SUPERNUMERARY PHANTOM LIMBS ASSOCIATED WITH
LEFT HEMISPHERIC STROKE: CASE REPORT AND REVIEW
OF THE LITERATURE
Nobuhiko Miyazawa, M.D.
Department of Neurosurgery,
Akiyama Neurosurgical Clinic,
Yamanashi, Japan

Masataka Hayashi, M.D.
Department of Neurology, Kofu
City Hospital, Yamanashi, Japan

Keiji Komiya, M.D.
Department of Rehabilitation, Kofu
Johnan Hospital, Yamanashi, Japan

Iwao Akiyama, M.D.
Department of Neurosurgery,
Akiyama Neurosurgical Clinic,
Yamanashi, Japan
Reprint requests:
Nobuhiko Miyazawa, M.D.,
Department of Neurosurgery,
Akiyama Neurosurgical Clinic,
1330 Wakao Ohkusa-cho, Nirasaki
City, Yamanashi 407-0037, Japan.
Email: akinouge@crux.ocn.ne.jp
Received, March 8, 2003.
Accepted, August 28, 2003.

OBJECTIVE AND IMPORTANCE: Supernumerary phantom limb (SPL) is extremely
rare. Literature reports noted 17 cases that occurred after right cerebral hemispheric
stroke and 2 cases that occurred after left cerebral hemispheric stroke, but without
imaging diagnoses.
CLINICAL PRESENTATION: A 45-year-old male patient complained of SPLs on the
right side after recurrent left thalamic hemorrhage. Computed tomography and magnetic resonance imaging demonstrated the lesion causing the left hemispheric stroke.
INTERVENTION: The patient was treated conservatively. Computed tomographic
scans demonstrated that the lesion had disappeared by 15 days after admission. The
sensation of SPLs disappeared after 28 days.
CONCLUSION: SPL may occur among patients with left hemispheric stroke, especially those with lesions in the thalamus, spastic paresis on the right side immediately
after stroke, and psychiatric disorders such as alcohol and tobacco intoxication.
KEY WORDS: Cerebral infarction, Intracerebral hemorrhage, Supernumerary phantom limb
Neurosurgery 54:228-231, 2004

DOI: 10.1227/01.NEU.0000097558.01639.F5

T

he phantom limb phenomenon commonly occurs after surgical or traumatic
amputations, in as many as 90 to 100%
of cases (8, 16). In contrast, the equivalent
phenomenon caused by cerebral lesions,
which is called supernumerary phantom limb
(SPL) (4) or phantom third limb (8, 16), is
extremely rare; it was first noted after right
hemispheric stroke (6) or left hemispheric
stroke (18), but without imaging diagnoses.
SPL may be caused by lesions in the right or
left cerebral hemisphere (7), but a predominance of right hemispheric stroke has been
reported (3, 10, 11). Here we describe a case of
SPL after left hemispheric hemorrhage, which
was identified on computed tomographic (CT)
and magnetic resonance imaging scans, and
we discuss the clinical and radiological
features.

CASE REPORT
A 42-year-old man was hospitalized on
March 20, 1999, because of right hemiparesis
and dysarthria. His history included alcohol
intoxication, excessive smoking, liver dysfunction attributable to alcohol consumption,
hypertension, hyperuremia, diabetes mellitus,
and hyperlipidemia. The patient had been

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treated medically, but his compliance was unusually poor. A neurological examination revealed consciousness disturbances, motor
aphasia, right hemiparesis involving the face
(spastic paresis), and superficial sensory disturbances, including an absence of sensitivity
to pain, temperature, and light touch (almost
anesthesia). No profound sensory disturbances, such as positional sense deficits, vibrational sense deficits, hemianopia, or right-side
neglect, were observed. CT scans obtained at
the time of admission revealed an area of left
putaminal hemorrhage measuring 3 â«» 3.5 cm
(Fig. 1A). The patient was treated conservatively, with hyperbaric therapy.
Two days after admission, the patient became alert and could report his hemiparesis.
He experienced kinetic hallucinations (movement of the right extremities) and macrosomatagnosia (ballooning of the face and body
on the right side, especially the face) 5 days
after admission. These phenomena tapered
and disappeared by 10 days after admission.
The symptoms gradually improved, and the
patient became independent in daily life, with
slight difficulty in the fine movements of the
fingers of the right hand. He was referred to a
rehabilitation hospital on May 22, 1999.

