schneider et al. () reported on a study of retrospective parental recall of the early occurrence of
stereotypical, self-stimulatory, self-injurious, and
related repetitive movements. Fifty-nine parents of
children with disabilities were interviewed and asked
to recall events salient to the early development of
their childs current SIB. Problems associated with
retrospective interviews notwithstanding, they found
that % (/) of parents reported age of onset
from between and years, their first reactions were
to caution and physically hold their child (%),
and % of parents reported some form of rhythmic
precursor (sucking, rubbing, fluttering, etc.). Specific
details of the age of onset between and were not
available.
Wehmeyer () documented age differences in
the onset of stereotyped behaviours using direct
observations repeated once per month for three
consecutive months. Based on comparisons with
Thelen (), similar developmental patterns were

151
observed, but with delayed onset and prolonged

duration of repetitive movements. Within the same
group, Wehmeyer () also documented individual
characteristics related to age of onset and developmental pattern. Consistent with previous findings
among typical and at-risk children, level of disability,
ambulation, and postnatal hospitalization were identified as factors related to the expression of atypical
stereotyped behaviours.
Hall et al. (b) reported data based on parent
interview on the estimated age of onset of self-injury
for three young boys with LNS. In each case, severe
self-injury appeared early in the childs development
with initial self-scratching first reported at months,
hair pulling by months, and injurious biting by
months. Early head banging and face scratching
were also reported to occur at or around these times
but with no specific age of onset noted.
Berkson et al. () included information regarding the mean age of onset of self-injury ( months),
transient-injury ( months), and proto-injury
( months) based on a combination of direct observation, parent report, and staff report. Within the
same group, mean age of onset for different forms of
self-injury/proto-injury could be determined from
data reported in a summary table that included
eye poking/pressing ( months), head banging
( months), face hitting/slapping ( months), hair
pulling ( months), self-scratching ( months),
head hitting ( months), self-hitting/slap
( months), self-biting ( months). It was not clear

from the data reported, however, whether the age of
onset for body rocking was recorded, thus making it
difficult to draw any conclusions about the relation
between onset of self-injury/proto-injury and more
general stereotyped movement (i.e. body rocking).
Behavioural topographies
Across the studies reviewed, a total of discrete
forms of stereotyped behaviour and broad but
discrete forms of SIB were studied. These discrete
behaviours are listed in Table . Although the forms
of self-injury studied were essentially identical in the
typical and atypical samples studied, there was a subset of stereotypic behaviours that were studied only
in at-risk children with developmental delays (see
Table ). This should be viewed cautiously though, as
there were few direct group-comparison studies and
as few studies used the same behavioural measurement procedures across the groups studied.
Genetic syndromes and behavioural phenotypes

Although almost no work of a developmental nature
specific to emergent self-injury within syndromes has
been performed, there were two exceptions including
LNS and FXS. In LNS, Hall et al. (b) reported
that the onset of SIB for three young boys was sudden
rather than progressive, severe (i.e. tissue damage),
Table 3 List of discrete forms of stereotypic and self-injurious behaviours examined in studies reviewed
Stereotypic behaviours
Self-injurious behaviours
Typical & at-risk

children
Typical children
only
At-risk children
only
Typical & at-risk

children
Typical
children only
At-risk
children only
Body rocking
Head rolling/tilting
Object manipulation
Hand/finger movement
Foot kicking
Facial grimace
Breathing/puffing noises
Tongue wagging
Hand gazing
Head shaking/nodding
Toe sucking
Object mouthing
Leg shaking
Rubbing body part
Toe wiggling
Arm waving
Neck extension
Eye blinking
Hand posturing
Grunting noises
Tip toe walking

Mouth opening
Lip sucking
Eyebrow movements
Mouth twitching
Smell/sniff movements
Covering eyes/ears
Head banging
Hand sucking
Hair twirling/pulling
Lip biting
Scratching
Pinching
Rubbing
Eye poking
Eye pressing
Self-biting
Self-hitting
Nail biting
Body throwing

152
and correlated with periods of low social interaction.

