COGNITIVE NEUROPSYCHiATRY

V D Psychology Press

2006. 11 (5). 465-479

A

Taylor6.Ffai,ci.C.oL,p

A case of evolving post-ictal language disturbance
secondary to a left temporal arteriovenous
malformation: Jargon aphasia or formal thought
disorder?
Adam Zeman
Western General Hospital UK

Alan Carson
Royal Edinburgh Hospital

Carly Rivers
Western General Hospital. UK

Uma Nath
Sunderland Royal Hospital, UK

Introduction. Wemickc's dysphasia and formal thought disorder are regarded as
distinct diagnostic entities although both are linked to pathology in the left superior
temporal gyrus (STG). We describe a patient with focal pathology in the left STG,
giving rise acutely to a fluent dysphasia, which gradually evolved into formal
thought disorder.
Method. Clinical, neuropsyehological, neuropsyehiatrie, and neuroradiologieal
assessment.
Correspondence should be addressed to Dr A. Zcnian, Consultant Neurologist, Department of
Clinical Neuroscicnccs, Western General Hospital, Crcwe Road South. Edinburgh, EH4 2XU. UK.
E-mail: az((i',skulI.den.ed.ae.uk
We thank Trudi Minns and Ann Marie Pringle for detailed language asses.>inicnts; Dr George
Masterton. Consultant Psyehiathst, Royal Infirmary, Edinburgh for his helpful adviee and for providing an audiotape of the patient's speech on admission; Professor Tim Crowe, Dr Stephen Lawric,
and Dr Loma Torrens for valuable discussion; Professor Joanna Wardlaw, Dr Paul Armitagc, and Dr
Don Collie, lor radiologieal adviee; two anonymous referees for exceptionally helpful comments.
Adam Zeman is supported by The Health Foundation.
The work described here was presented in part at a meeting of the British Neuropsyehiatry
Association and published in abstrael form under the following title: Evolving "jargon liysphasia"
seeondary to a Ict^ temporal lobe arteriovenous malformation: A disorder of thought, behaviour or
language? Nath, U., & Zeman, A. (1998). Journal of Neurology. Neurosurgery and Psychiatry, 64.
423.
© 2006 Psychology Press Ltd
http://www.psyprcss.coin/cogneuropsychiatry
DOI: 10.1080/13546800544000019

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ZEMAN ETAL

Results. A right-handed patient, AJ, presented acutely with a fluent dysphasia.
His speech output gradually evolved from undifTerentiated jargon, through neologislie jargon, to an intelligible but bizarre form of discourse. Comprehension v^'as
relatively well preserved. Radiology revealed an arteriovenous malformation in the
left middle, and inferior temporal gyri. with reduced perfusion of the left STG. Six
months later his overt dysphasia had recovered, but his speeeh retained some of its
previous characteristics, in particular a tendency to a loose association of ideas
which now suggested a disorder of thought.
Conclusions. AJ's ease illustrates that comprehension may be unexpectedly
preserved in jargon aphasia, and that an overtly linguistic impairment ean gradually evolve to an apparent disorder of thought. Indistinguishable formal thought
disorders can result from "structural" and "functional" pathology in the dominant temporal lobe.

