Brain Injury

ISSN: 0269-9052 (Print) 1362-301X (Online) Journal homepage: http://www.tandfonline.com/loi/ibij20

Case Study: Biopercular lesions and acquired
mutism in a young patient
Zeev Groswasser, Irith Groswasser-Reider & Cecilia Korn
To cite this article: Zeev Groswasser, Irith Groswasser-Reider & Cecilia Korn (1991) Case Study:
Biopercular lesions and acquired mutism in a young patient, Brain Injury, 5:3, 331-334, DOI:
10.3109/02699059109008104
To link to this article: http://dx.doi.org/10.3109/02699059109008104

Published online: 03 Jul 2009.

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BRAIN INJURY, 1991, VOL. 5, NO. 3,331-334

Case study

Biopercular lesions and acquired
mutism in a young patient

Downloaded by [McMaster University] at 14:55 07 April 2016

ZEEV G R O S W A S S E R t , I R I T H
G R O S W A S S E R - R E I D E R S and C E C I L I A K O R N t
tLoewenstein Rehabilitation Hospital Ra’anana and $Department of
Radiology/Section of Neuroradiology, Tel-Aviv Medical Center, Ichilov Hospital,
Sackler Faculty of Medicine, Tel-Aviv University, Israel
(Received 1 0 September 1990; accepted 18 October 1990)
A 13-year-old patient developed complete mutism and buccofscial apraxia following toxic vasculitis
due to a yellow scorpion sting. Language functions were preserved. A CT scan disclosed mainly
biopercular infarcts. It is suggested that the lost control of vocalization and speech is associated with
biopercular lesions and that a preserved right opercular region can take over this function in the
presence of damage to homologous left opercular region.

Introduction
The clinical phenomenon of long-standing mutism, defined as loss of vocalization and
speech, without involvement of language hnctions, due to lesions in the central nervous
system (CNS) has attracted considerable attention and has been described under many
names [l-91.
Most of the reported patients acquiring the syndrome were middle-aged or older and
had suffered &om an underlying vascular disease. Ths paper reports a case of a 13-year-old
patient who developed complete mutism with buccohcial apraxia (BFA) following vasculitis due to a toxic response to a scorpion bite, causing biopercular infarcts.
Case history

Patient presentation

A 13-year-old beduin boy was bitten by a yellow scorpion (Leirus quinquestriatus) on his
left hand. O n admission to hospital an hour and a half later, he was conscious but restless
and showed multiple toxic manifestations including di&se cold sweat, excessive sahvation,
vomiting, priapism and mydriasis. Blood pressure was 70/55. Therapy commenced
immediately with diazepam, atropin and fluids. Soon after, his temperature rose to 39.5OC,
he became unconscious and responded only to painful stimuli. Bilateral Babinski signs
were noted and the patient developed pulmonary oedema and respiratory failure and had
Address correspondence to: Dr Z. Groswasser, Loewenstein Rehabilitation Hospital, PO Box
3, Ra’anana 43100, Israel.
0269-9052/91 $3.00 0 1991 Taylor & Francis Ltd.

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Zeev Groswasser et al.

to be intubated and artificially ventilated. Cardiogenic shock appeared due to ventricular
tachycardia in spite of the prophylactic administration of lidocain. CT performed on the
third day showed patchy areas of cortical damage. Consciousness recovered gradually on
the fourth day and it became evident that the patient was unable to speak.
O n the fifih hospital day, a demarcation line appeared 7 cm above his right ankle.
Pulses were palpable at the right popliteal artery, but not below this level. Heparin was
started but the foot became gangrenous and a below-knee amputation was performed on
the 39th hospital day. Flexion contracture of the right knee developed in spite of intensive
physiotherapy and the stump did not heal, therefore an above-knee reamputation was
performed.
The patient was referred for rehabilitation during the 1l t h week of his illness. Physical
examination was normal apart from the above-knee amputation on the right. O n neurolog~calexamination he was conscious, alert and co-operative, obeyed commands but could
not speak. Mild central palsy of the right facial nerve was noted; gag reflex was elicited on
both sides. No other defects were found in the motor apparatus subserving speech production. The movements of the vocal cords were good. Some weakening of the right hand
was found, but tendon reflexes were normal and symmetrical, pyramidal signs were
negative. Sensation was normal and no extrapyramidal signs were found.
A CT scan performed at 4 months disclosed bilateral frontal opercular infarction
around the temtory of the prefrontal and insular branches of the middle cerebral arteries.
Widened ventricles and subarachnoid spaces were also noted (Figure 1).

Fkure 1 . Bilateraljiontal opercular infarcts in the tem.tories of the insular and prejiontal branches of the middlc
cerebral arteries. Widened ventricles and rubarachnoid spaces are noted.

Biopercular lesions and acquired mutism

333

Communication studies

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The patient was mute, but his receptive functions were intact as evaluated on tests for
aphasia, based on Eisenson’s test and the Boston Aphasia test. The patient communicated
effectively through gestures. His spontaneous orofacial movements were impaired in
range, especially the lateral movements of the tongue. He was unable to perform any
movement properly on command. He had swallowing difficulties and his co-ordination
was impaired. Severe disturbances were found when tested for BFA according to Tognola
and Vignolo [lo].
The patient was followed for 6 months as an in-patient; improvement of hs spontaneous tongue movements occurred, but his BFA did not change. On discharge, he was able
to laugh, cough and produce a few isolated vowels. The patient’s condition 2 years later
had not improved.

Discussion
The phenomenon of lasting mutism associated with BFA following brain damage without
any sign of language impairment are h r l y rare and of considerable theoretical interest.
Lecours et al. [ l l ] assumed that it could be attributed to damage in a specific neuronal
network which is functionally independent tiom the other cerebral systems underlying
speech and language. The role of bilateral opercular lesions in the development of this
specific clinical picture was clearly demonstrated by Cappa et al. [l],whose patient clearly
showed that only the second infarction, affecting the homologous opercular region of the
non-dominant right hemisphere, resulted in the clinical image of speechlessness and BFA
without any signs of aphasia, similarly to patient F1 described by Groswasser et al. [4].
Both patients were initially described as aphasics whose aphasia resolved completely within
a few weeks. Only the second stroke, affecting the non-dominant hemisphere, rendered
them mute.
Our patient had several specific features. He is, as far as we know, the youngest patient
ever reported to have acquired this syndrome having no underlying cardiovascular disease,
which is the most common underlying dsease [6]. The patient suffered fiom difise vasculitis and thrombotic phenomena as evidenced by brain infarcts in the territories of the prefrontal and insular branches of the middle cerebral artery and in his right leg. The
cardovascular and autonomic disturbances following scorpion stings are well known but,
to the best of our knowledge, diffuse vascdtis which led to the thrombosis of cerebral
vessels, has not previously been reported.
The symmetrical bilateral involvement of the frontal operculum are responsible for the
clinical picture of mutism and BFA in this patient. Patient F1 (described by Groswasser et
al. [4]) and the patient described by Cappa et al. [l] demonstrate clearly the importance of
the non-dominant right hemisphere in the development of a clinical picture identical to
that of the present patient. The CT findings of these three patients are similar.
It therefore seems reasonable to asume that the permanent picture of mutism and BFA,
without any other manifestations such as language impairment, may have developed in the
present patient due to the homologous involvement of both opercular regions. The fact
that the right frontal operculum was affected probably did not allow this region to take
over successfully and efficiently the control of speech production, usually performed by
the lefi hemisphere as was the case in the first stage of illness in the other two patients.

References
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