In FXS, Symons et al. () reported the prevalence, onset, form, function, location, and correlates
of SIB from a sample of elementary school-aged
students with full mutation FXS. Although the age
of the study sample was on average greater than
months (reported mean age of the boys at the time
of the survey was months), SIB occured for %
of the boys with a mean age of onset of months.
Biting was the most commonly reported form of selfinjury with the fingers and back of the hand disproportionately targeted as the most prevalent self-injury
body site. Using a hazard analysis, no linear increase
in risk of SIB with age past months was found.
Discussion
There is an extremely limited database directly
addressing the early development of stereotypy or
self-injury in children at risk for or identified with
developmental delay or disability. The studies that do
exist have been sporadic, used different definitions,
measurement strategies, and cohorts of children,
making direct comparisons difficult if not impossible
among at-risk groups or typically developing infants
and children. A number of tentative conclusions,
however, may be drawn regarding the delayed onset
but similar developmental pattern of early stereotyped movements in children at risk for delay or
disability. There is little to suggest that risk factors
were well understood for aberrant stereotyped movements or self-injury and almost no evidence concerning developmental or behavioural mechanisms in
relation to biology and disabling conditions.
A number of limitations of this review deserve
comment. First, studies were undoubtedly missed,
particularly those not in English. We did not review
animal research, nor did we consider treatment-only
studies, both of which may have been designed to
elucidate mechanisms relevant to developmental
issues. Second, because we were interested in the
nature of the evidence concerning the development
of atypical repetitive and self-injurious behaviour in
young children with intellectual and developmental
delays and disabilities, concept papers advancing theoretical perspectives or particular models (Lewis &
Baumeister ; Kennedy ), previous literature
reviews (Oliver ; Nijhof et al. ), treatment-
only studies (Horner ), studies of nonhuman

primates (Harlow et al. ) or other animals (Powell et al. ) were not included for review. Finally,
other primary disabling conditions also associated
with stereotyped behaviours, most notably visual
impairment, were not reviewed (for an earlier review
of children with visual impairments, see Eichel
and Troster et al. ).
Several measurement strategies were evident in the
studies reviewed: direct observation of repetitive
actions by body area (Thelen ; MacLean et al.
), direct observation of specific topographies
(Short & Simeonsson ; Berkson et al. ),
survey and checklists (Werry et al. ), or retrospective recall (Schneider et al. ) of a variety of
topography-specific forms of stereotypy and selfinjury (e.g. body rocking, head banging). The variety
of measurement procedures that we found may relate
to the absence of a standardized, psychometrically
sound scale for measuring early repetitive behaviours.
This is an important area for future research, as the
adoption of a valid instrument or set of procedures
across research groups would permit reasonable comparisons across studies.
Certain topographies have received more detailed
attention than others, most notably head rolling,
body rocking, and head banging. It is likely that
issues related to health, social concerns, and study
methodology determine, in part, this selection bias.
On the other hand, the small number of studies
reviewed have included a wide range of topographies
(see Table ), but it is difficult to draw conclusions
about the interrelations among stereotyped movements within and between individuals because the
appropriate comparison studies have not been carried
out.
There are only limited data on the differences
between typically and atypically developing children
and repetitive behaviour. There are apparently the
same or similar forms of early repetitive behaviour
and the same or similar sequence of development
through forms for typically developing children and
children at risk for delay or identified with disabilities
(MacLean et al. ; Wehmeyer ). There is also
limited evidence for a general delay in expression of
repetitive behaviour associated with atypical development (Wehmeyer ). No data were available to
allow direct comparisons on severity, risk factors, or
developmental impact of repetitive motor behaviour.

