430

STURMEY

maladaptive behaviour (Aman & Singh, 1985) rather than psychiatric disorder parse. The meaning of this finding is, however, unclear: it may reflect either a true correlation between these constructs, or artifacts, such as the response styles of informants and/or halo

effects operating when two measures are administered concurrently with one informant. Visual inspection of the data did reveal that raters had consistent styles of responding to both measures. Aman et al(1986) question the appropriateness of the current scoring method of the PIMRA, given the poor correspondence of the factor analyses to the eight DSM-III-derived

scales. This paper found that

AND LEY

more empirical approach to measuring psycho pathology in mentally handicapped people. References AMAN,

M.

0.

& SINCH,

N. N. (1985)

The Aberrant

Behaviour

Checklist.East Aurora, New York: Slossen.

—¿ ,

—¿ ,

STEw@aT,

A.

W.,

ci

al

(1985)

The

Aberrant

Behaviour

Checklist: a behaviour rating scale for the assessment of treatment

485-491. —¿ ,

effects. American

WATSON,

J.

E.,

SmeoH,

Journal

N.

N.,

ci

A.

K.,

of Mental Deficiency.

al(1986)

Psychometric

and

demographiccharacteristicsof the PsychopathologyInstrument for MentallyRetardedAdults. PsychopharmacologyBulletin, 22, 1072—1076.

such scales had internal consistencies which were of

—¿ ,

problematic as they currently stand. Nevertheless, these broader-band measures of psychiatric distur bance would appear to be robust since they were

FRAsER, W. I., LEUDAR,I., G@y, J., ci al (1986) Psychiatric

similar magnitude to the original scales and were thus

replicated by Aman et al (1986). However, the items

on the PIMRA which correspond to these three dimensions (affective, somatoform, and psychosis) appear to measure these constructs rather coarsely. Indeed, the issue of correspondence between check list scores and psychiatric diagnosis has become a major issue within the field of childhood psycho pathology (e.g. Kline, 1988). Future measures of psychopathology

in this population could extend and

refine measures of these constructs as a basis of a

91,

RICHMOND,

0.,

STEWART,

ci

al

(1987)

The

Aberrant

Behaviour Checklist: factorial validity and the effect of demographic/medical variables in American and New Zealand facilities. American Journal of Menial Deficiency, 91, 570—578. and

behaviour disturbance in mental handicap. Journal of Menial Deficiency Research, 30. 49-57.

KImIE, R. P. (1988) Methodological

considerations

in the evaluation

of theconvergenceof psychiatricdiagnosesandparent-informant

checklists. Journal of Abnormal Child Psychology, 16, 289-298. M@isoN, 1. L., SENATORE, V. & KAZDIN,A. E. (1984) Psychometric properties of the Psychopathology Instrument for Mentally Retarded Adults.App&d Researchin Mental Retwdaiion,5,81-89. NEWTON, 3. T. & STust@my, P. (1988) The Aberrant

Behaviour

Checklist:a Britishreplicationand extensionof itspsychometric

properties. Journal of Menial Deficiency Research, 32, 87-92.

SENATORE,V., M@@oN, 3. L. & KAZDIN,A. E. (1985) An inventory

to assess psychopathology in the mentally retarded. American Journal of Menial Deficiency, 89, 459—466.

5P. Sturmey, BSc, PhD, MClinPsychol,School of Psychology, University of Birmingham, P0 Box 363, Birmingham BiS 277; T. Ley, BSc, Department of Psychology, Plymouth Health Authority Correspondence

British Journal of Psychiatry (1990), 156, 430—433

Tourette-Like Disorder in Asperger's Syndrome CARL S. LITTLEJOHNS, DAVID J. CLARKE and JOHN A. CORBETT An eight-year-oldboy with Asperger'ssyndromewas given haloperidolto control agitation and aggressiveoutbursts.Withdrawal of the drugafter two yearswas followed by Tourette like symptoms. Subsequently neither haloperidol nor a second antipsychotic drug altered the

core features of Asperger's syndrome, despite suppressing the movement disorder. His first exposure to neuroleptics was in utero. Pervasive developmental disorders (PDDs) are characterised by qualitative impairment in the

development of reciprocal social interaction, verbal and non-verbal communication skills, and imaginative

