Thalamo-frontal psychosis.

I McGilchrist, L H Goldstein, D Jadresic and P Fenwick
BJP 1993, 163:113-115.
Access the most recent version at DOI: 10.1192/bjp.163.1.113

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Thalamo-frontal
lAIN McGILCHRIST,

Psychosis

LAURA H. GOLDSTEIN,

DANITZA

JADRESIC

and PETER FENWICK
A 43-year-oldmanpresentedwith an 18-monthhistory
of acute-onset cyclical behaviouralchange affecting
mood, appetite, sleep, and energy levels. This had
followed an initial episode of transient drowsiness which

lasted 24 hours. On examination, there was some
evidenceof visualmemoryand frontal lobe deficits. A
brainCT scanshowed bilateralthalamicinfarctsand a
brain SPECTscan showed bilateral hypoperfusion of the
frontal lobes. To our knowledge, this is the first reported
case of thalamic infarction associated with acute-onset

cyclical affective psychosiswith clinical and neuro
physiological features of frontal lobe syndrome. The
case also highlights the possible role of thalamo
frontal circuitsin the pathogenesisof the Kleine-Levin
syndrome.
British Journal ofPsychiatry (1993), 163, 113—115
Atypical psychiatric

syndromes

following

infarction

thalamic

have been reported
(Bogousslavsky

et al,

1988). There are often no neurological disturbances
of note, so that the infarcts,

which are uncommon,

may be overlooked.
There is evidence that damage
to the thalamus,
or to a sufficient
part of the
thalamocortical
projection,
plays a key role in the
occurrence
of ipsilateral
cortical hypometabolism,
and this in turn is probably
responsible
for the
psychiatric
disturbances
(Pappata et a!, 1990). We
report a case of bilateral thalamic
infarction
which

we believe

illustrates

both

diagnosis
and the possible
in such cases.

the complexities

underlying

by the council to close one of his shops. His wife, however,
described him as a happy man and commented that he had
been cheerful and lucid until the collapse, whereas he now
seemed apathetic, as well as fatuous, giggling, and treating
questions as a joke. He had mild left-sided pyramidal signs
and was referred to the neurologists. After four days he
was discharged, as his disorder was thought to be ‘¿non
organic' . There was a further brief readmission one month
later because of drowsiness, apathy, and memory impair
ment. A computerised tomographic (CT) scan at this point
showed a small infarct in the right thalamus.

Over the subsequent six months he was ‘¿like
a zombie',
sleeping and eating excessively (especially his children's
crisps and sweets), and behaving inappropriately (e.g. going
out in his slippers to get fish and chips). On average, every
three weeks, he would suddenly become alert and would
play with the children, begin hatching plans, sleep little
during the night, and the next day he would be excitable
and elated. After about 36 hours he would suddenly relapse
to his former apathetic condition. He was thought to be
malingering. A consultant psychiatrist to whom he was
eventually
referred
treated
him with lofepramine,
imipramine, and 12 sessions of electroconvulsive
therapy

(ECT) with little or no improvement.
There was a family history of cardiovascular disease and
depression on the father's side. There was no personal,
medical, or psychiatric history of note. He rarely drank
alcohol and did not smoke.

On admission he was overweightand normotensive. His
manner was histrionic and tearful, but he sometimes joked
or smiled somewhat incongruously. He was subjectively

of

mechanisms

Case report
A 43-year-old right-handed,marriedman who ran his own
business was referred to us by a consultant neurologist for
assessment of his mental state. He appeared to have
undergone a complete change of personality and it was not
clear whether this was a primary psychiatric disorder, or
whether there was an organic or, alternatively, a purely
behavioural,
basis for it.

depressed

and had ideas of guilt about the effect that his

condition

had had on work and family

life. He had poor

self-esteem but no feelings of hopelessness. There was no
diurnal mood variation; other biological symptoms have
been described above. He stated that he was suffering from
“¿psychologicaldepression― and complained that he was
like “¿an
engine that won't tick over―.

