J. Child Psychol. Psychiat. Vol. 40, No. 6, pp.

969–978, 1999
Cambridge University Press
' 1999 Association for Child Psychology and Psychiatry
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0021–9630\99 $15.00j0.00

A Case-comparison Study of the Characteristics of Children with a Short

Stature Syndrome Induced by Stress (Hyperphagic Short Stature) and a
Consecutive Series of Unaffected ‘‘ Stressed ’’ Children
Jane Gilmour and David Skuse
Behavioural Sciences Unit, Institute of Child Health, London, U.K.

Recently a type of growth failure (Hyperphagic Short Stature) has been described, in which
there is potentially reversible severe impairment of growth hormone secretion, in association
with excessively high levels of psychosocial stress. This condition is a variant of the disorder
formerly known as Psychosocial Dwarfism. In the present study we compared children with
Hyperphagic Short Stature (N l 25, aged 9n04 yearsp3n78, 72 % male) and a closely
matched sample with normal height, drawn from comparably stressful family circumstances
(N l 25, aged 10n61p3n04, 60 % male). Measures of the psychosocial environment,
anthropometry, and developmental history from infancy were obtained. Many symptoms
thought previously to be characteristics of psychosocial dwarfism were found to be
nonspecific stress responses. Hypotonia (p n05), enuresis\encopresis (p n01), and sleep
cycle disruption (p n05) did differentiate the groups. Growth, appetite, and sleep are all
influenced by hypothalamic nuclei, suggesting hypothalamic pathology could account for
most of the clinical features of Hyperphagic Short Stature.

Keywords : Behaviour problems, distress, eating behaviour, endocrinology, growth re-

tardation.

Abbreviations : EBD schools : schools for emotionally and behaviourally disturbed children ;
EE : expressed emotion ; HSS : Hyperphagic Short Stature ; HSSDI : Hyperphagic Short
Stature Diagnostic Interview ; PSS : Psychosocial Short Stature.

Introduction moved from adversity. However, the importance of the

symptom of hyperphagia as a predictor of children with
Linear growth may become impaired in some children that capacity for reversibility, and subsequent catch-up
as a consequence of psychosocial or maternal deprivation growth, was not recognised at the time.
and abuse, an observation first recorded over 50 years The diagnostic criteria for HSS are given in Table 1.
ago (Talbot, Sobel, Burke, Lindemann, & Kaufman, We estimate that the population prevalence is around 3 %
1947). This group of disorders has been known as of exceptionally short children on the basis of an
Psychosocial Dwarfism or as Psychosocial Short Stature epidemiological study (Skuse, Albanese, et al., 1996).
(PSS). Recently a novel variant of PSS has been described, Below we review the existing literature on the Hyper-
Hyperphagic Short Stature (HSS) (Skuse, Albanese, phagic Short Stature variant of PSS. Although the
Stanhope, Gilmour, & Voss, 1996). The clinical features condition of HSS was described in detail, with validation
of HSS include a retarded growth velocity, secondary to on the basis of physiological characteristics, only in 1996
growth hormone insufficiency, together with an insatiable (Skuse, Albanese, et al., 1996), it is possible retro-
appetite, or hyperphagia. The phenotype is found in the spectively to identify the syndrome in previously pub-
context of a chronically stressful (usually an abusive) lished literature. We will thus be referencing reports
environment (Skuse, Albanese, et al., 1996) and it appears which have provided sufficient symptomatic detail to
to manifest only in children with a congenital pre- make it possible to identify a subgroup of HSS cases
disposition, which is probably genetic in aetiology. A within broader clinical samples. In the review we will
series of children with stunted growth, some of whom had make reference to ‘‘ HSS ’’ where it has been possible
similar features, was originally described by Powell, retrospectively to make the diagnosis with a reasonable
Brasel, Raiti, and Blizzard (1967a, b). They emphasised degree of certainty, based on our clinical diagnostic
the fact that some of those children from unhappy home algorithm (Table 1).
circumstances had the capacity for spontaneous reversi-
bility of their ‘‘ idiopathic hypopituitarism ’’ when re-
Anthropometric Features
Requests for reprints to : Dr J. Gilmour, Institute of Child Short stature or poor growth velocity is always
Health, Behavioural Sciences Unit, 30 Guilford Street, London observed in HSS (e.g. Money, 1977 ; Mouridsen &
WC1N 1EH, U.K. Nielsen, 1990 ; Powell et al., 1967a, b). However, despite