153
One exception to the general lack of empirical data

on the developmental impact of early repetitive movement was reported recently in an investigation with
dependent measures outside those included in this
review. Kroecker et al. () examined the relationship between early rhythmic movements of month-old infants and cognitive outcomes at years
of age. These investigators coded rhythmic motor
behaviour in -month-old children born prematurely and at risk for developmental delay and a fullterm comparison group during -min segments
from two structured interaction contexts, snack and
free play. Few differences in number, duration, or
profile of rhythmic movement bouts were observed
between the two groups at months. After controlling for background variables (gender, prematurity
status, and socio-economic status), level, but not
duration of rhythmical movement at months was
significantly related to childrens performance on the
Bayley Scales of Infant Development (MDI) at
years of age. Moreover, the direction of this relationship varied depending on social context. Higher
levels of rhythmical movement in the free play context
were associated with lower MDI scores. In contrast,
higher levels of rhythmic movement exhibited in the
snack context predicted higher MDI scores at age ,
but only for boys. That the direction of the relationship between rhythmical movement and later outcomes differed by context suggests that these early
topographically similar forms may function differently depending on social variables.
The developmental relation between the sequencing of stereotypy and self-injury is not clear. Guess &
Carr () proposed a -stage model outlining the
possible relations among early rhythmic patterns of
behaviour and neurobiological and environmental
variables related to later aberrant stereotyped and
self-injurious behaviour. Consistent with Guess and
Carrs model, Kennedy () suggested several possible behavioural mechanisms through which stereotyped behaviour may evolve into SIB. Implicit in the
conceptual model is the assumption that rhythmic
stereotyped movements are precursors, under specific circumstances, for the later development of selfinjury. To date, no studies have been designed to
explicitly test these ideas and address this issue for
very young children (i.e. < years old). In the simplest scenario, it might be hypothesized that early
stereotypies are precursors to self-injury, or that
delayed onset stereotypies are related to later selfinjury. In either case, appropriately designed prospective studies that include measures of process and
outcome are needed to address the issue. It should
be noted that Murphy et al. () and Hall et al.
(a) are directly addressing this issue with prospective studies designed to document incidence and
applying sequential analytic techniques to direct
observational data to study behavioural processes.
With respect to the current review, however, their
study samples were outside the inclusion range used
for screening studies specific to children under
years of age.
Stereotyped movement disorders including selfinjury are associated with numerous syndromes and
aetiologies underlying ID and developmental disabilities (Stein et al. ). Baranek () recently completed a retrospective home video study examining
early sensory and motor signs of autism and found
that early object stereotypies (i.e. mouthing objects)
were relevant in discriminating among children with
autism, developmental disabilities, and typically
developing children. Stereotyped movements are
associated with FXS (Hagerman & Cronister ),
most notably hand flapping and hand biting
(Lachiewicz et al. ; Symons et al. ). Upward
of % of children and adults with PWS engage
in compulsive, repetitive skin picking (Dykens &
Cassidy ). In LNS, an X-linked recessive disorder of purine synthesis, the cardinal behavioural feature is loss of tissue through self-biting particularly of
the lips or fingers (Nyhan ). As other syndromes
continue to be identified and their corresponding
behavioural phenotypies characterized (Dykens ;
Hodapp ), opportunities to understand the early
development of self-injury may be realized. In the
majority of genetically identified cases, however, few
data exist concerning the early onset, developmental
sequence, severity, risk factors, or developmental
consequence of stereotypies or self-injury in syndromes associated with ID and related developmental
disabilities. The clear exception to this is the recent
Hall et al. (b) report of the early development of
self-injury in three young boys with LNS. Three
points from their work are particularly noteworthy.
First, in contrast to what we seem to know about the
early development of SIB in at-risk study samples,
the early self-injury evident in LNS is intense and
almost always associated with tissue damage from the

154
beginning (cf. Berkson et al. proto-SIB). Second, and probably because of this, initial interventions can be highly intrusive (e.g. tooth extraction).
Third, based on sequential analyses of prospectively
collected real-time direct observational data, it
appears that early self-injury may be more likely to
occur during time periods characterized by the
absence of social interaction suggesting that there
may, in part, be some level of environmental mediation of self-injury and a role for early behavioural
intervention.
Few studies have examined contextual determinants of moment-to-moment variability in early stereotypies and/or SIB. Thelen (, ) found that
with typically developing infants certain behavioural
states (nonalert) and environmental events (social
interaction) were reliably related to the occurrence
of stereotypies. Kroecker et al. () reported low
correlations between level of rhythmical movement
exhibited in two interactional contexts (snack and
free play) for -month-old infants. Baumeister et al.
() studied seven preschool children identified
with moderate to severe ID. Direct observation was
used during preschool activities across -week time
spans. Stereotypic motor movements including upper
torso rocking, head shaking, hand flapping, complex
hand movements, and hand/finger mouthing were
coded. Several related findings emerged most notably
that rate of stereotypies was context dependent
(actively engaged although individual differences
and daily variability were also reported). In addition,
the adult (teacher) interactions were quantitatively
and qualitatively different for children with and without stereotypies. Finally, there were sequential
dependencies between some types of teacher behaviour and the occurrence of stereotyped behaviour
(approach child, negative contact). In other words,
certain temporal sequences of adultchild interactions related to stereotyped behaviour occurred
beyond chance levels. Using similar sequential analytical methods, Hall et al. (a) found significant
sequential dependencies with specific environmental
or social contexts (e.g. low levels of social contact)
for a subset of elementary school-aged children with
self-injury that increased over time and three young
boys with LNS (Hall et al. b). Finally, in their
work on early development of SIB in elementary
school-aged study samples, Murphy et al. ()
showed that teacher concern was the only environ-
mental variable predicting change in self-injury over