The hallmarks of Wemicke's dysphasia are impaired comprehension, fluent
speech incorporating paraphasias atid neologisms which may render the output
unintelligible, poor repetition and impaired naming (Caplan, 2003). The bizarre
speech content of patients with Wemicke's dysphasia sometimes gives rise to
psychiatric diagnoses, before the true nature of the speech disturbance is
appreciated (Lishman, 1998). This confusion may reflect overlapping mechanisms, as recent work on the neural basis of formal thought disorder (FTD) in
patients with schizophrenia implicates subtle structural and functional lesions in
or close to Wemicke's area (Kircher et al., 2001, 2002; Rajarethinam,
DeQuardo, Nalepa, & Tandon, 2000; Shenton et al., 1992).
The term "jargon dysphasia" is most often applied to a variety of Wernicke's dysphasia in which speech output is especially prolific and faulty (Adair,
Schwartz, & Barrett, 2003). Kertesz and Benson, for example, applied the term
to patients who "produce a copious, largely unintelligible speech consisting of a
mixture of neologistic, paraphasic and appropriate words" (Kersetz & Benson,
1970). Other authors have emphasised the speaker's apparent lack of awareness
that his/her output is extraordinary (Aljouanine, 1956), Weinstein describing
jargon as "a parody or caricature of meaningful speech ... a mixture of aphasia
and anosagnosia' (Weinstein, 1974), Kinsboume highlighting the "large
quantities of faulty and circuitous speech ... produced by patients with anosagnosia for their aphasia" (Kinsboume & Warrington, 1963). However, several
authors have drawn attention to the surprising fact that this variety of dysphasia
can rarely occur in patients with entirely, or relatively, intact comprehension
(Adair, 2003; Aljouanine, 1956; Kinsboume & Warrington, 1963; Maher,
Gonzales, Roth, & Heilman, 1994; Marshall, Robson, Pring, & Chiat, 1998;
Shuren, Smith Hammond, Maher, Rothi, & Heiiman, 1995),
We describe a patient of this kind, in whom we were able to study the
evolution of jargon dysphasia from its acute "undifferentiated' phase, in which
speech consisted mainly of repetitive sounds, through a stage of fluent neologistic output, to a state of intermittently circuitous and bizarre but intelligible

JARGON APHASIA OR FORMAL THOUGHT DISORDER?

467

speech which struck fresh observers as reflecting a disorder of thought rather
than language. Throughout this evolution his comprehension of the speech of
others was relatively intact.

METHOD
Case report
On 10 October 1996, a 51-year-old right-handed man, AJ, was found by police
in the centre of a shopping precinct where he was apparently posing as a market
researcher. He also told the police he was an "agent", and that he was being
followed by a Customs van. He was taken to the local Casualty Department. On
arrival, he appeared to be having visual hallucinations, claiming that the Customs van was in the department, He was pyrexial and dehydrated. His creatine
kinase was elevated at 2515 (normal range <200). He was diagnosed as suffering
from an acute organic confusional state. During transfer to the ward his speech
became highly pressured and nonsensical. The admitting team revised the
diagnosis to an acute psychotic episode.
He was reviewed by a consultant psychiatrist who described his speech as a
hyperfluent "word salad" with neologisms, punning, clang associations, and
perseveration. His speech suggested emotional lability although he was otherwise calm. He could follow simple commands, but was unable to answer
questions or to provide any information. He tended to introduce words used by
others into his own sentences. There was no extralinguistic evidence of confusion. Some examples of AJ's speech were recorded at this time:
1.10.96

2.10.96

"It's unther ah excuse me it's garvo it's gungle black it's dim it's
clung cleetly aatly clung clu clat clee-artly and danzai duver cloutly
fouchy dill debs dill doot dilartly... clexus, elexus, elexus".
on hearing the examiner say "amo, amas, amat", he continued
"amarum, amavis, amant", subsequently incorporating Latin words
into his jargon.

He was treated conservatively with rehydration and began to improve over the
next few days. The psychiatrist suspected an underlying organic cause for his
disorder.
According to his ex-wife, who was contacted by the medical team, AJ had
suffered from epilepsy diagnosed at the age of 13, with infrequent generalised
seizures and tnore frequent complex partial events. These were episodes of
altered awareness lasting a few minutes during which he was unresponsive with
occasional limb twitching. Although she felt that his speech had been "strange
and circumlocutory" as long as she had known him, the seizures were always
followed by a worsening of his speech problems which could last for up to a few

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ZEMAN ET AL.