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SUPERNUMERARY PHANTOM LIMBS WITH HEMISPHERIC STROKE

FIGURE 1. A, CT scan obtained at the time of the first admission, showing a left putaminal area of hemorrhage measuring 3 â«» 3.5 cm. B, CT
scan obtained at the time of the second admission, revealing a left thalamic
area of hemorrhage measuring 1.5 â«» 2 cm. C, coronal magnetic resonance
imaging scan, showing left posteromedial thalamic hemorrhage. D, drawing of the SPLs by the patient. The abnormal upper and lower limbs
emerged inside the original limbs, and the distal SPLs were not clearly
drawn.

The patient experienced an onset of anesthesia on the right
side of the body and worsening of right hemiparesis after he
was surprised by a fire surrounding his garden on November
24, 2001. He was transported to our clinic by ambulance and
was immediately hospitalized. He was almost alert, with
anisocoria. A neurological examination demonstrated motor
aphasia, right hemiparesis (severe spastic paresis), right anesthesia to both superficial and deep sensations, and autotopagnosia (agnosia of his own body parts) on the right side of the
body without anosognosia (agnosia of hemiparesis). Axial CT
scans demonstrated a high-density area extending from the
thalamus to the posterior limb of the internal capsule in the
left hemisphere, measuring 1.5 â«» 2 cm (Fig. 1B), which was
considered to represent thalamic hemorrhage.
Two days after admission, the patient complained of complex visual hallucinations in his hemianoptic field (a number
of flying objects). He also complained of severe spasticity in
the right extremities. Three days after admission, he was alert
and fully oriented, and the visual hallucinations and hemianopia had disappeared. However, the patient felt the presence of another set of right upper and lower extremities, inside
the original extremities (Fig. 1D). He explained that he could
clearly see another upper arm, forearm, upper leg, and lower
leg but no hand or foot. He became upset when asked about

NEUROSURGERY

his phantom limbs. He did not feel the phantom limbs when
he used the left extremities. Five days after admission, axial
magnetic resonance imaging scans revealed a high-intensity
area extending from the thalamus to the internal capsule,
measuring 2.1 â«» 4 cm, and coronal and sagittal magnetic
resonance imaging scans demonstrated that the core of high
intensity was located in the ventroposterior medial and lateral
nuclei (Fig. 1C). Magnetic resonance angiography demonstrated no vascular abnormalities, such as an arteriovenous
malformation.
Fifteen days after admission, CT scans demonstrated that
the high-density area in the thalamus had completely disappeared. Seventeen days after admission, single-photon emission CT scans revealed hypoperfusion in the posterior thalamus, putamen, subcortical region, parietal cortex (including
the sensory cortex and motor cortex), and corpus callosum in
the left hemisphere and also indicated crossed cerebellar diaschisis. Cerebral blood flow in the middle cerebral artery
territory was 52 ml/100 g/min in the right hemisphere and 45
ml/100 g/min in the left hemisphere. Although the phantom
lower extremity had disappeared by 15 days after admission,
the patient experienced severe pain in the phantom upper
extremity 18 days after admission. The phantom upper extremity gradually tapered (telescoping), and it disappeared
completely 28 days after admission. No impairments of the
visual field and acuity or hearing were noted. Electroencephalography demonstrated no abnormalities. The patient was
transferred to the rehabilitation hospital on December 22,
2001.
After discharge from the rehabilitation hospital, the patient
was treated medically at another clinic. His compliance with
the medication regimen was poor. The patient drank volumes
of alcohol similar to those consumed previously. He complained of severe pain in the right upper arm and the spastic
paresis continued, but the muscle tone was more relaxed than
at the time of the second admission. No more episodes of SPL
occurred, but the patient clearly remembered the SPLs on the
right side during the second admission.

