Clinical Neurology and Neurosurgery 110 (2008) 190–193

Case report

Involuntary masturbation and hemiballismus after bilateral
anterior cerebral artery infarction
Yannick Bejot a,∗ , Marie Caillier a , Guy-Victor Osseby a , Roy Didi b ,
Douraied Ben Salem c , Thibault Moreau a , Maurice Giroud a
a Department of Neurology, University Hospital of Dijon, France
b Department of Psychiatry, Psychiatric Hospital of Dijon, France
c Department of Neuro-imaging, University Hospital of Dijon, France

Received 10 July 2007; received in revised form 11 September 2007; accepted 14 September 2007

Abstract
Ischemia of the areas supplied by the anterior cerebral artery is relatively uncommon. In addition, combined hemiballismus and masturbation
have rarely been reported in patients with cerebrovascular disease. We describe herein a 62-year-old right-handed man simultaneously
exhibiting right side hemiballismus and involuntary masturbation with the left hand after bilateral infarction of the anterior cerebral artery
territory. Right side hemiballismus was related to the disruption of afferent fibers from the left frontal lobe to the left subthalamic nucleus.
Involuntary masturbation using the left hand was exclusively linked to a callosal type of alien hand syndrome secondary to infarction of the
right side of the anterior corpus callosum. After 2 weeks, these abnormal behaviours were completely extinguished. This report stresses the
wide diversity of clinical manifestations observed after infarction of the anterior cerebral artery territory.
© 2007 Elsevier B.V. All rights reserved.
Keywords: Hemiballismus; Masturbation; Anterior cerebral artery; Stroke; Corpus callosum; Alien hand syndrome

1. Introduction
Infarction of the areas supplied by the anterior cerebral
artery is relatively uncommon, accounting for approximately
4% of all ischemic strokes [1,2]. Clinical features vary
according to the exact site of the occluded branches and the
spread of the ischemic lesion, but the most frequent signs
reported are contralateral crural monoplegia or hemiplegia
with predominant lower extremity involvement, forced grasping or groping of the ipsilateral upper limb, ideomotor apraxia
of the left arm, and dynamic aphasia, regardless of the side
of the anterior cerebral artery occlusion [3].
Hemiballismus and masturbation have rarely been
observed in patients with cerebrovascular disease. We report
herein the case of a patient with bilateral lesions in the vas-

∗ Corresponding author at: Dijon Stroke Registry (INSERM), Service de
Neurologie, CHU, 3 Rue du Faubourg Raines, 21000 Dijon, France.
Tel.: +33 3 80 29 37 53; fax: +33 3 80 29 36 72.
E-mail address: ybejot@yahoo.fr (Y. Bejot).

0303-8467/$ – see front matter © 2007 Elsevier B.V. All rights reserved.
doi:10.1016/j.clineuro.2007.09.007

cular territory of the anterior cerebral arteries who exhibited
these two very rare symptoms simultaneously.

2. Case report
A 62-year-old right-handed man was admitted to our
stroke unit because he had experienced difficulties in moving his left leg at night when he was trying to stand up. He
had controlled hypertension, was a current smoker, and did
not have a history of cerebrovascular accidents or alcohol
abuse. He was married and had two children. He was a retired
skilled worker. When he was examined 5 h after the onset of
the symptoms, he was oriented. He had paresis of the left
lower extremity, predominant in the proximal segment with
less strength than antigravity, in contrast with only a slight
deficit in the left upper extremity. Strength was normal on
the right side. Deep tendon reflexes were normal, but plantar
responses were extensor in both feet. Examination of sensory
and cranial nerves did not reveal any abnormalities. He had