days. His friends had often commented on his peculiarities of speech, but he had
consistently denied that his speech was in any way abnormal. He had been
admitted to hospital a few months earlier with a generalised seizure followed by
a speech disturbance which resolved over a week. He was known to be poorly
compliant with his anticonvulsant medication (phenytoin and primidone). A
previous electroencephalogram (EEG) had shown a left frontotemporal focus.
There was no history of contact with psychiatric services, although there was a
past history of frequent changes of employment and moderately heavy alcohol
intake.
The patient underwent an initial speech therapy assessment six days later.
He was orientated in time and person but not place. His autobiographical
memory was intact and immediate and delayed recall were normal. He could
calculate and problem solve effectively. He was able to follow complex spoken
or written commands. Repetition was normal. However, his spontaneous
speech was highly pressured with neologisms, perseveration, and circumlocution:
7.10.1996

To the question "What is the weather like today?" he replied
"October rains right converted musk usk might trite convert on the
av musk promised preserved from the Atlantic Melag port."
He described a volcano as "Vesuvius-type; an earth fire system of
energy which I haven't attended college to teach."
He defined a beaver as "a fal- tailed yellow type mongoose of the
USA", a rhinoceros as a "monohom only known by people in
North America".
On being asked the meaning of the proverb "A rolling stone gathers
no moss" he replied "stock that moves is heallhy and doesn't
accumulate explosive characteristics."

He showed significant naming difficulties. His written output was perseverative
and nonsensical. His mood appeared labile and he was easily distracted.
Despite continuing suspicions on the part of his physicians that the underlying diagnosis might be psychiatric, a range of further investigations was
performed. Cerebrospinal fluid examination performed the day after admission
was normal, but a computed tomographic (CT) scan showed a large calcified
lesion in tbe left temporal lobe. An magnetic resonance imaging (MRI) scan
performed three weeks later indicated that this was a arteriovenous malformation (AVM) involving the middle and inferior temporal gyri of the dominant
hemisphere (Figure I a, b, c). This was confirmed at angiograpby in January
1997 (Figure Id).
Once AJ's acute language disturbance had resolved, an intermittent but
persistent disorder of language—or thought—became apparent:

JARGON APHASIA OR FORMAL THOUGHT DISORDER?

469

Figure I. Cranial magnetic resonance imaging (MRl) showing left temporal vascular malformaiion
involving middle and inferior gyri of doniinani licniLsphcrc, in sagittal (a), transverse (b), and eorona!
(c), planes, with (d) angiogram eonfirming Ihe presence of a Ictl temporal arteriovenous malformation (AVM) supplied by the left carotid circulation.

November 1996

On being asked about his career plans: "I am hoping (o
produce a company producing commodity-branded packs
which will have a mark-up of 900% and supply limestone to
one person and cat-litter lo another"'.

December 1996

"I'm talking in French or, don't speak Belgian, or English
for that matter, Gallic is the in thing to talk these days."

January 1997

"Work seems to be springing up. and it might be if I get the
okay I'm sounding out a recycling agency to buy all the
yellow pages kicking round the Edinburgh borough, because
none of the Edinburgh merchants knew where the paper came
from and therefore didn't want to buy it. I could ofler £20
pounds per ton of pulp and this used to be SI50 a ton, maybe
this is more ethical."
"I'm completely separate from all my tests, oh dear, if 1 had
mentioned it when I came in I could have illuminated a lot
more paperwork."
Question: '"Do you have a problem with comnntnication?''