TOURETTE-LIKE

DISORDER

IN ASPERGER'S

431

SYNDROME

activity. Infantile autism (IA) is the only named sub class recognised in DSM-III-R (American Psychiatric Association, 1987).

night by three years six months. His adoptive parents described him as being reserved and ‘¿ loner'. a He ‘¿ didn't play' but rather would show excessive preoccupation with

under section 299.80('PDD

people, but had little interest in them. He would adopt odd

Asperger's syndrome in DSM-III—Ris classified

Not Otherwise Specified' ),

but is now more widely regarded as a second subclass

of PDD (Wing, 1986; Kerbeshian

& Burd, 1986a).

Patients have autistic features such as resistance to

change,

inappropriate

emotional

expression,

and

very poor use and comprehension of non-verbal language. However, their speech is grammatically

correct, though pedantic, and they mostly are not socially aloof. Interests are restricted and repetitive, and motor activity is stereotyped and clumsy.

Tourette's syndrome (TS) is a disorder consisting of multiple motor and vocal tics (sometimes including coprolalia) which can be suppressed voluntarily for short periods. It usually occurs between the ages of 2 and 15, is commoner in boys, and follows a fluctuating course. Tourette-like symptoms have been reported as a complication of

inanimate

household

postures

objects.

with his arms,

He did not

and in noisy

actively

or crowded

avoid

environ

ments would bang his head, howl, and rock himself. He developed secondary nocturnal

enuresis on starting school

at the age of five years, and became aggressive and

destructive, despite a behavioural an educational psychologist.

programme

devised by

Bythe age of sevenhis conversation, whichwasusually

very polite and grammatically correct, reflected an excessive preoccupation with a narrow range of inanimate objects

includingvacuumcleaners,musicalboxes,and insectivorous

plants, and he would talk of little else, often making cardboard models of them. This, together with increasingly frequent outbursts of temper, led to referral to a child psychiatrist, where imipramine(25 mg nocte) and bell and

pad training failed to resolve the nocturnal enuresis.

At the age of eight years the agitation and aggressive

behaviourledto the prescriptionof haloperidol(1mgt.d.s., subsequently

increased to 2.5 mg t.d.s.),

which had some

long-term neuroleptic therapy (Klawans et al, 1982), and can be confused with movements due to tardive dyskinesia (Mueller & Aminoff, 1982). The coexistence of a PDD with TS in a patient has been reported before (e.g. Kerbeshian & Burd,

calming effect. At the age of ten he was admitted to a

suggested that TS in patients with PDD may be a marker for improved developmental outcome and

A spoke little, but was very polite. He skilfully used a pair of scissors and some paper to make a model of a venus fly-trap, which he stuck to the end of a ruler. His face was

We describe a young man with AS, now aged 15, who developed a Tourette-like movement disorder at the age of 10, when treatment with haloperidol

giggling sounds, but no firm features suggestive of a

of language were not improved by neuroleptic

(EEG) were normal.

1986a,b). Language disorder and social dysfunction in atypical PDD is reported as responding favourably to drug treatment when coincident with TS (Fisher et al, 1986). The same group of workers has response to haloperidol

(Burd et a!, 1987).

residential

special school,

The preoccupations ritualistically

with ‘¿ lack of concentration'.

persisted.

He touched

objects

and insisted that things were not moved. Any

attempt to change the order of objects in his environment

provoked a tantrum.

