Clinical Neurology and Neurosurgery 103 (2001) 123– 126
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Case report

Aphemia: an isolated disorder of articulation
Robert J. Fox *, Scott E. Kasner, Anjan Chatterjee, Julio A. Chalela
Department of Neurology, Uni6ersity of Pennsyl6ania Medical Center, Philadelphia, PA, USA
Received 11 December 2000; received in revised form 27 April 2001; accepted 27 April 2001

Abstract
Aphemia is a disorder with prominent speech abnormality. Since its description by Broca, there has been debate regarding the
neuropsychological disorganization underlying aphemia: is aphemia an articulatory disorder or a language disorder? We describe
a patient with markedly impaired articulation, but preserved receptive and written language function and buccal–facial
coordination. The location of his stroke was in the left precentral gyrus, undercutting a small area of motor and premotor cortex.
This case suggests that aphemia can occur as an isolated articulation deficit without language involvement or more widespread
bulbar apraxia, and may be a severe form of apraxia of speech. © 2001 Elsevier Science B.V. All rights reserved.
Keywords: Agraphia; Aphemia; Stroke

1. Introduction
Aphemia, also termed simple aphasia, cortical
anarthria, pure word mutism, or ataxic aphasia, is a
rare articulatory disorder in which verbal motor output
is grossly distorted. Broca coined the term aphemia in
1861 when he treated Leborgne, a stroke patient with
complete articulatory failure. Broca described the disorder as a defect in the faculty of articulated language,
although his patient later progressed to have deficits
involving the arm and leg. Shortly after its first description, the term ‘aphemia’ was strongly criticized by
Trousseau, who preferred the term ‘aphasia’ and
claimed that the disorder extended beyond articulation
to include comprehension, reading and writing.
In 1887, Bastian revived interest in aphemia and
agreed with Broca’s original description of aphemia as
a disturbance in the organization of articulatory and
motor aspects of speech, with intact grammar, reading,
and writing. Marie and Dejerine continued the debate
at the turn of the century by coining ‘anarthria’,
* Corresponding author. Present address: Mellen Center — U10,
Cleveland Clinic Foundation, 9500 Euclid Avenue, Cleveland, OH
44195, USA. Tel.: + 1-216-4444232; fax: +1-215-4456259.
E-mail address: foxr@ccf.org (R.J. Fox).

thereby distinguishing between a linguistic disorder and
a motor programming disorder (for a historical review,
see Ref. [1,2]). Controversy has continued regarding the
true neuropsychological disorganization of aphemia: is
it a language disorder, or an articulatory disorder [3]?
Most modern descriptions suggest that aphemia involves a dysfunction of written language in addition to
verbal language, which contrasts with older
descriptions.

2. Case report
A 52 year old right-handed man with hypertension,
diabetes mellitus, and dilated cardiomyopathy presented with acute onset right facial weakness and inability to speak. On initial examination, he appeared
anxious and frustrated. He was completely mute but
tried to communicate desperately through gestures and
facial contortions. Propositional and automatic speech
were similarly affected. Comprehension of verbal and
written language was completely normal. He was able
to point to his needs using a pictorial communication
board. He was able to cough, swallow, smile, and blow
air without difficulty. He had mild right facial weakness
but no limb weakness, sensory loss, or limb apraxia. A

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R.J. Fox et al. / Clinical Neurology and Neurosurgery 103 (2001) 123–126

Fig. 1. Head CT on the day of symptom onset demonstrating a left precentral gyrus infarct that undercuts a small segment of motor and premotor
cortex (arrows). There was no infarct on cuts above or below those shown. The hypodensity in the left parietal lobe is a cortical sulcus.

CT of the head on the day of admission demonstrated a small linear infarct involving the precentral
gyrus about halfway up the lateral convexity and undercutting a small segment of motor (BA4) and premotor (BA6) cortex, using the lesion mapping
technique described by Damasio and Damasio [4]
(Fig. 1).
One day after admission, he began to form words.
Speech was effortful and explosive, and words came
out in a strangled fashion. Articulation was hesitant
and slow. His speech was monotonous with abnormal
prosody. Speech output was not facilitated by reading, repetition, naming, singing or counting. He was
able to hum a familiar melody with excellent accuracy. Although phonemes were distorted, there were
no semantic or literal paraphasias. Exclamatory
speech and propositional speech were similarly compromised. He was able to read without difficulty, but
reading out loud was severely impaired. He was able
to write both spontaneously and in reply to a question (Fig. 2). Handwriting was legible and neat. Written sentences were full, in contrast to his elementary
sentence structure in speech. Orthography, syntax,
and grammar were normal. No word omissions or
substitutions were present except for one homonym
spelling error (to for too) in his writing. Writing to
dictation was entirely intact and was executed with
normal speed and fluency. On the Halstead– Reitan–
Wepman screening test as modified by Reitan [5],
there were 10 errors, all related to dysarthric speech
when asked to read out loud or repeat. There were
no errors in writing, following written commands,

copying figures, or arithmetic. Over the next several
days, his speech improved moderately. Telephone follow-up several months later revealed mild residual
dysarthria.