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ZEMAN ETAL

Answer: "Now that the swelling has decreased you can sec
that I've bitten off approximately 15% of my tongue and this
caused the swelling and the speech problem."
Question: '"Do you remember me?''^
Answer: "Oh yes size 16 18 rather smart blond hair sorTy I
have been married!"
His speech disorder was not always in evidence, and indeed at times he could
have a perfectly normal conversation. Nevertheless, as the examples make clear,
his speech was frequently elliptical and bizarre despite the absence of neologisms by this time and the normality of his comprehension.
In March 1997, he underwent a further speech therapy assessment. He scored
at ceiling on the Western Aphasia Battery on all three auditory subtests and he
was able to comply easily with five-part sequential commands. His responses to
picture description were appropriate. He gave a full description of a composite
picture. Maximum scores were found for repetition and reading comprehension
even in lengthy and complex tests. Apart from omitting one word in a dictated
sentence his written language scores were at ceiling. Calculation, block design,
drawing and Raven's Coloured Progressive Matrices were all within normal
limits. His score on the Boston naming test was within normal limits. The only
possible indication of a language disorder was a mild reduction of verbal
fluency.
However, in the course of describing his prior admission he became verbose
and pedantic. His description of the composite picture, although full, incorporated some unnecessary and unusual inferences. He gave elaborate, tangential
word definitions:
March 1997 Asparagus: "a vegetable fully frozen. You dip it into a cheese sauce.
It is marketed under deep freeze."
During the session AJ introduced some completely unrelated ideas including
matters related to Customs and Excise, and the following examples:
Mareh 1997 "1 plan to get a loan in order to plant rowan trees on the riverbank
which would engage lots of unemployed people in jobs."
"Felt pens are the in-things nowadays and everyone is scared to use
them as they might blow up."
These remarks were taken to suggest a disorder of thought.
Treatment options for AJ's arteriovenous malformation including surgery
were discussed with him. He declined further treatment, partly in view of the
strategic location of his malformation.

JARGON APHASIA OR FORMAL THOUGHT DISORDER?

471

He has been reviewed regularly between 1997 and 2005. He has complied
with anticonvulsant treatment and has suffered only very occasional seizures. He
is living independently without mishap. His language function is superficially
nornial, with mild naming difficulties and reduced verbal fluency on formal
testing. His responses to questions are normally to the point, although, as before,
he occasionally digresses bizarrely. For example, on being asked how he was
occupying himself in October 1999, he replied:
"There's great potential for wastcpaper, mainly because sodium chlorate, put by
furnaces into 2-year-old or 3-year-old timber, when it enters the rcpulping process,
gives rise to swayback or DCN [the initials of our department] in cows."
In 2004, defming the phrase "Still waters run deep', he explained:
"The Union Canal may be still but it is 12 feet, 4 metre deep, and takes 5000
gallons/hour. On the marine side, if the wind isn't blowing 8 gale, 10 hurricane, 0
flat - deeper than 5 fathoms will be quieter, less pitcliable than shallow water."
Recent (2004) neuropsyehological assessment revealed a WAIS-III full scale
IQ of 102, verbal 104, performance 99. Processing speed was depressed relative to other indices at 73. WMS-II! immediate memory index was 102, general memory index 107; unexpectedly, verbal memory indices were higher than
visual (auditory immediate 117, delayed 117; visual immediate 84, delayed
81). This discrepancy between verbal and visual memory was reproduced on
the Warrington Recognition Memory Test: His scaled score was 13 for words,
5 for faces. He perfonned within the normal range on the Wisconsin Card Sort
Test (64 card version), but his performance on the Stroop test was impaired
(4th centile). On the Speed and Capacity of Language Processing, his scaled
score on the "spot the word" test, which requires discrimination of real words
from non-words, was 9, while his comprehension speed, on a test requiring
discrimination of true from false sentences, was impaired at 3. His scaled
scores on the Hayling and Brixton Tests were impaired at I and 2, respectively: These tests assess the abilities to inbibit automatic verbal responses in a
sentence completion task and to infer the simple but changing rules governing
the movements of a token in a numbered spatial array . In summary, psychometric testing revealed an average verbal, performance, and full-scale IQ, an
unexpected superiority of verbal over visual memory, low processing speed
and impairment on some tests of executive function, which could indicate a
primary disorder of executive function or possibly reflect AJ's low processing
speed and intermittent thought disorder.
Neuropsychiatric examination confirmed the presence of formal thought
disorder but was not suggestive of any additional psychotic symptoms. In particular there were no Schneiderian first-rank symptoms. There was no evi-