At 11 years he developed marked writhing movements of his limbs and trunk. He fidgeted constantly and ran round in circles, sometimes falling. He was admitted to an in-patient child psychiatric unit. During the initial interview

inexpressive

and

he occasionally

made

inappropriate

psychotic illness were observed. He was noted to be restless

was stopped. His social functioning and pedantic use

and overactive, with jerky, non-repetitive limb movements. Physical examination and an electroencephalogram

medication.

haloperidol was slowly withdrawn. Over the following

After a period

of observation

the

Case report

weeks a gradually progressive dystonia developed, which affected his whole body and interfered with his ability to walk. He also developed sudden facial grimacing and

A was born at term, with a birth weight of 3.18 kg. He was

grunting and barking noises, and was heard to utter obscene

of fluphenazine decanoate (dose unknown) throughout

of TS was made, and treatment with haloperidol resumed.

during his second day of life, and at ten days developed an E. coil gastroenteritis. Shortly afterwards his mother was admitted to psychiatric hospital and he was taken into

poor level of social functioning were not altered. At 13 years his reading age was eight years six months

the third child of a mother who had receivedinjections

repetitive tic-like neck movements, made involuntary words,mostlywhenhe felthe wasunobserved.A diagnosis

pregnancy for schizophrenia. A became hypoglycaemic The tics and the other abnormal movementsresolved,but A's preoccupations,inappropriatemonologues,and generally

care. Fullrecordsare not availablebut hismotherhad been convicted of infanticide after poisoning her first child, and it seems that A was returned to her and taken back into

and his mathematical ability was at an eight-year level. During

his 14th year he became

markedly

tearful

and

withdrawn. A diagnosis was made of Asperger's syndrome

care on severaloccasionsbefore being adopted at the age

and a dysphoricreactionto haloperidol(2.5mg t.d.s.). His

constructing simple sentences at two years. He was dry at

lifted withinweeksof the change,but he continuedto talk about vacuum cleaners and venus fly-traps. His socially

of nine months. A walked at II months, spoke at 12 months, and was

medication was changed to pimozide (2mg daily). His mood

432

LIULEJOHNS ET AL

unacceptablecommentsincludedinvitingothers to ‘¿ tickle' pimozide was substituted. We would point out that his genitalia without appreciating the reaction this produced, dysphoric reactions have been reported previously but he learned to reduce this behaviour after counselling (e.g. Bruun, 1982) and should be looked for. by school staff. The assessment and training of our patient was The Wechsler Intelligence Scale for Children (Revised)

when he was 14years showed a corrected verbal score of

62, a performance score of 72, and a full-scale IQ of 65.

A's teachersand parents have noticeda reduction in his

use of speech over the past 12 months, the content of which

continues to reflect his unusual preoccupations. He has succeeded in forming a good relationship with a key worker, but his apparent understanding of non-verbal cues such as facial expression is still poor.

by teachers skilled in the education of pupils with language delays and disorders who would have been especially sensitive to improvements in social

and verbal functioning of even a minor degree.

Reintroduction of haloperidol did suppress the Tourette-like symptoms, but did not improve social

or verbal functioning. Although this is in contrast to the previously mentioned studies of Fisher et a! (1986) and Burd et al (1987), it is compatible with

Discussion In children, haloperidol is effectiveand well tolerated (Serrano, 1981). Although some abnormal move ments were noted in our patient before the drug was stopped, the repetitive, tic-like movements began

after the drug was withdrawn. Tic-like movements settling after six to nine months have been noted before when neuroleptics are discontinued (Singer, 1981).

Stahl (1980) described a 28-year-old man with IA

the more recent observations of Kano et al (1988), and may imply that patients presenting with both a PDD and TS comprise a heterogeneous group, with

more research needed on their further subdivision. References AMERICAN PSYcHIATRIC ASSOCIAT)ON (1987) IY@agnostic and &atLstical

Manual ofMental Dsorders (3rd edn, revised) (DSM-III—R). Washington, DC: APA.