Y. Bejot et al. / Clinical Neurology and Neurosurgery 110 (2008) 190–193

neither cerebellar ataxia nor dysmetria, but he presented with
intermittent abnormal movements of his right side. They were
vigorous, rapidly executed, asynergic, complex and combined, and affected predominantly the proximal segments
of the right upper and lower extremities. The face and neck
were not involved. Right hemiballismus was evoked. In addition, his contralateral hand presented anarchic involuntary
movements, leading to difficulties in bimanual tasks, such
as eating or manipulating objects. Moreover, he exhibited
repeated exposure of his genitals and masturbation with his
left hand and affirmed that this behaviour was not under his
control. He was very self-critical with regard to this activity,
and according to his family, he had never experienced such
behaviour in the past. Further examination revealed bilateral forced grasping and the smooth and complex courses of
skilled movement were disturbed as were kinetic melodies.
Neuropsychologic assessment revealed marked reduction in
spontaneous speech with normal comprehension and repetition. There were difficulties in initiating talking, but oral
denomination, writing, and reading were normal. The intelligence quotient, as evaluated by a seven-subtest short form
of the Weschler Adult Intelligence Scale-Revised (WAISR) [4], was in the average range. The Mini-Mental State
Examination [5] score was 28/30. Anterograde and retrograde memory skills were normal. The Frontal Assessment
Battery (FAB) [6] total score was 12/18; he had difficulties in
motor programming and executive control of action (score:
2/3), resistance to interference (score: 2/3), inhibitory control (score: 1/3), and environmental autonomy (score: 1/3),
whereas conceptualization, abstract reasoning, and mental
flexibility were normal. Left ideomotor dyspraxia was noted,
whereas left agraphia, left visual, tactile anomia, and constructive apraxia were not observed. Both social cognition
and the patient’s judgment assessment did not reveal any
impairment.
Magnetic resonance imaging was then performed and
showed bilateral hyperintense signals in diffusion-weighted
images in the anterior cerebral artery territories involving the
right anterior corpus callosum, suggesting a hemodynamic
mechanism (Fig. 1). Trans-oesophageal echocardiography

191

was performed, but no cardio-embolic sources were detected.
No rhythm disturbances were apparent either on the repeat
EKG or on the Holter recording. Carotid and vertebral ultrasonography showed diffuse atheromas with no significant
arterial stenosis. Blood tests were normal. The patient was
admitted to the rehabilitation center after 2 weeks and his
abnormal movements and masturbation did not persist.

3. Discussion
The most interesting point of our observation is the association of two symptoms that are rarely described in ischemia
of the anterior cerebral artery: (1) hemiballismus and (2)
involuntary masturbation.
Hemiballismus is a rarely reported hyperkinetic disorder secondary to cerebrovascular pathologies with an
estimated annual incidence in the general population of
0.45/100,000 [7], accounting for 0.4% of all strokes [8].
The vascular lesion usually involves the contralateral subthalamic nucleus [9]. In the patient described herein, there
were no lesions in this cerebral area on diffusion-weighted
MRI. Nevertheless, hemiballismus has also been reported
after lesions involving the frontal and parietal lobes, the
thalamus, or the corona radiata [8,10,11]. From a pathophysiologic point of view, the disruption of the balance
between the direct (cortex–caudate–internal pallidum) and
the indirect (cortex–caudate–external pallidum–subthalamic
nucleus–internal pallidum) pathways modulating the glutamatergic activator thalamocortical retroactive pathway by
such lesions could be responsible for the induction of
hemiballismus, explaining these clinical features in our
patient [12].
The onset of involuntary masturbation following a cerebrovascular lesion is not common, whereas it is often
observed in patients with temporal lobe epilepsy [13]. Hypersexuality has been reported after bilateral thalamic infarction
associated with frontal hypoperfusion in SPECT imaging,
suggesting dysfunction of frontal–subcortical circuits [14].
Moreover, cases of hypersexuality have been described as

Fig. 1. Diffusion-weighted magnetic resonance imaging showing hyperintense signals over the right anterior corpus callosum and the two frontal lobes related
to bilateral anterior cerebral artery infarction.