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ZEMANETAL

dence of any hallucinations. The assessment of delusional ideation was compromised by his disorder of form of thought. Careful exploration of the content of thought did not suggest that a pathologically falsified judgement
was present (Jaspers, 1913/1963), but it was not possible to exclude the possibility of fleeting delusional ideas being present. There was no evidence to
suggest any well systematised delusional ideation. AJ was slightly dishevelled and poorly groomed. His affect did not vary with the content of his conversation: He maintained a slightly vacant, cheerful demeanour irrespective
of topic. This probably did not amount to affective blunting and fell within
nonnal limits.
Recent perfusion MRI scanning (Figure 2) shows increased blood flow in the
left inferior temporal lobe, corresponding to his arteriovenous malformation,
with a relative reduction of blood flow in the left superior temporal gyrus by
comparison with the right.

DISCUSSION
We interpret AJ's unusual case along the following lines. The evolving manifestations of his disorder can all be traced to dysfunction of the left temporal
lobe, for which his arteriovenous malformation (AVM) is ultimately responsible.
The acute exacerbations of his disorder in October 1996, and on previous
occasions, giving rise to "jargon dysphasia", were the result of epileptic activity
provoked by the AVM, or of a resulting post-ictal impairment of brain activity in
the region of the seizure focus, a "Todd's paresis". Some features of the presentation in October 1996, including his apparent elevation of mood, delusional
ideas, and hallucinations, may have been the result of an associated post-ictal
psychosis (Logsdail & Toone, 1988). Once AJ's acute illness and his severe
disturbance of language had resolved, he remained prone to bizarre, loosely
associated, and illogical utterances, exemplifying all the features incorporated
by Liddle et al. (2002) in their instrument designed to quantify formal thought
disorder (FTD), the Thought and Language index: looseness of the connection
of ideas, peculiar use of words which are invented or unusual, peculiar sentence
form and peculiar logic. We propose that AJ's liability to FTD is the result of
precarious function in his structurally compromised dominant temporal lobe and
its wider consequences for AJ's language processing and lateralisation of cerebral function.
We shall discuss several relevant issues: (1) the occurrence of fluent dysphasia as an ictal or post-ictal phenomenon; (2) the evidence that formal
thought disorder (FTD) is related to left temporal lobe dysfunction; (3) the
possibility that AJ's thought disorder might be due to an inter-ictal psychosis;
(4) the relationship between AJ's language disorder and "Wemicke's dysphasia".

JARGON APHASIA OR FORMAL THOUGHT DISORDER?

473

Figure 2. Pcrfiision MR scan (top), with corresponding T2-wcightcd axial MR images (bottom).
The left temporal atcriovenous malformation (AVM) has incrca.scd blood flow (arrow), white the left
superior temporal gyrus adjacent has redueed hlood flow (arrow head) compared with the opposite
superior temporal gyrus. [Colour figure available from authors.]

Occurrence of fluent dysphasia in temporal lobe
epilepsy
Although we cannot be certain that epileptic activity was the explanation for
AJ's acute dysphasia, the history of previous similar episodes following generalised seizures strongly suggested that this was so. Dysphasia has been
described both as an ictal and as a post-ictal manifestation of temporal lobe
epilepsy. Two reports document fluent dysphasia occurring as a circumscribed
ictal phenomenon. Bell reported a case of neologistic speech associated with
epileptic discharge in the left temporal lobe. The patient was unable to respond
to commands during the episode, for which she was subsequently amnesic.
There was no post-ictal dysphasia. The patient was subsequently found to have
an arteriovenous malformation in the left supramarginal and superior temporal
gyrus (Bell, Homer, Logue, & Radtke, 1990). Smith Doody, Hrachovy, and
Feher (1992) reported a 47-year-old man who developed a fluent dysphasia
characterised by hyperfluent neologistic jargon with a concurrent dominant
temporal lobe focus on EEG. Comprehension was severely impaired during the
episode. Prolonged dysphasia has also been described as a rare manifestation of
focal status epilepticus in the dominant temporal lobe: In one case, episodes