969
970 J. GILMOUR and D. SKUSE

Table 1 behavioural features (Skuse, Albanese et al., 1996 ; Skuse,

Clinical Criteria for a Diagnosis of Hyperphagic Short Gilmour, et al., 1996). Intrafamilial stressors do not
Stature necessarily implicate or involve the child ; marital conflict
Cases should show at least two of the following behaviours may be sufficient to provoke expression of the phenotype
from group A and at least one from group B. Evidence that in vulnerable children. However, emotional (e.g. Bowden
the behaviours have persisted over a period of at least 3 & Hopwood, 1982), physical (e.g. Money, 1977), and
months is essential. sexual abuse (e.g. Money, Annecillo, & Lobato, 1990 ;
Group A Stanhope et al., 1988) have frequently been found in
$ gorging and vomiting association with HSS and are probably the usual reasons
$ stealing food at home for its manifestation in those with the predisposition.
$ stealing food at school Accordingly, abuse is not the only cause of the disorder,
Group B but it is the most frequent cause of severe and chronic
$ stealing food at night or early morning stress in childhood.
$ hoards food
$ forages for discarded food
$ eating excessively
Other Behavioural Features
$ drinking excessively Links have been found between sleep and growth
Affected children should be : hormone regulation in childhood (e.g. Friess,
$ between 3rd and 97th centile for weight, according to Wiedemann, Steiger, & Holsboer, 1995 ; Hayashi,
national standards Shimohira, Saisho, Shimozawa, & Iwakawa, 1992). Night
$ below the 3rd centile in height or below the 3rd centile in
roaming has frequently been associated with HSS
height in relation to parental height, or a consistent height
velocity below the 25th centile for at least 1 year prior to
(Bowden & Hopwood, 1982 ; Green, Campbell, & David,
presentation. 1984 ; Money, 1977 ; Money, Annecillo, & Hutchinson,
1985 ; Mouridsen & Nielsen, 1990 ; Powell et al., 1967a)
and it has been suggested the growth disorder is secondary
their bizarre food-seeking behaviours (e.g. Money, 1977 ; to a deficiency of stage III\IV sleep, that period of sleep
Mouridsen & Nielsen, 1990 ; Powell et al., 1967a, b ; Silver when growth hormone is normally secreted in a pulsatile
& Finkelstein, 1967 ; Wolff & Money, 1973), children fashion (Van Cauter & Plat, 1996). However, there is no
with HSS are not malnourished. A low body mass index, evidence that sleep disturbance in general is associated
which would indicate chronic undernutrition (Skuse, with impaired growth (Gulliford, Price, Rona, & Chinn,
Gilmour, Stanhope, Albanese, & Voss, 1996), is rarely 1990), and our own research has not supported the
found (Bowden & Hopwood, 1982 ; Money, 1977 ; earlier findings (Stanhope et al., 1988).
Mouridsen & Nielsen, 1990 ; Skuse, Albanese, et al., Enuresis and encopresis have also been associated with
1996). HSS (e.g. Blizzard & Bulatovic, 1992 ; Money et al., 1985 ;
Skuse, Albanese, et al., 1996). Bizarre patterns of be-
haviour have included urination over belongings or
Hyperphagia and Appetite Disturbance smearing of faeces (e.g. Money, 1977). HSS has been
Children with HSS have been described as : eating associated with polydipsia (Bowden & Hopwood, 1982 ;
excessively requiring restraint, gorging and vomiting, Powell et al., 1967a, b ; Skuse, Albanese, et al., 1966 ;
stealing or hoarding food, eating discarded food, and Swanson, 1994), and it is possible that the unusual
displaying pica (Bowden & Hopwood, 1982 ; Hopwood & patterns of urination may be, in part, the physiological
Becker, 1979 ; MacCarthy, 1974 ; Money, 1977 ; response to an excessive fluid intake.
Mouridsen & Nielsen, 1990 ; Reinhart & Drash, 1969 ; Oppositional and challenging behaviour (Blizzard &
Swanson, 1994). Bulatovic, 1992), poor social relationships (Green et al.,
1984), temper tantrums and apathy (Green, Deutsch, &
Stress Campbell, 1987), and depression (Blizzard, 1997 ; Ferholt
et al., 1985) have all been noted in children with HSS.
Former reports assumed stress was coincidental to the Hyperactivity and poor attention were frequently noted
intrafamilial circumstances found in association with in our own series of cases (Skuse, Albanese, et al., 1996).
psychosocial dwarfism, and that the condition was
specifically due to a disorder in the mother–child re- Cognitive Ability
lationship (e.g. MacCarthy, 1974). In our programme of
research into HSS we aimed to test the hypothesis that Children with HSS are often reported to have global
nonspecific stressors, rather than abuse or emotional cognitive deficits (Bowden & Hopwood, 1982 ; Ferholt et
deprivation, play a causal role in its aetiology. Janis and al., 1985 ; Green et al., 1984 ; Money, 1977 ; Money,
Leventhal (1968) define a stressor as that which : Annecillo, & Kelly, 1983 ; Silver & Finkelstein, 1967 ;
‘‘ typically … induces a high degree of emotional tension Skuse, Albanese, et al., 1996), although there are
and interferes with normal patterns of response ’’ (p. exceptions (Blizzard & Bulatovic, 1992).
1043).
The strongest evidence indicating HSS to be a stress- Characteristics of Infant Development
responsive condition is that removal from stress (for
example, from abusive home circumstances into hospital) Low birthweight is not associated with HSS
leads to the rapid resolution of the core physiological and (Mouridsen & Nielsen, 1990 ; Silver & Finkelstein, 1967).
 CHILDREN WITH HYPERPHAGIC SHORT STATURE : A CASE-COMPARISON STUDY 971