DISCUSSION
The 20 cases of SPL attributable to cerebral stroke that have
been reported in the past 70 years, including our case, are
summarized in Table 1 (1–7, 9–15, 18–21). The 14 male and 6
female patients were 35 to 80 years of age (mean, 58.1 yr).
Right cerebral hemispheric stroke was the cause in 17 cases
(2–7, 9–15, 19–21), and left hemispheric stroke was the cause in
only 3 cases (1, 18). A 69-year-old female patient experienced
SPL on the right side after encephalomalacia at the left claustrum to the internal capsule (18). Her right arm lay on her
chest, and her hallucination was that she lifted her right arm
20 cm. A 60-year-old male patient experienced SPL on the
right side after a cerebral infarction in the left thalamohypothalamic region (1).
The causative event, as demonstrated with neuroimaging,
was subarachnoid hemorrhage in one case (12), ischemic

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MIYAZAWA ET AL.

TABLE 1. Reports of supernumerary phantom limbs attributable to cerebral strokea
Side of stroke
Left

Right

With imaging diagnosisb

Without imaging diagnosis
Series (ref. no.)

Age (yr)/sex

Pinéas, 1932 (18)

69/F

Bogaert, 1934 (1)

60/M

Ehrenwald, 1930 (6)

Series (ref. no.)

Age (yr)/sex

Diagnosis

Present case, 2002

45/M

ICH

59/M

Hohjoh et al., 1989 (14)

49/M

ICH

Hecaen et al., 1952 (13)

64/M

Halligan et al., 1993 (11)

65/M

ICH

Gilliatt and Pratt, 1952 (9)

62/F

Halligan and Marshall, 1995 (10)

80/M

CI

Critchley, 1953 (4)

52/M

Sellal et al., 1996 (19)

77/M

CI

Unknown / M

a
b

Weinstein et al., 1954 (20)

57/F

Worthington and Beevers, 1996 (21)

72/F

CI

Brock and Merwarth, 1956 (2)

58/M

Donnet et al., 1997 (5)

35/M

ICH

Fredericks, 1963 (7)

36/M

Mazzoni et al., 1997 (15)

66/M

CI

Hari et al., 1998 (12)

37/F

SAH

Canavero et al., 1999 (3)

61/F

ICH

ICH, intracerebral hemorrhage; CI, cerebral infarction; SAH, subarachnoid hemorrhage.
Based on computed tomographic and/or magnetic resonance imaging findings.

stroke in four cases (10, 15, 19, 21), and intracerebral hemorrhage in four (3, 5, 11, 14). Our case was interesting because no
SPL occurred after the first hemorrhagic episode, whereas the
SPLs persisted for a rather short period after the second hemorrhagic episode.
Alcohol intoxication was noted in the histories of four patients, including our patient (5, 11, 14). A 49-year-old male
patient experienced SPL after right putaminal hemorrhage
(14). He had a history of alcohol intoxication and was hospitalized and treated. A 65-year-old male patient experienced
SPL after a right basal ganglia hematoma (11). He also had a
history of excessive alcohol consumption, which had ceased 3
years before admission. A 35-year-old male patient experienced SPL after a right capsulolenticular hematoma (5). He
also had a history of alcohol and tobacco intoxication. Our
patient had demonstrated severe alcohol and tobacco intoxication for nearly 20 years and was drinking two bottles of
whiskey every night and smoking 60 cigarettes every day.
These psychiatric factors might have influenced the occurrence of SPL.
Cases of SPL on the left may present with various degrees of
sensory disturbances, such as sensory loss (11), impairment of
sensitivity to light touch and proprioception (15), left hemisensory disturbances in all modalities (5), and tactile-pinprick
anesthesia on the left (3). Previous cases of SPL on the right
involved no precise description of sensory disturbances (1)
and sensory disturbances less severe than anesthesia (18). In
our case, severe anesthesia to superficial and profound stimuli

