710 Maeshima et al.

been reported to recur between 2 months and 14 years after
initial treatment; 80% of recurrences have been within 5
years.3,6±8 To our knowledge, the present case represents the
longest period between prior treatment and recurrence reported to date.
The radiosensitivity of germinomas has been well documented.2,3,5,9 The 5-year survival rates of patients with
germinomas after radiotherapy have been reported to be
95±100%.2,8,9 The rate of recurrence of CNS germinomas after
irradiation has been reported to be 6±17%.2,3 Ono et al.3
described 4 patterns of recurrence: an inadequate initial irradiation field, a benign teratoma caused by late growth of the
teratoma component, -fetoprotein- or human chorionic
gonadtropin-producing tumours of extraembryonic origin,
and extraneural metastasis without evidence of intracranial
germinoma. The pattern of first recurrence in this case may
have been an inadequate initial irradiation field, as this targeted only the pineal region. Ono et al.3 noted that no pure
germinoma recurred as the same histological tumour within
the irradiated field. Our patient received 50 Gy of irradiation
to the pineal region to target the primary germinoma. Seventeen years later, the germinoma recurred in the suprasellar
region. The patient received 50 Gy of irradiation to the
suprasellar region, because of the danger to the pineal region
posed by irradiation of the whole brain. A further recurrence
developed in the right frontal lobe along the catheter 4 years
after irradiation to the suprasellar region.
We were intrigued that the recurrent tumour disseminated
along the ventricular catheter. Although abdominal metastases
of brain tumours via ventriculoperitoneal shunts have been
reported,3,4,6 there have been no reports of specific dissemination along the catheter. We consider that the tract
around the catheter, created when the catheter was inserted,
caused this pattern. Tumour cells brought together by the
shunt flow lodged on the brain tissue near the tract and multiplied, subsequently disseminating into the ventricle.
In conclusion, recurrence after such a long period calls
attention to the need for long term follow up of patients with
germinoma. It is considered that irradiation to the whole
ventricular field is necessary as a primary treatment. Also, it is
possible that CNS germinomas can recur by dissemination
along the ventricular catheter.
CONCLUSION
We have reported a rare case of intracranial germinoma that
recurred along the shunt tube 17 years after prior treatment.
Recurrence after such a long period highlights the necessity of
long term follow up for patients with germinoma.
REFERENCES
1.

Jennings MT, Gelman R, Hochberg F. Intracranial germ-cell tumors:
Natural history and pathogenesis. J Neurosurg 1985; 63: 155±167.
2. Matsutani M, Sano K, Takakura K et al. Primary intracranial germ
cell tumors: a clinical analysis of 153 histologically verified cases.
J Neurosurg 1997; 86: 446±455.
3. Ono N, Isobe I, Uki J, Kurihara H, Shimizu T, Kohno K. Recurrence
of primary intracranial germinomas after complete response with
radiotherapy: Recurrence patterns and therapy. Neurosurg 1994; 35:
615±621.
4. Kun LE, Tang TT, Sty JR, Camitta BM. Primary cerebral germinoma
and ventriculoperitoneal shunt metastasis. Cancer 1981; 48: 213±216.
5. Merchant TE, Davis BJ, Sheldon JM, Leibel SA. Radiation therapy
for relapsed CNS germinoma after primary chemotherapy. J Clin
Oncol 1998; 16: 204±209.
6. Rickert CH. Abdominal metastases of pediatric brain tumors via
ventriculoperitoneal shunts. Child's Nerv Syst 1998; 14: 10±14.

Journal of Clinical Neuroscience (2002) 9(6)

7.

8.
9.

Shirato H, Nishino M, Sawamura Y et al. Analysis of long-term
treatment of intracranial germinoma. Int J Radiat Oncol Biol Phys 1997;
37: 511±515.
Sugiyama K, Uozumi T, Arita K et al. Clinical evaluation of 33 patients
with histologically verified germinoma. Surg Neurol 1994; 42: 200±210.
Linstadt D, Wara WM, Edwards MSB, Hundgins RJ, Sheline GE.
Radiotherapy of primary intracranial germinomas: the case against
routine craniospinal irradiation. Int J Radiat Oncol Biol Phys 1988; 15:
291±297.