BRUUN. R. D. (1982) Dysphoric

first appeared when thioridazine, which had been

given for 13 years, was withdrawn.

Stahl noted that

his case differed in some ways from classic TS. The age of onset was much later than usual, the tics could not be voluntarily suppressed and they fluctuated relatively little over time. He proposed that the term ‘¿ tardive' Tourette syndrome be used when Tourette like symptoms follow withdrawal of neuroleptics. Neuroleptic-related Tourette-like symptoms have been reported elsewhere in people with disorders other than PDD (e.g. Klawans et al, 1978, 1982). Gualtieri & Guimond (1981) refer to a neuroleptic ‘¿ withdrawal dyskinesia' or ‘¿ withdrawal emergent symptom' which may be misdiagnosed as tardive

of neuroleptics

to diagnose

this,

which would suggest a similar period might be useful when a tic-like disorder appears on discontinuing a neuroleptic. It is interesting to speculate on the relevance of

the fact that our patient's first exposure to neuro

leptics was in utero. A previous report of a patient

with TS and a PDD (in this case IA) also described the mother as suffering from schizophrenia, but prescribing during pregnancy was not recorded (Realmuto & Main, 1982). The dysphoric state which occurred on resuming

treatment

with

haloperidol

resolved

rapidly

when

with

patients with autism and other pervasive developmental disorders?

Journal of the American Academy of Child and Adolescent Psychiatry, 26, 162-165.

FISHER, W.,

KERBESHIAN, 3. &

BURD,

L.

(1986)

A

treatable

language disorder: pharmacological treatment of pervasive developmental disorder. Journal ofE@velopnenta! and BehaviOral Pediatrics, 7, 73—76.

GUALTIERI,C. T. & GUIMOND,M. (1981) Tardive dyskinesia

and

the behavioural consequences of chronic neuroleptic treatment. Developmental Medicine and Child Neurology, 23, 255-258.

KAN0, Y., OIrrA, M., NAGAI, Y., ci a! (1988) Tourette's

disorder

coupled with infantile autism: a prospective study of two boys. Japanese Journal of Psychiatry and Neurology,42, 49-57.

KERBESHIAN, 3. & BURD, L. (1986a)

A second

visually

impaired,

mentally retarded male with pervasive developmental disorder, Tourette disorder and Gamer's syndrome: diagnostic classification and treatment. International Journal of Psychiatry in Medicine,

by symptoms of nausea, vomiting, and weight loss.

administration

associated

Buiw, L., Fisiwit, W. W., KERBESHIAN,3., ci a! (1987) Is development of Tourette disorder a marker for improvement in

dyskinesia, althoughsuchcasesareoftenaccompanied They proposed a 12-week period of observation after

phenomena

haloperidol treatment of Tourette syndrome. Advances in Neurology, 35, 433—436.

and a two-year history of motor and vocal tics which

16, 67—75.

—¿

&

—¿

(l986b)

Asperger's

syndrome

and

Tourette

syndrome:

thecaseof the pinballwizard.BritishJournalof Psychiatry,148, 731—736.

KIAwANs,H. L., F@tK,D. K., NAUSLEDA, P. A., ci a! (1978) Gilles de Ia Tourette syndrome after long-term chiorpromazine

Neurology, 28, 1064-1065. —¿ ‘

NAUSIEDA,

P.

A.,

GCIETZ,

C.

C.,

et

a!

(1982)

therapy.

Tourette-like

symptoms following chronic neuroleptic therapy. Advances in

Neurology,35, 415—418.

MUELLER, 3. & AMINOFF, M. (1982) Toureue-like

syndrome

after

longtermneurolepticdrugtherapy.BritishJournalof Psychiatry, 141, 191—193.

Rp.,u.t@uro, G. M. & Mami,B. (1982)Coincidenceof Tourette'sdis

order and infantile autism. Journal of Autism and Developmental DIsorders, 12, 367—372.