Failure to thrive or infantile feeding problems are Methods

relatively common (Blizzard, 1997 ; Bowden & Hopwood,
1982 ; Hopwood & Becker, 1979 ; Reinhart & Drash, Subjects
1969 ; Silver & Finkelstein, 1967), as is motor milestone The case sample comprised children who met criteria for a
delay (Ferholt et al., 1985 ; Hopwood & Becker, 1979 ; diagnosis of HSS (N l 25) (see Table 1). They were recruited
Money, 1977). specifically for the current study and are not the same subjects
described by Skuse, Albanese, et al. (1996). The diagnosis of
Prader-Willi syndrome in the HSS group was excluded using a
Chronic Stress among Children in the General methylation status test (Dittrich et al., 1992) that is 99 %
Population sensitive in detecting the Prader-Willi syndrome genotype (Reis
et al., 1994). The comparison group were living in chronically
Children from the general population who are living in stressful circumstances (N l 25). Sixty-four per cent of children
adverse circumstances are vulnerable to a range of with HSS were recruited at Great Ormond Street Hospital and
psychopathologies, about which there is a considerable a further 24 % from other tertiary referral centres. Advertise-
literature. These include withdrawal (Kolko, Moser, & ments in magazines were used to contact the remainder.
Weldy, 1988 ; Livingston, 1987), acting out (Jensen, Families were invited to respond if their child had a persistently
excessive appetite. A total of 236 responders were screened
Richters, Ussery, Bloedau, & Davis, 1991 ; Watkins &
by means of a questionnaire survey (The Hyperphagic Short
Bentovim, 1992), conduct disorder and hyperactivity Stature Checklist ; Gilmour & Skuse, 1993—unpublished),
(Sirles, Smith, & Kusama, 1989), poor peer relationships which was conducted by telephone or post. The majority of
and aggression (Holden & Ritchie, 1991 ; Skuse & children ascertained using this method either had Prader-Willi
Bentovim, 1994), low self-esteem (Watkins & Bentovim, syndrome or were normal obese children who ate inappropriate
1992), somatic complaints (Rimsza, Berg, & Locke, foods such as sweets. Two children who were probable HSS
1988), and secondary enuresis (Felman & Nikitas, 1983) cases were then seen personally in order to apply the diagnostic
or encopresis (Feehan, 1995). Living in psychosocial algorithm. All but one family ascertained as belonging to the
adversity is also associated with poor cognitive per- HSS group participated in our study.
formance (Kolko, 1992 ; Rutter, 1985c ; Skuse & Fifty-two per cent of the comparison group were recruited
Bentovim, 1994). In contrast, little has been written on through the Child Care Consultation Team and Child Sexual
Abuse Team at Great Ormond Street Hospital. Other families
the impact of chronic emotional stress on appetite were contacted through the Children and Families Units of five
regulation (e.g. Morley, 1987). London boroughs. Information sheets were passed to candidate
families by social workers. The remainder were recruited by
means of a screening questionnaire from schools for emotion-
Summary ally and behaviourally disturbed children (EBD schools). In
There are parallels between the behavioural profile of these cases, names only became known to the authors if the
family responded. Families recruited through social services
HSS and symptoms found in stressed children in the
and EBD schools were offered expenses in return for their
general population. On the other hand, there are certain participation. Owing to the methods of recruitment we were
features characteristic of HSS that have not been de- required to employ, we are unable to report how many families
scribed in the general population of stressed children, were approached and thus we do not know the exact proportion
such as an excessive appetite. In order to show that the who agreed to take part.
clinical features described in our diagnostic algorithm are All 19 comparison children who were recruited directly or
specific to HSS, over and above those which are attribut- indirectly from our primary source (social services departments)
able to a nonspecific stress response, we needed to identify were on an ‘‘ at risk register ’’ or were the subjects of ‘‘ grave
whether such features would also be seen in a sample of concern ’’ about persistent emotional (six cases), sexual (three
children of normal stature who were living in comparably cases), or physical (five cases) abuse or neglect (five cases). Some
adverse circumstances. In view of the fact that the children appeared in more than one category. On the other
hand, it was a condition of the EBD schools’ participation that
majority of children we identified with HSS in the original the precise nature of the families’ circumstances would remain
survey were subject to social services intervention on undisclosed. However, following careful consultation with
account of abuse or neglect, independent of our diagnosis social workers involved in the recruitment from this source, and
(Skuse, Albanese, et al., 1996), we chose to recruit our following our assessment, we are confident that the EBD school
comparison subjects from the same sources, so far as families also met our criteria for chronic and severe stress. In
possible. this group, there were features of physical abuse (three cases),
emotional abuse (seven cases), sexual abuse (one case), and
neglect (three cases).
Aims
In the present study we systematically compared the Measures
parenting of a sample of children with HSS with a closely
matched comparison group of children from similarly Anthropometric measures were obtained using standard
stressful\abusive environments who had not been re- clinic equipment by trained personnel. A software programme
(Boyce & Cole, 1993) was used to convert heights and weights
ferred for the investigation of a growth disorder. We also into standard deviation scores, corrected for age according to
aimed to gather data on the children’s general state of national growth standards. The short form (Kaufman,
health, their developmental history, and their Kaufman, Balgopal, & McLean, 1996) of the Wechsler In-
behavioural, emotional, cognitive, and anthropometric telligence Scales for Children III UK (WISC-III-UK ; Wechsler,
characteristics in order to identify stress-associated 1992) and the Wechsler Preschool and Primary Scale of
features that were specific to HSS. Intelligence-Revised (WPPSI-R ; Wechsler, 1990) were used to
972 J. GILMOUR and D. SKUSE

