Development of the Autistic
Catatonia Questionnaire – an
empirical investigation of
catatonia in people with ASD.
Jennifer Breen (RHUL),
Dougal Julian Hare (UoM) &
Penny Trayner (UoM)
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Revision of DSM- V means that sensory and motor features now central to the presentation of ASD
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DSM-V Catatonia describes a cluster of abnormalities in speech, movement and behaviour,
characterised by the presence of at least three of the following:
 Catalepsy
 Waxy flexibility
 Stupor
 Sgitation
 Mutism
 Negativism
 Posturing
 Mannerisms
 Stereotypies
 Grimacing
 Echolalia
 Echopraxia.
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Increasing recognition that catatonia presents as a co-morbid syndrome in
ASD in 12–18 % of young people with ASD
Insidious onset between 10 to 19 years – initial presentation of isolated
symptoms with progression to full catatonic syndrome (Wing & Shah 2000).
Diagnosis of catatonia in ASD complicated by the overlap in symptoms e.g.
mutism, echolalia, stereotyped/repetitive behaviours
Are catatonic symptoms in ASD are akin to catatonic states associated with
other conditions ?
“Autistic catatonia” not a co-morbid condition but extreme manifestation of
ASD in a subgroup of this population (Hare and Malone 2004).
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Specific criteria to define what has been termed ‘autistic catatonia’ or
‘catatonia-like deterioration ’:
 increased slowness affecting movement and verbal responses,
 difficulty in initiating and completing actions, I
 increased reliance on physical or verbal prompting from others,
 increased passivity
 apparent lack of motivation
Wing & Shah 2000; Hare & Malone 2004: Wing 2005
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Given phenomenological overlap, key indicators of autistic catatonia are the
emergence of new symptoms / change in the pattern of pre-existing
symptoms.
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Autistic catatonia is challenging to treat and symptoms may persist over
many years.
Published treatment guidelines propose that psychological approaches, high
doses of lorazepam and bilateral ECT are the current treatments of choice
(Dhossche et al. 2006; Fink et al. 2006).
Recent systematic review (DeJong, Hare & Bunton 2014) of 22 papers
(N=28 cases) indicated that the evidence for the efficacy or effectiveness of
such interventions is extremely limited given the low quality of most reports
Interventions were frequently only partially described and outcome
reporting relied heavily on clinician impressions, rather than objective
measures of change.
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ECT and high dose lorazepam may have some acute partial effect
on catatonic symptoms in ASD.
Long-term maintenance of any improvement seemed reliant on
either maintenance ECT or continued medication.
Behavioural treatments show some positive outcomes in relation to
symptom reduction, although no case had complete resolution of
symptoms.
Behavioural treatments are comprised of several components and
unclear which elements are needed for change.
Following all types of treatment, patients may continue to display
catatonic symptoms and are unlikely to return to baseline levels of
function. There is some indication that early intervention may be
beneficial.
Developing ASD-specific measures of catatonic
symptoms
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Although accurate measurement of presenting symptoms is crucial for
evidence-based intervention and research, limited options for clinical
assessment tools for IDD populations.
Many extant measures are un-validated
Practical issues associated with importing catatonia rating scales to IDD
(Carroll et al., 2008; Heckers et al., 2010):
 Affective alterations that are difficult to accurately identify via third
party measures
 Altered speech or tone of voice that may be abnormal/ absent in IDD
 Non-typical movement in neurotypical populations = typical noncatatonic presentation in ASD (e.g. repetitive stereotyped movements).
Present study aimed to empirically investigate the phenomenon of
‘autistic catatonia’ in children and adolescents with ASD to:
 Develop a valid and reliable clinical measure which can be used
for assessment, research and to monitor the progression of the
condition and the effectiveness of interventions.
 Investigate the prevalence of catatonic symptoms in young
people with ASD.
 Develop better theoretical understanding of autistic catatonia.
Present study – development of the ACQ
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34-item Autistic Catatonia Questionnaire (ACQ) was developed from extant
reports of autistic catatonia:
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6 core features of autistic catatonia
28 supplementary items covering repetitive, motoric and sensory functioning.
ACQ items scored for presence, frequency and severity.