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ZEMAN ETAL

were misdiagnosed as "psychotic breaks" for 15 years (Knight & Cooper,
1986). To our knowledge, jargon dysphasia with relatively well-preserved
comprehension, similar to AJ's, has not previously been reported as an ictal, or
indeed post-ictal, phenomenon.
Post-ictal dysphasia is a common occurrence following temporal lobe seizures and is usually associated with a focus in the dominant hemisphere
(Koemer & Laxer, 1988; Gabr, Luders, Dinner, Morris, & Wyllie, 1989). It can
be "expressive" or "receptive" (Gabr et al., 1989). Its duration is typically
brief. Privatera, Morris, and Gilliam (1991) studied 105 complex partial seizures
in 26 patients measuring the interval between the end of an ictal discharge and
the ability to read a phrase clearly and correctly. This interval was less than 1
minute in 42/43 seizures originating in the right temporal lobe, whereas the
interval following 62 left temporal lobe seizures was always greater than 1
minute, with a mean of around 5 minutes. The slow recovery of AJ's overt
dysphasia, over a matter of weeks, at a time when his scalp EEG showed only
minor changes, suggests either that haemodynamic disturbance, caused by his
AVM, was compounding the effects of recent epileptic activity in the left
temporal lobe, or the occurrence of continuing focal epileptic activity which can
escape detection by surface EEG. (Hirsch, Emerson, & Pedley, 2001).

Role of left temporal lobe dysfunction in formal
thought disorder
An early functional imaging study by Liddle et al. (1992) demonstrated a
complex pattern of correlations between three groups of symptoms in schizophrenia (psyehomotor poverty, disorganisation, and reality distortion) and
alterations of regional cerebral blood flow as revealed by positron emission
tomography (PET). Recent structural and functional studies have more specifically linked pathology in the left superior and middle temporal gyri to FTD
(Crow, 2004; Kircher et al., 2001, 2002; McGuire et al., 1998; Rajarethinam et
al., 2000; Shenton et al., 1992), although there have also been some negative
reports (Barta, Pearlson, Powers, Richards, & Tune, 1990; Flaum et al., 1995;
Zipursky et al., 1994). The positive studies suggest that schizophrenia is
associated with reduced or reversed hemispheric asymmetry of the posterior
superior temporal gyrus (STG), which is normally larger on the left (Rajarethinam et al,, 2000; Shenton et ai., 1992). The magnitude of this abnormality
may be correlated with the severity of FTD (Rajarethinam et al., 2000; Shenton
et al., 1992). Using functional MRI, Kircher et al (2001) found that the severity
of FTD in a small group of patients with schizophrenia was inversely associated
with blood oxygen level dependent (BOLD) signal in the left (L) STG and
middle temporal gyrus (MTG). The amount of thought-disordered speech was
correlated with BOLD signal in the right (R) STG in patients with schizophrenia
(Kircher et al., 2002). In contrast, in healthy controls given the same descriptive

JARGON APHASIA OR FORMAL THOUGHT DISORDER?