Routine investigations, including full blood count,
erythrocyte sedimentation
rate, full biochemical profile,
thyroid function tests, serum Bl2, folate, and syphilis
serology, were normal or negative. A pituitary endocrine

One and a half years previously, apparently quite well,

screen was normal but fasting cholesterol was slightly raised,
and he was placed on a low-cholesterol diet. Autoimmune

wife, noticing that he slumped, found him unrousable, with
some mild drooping of the left side ofthe mouth. In hospital
he was drowsy but resisted some parts of the examination.

screening revealed raised anti-nuclear factor and DNA
binding but when these were repeated, they were normal;
cardiolipin levels were also normal. An echocardiogram
showed
a small pericardial
effusion
which
was
asymptomatic.
Routine and activated electroencephalo

On review by a psychiatrist the next morning, he was alert

grams (EEGs) were normal, as were brain-stem evoked

but complained of having been depressed and lacking in
energy over the previous year, since he had been compelled

potentials.
A CT scan of the head showed bilateral
thalamic infarcts in the region of the midline i @iclei

he was watching

television when he suddenly

felt himself

‘¿going'
for a fewsecondsand then lost consciousness.His

113

McGILCHRIST ET AL

114

marked pressure of speech and flight of ideas, lack of sleep,
and increased appetite.

Although

his mental state was not

accessible at the time, it has since transpired that during
this episode he suffered delusions of self-reference and
grandiosity and visual hallucinations. From this point his
mood gradually settled, he became sociable and cooperative,

his sleep and appetite reverted to normal, and on discharge
he had been euthymic

for five weeks. A month

later,

however, he had a final brief episode of increased activity.
He was then started on lithium therapy and has for the last
six months remained euthymic.
unable to return to work.

He still feels, however,

Discussion
It is probable that the infarct to the right (and less
certainly the left) thalamus occurred at the time of
the original episode. Aetiological factors for the
infarcts could have been hypercholesterolaemia

a
S

against

the

background

of

a family

history

of

cardiovascular disease. The mild, transient left-sided
pyramidal signs may represent an acute oedematous
reaction affecting the adjacent posterior limb of the

internal capsule. A similar picture was described by

80
S 28

Fig. I
(see

Fig.

Po@

Brain CT scan showing bilateral thalamic infarction.
1).

A

single-photon

emission

computerised

tomography (SPECT) scan (using labelled HMPAO)
showed

severe

hypoperfusion

of both

frontal

lobes

(left

more marked than right) and of both thalami. There was
also a mild increase in tracer deposition in the upper mesial
parietal cortex bilaterally. A cerebral digital subtraction
angiogram was normal.
Neuropsychological
assessment revealed a full-scale IQ
of 107 and mixed performance on memory tests. The main
deficit was in delayed recall of the Rey-Osterrieth figure
(Ostemeth, 1944/45). There were no language or perceptual
deficits. Frontal testing was within normal limits apart from
a particularly poor performance in both parts of the Trail
Making Test (Armitage, 1946), and the Cognitive Estimates
Test (Shallice & Evans, 1978).
Despite an appearance of depression, his libido remained
intact, and he was sexually disinhibited in his approaches
to women on the ward. The most striking feature of his
mood was profound apathy, and he became quite irritable
when prompted to get out of bed. His self-care was poor,
and he ate in excess. He was treated with imipramine and
a strict behavioural programme. There were two episodes
of elation: the first, lasting about two days, occurred six
weeks after admission; and the second, which was more
prolonged, at 12 weeks. This episode was frankly manic;
he laughed, prayed and cried, and had a labile affect,