time. Taken together, it appears that more detailed
study of change in self-injury/stereotypy over time
in relation to static (i.e. nature of disability) and
dynamic (i.e. social interaction) variables is
warranted.
The issue of normal vs. abnormal movements
related to repetitive behaviour remains to be more
fully addressed. Is abnormal movement related to
increased frequency, increased severity, multiple
numbers of atypical forms, delayed onset or
sequence, or adverse effect on development? In an
older group of children, Smith & Van Houten ()
showed that although few differences existed between
matched children with and without disabilities in the
per cent of time engaged in stereotyped movements,
the children with delays were characterized by higher
levels of obvious gross motor behaviours, and as
being visually oriented toward their stereotyped
behaviour. In turn, the children with delays and obvious gross motor stereotyped behaviours were more
likely to receive a strange or bizarre rating based on
these differences by blind, naive raters. The finding
related to visual orientation is consistent with the
results from Schwartz et al. () with much
younger children with disabilities. From a clinical
perspective, distinguishing between those behaviours
that are part of the normal developmental repertoire
and will eventually disappear and those that are
abnormal, likely to persist, and become pathological
remains a critical issue for the timing and intensity of
treatment (Tupa & Berkson ).
Knowledge of the early developmental course with
respect to precursors and correlates could also
inform the construction of traditional psychiatric
diagnostic categories. In the current version of the
American Psychiatric Associations diagnostic and
statistical manual (DSM IV), stereotypic movement
disorder is linked with SIB. Implicit in this taxon is
an assumption about a significant relation between
stereotyped movement and SIB. Although the two
behaviour disorders tend to co-vary in study samples
of adults with intellectual impairments and related
developmental disabilities (Bodfish et al. ),
more work needs to be conducted that informs diagnostic categories with respect to developmental
issues and emergent categorical disorders among
individuals at risk for ID or with identified neurogenetic syndromes.

155
No studies have been conducted that prospectively

examine risk factors for the early development of
aberrant stereotyped or self-injurious behaviour in atrisk children under years of age. The findings from
Field et al. () suggested that the onset of
rhythmic behaviours was unaffected by perinatal
complications of respiratory distress syndrome and
post-maturity. Earlier reports with older samples
have noted a range of possible factors correlated with
stereotypies and self-injury including crib confinement (Warren & Burns ), communication deficits (Schroeder et al. ), age (Berkson et al. ),
sensory deficits (Jan et al. ), and early rearing in
impoverished environments (Ames ). The majority of these findings were correlational and limited to
retrospective designs. Wehmeyer () made the
point elsewhere that the effects of early experience
and environmental factors remain to be fully explored
in relation to the development of atypical repetitive
movements. We still lack direct evidence that developmentally typical repetitive movements observed in
young children at risk for or identified with delay or
disability evolve into atypical repetitive movement
disorders.
In sum, many of the data regarding the prevalence,
phenomenology, and early development of self-injury
and related stereotypic behaviours in infants, toddlers, and children at risk for or identified with ID or
related developmental disabilities are dated and
sparse. Methodological differences limit direct comparisons across many of the studies. There are, however, a number of areas in which consensus has been
established and tentative conclusions may be made.
First, it is clear that a variety of repetitive behaviours
do occur during the early development of infants. In
the case of typical development, there is strong evidence that rhythmic repetitive behaviours occur in all
infants (Thelen , ), and that SIB can occur
in a substantial minority of infants (Kravitz & Boehm
). There is also some evidence that the same is
true for stereotypic (MacLean et al. ) and possibly self-injurious (Kravitz & Boehm ; Schneider
et al. ; Berkson & Tupa ) behaviours in atrisk infants. Despite the apparent ubiquity of these
behaviours we know very little about the developmental, functional, or diagnostic significance of early
repetitive behaviours in at-risk populations. Consequently, questions about the early identification of
self-injury and prevention strategies targeting risk
factors remain unanswered. Only rarely are these

problems identified early on although early intervention services are assumed to be more effective when
begun sooner than later (Guralnick ). Thus,
there remains a need for appropriately designed comparison studies that include uniform quantitative
measures of the early development of stereotypic and
self-injurious behaviours.
Acknowledgements
This work was supported, in part, by PHS Grant No.
HD and HD from the National Institute
of Child Health and Human Development.
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development of stereotypy and self-injuryF. J. Symons et al.