ItaL J. Neurol. Sci V: 77-83, 1984

Speech suppression without aphasia
after bilateral perisylvian softenings
(bilateral rolandic operculum
damage)
Villa G., Caltagirone C.
Clinica Neurologica, Universit3 Cattolica del S. Cuore di Roma.

The authors describe a patient who suffered two successive, right and left,
strokes that caused bilateral rolandic operculum damage. The clinical picture
was characterized by selective impairment of volitionalfacio-pharyngo-glossomasticatory movements with sparing of automatic and reflex motor activity
(Foix-Chavany-Marie syndrome). Though completely speechless, the patient
was not aphasic. This dissociation is discussed in the light of the peculiar
localization of lesions evidenced by CT-scan.
Key-Words : Bilateral rolandic operculum softenings - - Foix-Chavany-Marie syndrome - - speech
suppression without aphasia - - CT-scan.

Introduction

Bilateral destruction of the rolandic operculum
caused by perisylvian infarctions may result in
the so-called 'bilateral anterior opercular syndrome' [9] or Foix-Chavany-Marie (FCM) syndrome [5]. This uncommon neurological disorder is characterized by the selective impairment
of voluntary movements - - with preservation of
automatic and reflex motor activity - - in the
lower cranial nerve distributions (V, VII and IX
through XII) of both sides. It was first described
by Magnus in 1837 [8] and then reported almost
exclusively in the French literature [1, 3, 5, 11]
with some exceptions [2, 4, 9].
We report a typical case of FCM syndrome. It
presented with an acute facio-pharyngo-glossomasticatory diplegia and automatic-voluntary
dissociation of the affected muscles. CT-scan
showed bilateral damage: the left hemisphere
lesion was confined to the rolandic operculum;
in the right hemisphere the whole territory of the
middle cerebral artery was involved.

There are two main reasons for the present case
report:
Firstly, even if FCM syndrome has a localizing
value, it is often misdiagnosed because the neurological picture is very similar to that of the
more frequent bulbar and peripheral palsies.
Thus, from a clinico-methodological point of
view, the procedures that led to the diagnosis in
our case are worth emphasizing. Briefly: a)
FCM syndrome, being the cortical type of pseudobulbar paralysis, can be differentiated from
peripheral and bulbar syndromes on the basis of
clinical and electromyographic signs of supranuclear neuronal damage; b) beyond the general
characteristics of a pseudobulbar deficit, some
peculiar clinical features of FCM syndrome
point to a cortical localization of lesions; c) CTscanning clinches the diagnosis by disclosing the
bilateral perisylvyan softenings including the
rolandic operculum of both sides.
Secondly, the patient we describe was completely speechless, but not aphasic, as revealed by
neuropsychological examination. Her speech
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The Italian Journal of Neurological Sciences

was totally disrupted while her ability to communicate was normal due to normal language
organization. This dissociation is particularly
interesting in the light of the CT-scans, because
it confirms the hypotheses, recently advanced
by some authors [6, 10, 12], that speech may be
sprit into different components, which are differently localized in the left hemisphere. In summary: a) speech may be considered from two
main points of view: the effectiveness with
which information is transmitted (i.e.: 'communication') and the distinctness of enunciation
(i.e.: 'articulation') [6]; b) impairment of communication and impairment of articulation are
generally tightly interwoven in patients with
anterior (pre-rolandic) left hemisphere damage

(Broca's aphasia); yet, as shown by a few cases
reported in the literature [10, 12], they may be
completely dissociated; c) there seems to be an
anatomical basis for this dissociation: within the
left hemisphere, lesions restricted to the foot of
F3 (area 44 or Broca's area) transiently impair
some 'linguistic' aspects of speech, but do not
alter articulatory agility; by contrast, the destruction of the lower motor strip (with the rolandic operculum) exclusively affects articulatory
abilities without involving language organization.
Case report
This 47-year-old right-handed female is a dress-

Fig. 1. Photographic record of the case, showing some of the clinicalfeatures described in the text. Apart
from a congenital divergent strabismus, gaze to right (A), to left (B) and upward (C) are not defective.
The facial-masticatory muscles and the tongue are motionless. Knitting and unknitting of the brows only
appear as synkinetic movements in (A ) and (C). The deficit of voluntary eyelid movements is documented
in (D): the patient was asked to close her eyes," her unsuccessful attempts only resulted in knitting of the
brows (synkinetic movement of the frontal musculature).

