Acute infarct of the corpus callosum presenting as
alien hand syndrome: evidence of diffusion weighted
imaging and magnetic resonance angiography
Yuan et al.
Yuan et al. BMC Neurology 2011, 11:142
http://www.biomedcentral.com/1471-2377/11/142 (9 November 2011)

Yuan et al. BMC Neurology 2011, 11:142
http://www.biomedcentral.com/1471-2377/11/142

CASE REPORT

Open Access

Acute infarct of the corpus callosum presenting
as alien hand syndrome: evidence of diffusion
weighted imaging and magnetic resonance
angiography
Jun Liang Yuan1, Shuang Kun Wang2, Xiao Juan Guo2 and Wen Li Hu1*

Abstract
Background: Infarcts of the corpus callosum are rare and have not been well documented previously. As for a
variety of signs and symptoms presented, alien hand syndrome (AHS) can be easily overlooked.
Case presentation: In this report, we present a patient with a mixed types of AHS coexistence secondary to the
corpus callosum infarction, including a motor type of AHS by intermanual conflict (callosal type AHS) and a sensory
type of AHS by alien hand and left hemianesthesia (posterior AHS).
Conclusions: Our case may contribute to the early recognition of AHS and to explore the abnormal neural
mechanism of AHS. To our knowledge, rare reports have ever documented such mixed AHS coexisting secondary
to the callosal lesion, based on advanced neuroimaging methods as in our case.

Background
Alien hand syndrome (AHS) is a rare clinical syndrome,
characterized by involuntary, uncontrollable and purposeless movements of one upper limb, and the patient denies
ownership of the limb when touching it without visual guidance [1]. Such movements are aimless and differentiable
from choreic and athetotic movements [2]. AHS is usually
considered as a type of interhemispheric disconnection
syndrome resulting from several lesions involving supplementary motor area, corpus callosum, medial frontal
cortex, frontal, posterior parietal [3], or thalamus [4].
However, the neural mechanisms of AHS have remained
controversial. Here, in this report, we describe a recent
case of mixed types of AHS secondary to the infarction of
the corpus callosum. Examinations of 3-Tesla magnetic
resonance imaging (MRI) and magnetic resonance angiography (MRA) were used to assist early diagnosis of such a
callosal infarct and to identify the abnormal neural processes of AHS. We briefly discuss the complex varieties of
AHS, their anatomic substrates and the underlying neural
* Correspondence: huwenli@sina.com
1
Department of Neurology, Beijing Chaoyang Hospital, Capital Medical
University, No. 8 South Gongti Road, Beijing 100020, China
Full list of author information is available at the end of the article

mechanisms. To our knowledge, rare reports have documented such mixed types of AHS coexistence secondary
to the corpus callosum infarction based on MRI, as
reported in our case.

Case presentation
A 71-year-old right-handed woman was admitted to our
hospital because of urgent onset of paraphasia and slight
weakness of her left hand for 13 days. The patient had a
20-year history of hypertension, coronary heart disease
and insulin-dependent diabetes mellitus, with no history
of drinking or smoking, and negative personal or family
history of neurological diseases. Educational history of the
patient was five years.
She complained of involuntary and purposeless movements of the left hand and abnormal feelings in the left
upper limb. She claimed that the left hand did not belong
to herself but to her grandson (alien hand). She also presented that her left hand was controlled by someone else
instead of herself, and the two hands always “fought” with
each other during bimanual coordination, as the left hand
performed movements opposite to the right hand (intermanual conflict). For instance, when she used her right
hand to unbutton her clothes, the left hand interfered by

© 2011 Yuan et al; licensee BioMed Central Ltd. This is an Open Access article distributed under the terms of the Creative Commons
Attribution License (http://creativecommons.org/licenses/by/2.0), which permits unrestricted use, distribution, and reproduction in
any medium, provided the original work is properly cited.