Transient aphasia and
persistent amnesia after
surgery for internal carotid
artery ± posterior
communicating artery
aneurysm
Shinichiro Maeshima1 MD, Akitaka Ueyoshi1 MD,
Mitsuharu Tsuura 2 MD, Hideki Takemoto 2 MD,
Toru Itakura2 MD, Munehito Yoshida1 MD,
Tomoko Matsumoto1
1

Department of Physical Medicine and Rehabilitation and 2Department of
Neurological Surgery, Wakayama Medical University

Summary We report a case of transient aphasia and persistent
amnesia after clipping of a ruptured cerebral aneurysm to treat a
subarachnoid haemorrhage. Postoperatively, aphasia was identified and magnetic resonance imaging (MRI) showed an abnormal
intensity area in the left anterior thalamus. Single photon emission
computed tomography (SPECT) revealed a wider area of low perfusion surrounding the left thalamus and left frontotemporal lobe
than that shown by the MRI. His aphasia resolved over the subsequent 12-week period. He was left with an isolated disturbance
of memory; in the absence of any dementia, aphasia or disturbance of consciousness, his condition was classified as one of
amnesia. SPECT 14 weeks after admission revealed an area of
low perfusion limited to the left thalamus. These findings suggest
that the persistence of amnesia in this case was caused by the
infarction of the mammillothalamic tract, and the recovery from
aphasia may have resulted from the disappearance of surrounding
edema. & 2002 Published by Elsevier Science Ltd.
Journal of Clinical Neuroscience (2002) 9(6), 710±713
& 2002 Published by Elsevier Science Ltd.
DOI: 10.1054/jocn.2001.1065, available online at http://www.idealibrary.com on

Keywords: aphasia, amnesia, memory, subarachnoid hemorrhage
Received 13 August 2001
Accepted 22 October 2001
Correspondence to: Shinichiro Maeshima, MD, PhD, Department of
Physical Medicine and Rehabilitation, School of Medicine, Wakayama
Medical University, 811-1 Kimiidera, Wakayama 641-0012, Japan.
Tel.: ‡81 734 47 2300; Fax: ‡81 734 41 0509.

& 2002 Published by Elsevier Science Ltd.

Transient aphasia and persistent amnesia after surgery 711

INTRODUCTION
It is well known that left unilateral thalamic damage is
associated with disturbances of language and memory.1
Although there have been many patients with aphasia and/or
amnesia due to thalamic infarction2±4 or haemorrhage,5 few
patients with aphasia and amnesia after clipping of an aneurysm in a subarachnoid haemorrhage have been reported.
We recently had an opportunity to examine a patient who
experienced mild transient aphasia and persistent amnesia
after a direct operation for an internal carotid-posterior
communicating artery (IC±PC) aneurysm.
CASE REPORT
A 48 year old man with a history of hypertension experienced a
sudden attack of severe headache with vomiting and was
admitted to our hospital. He had no focal neurological signs
except for mild neck stiffness. A CT scan showed a
subarachnoid haemorrhage (SAH) and an angiogram revealed
a left IC±PC aneurysm (Fig. 1A), a left middle cerebral artery
(MCA) aneurysm and a distal anterior cerebral artery aneurysm. During the operation, the IC±PC aneurysm was determined to be ruptured, and both the IC±PC and MCA
aneurysms were clipped via a left frontotemporal approach.
Postoperatively, aphasia was identified and a CT scan showed
a low density area in the left anterior thalamus (Fig. 2).
The patient's spontaneous speech was fluent after the
operation. He had no difficulty initiating speech or articulating
normally, and he did not have logorrhea. Echolalia was not
observed, and the patient's phonological structure was clear.
Confrontation naming was moderately impaired, with