Boiten & Lodder in 1990.
The sudden onset of grossly altered mood and
behaviour is not typical of a functional depressive
disorder, and taken with evidence of periodic
hypersomnia,
hyperphagia,
and possibly hyper
sexuality is suggestive of an organic cause.
Additional evidence for an organic basis is
provided by the poor performance on some tests of
fronto-subcortical
function and by problems with
visuo-spatial recall. The latter have been reported in

association with non-dominant

thalamic lesions

(Speedie & Heilman, 1982).
Affective disturbance is highly characteristic of
non-dominant thalamic infarction and usually takes
the form of apathy, lack of initiative, and some
irritability (Bogousslavsky et a!, 1988). Our patient
conformed to this pattern but, in addition, showed
evidence of facetiousness and inappropriate behaviour

which on occasion became frankly manic. Only
one case with similar features has been reported
(Bogousslavsky et a!, 1988). The cyclical or periodic
nature of the disorder in our patient was in many
ways suggestive of the Kleine-Levin syndrome,
although some authors would exclude obvious
organic factors for this diagnosis. If this premise is
rejected, however, this case would provide fresh

evidence for the role of the thalamus rather than the
hypothalamus in the pathogenesis of this disorder
(Smolik & Roth, 1988).
The mechanism is likely to involve the concept of
diaschisis, first described by von Mannikow in 1914

(Szeier et a!, 1991). The term refers to a reduction

THALAMO-FRONTAL

of metabolic activity in distant brain structures which
is most

often

implicated

in subcortical

stroke

(Pappata et a!, 1990).
The advent of functional imaging has made this
phenomenon easier to study. Scans of patients with
thalamo-capsular or pure thalamic stroke reveal
ipsilateral cortical hypometabolism and this is
associated with neuropsychological deficit (Baronet
a!, 1989). As neither phenomenon occurs in pure
capsular infarction,

both appear to be dependent on

thalamic involvement (Pappata et al, 1990). Baron
noted that thalamo-cortical pathways include non
specific systems which project diffusely in the
ipsilateral cortex. One of the functions of these
pathways is to relay the ascending reticular activating
system which modulates hemisphere arousal:
disruption might lead to dysfunction and hence
hypometabolism in cortical neurones.
A more recent study suggests that infarcts in the

medial thalamic nuclei may lead to particularly
marked hypoperfusion of the frontal lobes and basal
ganglia (Szelier eta!, 1991). Both the clinical picture

and the SPECT phenomena in our patient accord
with this finding. Disruption of specific basal
ganglia-thalamo-frontal circuits, which have been
extensively

reviewed

elsewhere

(Alexander

et a!,

1990), probably mediates this more specific effect
on the frontal lobes.
Although neuroimaging in affective disorders
have yielded only tentative conclusions, frontal
involvement has been a consistent finding, and
hypometabolism in the frontal region, especially in
the left dorsal antero-lateral prefrontal cortex, has
been shown in patients with depression (Baxter et al,
1989).
A further unusual feature of this case is the length

of time over which the clinical picture extended, as
most patients show almost full recovery at one year
(Cambon et a!, 1987).

PSYCHOSIS

In addition, these psychiatric disturbances, in the
context of periodic hypersomnia and hyperphagia,
point to the role of the thalamofrontal axis in the
pathogenesis of the Kleine-Levin syndrome.
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Conclusion

There has been one previous case report of thalamic
infarction associated with an acute-onset frontal lobe
syndrome. Like ours, this began with transient
impairment

of consciousness

and eventually

resolved.

Other cases have been associated with apathy.
However, to our knowledge, this is the first reported

case of a cyclical psychosis with distinct manic and
depressive
affective

features.
disturbance

Taken

with other reports

in thalamic

infarcts,

lain McGilchrist, MA, BM, MRCPsych,Registrar in
Neuropsychiatry; Laura H. Goldstein, PhD, MPhil,
AFBPsS, Lecturer

in Clinical

Psychology;

Danitza

Jadresic, MB, BS, MRCPsych,Senior Registrar in
Neuropsychiatry; Peter Fenwick, MB, BChir,DPM,
FRCPsych,

Consultant

Neuropsychiatrist,

Neuro

psychiatry and Epilepsy Unit, Maudsley Hospital,
Denmark Hill, London SES 8AZ

of

it suggests

the concept of a ‘¿thalamo-frontalpsychosis' which
may be mistaken for a functional affective disorder.

5Correspondence:

University Department

of Psychiatry,

ManchesterRoyal Infirmary, Oxford Road, Manchester
M13 9PL