230 | VOLUME 54 | NUMBER 1 | JANUARY 2004

was present; therefore, whether any sensory disturbance is
characteristic of SPL remains unknown.
All three patients with left hemispheric stroke experienced
severe spastic right hemiparesis. Previous patients exhibited
spastic paralysis of the right arm (18) and left spastic hemiplegia (1). Our patient exhibited severe spastic paresis in the
right arm and leg, which continued during the episode of SPL.
The development of SPL may be determined more by motor
loss than by sensory loss (3, 11, 19). Therefore, spastic hemiparesis may be involved in SPL.
The main site of damage was the lenticular nucleus in cases
with right hemispheric hemorrhage causing left SPL (5, 11, 14)
and the middle cerebral artery territory in cases with right
hemispheric infarction (10, 15, 19, 21). Thalamic damage as the
cause of left SPL was rare (3, 10, 11). In contrast, autopsies in
two cases of left hemispheric stroke causing right SPL revealed that the main sites of damage were the thalamus and
internal capsule (1, 18). In our case, the first episode of hemorrhage (in the putamen) was not associated with SPL,
whereas the second episode of hemorrhage (in the thalamus)
resulted in SPL.
The reason why SPL is more likely to be associated with
right hemispheric stroke than left hemispheric stroke remains
unclear. This association may occur partly because of the
functional differences between the right and left hemispheres
and partly because of the prevalence of right hemispheric
stroke. The right hemisphere seems to be involved in controlling sensorimotor integration (3) and is dominant in maintain-

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SUPERNUMERARY PHANTOM LIMBS WITH HEMISPHERIC STROKE

ing the internal representation of the body state (3). The attentional network may also be controlled by the right
hemisphere (5). The incidence of right thalamic hemorrhage is
significantly higher than that of left thalamic hemorrhage (17).
Our case illustrates some possible causative factors of SPL
among patients with left hemispheric stroke, including lesions
located in the thalamus, spastic paresis immediately after
stroke, and psychiatric disorders, such as alcohol and tobacco
intoxication. SPL may be more common than reported (7, 8, 10,
11); therefore, thorough examinations are essential for the care
of stroke patients.

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Wahnbildung bei linkseitiger Hemiplegie. Monatsschr Psychiatr Neurol
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Elsevier, 1985, vol 1, pp 373–393.
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COMMENTS

I

n this report, the authors describe a rare case of supernumerary phantom limbs (SPLs) after left cerebral hemisphere
stroke with imaging diagnosis. Computed tomography and
magnetic resonance imaging demonstrated a left thalamic
hemorrhage. Such lesions may occur in the right or the left
cerebral hemisphere, but there is a predominance of right
hemisphere strokes in association with SPL. The reason why
SPL is more likely to be associated with right hemisphere
stroke than left hemisphere stroke remains unclear. The fact
that SPLs are experienced more frequently after right-sided
brain stroke is closely akin to the higher proportion of central
pain after right thalamic stroke: the right hemisphere seems to
have a role in monitoring somatic states and a dominant role
in processing somatic representation.
In a recent article reporting the findings of a functional
magnetic resonance imaging study, McGonigle et al. (1) suggested that activity within areas traditionally classified as
parts of the motor system (supplementary motor area) can
influence the conscious perception of the body under pathological conditions. Previously, only two cases of SPLs had
been reported after left-sided brain stroke. As the first report
of SPLs after left-sided brain stroke accompanied by neuroimaging, this article is unique and important.
Akira Yamaura
Chiba, Japan
1. McGonigle DJ, Hanninen R, Salenius S, Hari R, Frackowiak RS, Frith CD:
Whose arm is it anyway? An fMRI case study of supernumerary phantom
limb. Brain 125:1265–1274, 2002.

I

n this well-documented report, the authors provide interesting information about the neuroanatomic substrate of the
rare phenomenon of SPLs after right and left hemisphere
stroke. Right hemisphere damage with left-side SPL is most
commonly observed. In the patient reported in this article (and
in two patients in the literature) with left-sided stroke and
right SPL, the stroke involved the thalamus and internal capsule, and the patients also exhibited spastic paresis of the right
limbs. The authors hypothesize that the experience of SPL is
related to motor impairment as well as sensory loss and psychotic states caused by toxins.
Robert G. Grossman
Houston, Texas

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