assess IQ. The Hyperphagic Short Stature Diagnostic Interview p l n29] or fathers [χ#(1) l 0n52, p l n47], or in housing
(HSSDI) was designed specifically to identify children with HSS conditions (rented\owner-occupier) [χ#(1) l 0n44, p l
as no existing published measure was suitable. The HSSDI n51]. In both groups, the largest proportion of families
included questions assessing the types of discipline used by lived in rented accommodation. Sixty per cent of the HSS
parents. An abusive punishment was defined as being hit with and 72 % of the comparison group were single-parent
an implement, locked in a room for a substantial period of time,
or punishments that were considered by the professionals
families [χ#(1) l 0n81, p l .67]. Although there was a
reviewing the case as emotionally damaging—such as a care- trend for comparison children to be younger than HSS
taker destroying one of the index child’s favourite toys. Copies children [t(48) lk1n62), p l n11] and for a larger
of the interview are available from the authors. proportion of girls in the comparison group [χ#(1) l
The Camberwell Family Interview (CFI ; Vaughn & Leff, 0n80, p l .37], these differences did not reach statistical
1976) was incorporated into the HSSDI. JG completed the CFI significance.
training course at the Institute of Psychiatry, London. The
rating of expressed emotion (EE) produces a number of scales :
critical comments and positive remarks (simply frequency Stress and Psychosocial Environment
counts from the interview) and emotional over-involvement,
hostility, and warmth, all of which are global ratings. The scales The HSS group were recruited, consecutively, accord-
are not mutually exclusive. From these scales, care-givers are ing to the signs and symptoms in the diagnostic algorithm
rated as either high or low in terms of their expressed emotion. (Table 1). No preselection was made on the basis of
Care-givers who score over 3 on emotional over-involvement, children’s stressful home circumstances. Despite this,
are rated 1 and above on hostility, or make 6 or more critical 68 % of HSS families had social services involvement,
comments, are regarded as high EE (Vaughn & Leff, 1976). which concerned the parenting that the HSS index child
Ratings were made from audio tapes and transcripts of the
(or in one case, their sibling) was receiving. In the
interview, by a rater who was unaware of case status. The only
information available to the rater was the child’s IQ and age. IQ
remaining seven HSS cases, six were identified by pro-
was considered a relevant factor for rating emotional over- fessionals as having a poor quality of parenting and
involvement, which includes over-protection. Children with emotional environment. Confirmed or suspected physical
severe learning difficulties were judged to require a level of abuse (4 cases), emotional abuse (17 cases), or sexual
protection beyond that normally expected for children of the abuse (3 cases) or neglect (9 cases) was evident in all but
same age. 1 case, with some children appearing in more than 1
We used the Child Behavior Checklist (CBCL ; Achenbach, category. The exception concerned a child who had
1991a), a questionnaire completed by parents, which enquires experienced repeated civil war bombing and frequent
about both internalising and externalising difficulties, and the emigrations whilst living in the Middle East. There were
parallel Teacher Report Form (TRF ; Achenbach, 1991b), no differences in the frequency of confirmed or suspected
which was rated by teachers. Psychometric properties of both
physical abuse [χ# (1) l 2n60, p l n11], sexual abuse [χ# (1)
questionnaires include good test-retest reliability (Achenbach,
1991a, b). Teachers also completed the Difficult Behaviour
l 0n16, p l n68], emotional abuse [χ# (1) l 2n05, p l 15],
Questionnaire (DBQ)—an instrument, based on a questionnaire or neglect [χ#(1) l 0n35, p l n56] between the groups.
developed at the Institute of Psychiatry, London, designed Seven children in the HSS and four from the stressed
to screen for HSS symptoms, and questionnaires enquiring comparison group were removed from home subsequent
more generally about progress and behaviour at school (Skuse, to the assessment, voluntarily, or by Care Order. The
Albanese, et al., 1996). Copies of this questionnaire are available proportion of children removed from home was not
from the authors. significantly different between groups [χ#(1) l 1n05, p l
n30]. Twenty-eight per cent of the HSS, and 20 % of the
comparison care-givers described using frankly abusive
Statistical Testing punishments with their children [χ#(1) l 0n43, p l n51].
Where categorical data were concerned, the groups have been Expressed emotion (EE) ratings provide a description of
compared using conventional nonparametric tests. Analyses of the quality of emotional response the index children are
variables meeting criteria for at least an interval scale of receiving. Fifty per cent of the HSS group care-givers
measurement were undertaken with parametric statistical were rated as high EE as compared to the 54 % of the
analysis. We took cognisance of the fact that in some analyses comparison group [χ#(1) l 0n08, p l n50].
there were a large number of statistical comparisons on the
same data set ; consequently there was an increased chance of
making a type I error if we had employed a series of independent Neonatal and Child Health
univariate statistical procedures. To take this potential problem
into account we employed Multivariate Analysis of Variance, Sixteen per cent (four cases) of the HSS group reported
which controls for the internal relationship between variables difficulty feeding or cuddling their child when an infant
and thus for the possibility of collinearity. Age was used as a owing to floppy limbs, which was interpreted as hypo-
covariate in parametric analyses. tonia. None of the stressed comparison caregives reported
any degree of hypotonia [χ#(1) l 4n56, p l n03], which
can be a nonspecific indicator of developmental delay
Results (e.g. Georgieff, Bernbaum, Hoffman-Williamson, &
Demographic Variables Daft, 1986). The children with reported hypotonia had a
mean IQ of 64n5 (p13n08), ranging from 50 to 77. Thirty-
Socioeconomic status was balanced across groups. seven per cent (N l 8) of the HSS group care-givers
There were no significant differences in occupation reported that their child had failed to thrive. Of these,
(manual or nonmanual) between mothers [χ#(1) l 1n10), three had been admitted to hospital. Two comparison
 CHILDREN WITH HYPERPHAGIC SHORT STATURE : A CASE-COMPARISON STUDY 973