A main caregiver or parent completed the ACQ together the Repetitive
Behaviour Questionnaire (RBQ) (Moss, Oliver, Arron, Burbidge & Berg,
2009) and the Carer Supplement to the Glasgow Depression Scale for
People with Learning Disability (GDS-CS) (Cuthill, Espie & Cooper, 2003)
Parents and carers of individuals with ASD were recruited via UK charities
and support organisations
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ACQ developed via an iterative process:
List of reported symptoms extracted from a literature review
and the following excluded:
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‘Singleton’ symptom s e.g. finger tapping, diaphoresis)
Symptom associated with ASD (e.g. echolalia) and that did
show change to the individual’s previous presentation
Any symptom considered vague or speculative (e.g. auditory
hallucinations, anxiety, visual hallucinations)
Any item on the GDS-CS, RBQ or Sensory Behaviour
Scale (Harrison & Hare 2004)
N=8 reported symptoms thus excluded from the measure
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The conceptual structure of extant non-ASD measures of
catatonia was replicated as appropriate (Northoff et al. 1999).
ACQ comprises 34 items categorised into motor symptoms
(n=15), affective alterations (n=5) and behavioural
alterations (n=14)
Each ASQ symptom is clearly defined with most having
specific examples and descriptors (Carrol et al. 2008).
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All participants indicated that they were reporting symptoms
presenting in a young person with an existing diagnosis of ASD.
41.6% reported additional diagnoses in addition to ASD - 77
different co-morbid diagnoses (excluding autistic catatonia).
N=20 (14♂/6 ♀) had an existing diagnosis of autistic catatonia,
nine of whom had additional diagnostic labels
Not possible to complete statistical analysis for co-morbid
conditions other than autistic catatonia due to small numbers of
each additional diagnosis .
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High level of catatonic symptomatology
present in current sample.
All participants displayed at least four
of the broad range of symptoms
Eighty-four participants (85%) had
displayed at least one of the core
symptoms associated with autistic
catatonia (ACQ items 1-6) either
currently or in the past (mean=2.38,
sd=1.85).
Potential bimodal distribution that may
indicate that autistic catatonia becomes
a syndrome when core symptoms ≥ 3 ?
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All six of the core symptoms
commonly reported in the
sample
Difficulty initiating movement
= least frequently reported
symptom (n=18)
Requiring physical and/or
verbal prompts = most
frequently reported (n=60).
70
60
50
frequency
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40
30
20
10
0
core symptom
The ACQ as a clinical measure
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Two options for exploring the data set:
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Existing autistic catatonia diagnosis
Presentation of autistic catatonia symptoms
Problem of under-diagnosis of catatonia in ASD
(Wing & Shah, 2000) means using extant diagnosis
may exacerbate between-group differences
An empirical approach to between-group analysis using
a clinical cut-off should provide a more accurate and
robust measure of autistic catatonia in the current
sample.
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Core Autistic Catatonia Score - total number of the six core
symptoms with total score summed to obtain the Core
Autistic Catatonia Score [0-6]
Core Severity Score – sum total of the severity of the
currently presenting core symptoms [0-24]
Core Frequency Score – sum total of the frequency of the
currently presenting core symptoms [0-24]
Core Autistic Catatonia Score
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Mean Core Autistic Catatonia Score for the
sample =7.48 (sd=5.70; 0-24)
N=15 had a Core Autistic Catatonia Score
= 0 (15.2%).
No significant difference in Core Autistic
Catatonia Score between those with
(x=9.35;sd=6.00) and those without
(x=7.01, sd=5.57) extant autistic catatonia
diagnosis [t(97)=1.65, p=0.10]
Individuals with three or more core
symptoms had significantly higher Core
Autistic Catatonia Scores (x=12.04;
sd=4.24) than those with less than three
core symptoms (x=3.2; sd=2.86); [t(97)=12.23, p<0.01]
Core Severity Score
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Mean Core Severity Score = 5.23
(sd=4.81; 0-23)
N=22 had a Core Severity Score = 0
(22.2%).
No sig. diff. in Core Severity Score
between those with (x=6.95; sd=4.70)
and those without (x=4.80;sd=4.77) an
extant diagnosis of autistic catatonia [
t(97)=1.81, p=0.07]
Individuals currently displaying three or
more core symptoms had significantly
higher Core Severity Score
(x=8.94;sd=4.09) than those displaying
less than three (x=1.75; sd=2.01)
[t(97)=-11.20, p<0.01]
Core Frequency Score
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Mean Core Frequency Score = 4.73
(sd=3.87; 0-15).