considerable paraphasia. His repetition ability was excellent
and his ability to read aloud was good. His writing was
impaired, and considerable paragraphia was seen. On the
Wechsler Adult Intelligence Scale-Revised (WAIS-R), his
performance intelligence quotient (IQ) was 75. His verbal IQ
was unmeasurable because of the aphasia. His score on
Raven's coloured progressive matrices (RCPM) was 28/36.
Magnetic resonance imaging (MRI) 4 weeks after onset
showed a lesion in the left anterior thalamus including
the anterior ventral nucleus (Fig. 3). Single photon emission
computed tomography (SPECT) revealed a wider area of
low perfusion surrounding the left thalamus and left frontotemporal lobe than that revealed by CT/MRI (Fig. 4A).
Although postoperative internal carotid angiography
showed the posterior cerebral artery via the left posterior
communicating artery, neither the left tuberothalamic artery
(polar artery) nor the thalamoperforating artery was demonstrated (Fig. 1B).
Although his aphasia had resolved almost completely
12 weeks after the operation, the amnesia has persisted. On the

(A)
Fig. 2 Postoperative CT scan showed a low density area in the left
anterior thalamus.

(B)

Fig. 1 (A) Cerebral angiography of the left internal carotid artery revealed
a left IC±PC aneurysm. (B) Postoperative internal carotid angiography showed
the posterior cerebral artery via the left posterior communicating artery.

& 2002 Published by Elsevier Science Ltd.

Fig. 3 Magnetic resonance imaging 4 weeks after onset showed a lesion
in the left anterior thalamus including the anterior ventral nucleus.

Journal of Clinical Neuroscience (2002) 9(6)

712 Maeshima et al.

A

B
Fig. 4 (A) Single photon emission computed tomography revealed a
wider area of low perfusion surrounding the left thalamus and left
frontotemporal lobe than that revealed by CT/MRI. (B) Single photon
emission computed tomography 14 weeks after onset revealed an area
of low perfusion limited to the left thalamus.

WAIS-R, his verbal IQ was 84 and his performance IQ had
risen to 86, with an overall IQ of 85. His score on RCPM was
30/36. His digit recollection span was seven, and his word
fluency in naming animals was 8 words per minute. Despite
these preserved functions, his memory remained clearly disturbed. Although he did not show confabulation, he remained
disoriented with respect to day and place. The Benton visual
recognition test (BVRT) showed an immediate recall score of
6/10; delayed recall, 2/10; and copying, 10/10. On the auditory
verbal learning test (AVLT), his immediate recall performance
was 2, 2, 5, 3, 3/15; his delayed recall performance was 0/15;
and his recognition performance was 8/15. SPECT 14 weeks
after onset revealed an area of low perfusion limited to the left
thalamus (Fig. 4B).

suggest that neither is solely of neuropsychological origin. The
important role of the ventrolateral nucleus (VL) in thalamic
aphasia and amnesia has been extensively discussed.6 Ojemann
et al.9 found that electrical stimulation of the VL facilitated
recording of verbal information but caused deterioration in the
ability to retrieve verbal memories and disturbed object
naming. Thalamic aphasia is often accompanied by amnesia,10
suggesting that the VL may be involved in both aphasia and
amnesia.
On the other hand, some investigators have reported that
anteriorly located lesions are necessary for amnesia.4 Damage
to the mammillothalamic tract, anteriorly located structures,
relates to amnesia.
In this case, one possible reason for the recovery from the
aphasia and the persistence of the amnesia may be that the
anterior thalamus was affected not only by the infarction itself
but also by surrounding oedema, which gradually resolved in
the weeks following the damage.
Hunter et al.11 reported that patients after SAH with a
late result of either good or fair frequently had cognitive
deficits such as disturbed memory, concentration or language.
As a result of surgery, our patient had amnesia and aphasia
caused by left thalamic damage to the tuberothalamic artery
or the thalamoperforating artery, both of which are perforating branches of the PC. Three possible causes for the syndrome
are: the mechanical obstruction of these arteries by clipping,
the thromboembolism caused by manipulation ± e.g., a
temporary clipping ± or the distortion of the clip due to
postoperative oedema of neighboring structures. An infarct
may appear as a result of sacrificing the artery during surgery,
but a clear cause was not apparent in our case. Many authors
have emphasised that meticulous caution is necessary to
preserve the various perforators of the PC in neurosurgical
operations such as ICA aneurysms. We also insist that a
neurosurgeon needs to explain to the patient that he or she
may have neuropsychological deficits after surgery.