Table 2
Factor Analysis Coefficient Loadings on Hyperphagia Factors
Factor 1 : Factor 2 : Factor 3 :
General School Pica\
Variable hyperphagia hyperphagia polydipsia
Behaviour at home
Stealing food : kitchen cupboardsa n91 n13 n04
Stealing food : fridgea n92 n11 n09
Stealing food : plates at mealtimesa n63 n14 n34
Hoarding fooda n55 kn04 kn15
Overeating requiring restrainta n69 n13 n003
Gorging and vomitinga n28 n31 n11
Polydipsiaa n11 kn03 n78
Picaa n55 kn04 n44
Chewing nonfood itemsa kn05 kn11 n21
Behaviour at school
Foraging in rubbish binsb kn003 n50 n72
Picab kn03 n70 n54
Stealing food at schoola n39 n55 kn12
Stealing food at schoolb n14 n88 n02
a Parent report.
b Teacher report.

children (8 %) failed to thrive and both had been admitted versus Performance IQ discrepancy [χ#(1) l 0n33, p l
to hospital. Sixty-one per cent of HSS group biological n56]. Wechsler (1992) notes that such discrepancies are
mothers and 76 % of comparison group mothers smoked not uncommon in the general population and are not
during their pregnancy [χ#(1) l 0n96, p l n33] ; 17 % and necessarily clinically significant. Of those children with
35 % of HSS and comparison group mothers, respec- significantly different scores, 78 % of the children with
tively, reported that they drank alcohol during their HSS and 58 % of the comparison group had a Per-
pregnancy [χ#(1) l 1n59, p l n21]. formance score 11n5 IQ points or more greater than
Verbal ability [χ#(1) l 0n09, p l n76]. This pattern is
often demonstrated in children who have emotional or
Hyperphagia and Appetite Disturbance behavioural difficulties (Wechsler, 1992).
Thirteen variables relating to appetite disturbance,
taken from the HSSDI and the school report, were Multivariate Analyses
entered into a Principal Components Analysis (PCA),
with varimax rotation. Three factors were identified : We aimed to analyse our data in such a way that we
‘‘ general hyperphagia ’’ explained 33n2 % of the variance, could allow for any intercorrelation between variables in
‘‘ school-specific hyperphagia ’’ explained a further a conservative manner, and therefore a Multivariate
15n7 %, and ‘‘ pica\polydipsia ’’ explained 9n7 % (Table Analysis of Variance (MANOVA) was employed. Seven
2). Scale scores were compiled for the three variables dependant variables were entered, with group as a factor
derived from this analysis on the basis of adding together (at two levels). Only the three summed hyperphagia
the untransformed values for all relevant questions, scores described in Table 2 [F (1,48) l 35n38, p n001]
rather than using the factor scores as variables in the and standardised height [F (1,48) l 31n41, p n001]
analyses. Cases’ summed scores were 5n52 (p2n57), 11n0 significantly discriminated the groups. Given that these
(p5n00) and 5n61 (p2n78) as compared to comparisons’ two variables were so discriminating, the same procedure
scores of 2n80 (p1n35), 2n79 (p3n24), and 3n07 (p1n47) was repeated with summed hyperphagia and standardised
for school hyperphagia, general hyperphagia, and pica\ height entered as covariates (along with age). The
polydipsia, respectively. This indicates that hyperphagia remaining five variables were entered as dependant
may be a specific reaction to stress and not a normative variables. None of these other variables was found to
response, even in children with a relatively low IQ. With explain a significant proportion of the remaining vari-
one exception (a child whose height was at the 75th ance : Full Scale IQ [F (1,48) l 0n01, p
n10], summed
centile), none of the stressed comparison children fitted motor milestones (smiling, sitting and walking unsup-
the HSS criteria for hyperphagia. ported) [F (1,48) l 0n49, p
n10], birthweight [F (1,48) l
0n81, p
n10], gestational age [F (1,48) l 0n91, p
n10],
and Body Mass Index [F (1,48) l 0n07, p
n10].
Cognitive Ability
A discrepancy between Verbal and Performance IQ Psychosocial Profile (Parent and Teacher Report)
scores of 11n5 points or more is reliable at p l n05
(Wechsler, 1992). Sixty per cent of the HSS group children There were no significant differences between groups
and 50 % of the comparison children showed a Verbal according to parent report. Teachers reported the stressed
974 J. GILMOUR and D. SKUSE