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Y. Bejot et al. / Clinical Neurology and Neurosurgery 110 (2008) 190–193

part of an orbitofrontal syndrome [15]. Nevertheless, whereas
our patient exhibited symptoms, such as a disturbance of
skilled movements, forced grasping, a marked reduction in
spontaneous speech, or apathy, and had a pathologic FAB
score, we could not attribute the occurrence of masturbation to a loss of control of social behaviour in the case of a
frontal syndrome. Indeed, this right-handed patient always
used his left hand to masturbate, and this act appeared to
be independent of the patient’s will. Moreover, he was selfcritical with regard to this involuntary compulsive behaviour
and felt alienation, suggesting that it could be related to
a motor alien hand syndrome. Two types of motor alien
hand syndrome have been distinguished by Della Sala et
al. [16]. The first and most frequent affects the dominant
hand, the so-called “frontal alien hand syndrome,” and is
characterized by reflexive grasping, groping, and compulsive manipulation of tools, and results from damage to the
supplementary motor area, the anterior cingulate gyrus, the
medial prefrontal cortex, and the anterior corpus callosum of
the dominant hemisphere. The second type, or “callosal type,”
is described in patients suffering from lesions involving the
corpus callosum with or without associated frontal lesions of
the non-dominant hemisphere, especially in the supplementary motor area. Clinically, intermanual conflict and apraxia
of the non-dominant limb are observed. A unique case of
involuntary masturbation associated with intermanual conflict and apraxia secondary to ischemia of the right medial
frontal lobe and anterior corpus callosum on imaging similar
to that observed in our patient has been reported [17]. Like
that noted in our patient, this activity was limited to the left
hand and consequently, it can be regarded as a manifestation of a callosal type of alien hand syndrome. Classically,
other signs of callosal disconnection, such as left ideomotor
apraxia, left agraphia, left visual, or tactile anomia can be
found. We did not observe any such manifestations except
for left ideomotor apraxia.
Infarction of the anterior cerebral artery territory is
unusual, accounting for only 1.3–4% of all strokes [1,2,18]
and bilateral infarction in this territory is very rare, affecting only 4% of all anterior cerebral artery infarcts [18]. Very
few cases of bilateral infarction have been reported in the
literature and neuropsychological evaluation has not been as
extensive as the description of neurological signs. Hence, the
classic picture including akinetic mutism, paraplegia, incontinence, bilateral grasp reflex, and amnesia with apathy [18,19].
Boccardi et al. reported the case of a patient suffering from
bilateral infarction that damaged the supplementary motor
areas and who presented severe utilization behaviour [20].
They considered this pathologic behaviour as a double anarchic hand due to damaged balance between the premotor
cortices, responsive to environmental triggers, and the supplementary motor areas, which modulate actions and inhibit
them. Some authors suggested that bilateral frontal lesions
may cause an imbalance between impaired frontal and intact
parietal lobes, and released activity of parietal lobes may
yield to utilization behaviour and environmental-dependency

syndrome [21]. Bilateral lesions of the supplementary motor
areas have also been associated with gait apraxia, suggesting that these regions are required for the complexity of the
mostly automatic gait movements [22]. In a unique detailed
neuropsychological observation, Bird et al. observed a dysexecutive syndrome, characterized by a severe planning and
prospective memory impairment associated with confabulatory and amnestic syndrome, following bilateral medial
frontal lobe infarction, but they failed to demonstrate that this
region plays a critical role in the “Theory of Mind” network
in the human brain [23]. Neither involuntary masturbation
nor hemiballismus have been reported after bilateral anterior
cerebral artery infarcts.

4. Conclusion
To our knowledge, our patient is the first case of bilateral
lesions involving the anterior cerebral artery territory presenting with two rare symptoms simultaneously. The right
side hemiballismus was related to disruption of afferent fibers
of the left subthalamic nucleus from the left frontal lobe,
whereas the involuntary masturbation with the left hand was
linked to a callosal type of alien hand syndrome secondary
to infarction of the right side of the anterior corpus callosum.

Conflict of interest
None.

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