78

Villa G.: Speech suppression in bilateral rolandic strokes

maker. In May 1978 she sustained a moderate
left facio-brachio-crural hemiparesis which
cleared almost completely in two months. A
mild articulatory difficulty was present in the
acute stage for a few days. In the same year she
was found to have a mitral stenosis and atrial
fibrillation, previously unrecognized. In March
1977 she underwent valvular commisurotomy,
after which prophylaxis with digitalis and anticoagulants was started. The patient enjoyed
relatively good health till February 1979, when
she suddenly became totally unable to move her
facial and masticatory muscles, to stick out her
tongue, to swallow or to utter articulate sounds.
Admission neurological examination revealed a
facio-pharyngo-glosso-masticatory diplegia, a
moderate motor and sensory deficit in the right
arm, no visual field disturbances. In the following days the syndrome was examined in detail:
- - T h e facial diplegia involved the lower and the
upper portion of the VII nerve, yet without a
Bell's sign. The facial musculature was motionless and hypotonic; the mouth was half-open
(Fig. 1 A, B, C, D); voluntary eyelid movements
could not be performed (Fig. 1 D), but the corneal reflex was spared and a brisk glabellar
reflex could be elicited.
- - Ocular movements were possible in all directions, but adduction of the left eye on gaze to the
right appeared to be defective, owing to a congenital divergent strabismus (Fig. 1 A).
- - Impairment of chewing, due to bilateral masticatory paralysis, was not associated with trigeminal sensory deficit. The masseteric reflex
(jaw jerk) was exaggerated with a tendency to a
clonic response.
- - The tongue was completely immobile, flaccid, but not hypotrophic or fibrillating.
- - The soft palate and fauces were also immobile. There was no "curtain-movement"; the
palatal and pharyngeal reflexes (gag reflex)
could not be elicited.
Dysphagia was one of the most striking features, especially in the acute stage. The patient
was unable to perform the first (voluntary)
phase of deglutition, whereas the second (reflex)
phase was intact. There was continuous drooling
and saliva frequently ended in the trachea or in
the nose.
- - No paralysis of the vocal cords was found on
laryngoscopy.
movements were spared: the patient could knit and unknit her brows on looking
upward (Fig. 1 C), blink, close her eyes while
sleeping.
- - Reflex movements such as coughing, sneezing, belching were also present.
- - The patient could not communicate orally:
she produced only inarticulate, unmodulated
- -

- - A u t o m a t i c

sounds and answered questions only by moving
her head and eyes or by writing.
Electromyographic investigations, performed
with coaxial needle electrodes, on the orbicularis
oculi, orbicularis otis and tongue muscles of
both sides showed: no fibrillation at rest; complete absence of motor unit potentials during
attempts at voluntary contractions whereas
some motor unit potentials could be recorded
from the orbicularis oculi during reflex and
automatic movements of the eyelids.
The excitability of facial and hypoglossus nerves
was normal and the evoked motor action potentials appeared to be normal in amplitude, form
and latency.
Both early and late responses of the blink reflex,
elicited with mechanical and electrical (0.5 msec
duration, double threshold intensity) stimulations, were normal. The jaw reflex appeared after normal latency.
In conclusion: the electrophysiological study
demonstrated lack of voluntary activity but no
signs of peripheral motor neurone damage.
CT-scan showed two areas of decreased density:
the left lesion destroyed the inferior portion of
the rolandic cortex, partially extending deeply
to the subcortical white matter; damage to the
right hemisphere involved cortical, subcortical
and deep structures of the frontal, parietal and
temporal lobes (Fig. 2).
From the lateral reconstruction of CT maps
(Fig. 3) it could be seen that the left lesion was
circumscribed to the lower third of the frontoparietal gyri, including the rolandic operculum:
the pars triangularis and the pars opercularis
(area 44 or Broca's area) of the third frontal
gyrus were spared. The right lesion extended
over the whole territory of the middle cerebral
artery.