Table 3
Child Behavior Checklist (CBCL ; Parent Report) and Teacher Report Form (Teacher Report) Population T Scores :
Group Means
TRF (teacher report) CBCL (parent report)

Stressed Stressed
HSS comparisons HSS comparisons
(N l 24) (N l 22) (N l 17) (N l 18)

Population T score Mean (SD) Mean (SD) Mean (SD) Mean (SD)
Total problems 60n88 (9n60) 65n95 (9n19) 65n29 (11n30) 70n22 (10n40)
Internalising 56n21 (11n28) 61n59 (9n64) 60n29 (12n30) 63n89 (11n83)
Externalising 59n83 (9n92) 67n23 (9n38)* 61n00 (12n90) 70n94 (9n56)
Withdrawn 58n58 (10n49) 57n68 (6n31) 62n47 (11n45) 61n55 (9n91)
Somatic complaints 56n13 (9n11) 55n45 (6n80) 60n06 (8n68) 58n61 (8n43)
Anxious\depressed 57n79 (7n08) 64n36 (10n27)* 59n53 (11n39) 66n50 (14n70)
Social problems 62n17 (5n74) 67n18 (9n48)* 64n17 (9n19) 66n44 (13n10)
Thought problems 57n92 (9n38) 58n73 (10n84) 60n94 (7n81) 63n22 (9n37)
Attention problems 61n46 (9n77) 61n04 (8n38) 70n00 (11n71) 69n00 (13n10)
Delinquent behaviour 59n21 (7n06) 64n95 (8n23)* 62n76 (10n45) 68n94 (9n82)
Aggressive behaviour 60n83 (9n48) 67n95 (11n49)* 62n70 (11n26) 72n17 (11n19)
Sex problems n\a n\a 54n17 (7n64) 55n89 (13n02)
*p n05.

comparison children to have more externalising problems comparison children respectively [χ#(1) l 0n72, p l .40].
(F l 6n72, p n05), anxious\depressed features (F l Children with HSS had significantly greater sleep
6n47, p n05), social (F l 4n08, p n05), delinquent problems than the comparison group, according to care-
(F l6n49, p n05), and aggressive problems (F l 5n30, givers [t(48) l 2n14, p n05] ; roaming at night was
p n05). All other comparisons were insignificant. Means reported in 44 % of the HSS group and 20 % of the
are reported in Table 3. The Clinical T score gives an in- comparison group [χ#(1) l 3n30, p l n06].
dication of the probability that the child in question has a
behavioural or emotional disorder of clinical significance
and equates approximately to the 90th population centile. Discussion
According to parents, 29 % of HSS as compared to 50 % Stress and Psychosocial Environment
of stressed comparisons scored in the clinical range for
total problems. The other proportions scoring in the sub- Although HSS cases were recruited according to the
scales were as follows (HSS vs. comparisons) : external- presence of key behavioural and anthropometric charac-
ising problems (35 % vs. 77 %), internalising problems teristics, without regard to psychosocial adversity, 68 %
(29 % vs. 44 %), withdrawn (30 % vs. 28 %), somatic of the HSS group (17 cases) were subsequently found to
(24 % vs. 17 %), anxious\depressed (23 % vs. 44 %), social be known to social services, compared with 4 % in the
problems (47 % vs. 56 %), thought problems (41 % vs. general population (Skuse & Bentovim, 1994). We ac-
38 %), attention problems (71 % vs. 56 %), delinquency knowledge that the categories of abuse into which
(47 % vs. 67 %), aggression (24 % vs. 72 %), and sex children are put on ‘‘ at risk ’’ registers can be rather
problems (6 % vs. 6 %). According to teachers, 21 % of arbitrary. The central issue is usually that they have
HSS compared to 41 % of stressed comparisons scored in become a cause for concern at all. In no instance was the
the clinical range for total problems. The other subscale concern expressed by social services in an HSS child a
proportions rated in the clinical range for cases and consequence of our own assessment, nor were the criteria
comparisons were as follows (HSS vs. comparisons) : we used to confirm case status the criteria that were used
externalising problems (25 % vs. 54 %), internalising to justify their involvement with the family. Bearing these
problems (25 % vs. 32 %), withdrawn (12 % vs. 9 %), limitations in mind, there was no evidence that the type of
somatic (16 % vs. 9 %), anxious\depressed (21 % vs. abuse (confirmed or suspected) differentiated the HSS
50 %), social problems (17 % vs. 50 %), thought problems and comparison groups. This finding appears to exclude
(25 % vs. 23 %), attention problems (17 % vs. 28 %), the possibility that HSS is a specific reaction to a
delinquency (17 % vs. 50 %), and aggression (17 % vs. particular type of stress.
50 %). High EE is not synonymous with abuse but it is
Of those children who showed encopresis or enuresis, indicative of the degree of emotional stress experienced
52 % of the HSS group appeared to urinate in inap- by a key individual (Koenigsberg & Handley, 1986). High
propriate places deliberately, including over furniture or EE, in the sense of criticism or other negative communi-
belongings, compared with 16 % in the comparison group cations, is associated with family conflict and parental
[χ#(1) l 7n21, p
n01]. No differences were found in the mental health difficulties (Hibbs, Hamburger, Markus,
frequency of self-injurious behaviour, with at least one Kruesi, & Lenane, 1993). Care-givers from both groups
episode having occurred in 44 % and 56 % of HSS and were equally likely to show high EE in their reports of
 CHILDREN WITH HYPERPHAGIC SHORT STATURE : A CASE-COMPARISON STUDY 975