Yuan et al. BMC Neurology 2011, 11:142
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buttoning the clothes up. Also, she had visual hallucination at admission, such as her grandson standing beside
her bed. Although frustrated with her alien hand, she was
not afraid of her abnormal hand.
On her bedside mental examination, she had no symptoms of amnesia, disorientation, agraphia, tactile naming
in left hand, alexia in left visual field, hemispatial neglect
and hemiballismus. The scale of the Mini-mental state
examination was 24 when she was admitted to our hospital. She comprehended language well, but could not find
appropriate words to answer questions, frequently using
wrong words. Left hemiasomatognosia and apraxia were
also noted. For instance, she did not perceive the existence
of her left hand, with the abnormal perception that it was
her grandson’s hand (hemiasomatognosia). She had
apraxia when miming how to use tools with her left hand,
such as dressing, brushing teeth, combing her hair (ideomotor apraxia). She also had some difficulties in purposefully picking up objects (motor apraxia). A neurological
examination showed mild left hemiparesis with slight
clumsiness, and left hemianesthesia. Babinski sign was
positive on the left. Activities of daily living (ADLs) were
severely impaired, such as bathing, dressing and transferring, but the self feeding, bladder and bowel control were
not significantly affected. The impairment for ADLs may
be attributed to the left hemiparesis, apraxia, and intermanual conflict, with their disabling impact on everyday
life.
Carotid ultrasound demonstrated atherosclerosis plaques bilaterally in carotid artery. A brain 3-Tesla MRI was
performed 3 days after admission, including conventional
MR sequences: sagittal T1, axial T2-weighted, axial fast
fluid attenuated inversion recovery (FLAIR), along with
diffusion weighted imaging (DWI). Conventional
sequences of MRI indicated regions of low signal intensity
in the genu, body and splenium of the corpus callosum on
the T1 weighted image (Figure 1e) and high signal intensity in the same area on T2 weighted and FLAIR images
(Figure 1a-b). DWI demonstrated high signal intensity in
the observed T2 abnormality of the lesions, with corresponding restricted diffusion on the apparent diffusion
coefficient (ADC) maps (Figure 1c-d, Figure 2). MRA
showed the absence of the A1 segment of the bilateral
anterior cerebral artery (ACA). Atherosclerotic segment of
stenosis in the area of the bilateral middle cerebral artery
(MCA) and left posterior cerebral artery (PCA) were also
noted (Figure 1f).
Two weeks later after treatment (platelet aggregation
inhibitor and rehabilitation), the patient’s aphasia,
apraxia and hemiasomatognosia disappeared and ADLs
were significantly improved. At discharge, this patient
could perform routine activities independently and the
behaviors of AHS were completely resolved.

Page 2 of 5

Discussion
We described a recent case of an acute infarct of the corpus callosum presented with mixed types of AHS. AHS
was originally described by Brion and Jedynac in 1972,
characterized by the denial of ownership of one hand
when it was held by the other hand behind the back, due
to the lesions of the corpus callosum [5]. Two types of
AHS were proposed by Feinberg in 1992, including frontal
and callosal types [6]. Frontal AHS always occurs in the
dominant hand, characterized by reflexive grasping, groping, and compulsive manipulation of tools, and is usually
associated with frontal release signs. The supplementary
motor area, anterior cingulate gyrus, medial prefrontal
cortex and anterior corpus callosum have been described
in this type of AHS [7]. The second type or callosal AHS
is characterized primarily by the presence of intermanual
conflict and the absence of frontal release signs. This type
of AHS occurs in the non-dominant limb and may result
from only a corpus callosal lesion [8].
Based on previous classifications, Kato et al reported
three types of AHS [9]. The first and second types were
similar to the frontal and callosal type of AHS, as
described by Feinberg. The third type of AHS or posterior AHS was due to lesions in cortical and subcortical
structures supplied by the posterior cerebral artery, characterized by sensory impairment such as left hemianesthesia, left homonymous hemianopia and left spatial
neglect. The first two types of AHS are classified as an
“anterior” or “motor” form of AHS, whereas the posterior
AHS is considered as a “sensory” form of AHS. However,
Aboitiz et al strongly proposed that the cases of AHS
should be classified into at least five broad categories
[10]: diagonistic dyspraxia and related syndromes, alien
hand, way-ward hand and related syndromes, supernumerary hands, and agonistic dyspraxia.
Although different subtypes of AHS have been distinguished, these classifications obviously can not involve
the wide clinical variety of abnormal behaviors of the
upper limb. Up to now, the classifications of AHS are
still inconsistent and have been remain disputable. It is
regrettable because different pathophysiological mechanisms probably underlie the diverse behaviors in alien
hand. As for lack of uniformity in assessment methods
(behavioral tests, neuroimaging), Scepkowski indicated it
may be difficult to establish clear subtypes of alien-hand
phenomena [11].
Our patient in this report presented with mixed types of
AHS, including a motor type of AHS by the presence of
intermanual conflict (callosal type of AHS) and sensory
type of AHS by sensory impairment such as alien hand
and left hemianesthesia (posterior AHS). The neuroanatomic substrate of AHS and its explanatory models have
remained controversial due to the noncircumscribed

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Page 3 of 5

Figure 1 Multi-modal brain MRI. Axial T2-weighted (a) and FLAIR images (b) depicted T2 hyper-intensity and sagittal T1-weighted MRI (e)
showed hypo-intensity in the genu, body and splenium of the corpus callosum. Axial DWI demonstrated high signal intensity in the observed
T2 abnormality of the lesions of the corpus callosum (c). Corresponding ADC map illustrated matched abnormality, confirming restricted
diffusion in the corpus callosum (d). MRA showed the absence of the A1 segment of the bilateral anterior cerebral artery. Atherosclerotic
segment of stenosis in the area of the bilateral middle cerebral artery and left posterior cerebral artery were noted (arrows) (f).