Neuropsychological Examination
Language: speech was completely suppressed,
the patient being unable even to vocalize. But
her speech comprehension appeared to be intact. She scored 35/36 on a shortened version of
the Token Test.
Phonemic, semantic and syntactic levels of language integration were explored for the receptive route, by means of three different tests:
Semantic discrimination test (including subtests of auditory language comprehension and
reading comprehension): the patient correctly
selected, 39 out of 40 times, the picture corresponding to a word - - read aloud by the examiner or printed on a sheet - - among three pictured alternatives, strictly related from a semantic-conceptual point of view.
Phoneme discrimination test: the patient
could discriminate with 99% accuracy between
two non-sense CCVC syllables pronounced
aloud by the examiner.
- -

- -

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The Italian Journal of Neurological Sciences

Fig. 2. CT-scan sections." two areas of decreased density are recognizable as ischemic lesions. The left
lesion is confined to the rolandic operculum whereas the right lesion involves the whole territory of the
middle cerebral artery.
Syntax Battery: requested to point, out of 2 or
4 alternatives, to the picture representing a syntactically complex sentence, the patient showed
normal comprehension of reversible sentences
(active, passive, temporal relations - - b e f o r e /
after - - , embedded active and passive).
Spontaneous writing or writing about a subject
set by the examiner was performed slowly but
without phonemic and semantic paragraphias
and with good syntactic construction. The patient was also capable of writing under dictation
and of copying a block-letter text perfectly.
Written naming of pictures was prompt and
accurate.
Memory: digit-span (tested by pointing to the
correct alternatives in a 10-choice device) was 6
forwards and 4 backwards. Spatial memory
span (Corsi's Block Tapping Test) was 5 forwards and 4 backwards. Visual memory of
meaningless shapes and of object drawings was
normal (both immediate and delayed).
Praxis: the impairment of volitional movements
did not allow testing for bucco-facial apraxia.
-

-

80

Transitive and intransitive gestures (including
two and three element sequences), both on oral
order and on imitation, were accurately performed with the right arm. More difficulty was
noted for the left, owing to the slight pyramidal
deficit. Constructional apraxia was absent, since
the patient was capable of reproducing geometrical figures with a very good accuracy.
Visuo-spatial abilities: the patient had no difficulty finding her way about; could accurately
copy geometric and figurative designs and arrange elements in their proper spatial relationship. Left-sided spatial neglect and other disturbances of visuo-spatial exploration of the extrapersonal space (usually related to a massive
right hemisphere damage) could not be evidenced.
General intelligence: In Raven's Coloured Matrices (1947), she gave 30 correct responses out
of 36.
In conclusion: no neuropsychological deficit
could be detected beyond the complete suppression of speech.

Villa G.:

Speech suppression in bilateral rolandic strokes

Fig. 3. Lateral reconstruction of the CT-scan sections shown in fig. 2. The precise boundaries of the
infarctions in the cortex of right (top) and left (bottom) hemispheres are shown.

l J
J
J

jl s

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The Italian Journal of Neurological Sciences