3. Discussion
We have described a patient who was completely
mute, but demonstrated intact receptive language
function and writing, as well as normal bucco-facial
coordination. His case suggests that aphemia can be
due to a defect in motor control of articulation independent of both bucco-facial coordination and language processing. The aphemia observed in our
patient contrasts with modern descriptions of
aphemia. Although detailed linguistic testing was not
performed, there was a clear contrast between his
severely impaired speech output and preserved writing.
The review by Schiff et al. [6] describes aphemia as
arising from an impaired sound production motor
system but normal lexical and syntactic language
function.

Fig. 2. Writing sample demonstrating full sentence structure with
normal orthography, syntax, and grammar and neat print. There was
one spelling error, which was a homonym error (to instead of too).

R.J. Fox et al. / Clinical Neurology and Neurosurgery 103 (2001) 123–126

However, three of their four cases had writing problems, indicating a more profound dysfunction involving
language. Their fourth patient had normal writing, but
was not examined until 4 weeks after symptom onset,
which may have been after resolution of some aspects
of language dysfunction.
Other case reports of aphemia describe similar language dysfunction. Alexander et al. described five patients with single left hemispheric stroke and
predominant articulation and prosody deficits [7]. All
five patients had severely impaired written language,
indicating a pervasive language dysfunction. Furthermore, all five patients had substantial bucco-facial
apraxia. The writing of a patient with aphemia from a
right frontal hematoma was characterized by frequent
omissions of prepositions or connectives, and frequent
spelling errors [8]. David and Bone described a patient
with total mutism following a left hemispheric infarction [9]. Although they reported that mutism was secondary to speech apraxia, their patient’s writing had
paraphasic errors that rendered writing only ‘adequate
for day-to-day communication’. In a patient with pure
progressive aphemia, writing had occasional omissions
of short words [10]. Although the predominant language dysfunction in all of these patients involved
speech production, other areas of language were clearly
affected.
In contrast to these modern reports of aphemia, our
patient had preserved written expression. Our case resembles Broca’s original description of aphemia in
which a disturbance in articulation was observed in
isolation from any other language dysfunction. In our
patient, the only evidence of written language disruption was a homonym error, which may have been due
to educational limitations. His handwriting was neat,
which is in contrast to the messy handwriting common
in aphasic patients.
During the first several days after presentation, spoken language began to return, although a marked disturbance in speech output persisted. Disturbances in
speech output have been divided into three broad classifications: dysarthria, which is usually secondary to
facial or tongue weakness or cerebellar dyscoordination; apraxia, which is due to a motor programming
disorder; and phonological disorder, which is usually
characterized by phonemic substitutions, transpositions, and sequencing errors [11]. Although these distinctions can be difficult to make in practice, the first
two would be considered disorders of oral verbal output, and the last a linguistic disorder. When applied to
our patient, there was no evidence of oro-lingual-buccal
weakness or dyscoordination to suggest dysarthria. He
had normal control of his lips, tongue, and pharynx
when asked to perform non-speech tasks such as blowing air or humming, making dysarthria an unlikely
explanation.

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Distinguishing between speech apraxia and phonological defect is more difficult. Our patient demonstrated sound distortions, as well as articulatory
hesitancy, struggle, and groping, which is typical of
apraxia of speech [12]. Although he did not appear to
have inappropriately selected or sequenced phonemes,
his speech was so contorted that we cannot definitively
distinguish between the phonemic distortion of apraxia
and the phonemic substitution of phonological disorders. None the less, contortions are usually considered
a praxic rather than a linguistic disorder [11], and the
other predominant speech features during recovery appear most consistent with apraxia. Therefore, his speech
apraxia during recovery was most likely a disorder of
oral verbal output and not linguistic. Apraxic features
during recovery also suggest that the aphemia observed
at presentation may have been a severe form of apraxia
of speech.
The location of our patient’s stroke is similar to that
described by Kreisler et al. [13], who found frontal
gyrus and putamenal involvement in eighteen patients
with mutism. However, this study did not describe
whether their patients with mutism had either writing
difficulties or bulbar apraxia, making it unclear whether
these patients had isolated articulation deficits.
Our case suggests that verbal motor output can be
selectively impaired without involvement of other language functions or a more generalized bucco-facial
apraxia. The lesion in our patient was limited to a small
area of the precentral gyrus with undercutting of motor
and premotor cortex. Although we cannot generalize to
all cases of aphemia, our patient’s unequivocal dissociation between spoken and written language provides
support of Broca’s original contention that aphemia
can exist as a pure articulation disorder without language dysfunction. Our patient’s speech apraxia during
recovery suggests that aphemia can present as a severe
form of apraxia of speech.

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