their relationship with the index child. Living with high described in association with severe child abuse (Felman
EE relatives may have a causal link, as opposed to simply & Nikitas, 1983). Previous reports have characterised
an association, with psychopathology (Vaughn, 1989). children with HSS as showing ‘‘ aggressive ’’ patterns of
We chose to use a measure of EE in order to test our enuresis and encopresis (e.g. Skuse, Albanese, et al.,
hypothesis about the psychological mechanisms by which 1996 ; Money, 1977). These behaviours may not be
the HSS phenotype arises. However, high levels of EE syndrome-specific, although it is plausible that in cases of
cannot be the only factor that precipitates the onset of the HSS where polydipsia is a major part of the clinical
condition, as some families in the HSS group were not picture enuresis could be consequentially related.
high EE and many frankly abusive families would not be Psychosocial profile (parent and teacher report).
rated as high EE. Although children with HSS are more emotionally
disturbed than the general population (20–30 % of
Identification of the HSS-Specific Features individual children within the HSS group fell into the
clinical range on the basis of independent reports from
Anthropometry. Previous reports have identified HSS parents and\or teachers), they have comparable profiles
on the basis of growth failure. We have shown that among according to parents and are significantly less disturbed
children living in conditions of high stress, which is than the stressed comparison group, according to teacher
usually but not invariably emotional in origin, short report. Previous studies have reported that depressive
stature can be associated with hyperphagia but it is symptoms (Blizzard, 1997 ; Ferholt et al., 1985),
unusual to find hyperphagia in the absence of short oppositional behaviour (Blizzard & Bulatovic, 1992), and
stature. Our design provided strong evidence that short social problems (Green et al., 1984) are characteristic of
stature is not a normative response to chronic stress. children with HSS. Our data suggest that such psycho-
There was no evidence from social services information social problems are not specifically associated with HSS.
or parental report to indicate that case children had Design, measures, and population sampled. There was
experienced stress at critical points in development or for probably a high refusal rate in families approached to
longer periods of time than comparisons, yet comparisons participate from the stressed comparison group, com-
were not growth retarded. However, the presence of a pared to the HSS group. Parents who were most
partial phenotype among the comparisons raises the distressed by their child’s behaviour may have been most
possibility that for some children hyperphagia may be likely to volunteer for our study. Accordingly, any
evoked without any impact on growth. Clearly here the selection bias would have resulted in an ascertainment by
crucial question is how long had the child been exposed to which the most overtly disturbed stressed children would
that stressor ? For there to be a marked impact on stature comprise the comparisons. If this was indeed the case, our
it would have to be long standing—at least 6 months and design was a robust test of the hypothesis that HSS is a
usually far longer. specific stress response, and not a normal reaction to
Neonatal and child health. Hypotonia has not pre- particularly adverse conditions.
viously been described in association in HSS. We assessed Further evidence in support of the external validity of
retrospectively the presence of this symptom on the basis the diagnosis comes from an epidemiological study of
of operationally defined criteria, from parental report. It children with short stature, in which screening for HSS
was reported in only four cases (16 %). Further clinical was one component. Voss, Walker, Lunt, Wilkin, and
assessment is required to follow up this finding, which Betts’ (1989) investigation supported our conclusion that
may be of significance in that many of the symptoms of HSS is a stress-responsive syndrome with specific
HSS (hyperphagia, low IQ, behavioural characteristics) behavioural features. Of the 148 short children who were
are reminiscent of Prader-Willi syndrome, in which identified in that survey, whose condition was not due to
infantile hypotonia and failure to thrive are very common organic disease, 4 probable cases (diagnosed on
(Holm, Butler, Hanchett, Greenberg, & Greenswag, behavioural criteria) were found. In each instance there
1992). were very high levels of intrafamilial stress.
Hyperphagia and appetite disturbance. Literature des-
cribing the eating behaviour of the general population of Implications
children living in severely stressful conditions is lacking.
Our findings provide evidence that hyperphagia is not The condition of HSS appears to be characterised by a
common among children living in adverse circumstances, combination of growth failure, developmental delay, and
even among those with low IQ. None of the stressed hyperphagia. Children with the condition manifest these
comparison children, according to parents, gorged and symptoms in response to high levels of stress, but not all
vomited or had stolen food from school, while 48 % had children exposed to stress develop the disorder. Evidently
gorged and vomited, and 52 % had stolen food from some aspects of the condition will appear relatively
school in the HSS group. That said, we acknowledge this acutely (the appetite disturbance, and the polydipsia
form of eating disorder was a prerequisite for the where present) whereas others will only manifest if the
diagnosis. Hyperphagia, together with short stature and stressors have been present for some time (the growth
hypotonia, is a clinical picture that fits with Prader-Willi disorder). The symptoms and the physical features of the
syndrome (Holm et al., 1992). condition are, however, not seen in the great majority of
Enuresis and encopresis. Care-givers in the HSS group very stressed children. Accordingly there seem to be two
reported that when their child showed enuresis or alternative explanations for the onset of the disorder.
encopresis, the majority of children were behaving in- Either affected children are in some way peculiarly
tentionally. Secondary enuresis and encopresis has been vulnerable to stress (because of a genetic predisposition
976 J. GILMOUR and D. SKUSE