Discussion

The clinical pattern was indicative of a pseudobulbar palsy, resulting from bilateral breakdown of the connexions between the cortical
motor areas and the nuclei of the lower cranial
nerves.
Peripheral palsy was ruled out by the absence of
muscle denervation phenomena, such as fasciculation and hypotrophy, the presence of the
blink reflex, the augmentation of the masseteric
reflex, the sparing of some automatic motility.
Likewise, bulbar damage (due to a vertebrobasilar ischemic stroke) would be incompatible
with the lack of massive paralysis of the limbs, of
sensory and internuclear signs. The electromyographic data, supplemented by blink and jaw
reflex recordings, confirmed the clinical evidence of "upper" rather than "lower" motor
neurone damage.
Other clinical features were then specific for a
cortical localization of the lesions, thus suggesting the diagnosis of 'opercular syndrome', that is
the cortical type of pseudobulbar paralysis:
- - the association of lower cranial nerve involvement with a transient motor and sensory deficit
of the right arm;
- - the dissociation between the severe motor
deficit and the absence of sensory symptoms in
the territory innervated by the trigeminal
nerve;
- - the history of the patient, showing repeated
CVAs with pyramidal motor weakness involving both sides alternately.
CT-scan supported a definite diagnosis of FCM
syndrome by disclosing the cortical level of the
bilateral lesions.
The second interesting feature in the present
case is the dissociation between the normal language organization and the complete suppression of speech - - that is, following Levine and
Mohr (1979), between normal 'communication'
and totally disrupted oral 'articulation' - - and
its CT-scan correlate.
It has recently been suggested [ 10, 12] that lesions
confined to posterior F3 of the left hemisphere (area 44 or Broca's area) do not produce
either Broca's aphasia or any persisting aphasic
syndrome. They only give rise to mild, transient
language disorders, word finding and naming
difficulties, literal and verbal paraphasias, shor-

tening and simplification of sentences, moderate comprehension disturbances [12], without
loss of articulatory agility.
On the other hand, selective damage to the left
lower motor strip (including the rolandic operculum) only affects articulation causing a 'cortical dysarthria' (slowness and dysprosodic alteration of speech-melody with both stuttering
and poor control of pitch [12]).
Our case lends further support to the notion that
a left hemisphere lesion confined to the rolandic
operculum and sparing Broca's area is not associated with a 'linguistic' impairment of speech,
but only with a 'more peripheral' deficit of
speech articulation. In our case the latter deficit
was much more severe than the 'cortical dysarthria': since the left rolandic operculum lesion
was superimposed on preexisting right hemisphere damage (including the right rolandic
operculum), the typical clinical pattern of FCM
syndrome (with complete inabihty even to vocalize) had developed.
The successive involvement of the right and left
rolandic opercula by CVAs is also relevant to the
notion of a different role of the fronto-parietal portions of the two hemispheres in speech.
It has been claimed that fronto-parietal lesions
of the non-dominant hemisphere, although massive, do not affect the efficiency of 'communication': they may impair 'articulation', but mildly and transiently. However, if a small frontoparietal lesion of the left hemisphere - - incapable in itself of producing aphasic disturbances
or severe articulatory defects - - follows this primary right hemisphere lesion, a very severe
'pseudobulbar dysarthria' takes place [6, 7]. On
this interpretation, our patient's first stroke,
though extensively involving the right hemisphere, only produced a very mild, transient
dysarthria; it was the second, less severe, contralateral stroke (with an ischemic lesion circumscribed to the rolandic operculum) that
caused suppression of speech, yet without aphasia.
A complete facio-pharyngo-glosso-masticatory
diplegia, rather than a pseudobulbar dysarthria,
occurred since the primary cortical motor areas
for the lower cranial portions were destroyed in
both hemispheres. Thus, the oft-reported dissociation between 'communicative' and 'articulatory' aspects of speech was displayed by our
patient in the highest degree.

Sommario

Viene descritta una paziente portatrice di due lesioni ischemiche coinvolgenti l'opercolo rolandico in
entrambi gli emisferi. II quadro clinico era caratterizzato da una compromissione selettiva dei movimenti
volontari (con integrit5 di quelli autornatiei e riflessi) bilateralmente a livello facio-faringo-glosso82

Villa G.: Speech suppression in bilateral rolandic strokes

masticatorio (sindrome di Foix-Chavany-Marie). La paziente, del tutto incapace di articolazione e
fonazione, non risultava afasica ad una valutazione neuropsicologica.
Questa dissociazione viene diseussa in riferimento alia particolare localizzazione delle lesioni alia
T.A. C. cerebrale.

Address reprint requests to: Dr. G i a m p i e r o Villa
Clinica N e u r o l o g i c a
Policlinico "A. G e m e l l i "
Largo A. Gemelli, 8 - - 00168 R o m a

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