475

task, the amount of speech correlated with BOLD signal in the contralateral L
STG (Kircher et al., 2002). Thus there is converging evidence for alteration of
structure and function in the L STG and MTG in patients with schizophrenic
FTD. Related work suggests that the left and right hemispheres employ contrasting modes of semantic processing; Whereas in the left hemisphere semantic
activation is focused and short-lived, in the right it is more difftise and prolonged
(Faust & Chiarello, 1998). The creates potential for "overlap of distantly related
concepts" in the right hemisphere, one of the features of FTD.
Given that AJ is right-handed, and that his seizures, associated with a left
hemisphere focus on EEG, have given rise to dysphasia, it is likely that his left
hemisphere is dominant for language. His AVM, a developmental abnormality
centred, in his case, on the left middle, and inferior temporal gyri and associated
with reduced perfusion of the left STG, is strategically placed to disrupt the
processes of lexical access and retrieval which normally ensure semantic coherence. We suggest that the chronic effects of this overt structural lesion, and its
haemodyamic and physiological results, mimic those of the elusive developtnental
or functional lesions that cause the varieties of FTD seen in in schizophrenia.

A case of inter-ictal psychosis?
There is a putative association between schizophrenia-like psychosis and temporal lobe epilepsy, particularly in patients with foci in the dominant temporal
lobe (Lambert & David, 2000). The occurrence of apparently bizarre and
inappropriate mentation in a patient with an epileptic focus in the dominant
temporal lobe therefore raises the possibility of an underlying inter-ictal psychosis. Despite the curious circumstances of his presentation, several observations argue that AJ does not suffer from a full-blown inter-ictal psychosis. First,
although the content of his speech is often odd and at times bizarre, he has never
displayed any "first-rank" symptoms of schizophrenia. Second, by the stage at
which the formal characteristics of his speech had returned to normal, his
manner was reasonably appropriate. Finally, he has been able to organise an
independent life without undue difficulty, arguing somewhat against the presence of any active psychotic disorder. However, our interpretation of AJ's case
is precisely that the structural lesion in his left temporal lobe predisposes him to
one prominent manifestation of psychosis, namely formal thought disorder
(FTD). The possibility that some other cases of presumed inter-ictal psychosis
may in fact represent instances of isolated FTD due to focal left temporal
dysfunction should be considered.

A case of "Wernicke's aphasia"?
Was AJ genuinely dysphasic at any stage of his illness? AJ's initial speech
disorder had several characteristics pointing towards a Wernicke's dysphasia.
His speech was ftuent, indeed hyperfluent, and consisted for a while almost

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ZEMAN ET AL

entirely of meaningless phonemes and neologisms. This exclusively neologistic
output evolved over days into hyperfluent speech with numerous neologisms,
paraphasias, and prominent "clanging". AJ appeared to have no insight into his
speech disorder. These features are all in keeping with a classical "Wemicke's
dysphasia". Tvi'o findings, however, are unexpected for this disorder: his preserved repetition and his relatively well-preserved comprehension. Preserved
repetition is the hallmark of the transcortical sensory type of dysphasia, a close
relative of Wemicke's dysphasia (Caplan, 2003). But the relative sparing of
comprehension is not in keeping with a transcortical sensory dysphasia, and
requires some other explanation.
Although it is exceptional, fluent dysphasia with relative sparing of comprehension has been described repeatedly (Aljouanine, 1956). An early case, EF,
reported by Kinsboume and Warrington (1963) is reminiscent of AJ. Following
probable lef^ intemal carotid artery occlusion, EF spoke incoherently yet
"rapidly, with hardly a pause for breath, and with considerable emphasis, as
well as an air of self-satisfaction ... he was able to maintain a considerable
pressure of speech, and both in rate and content his speech resembled that of
patients with manic psychosis exhibiting flight of ideas" (Kinsboume & Warrington, 1963). Nevertheless, "where any instmction required a motor rather
than a verbal response it was quickly executed", to single, double, and treble
commands, and "he gave evidence of good comprehension for written material". Several other, more recent, reports have documented a dissociation
between normal or relatively normal comprehension and highly abnormal output, in occasional patients, using standard aphasia batteries (Maher et al., 1994;
Marshall et al., 1998; Shuren et al., 1995).
The combination of preserved comprehension and fluent highly disordered
speech in an "organic" dysphasia is puzzling at first sight. The most straightforward explanation for the abnormalities of speech in patients with Wemicke's
dysphasia is an inability to monitor speech due precisely to the loss of access to
lexical representations which impairs comprehension (Adair et al., 2003). In
patients where comprehension is more or less intact, it appears that "an
apparently viable monitoring mechanism is being either ignored or overridden"
(Marshall et al., 1998).
Two kinds of explanation tend to be offered for such cases. The first type
suggests that jargon dysphasia, in patients with preserved comprehension, is an
effort to bolster denial of a language deficit, an adaptive response to a disquieting disorder. On this view the emphatic production of fluent speech, albeit
meaningless, restores the speaker's confidence in his/her own linguistic powers,
allowing him/her to regain a ^\feeHng of meaning ... by virtue of the way his
words identify him as a member of a social group" (Weinstein & Puig-Antich,
1974). This line of thought provides a valuable reminder that language output is
influenced by a range of factors beyond the brain areas and processes that are
most closely associated with language processesing: These include diffuse