nature of cerebral injuries present in most cases [12].
Several hypotheses have been suggested, but the most
accepted one seems to be the disconnection syndrome
[13,14], secondary to the lesions in the medial frontal lobe,

the supplementary motor area, anterior cingulate gyrus,
medial prefrontal cortex, anterior corpus callosum and
posterior parietal lobe. Our patient demonstrated alienation and loss of voluntary control of the left hand, which is

Yuan et al. BMC Neurology 2011, 11:142
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Page 4 of 5

Figure 2 Axial DWI showed high signal intensity in all the slices of the lesions of the corpus callosum.

consistent with the above anatomical explanation. Our
case also provides supportive evidence for damage to the
corpus callosum as the anatomic basis of nondominant
AHS and conforms to a model of interhemispheric
disconnection as the essential component of this unusual
behavioral syndrome. We emphasize the importance of
distinguishing different features of abnormal behaviors in
AHS because it may result in different treatment strategies
[15]. However, its significance as a sign of disconnection
syndrome is not well validated and needs further
investigation.
Neuroimaging studies in our case identified the responsible lesions for the interhemispheric disconnection syndrome in the corpus callosum. This report is explicit in
manifestations and the neuroimaging results. The cause

of this infarct in our case was possibly due to the atherosclerotic fibrous plaques of the carotid artery, with longstanding hypertension, coronary heart disease and diabetes mellitus of 20 years. As a result, the collective evidences of DWI, MRA should have the advantages of
differentiating infarction from other pathologies. Meanwhile, the combination of modern advanced imaging
techniques makes the accurate diagnosis of corpus callosal infarction possible, biopsy not necessary any more
[16]. In addition, more comprehensively neuropsychological tests are needed to evaluate the brain function. The
combinations of functional MRI (such as task-related and
resting state fMRI), CT angiography (CTA), CT perfusion
(CTP), perfusion weighted imaging (PWI), magnetic
resonance spectroscopy (MRS), and digital subtraction

Yuan et al. BMC Neurology 2011, 11:142
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angiography (DSA) along with follow-ups should be
employed as supplement to the original studies.

Page 5 of 5

5.

6.

Conclusions
In conclusion, infarcts of the corpus callosum are not
common, which is most likely due to its rich blood supply from three main arterial systems, specifically the anterior cerebral, anterior communicating, and posterior
cerebral arteries [17]. The isolated infarcts of the anterior
cerebral arteries are uncommon (Figure 1f), accounting
for only 0.6% of all ischaemic infarcts [18]. As a result,
the patient described in our case is unique in exhibiting a
complete corpus callosum infarction with a mixed types
of AHS, including a motor type of AHS by intermanual
conflict (callosal type AHS) and a sensory type of AHS
(posterior AHS) by alien hand and left hemianesthesia.
To our knowledge, rare reports have ever documented
such mixed AHS coexisting secondary to the callosal
lesion without evidence of cortical involvement, based on
advanced neuroimaging methods as in our case.
Consent
Written informed consent was obtained from the patient
for publication of this case report and accompanying
images. A copy of the written consent is available for
review by the Editor-in-Chief of this journal.

7.
8.
9.

10.

11.
12.

13.
14.

15.

16.

17.

18.
Acknowledgements and Funding
The authors thank this patient for her participation in our study.
Author details
1
Department of Neurology, Beijing Chaoyang Hospital, Capital Medical
University, No. 8 South Gongti Road, Beijing 100020, China. 2Department of
Radiology, Beijing Chaoyang Hospital, Capital Medical University, No. 8 South
Gongti Road, Beijing 100020, China.
Authors’ contributions
JLY examined, evaluated the patient and drafted the manuscript. SKW and
XJG performed and interpreted the MRI studies. WLH participated in the
design of the case-report and helped to draft the manuscript. All authors
read and approved the final manuscript.

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Pre-publication history
The pre-publication history for this paper can be accessed here:
http://www.biomedcentral.com/1471-2377/11/142/prepub
doi:10.1186/1471-2377-11-142
Cite this article as: Yuan et al.: Acute infarct of the corpus callosum
presenting as alien hand syndrome: evidence of diffusion weighted
imaging and magnetic resonance angiography. BMC Neurology 2011
11:142.

Competing interests
The authors declare that they have no competing interests.
Received: 18 July 2011 Accepted: 9 November 2011
Published: 9 November 2011
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