N=21 had a Core Frequency Score = 0
(21.2%)
Individuals with an extant diagnosis of
autistic catatonia had significantly
higher Core Frequency Scores (x=6.45,
sd=3.90) than those without (x=4.34,
sd=3.72) [ t(97)=2.24, p<0.05)]
Individuals currently displaying three or
more core symptoms had significantly
higher Core Frequency Scores (x=7.85;
sd=2.78) than those displaying less than
three core symptoms (x=1.86;
sd=1.93)[t(97)=-12.51, p<0.01]
Total ACQ score
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Analysis including all 34 ACQ items on the ACQ measure (0136) yielded a mean Total ACQ Scores of 54.28 (sd=21.23; 5110)
No statistically significant difference between those with
(x=59.33, sd=19.77) and those without (x=52.96, sd=21.54) an
extant diagnosis of autistic catatonia [t(85)=1.14, p=0.259]
Individuals currently displaying three or more core symptoms
had significantly higher Total ACQ Score (x=61.04; sd=21.07)
than those displaying less than three core symptoms (x=44.33;
sd=22.02)[t(82)=-3.53, p<0.01]
Scoring recommendation for the ACQ
measure
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Recommended primary scoring strategy for the ACQ as a
diagnostic screening tool is Core Autistic Catatonia Score.
Receiver Operating Curve (ROC) analysis was conducted to
determine the ability of Core Autistic Catatonia Score ≥ 3 to
identify individuals with extant diagnoses
Area under curve = 62.4% with extant diagnosis sub-group
identified at significantly higher than chance probability rates
(p<0.05).
An alternative clinical cut-off of between 7-8 is also possible,
based on a analysis of the co-ordinates of the ROC curve
(sensitivity and specificity values).
Age
 No significant different in age between those with (x=16.20,sd=4.42) and
those without (x=15.55,sd=3.89) an extant diagnosis of autistic catatonia
[t(96)=-0.647, p=0.519], nor between those displaying three or more
(x=16.32, sd=4.57) and those displaying less than three core symptoms
(x=15.10; sd=3.29)[t(96)=-1.53, p=0.130].
 Caveat of skewed distribution of age within the study sample.
Gender
 Extant diagnosis of autistic catatonia and gender were not associated [χ²(1,
n=99)=0.45, p=0.501] and there were no significant differences in
 Independent samples t-tests reveal that there were no significant differences
in Core Autistic Catatonia Score, Core Severity Score or Core Frequency
Score between males and females in the sample
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Core ASQ symptoms and existing diagnoses
of autistic catatonia
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The presentation of the six core
ASQ symptoms in people with
extant diagnoses of autistic
catatonia (N=20) was
examined
No clear pattern could be
identified in the current sample
16
14
12
frequency
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10
8
6
4
2
0
core symptom
GDS-LD scores
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There was no significant difference in
GDS-CS between those with (x=13.33,
sd=5.02) and without (x=12.77,
sd=4.17) extant diagnosis of autistic
catatonia [t(85)=-0.491, p=0.625].
Individuals displaying three or more
core symptoms had a significantly
higher GDS-CS Score (x=12.74;
sd=5.07) than those displaying less than
three (x=8.43; sd=6.09)[t(85)=-3.586,
p=0.01]
Linear regression analysis indicated that
Core Autistic Catatonia Score
accounted for 15% of GDS-CS Score
[r2=0.15, p<0.001]
RBQ scores
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Mean RBQ score for the sample was
32.82 (sd=16.73; 1-68).
Individuals with extant diagnoses of
autistic catatonia had higher RBQ total
scores (x=40.56, sd=15.73) than those
without (x=30.80, sd=16.49)
[t(85)=2.256, p<0.05]
Individuals presenting with three or
more core symptoms had RBQ total
scores (x=39.58, sd=13.74) than those
with less than three (x=26.20,
sd=16.86)[t(85)=-4.05, p<0.01]
Linear regression analysis indicated that
Core Autistic Catatonia Score
accounted for 12% of the variation in
RBQ Total Score [r2=0.12, p<0.001]
Future directions
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ACQ appears to be a valid measure for both research and clinical
assessment of autistic catatonia
Further work required to refine psychometric properties of the ACQ
Ongoing study at Manchester and Birmingham Universities
examining catatonic symptomatology in Tuberous Sclerosis, Fragile
X, Retts and Cornelia de Lange syndromes