SERRANO,A. C. (1981) Haloperidol

- its use in children.

of Clinical Psychiatry, 42, 154—156.

Journal

TOURETFE-LIKE SINGER, W. D. (1981) Transient

DISORDER

Gilles de Ia burette

syndrome

after chronic neuroleptic withdrawal. Developmental Medicine and Child Neurology, 23, 518—521.

STAHL, S. M. (1980) Tardive burette's

*Carl S. Littlejohns,

5SS, formerly

syndrome

in an autistic

IN ASPERGER'S

433

SYNDROME

patient after long-termneurolepticadministration.American

Journal of Psychiatry, 137, 1267-1269. Wu@o,L. (1986) Clarification on Asperger's syndrome. Journal of Autism and Developmental Disorders, 16, 513-515.

MBChB, MRCPsych, Senior Registrar,

North

Wales Hospital,

Denbigh,

Clwyd LLJ6

registrar, Monyhull Hospital, Kings Norton, Birmingham; David J. Clarke, MBChB,

MRCPsych, Clinical Lecturer,

University

of Birmingham,

Department

of Psychiatry,

Queen Elizabeth

Hospital, Edgbaston, BirminghamB15 2TH; John A. Corbett, MBBS,FRCPsych, Professor of Mental Handicap, University of Birmingham, Department of Psychiatry, Leacast!e Hospita!, Wo!verley, Kidderminster DYJO 3PP Correspondence

British Journal of Psychiatry (1990), 156, 433—435

Temporary Remission of Tardive Dystonia Following Electroconvulsive Therapy ADITYANJEE, S. K. JAYASWAL, T. M. CHAN and M. SUBRAMANIAM An improvementin tardive dystonia in a patient who had receivedECT for a schizophrenic psychosis is reported. The improvement suggests that the pathophysiology of tardive dystonia may involve neurotransmitter receptor changes similar to those seen in schizophrenia.

Although the first series of six cases of late-onset persistent dystonia associated with neuroleptic treatment was described as early as 1967 (Harenko,

1967), the term ‘¿ tardive dystonia' was coined by Keegan & Rajput (1973) to designate drug-induced torticollis of a persistent nature. There were very few case reports until Burke et a! (1982) described the clinical characteristics of tardive dystonia in 42

patients and reviewed 15 cases reported in the literature.

Tardive

dystonia

differs

from

choreic

tardive dyskinesia in its clinical phenomenology, epidemiology, and clinical pharmacology (Burke,

1982). Similarly, Gimenez-Roldan et a! (1985) found

important differences and tardive dyskinesia

between tardive dystonia in terms of risk factors and

prognosis. About 1.5-2.0% of patients treated with

neuroleptic

drugs develop this iatrogenic

condition

(Yassa eta!, 1986; Friedman eta!, 1987). Risk factors for tardive dystonia appear to be youth, male sex, mental retardation, and convulsive therapy (Burke

et al, 1982; Gimenez-Roldan 1986; Friedman

eta!,

eta!, 1985; Yassa eta!,

1987). A few isolated

case reports

have also appeared in the literature commenting on its

phenomenology,

treatment, and differential diagnosis

(e.g. Kwentusetal, 1984;Ananth eta!, 1988;Falketal,

1988; Lal et a!, 1988; Adityanjee et a!, 1989). Most

often it has been misdiagnosed as conversion hysteria (Kwentus et a!, 1984; Yassa et a!, 1986).

We describe a case of tardive dystonia and its short-term remission with electroconvulsive therapy (ECT). This is the second case report in the literature in which tardive

dystonia

has been successfully

treated with ECT (Kwentus et a!, 1984). Case report

A 30-year-oldman presentedin April 1987witha two-week historyof sustainedcontractionof the sternocleidomastoid muscles and anterior

fiexion of the neck.

At the age of 15 he had manifested insidiously, for

the first time,

signs and symptoms

of mental

illness