JARGON APHASIA OR FORMAL THOUGHT DISORDER?

477

disturbances of neurological function giving rise to organic confusional states,
impairments of executive function and personality traits which create a "strong
urge to speech" (Curran & Schilder, 1935; Weinstein and Kahn, 1952).
The second type of explanation remains within the realm of conventional
language processing. A patient with preserved comprehension for the speech of
others might nonetheless become unable to monitor his/her own speech if his/her
deficit rendered him "unable to be a speaker and a listener at the same time"
(Lebrun, 1987). This could occur if reduced attentional capacity prevented
concurrent speech production and speech monitoring, or if the process of speech
production interfered in some way with speech reception There is experimental
support for both these possibilities (Marshall et al., 1998; Shuren et al., 1995).
The relative preservation of comprehension in AJ's case would not therefore
exclude an account of his case in terms of Wemicke's dysphasia, although it is
certainly atypical. What of the evolution of his disorder from an overtly "linguistic" disorder to a "disorder of thought"? A similar pattem has been
described in some previous cases of "jargon aphasia" (Alajouanine, 1956; Cohn
& Neumann, 1958). Alajouanine described an evolution from "undifferentiated" jargon, consisting of a continuous flow of stereotyped unintelligible
phonemes, through "asemantic" jargon, consisting substantially of neologisms,
to "paraphasic" jargon, in which output consists of real words used inappropriately. AJ's recovery proceeded along these lines: from the production of
unintelligible phonemes, through a stage of neologism and paraphasia, to a stage
of apparently faultless linguistic competence flawed by a loss—albeit intermittently^of "contextual thread". In this respect, as in his preserved comprehension at the earlier stages of his illness, AJ's linguistic disorder is strongly
reminiscent of the "schizophasia" described in a recent study by Oh, McCarthy,
and Mckenna (2002) and compared by these authors to a "forgotten type of
aphasia. Head's (1926) syndrome of "semantic aphasia' ... a disorder associated with psoterior left hemisphere lesions which affected the assembly of
meanings at a level above that of the single word and which spared the retrieval
and comprehension of single words".
The variability of features in dysphasia and the limitations of syndromic
classification are now well recognised (Caplan, 2003). "Typical" cases are rare,
and indeed AJ's case is neither typical of Wemicke's nor of jargon aphasia.
However, it is noteworthy that features similar to those we encountered in AJ
have been reported previously in the dysphasia literature.

Conclusion
We have described a patient in whom a hyperfluent "disorder of language" was
replaced by a "disorder of thought". We have argued that his case exemplifies
the evolving manifestation of an underlying disorder of structure in the dominant
temporal lobe. The difficulty his doctors encountered in deciding whether the

478

ZEMAN ET AL.

disorder was fundamentally one of thought or of language reflects their intimate
relationship. Indistinguishable "formal thought disorders" can result from
''structural" and "functional" pathology in the dominant temporal lobe.
Manuscript received 23 July 2004
Revised manuscripl received 10 February 2005

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