or because of some environmental event, which could of alcohol, caffeine, socio-economic factors and psychological
course be prenatal), or there is something characteristic stress. British Medical Journal, 298, 795–801.
about the nature of the stressors to which affected Dittrich, B., Robinson, W. P., Knoblauch, H., Buiting, K.,
children have been exposed, stressors which are not Schmidit, K., Gillessen-Kaesbach, G., & Horsthemke, B.
(1992). Molecular diagnosis of the Prader-Willi and
experienced by stressed children who do not have the
Angelman’s syndromes by detection of parent-of-origin
condition. All the evidence we have accumulated so far specific DNA methylation in 15q11-13. Human Genetics, 90,
points to the probability that there is a genetic pre- 313–315.
disposition, which is inherited according to simple Eckenrode, J., Laird, M., & Doris, J. (1993). School per-
Mendelian principles (Skuse, Albanese, et al., 1996). formance and disciplinary problems among abused and
HSS is the only behavioural disorder of childhood in neglected children. Developmental Psychology, 29, 53–62.
which the clinical phenotype is associated with a path- Feehan, C. J. (1995). Encopresis secondary to sexual assault.
ognomonic physiological substrate (in respect of the Journal of the American Academy of Child and Adolescent
unique disorder of growth hormone dynamics). The Psychiatry, 34, 1404.
diagnosis meets conventional criteria for validity and Felman, Y. M., & Nikitas, J. A. (1983). Sexually transmitted
should be recognised within conventional classifications diseases and child sexual abuse. Part 1. New York State
Journal of Medicine, 83, 341–343.
of disease such as ICD-10 (World Health Organisation,
Ferholt, J. B., Rotnem, D. L., Genel, M., Leonard, M., Carey,
1988) and DSM-IV (American Psychiatric Association, M., & Hunter, D. E. K. (1985). A psychodynamic study of
1993). The specific clinical and physiological features of psychosomatic dwarfism : A syndrome of depression, per-
HSS, which include impaired growth, excessive appetite sonality disorder, and impaired growth. Journal of the
(thirst), and frequently a sleep disorder too, share similar American Academy of Child Psychiatry, 24, 49–57.
neurophysiological pathways ; they are all influenced by Fix, J. D. (1995). Hypothalamus. In E. A. Nieginski, D. R.
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implying that hypothalamic pathology underlies the (pp. 84–87). Baltimore, MD : Williams & Wilkins.
predisposition to the disorder, but this only becomes Flint, J. (1996). Annotation : Behavioural phenotypes : A
manifest in particular environmental conditions. window onto the biology of behaviour. Journal of Child
Psychology and Psychiatry, 37, 355–367.
Friedrich, W. N., Einbender, A. J., & Luecke, W. J. (1983).
Cognitive and behavioural characteristics of physically
Acknowledgements—The authors would like to thank Jim abused children. Journal of Consulting and Clinical Psy-
Stevenson, Linda Voss, Marcus Pembrey, and Joanne Newbolt. chology, 51, 313–314.
They are grateful to Jennifer Smith for her administrative Friedrich, W. N., & Wheeler, K. K. (1982). The abusing parent
support and to the families who took part in the study. Jane revisited : A decade of psychological research. Journal of
Gilmour was supported by the Wellcome Trust. Nervous and Mental Diseases, 170, 577–587.
Friess, E., Wiedemann, K., Steiger, A., & Holsboer, F. (1995).
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