DISCUSSION
Although this patient experienced thalamic aphasia immediately after the surgery, the aphasia resolved over the subsequent 12-week period. He was left with an isolated
disturbance of memory; in the absence of any dementia,
aphasia or disturbance of consciousness, his condition was
classified as one of amnesia.
Earlier reports have noted that cases of thalamic ischaemia
that produce aphasia can be divided into either occlusions of
the posterior thalamosubthalamic paramedian artery, which
supplies blood to the lateral medial corpus of the thalamus,6 or
occlusions of the tuberothalamic artery, which supplies the
anterior and central thalamic corpi.7
Recent discoveries indicate that damage to the thalamus
may cause secondary impairment of cortical function.8 In some
cases, thalamic aphasia may be correlated with cortical hypometabolism rather than with the extent of the lesion itself. In
our case, the lesion as seen in CT/MRI appeared restricted to
the anterior section of the thalamus, perfused by a branch of
the tuberothalamic artery or the thalamoperforating artery.
Examination by SPECT of the area extending from the left
frontal lobe to the temporal and parietal lobes also confirmed
a regional decline in cerebral circulation.
In cases of thalamic lesions, amnesia tends to persist even in
cases where the associated aphasia resolves or improves to a
considerable degree. The discrepancies between the characteristics of thalamic aphasia and those of thalamic amnesia
Journal of Clinical Neuroscience (2002) 9(6)

REFERENCES
1.

2.

3.
4.

5.
6.

7.

8.

9.

10.

Cambier J, Graveleau Ph. Thalamic syndromes. In: Vinken PJ, Bruyn GW,
Klawans H, Frederiks JAM (eds). Handbook of Clinical Neurology
(Vol. 45, Revised series 1 Clinical Neuropsychology). Amsterdam: Elsevier,
1985: 87±98.
Speedie LJ, Heilman KM. Anterograde memory deficits for visuospatial
material after infarction of the right thalamus. Arch Neurol 1983; 40:
183±186.
Goldenberg G, Wimmer A, Maly J. Amnestic syndrome with a unilateral
thalamic lesion: a case report. J Neurol 1983; 229: 79±86.
Mori E, Yamadori A, Mitani Y. Left thalamic infarction and disturbance
of verbal memory: a clinicoanatomical study with a new method of
computed tomographic stereotaxic lesion localization. Ann Neurol 1986;
20: 671±676.
Choi D, Sudarsky L, Schachter S, Biber M, Burke P. Medial thalamic
hemorrhage with amnesia. Arch Neurol 1983; 40: 611±613.
Bogousslavsky J, Miklossy J, Deruaz JP, Regli F, Assal G. Unilateral
left paramedian infarction of thalamus and midbrain: a clinico-pathological
study. J Neurol Neurosurg Psychiat 1986; 49: 686±694.
Graff-Radford NR, Eslinger PJ, Damasio AR, Yamada T.
Nonhemorrhagic infarction of the thalamus: behavioral, anatomic,
and physiologic correlates. Neurology 1984; 34: 14±23.
Baron JC, Levasseur M, Mazoyer B et al. Thalamocortical
diaschisis: positron emission tomography in humans. J Neurol
Neurosurg Psychiat 1992; 55: 935±942.
Ojemann GA, Blick KL, Ward AA Jr. Improvement and disturbance
of short-term verbal memory during human ventrolateral thalamic
stimulation. Trans Am Neurol Assoc 1969; 94: 72±75.
Archer CR, Illinsky IA, Goldfader PR, Smith KR Jr. Aphasia in
thalamic stroke; CT stereotactic localization. J Comput Assist
Tomogr 1981; 5: 427±432.

& 2002 Published by Elsevier Science Ltd.

Intratumoral hemorrhage of C5 root hemangioblastoma 713
11.

12.
13.

Hutter BO, Gilsbach JM, Kreitschmann I. Quality of life and
cognitive deficits after subarachnoid haemorrhage. Br J Neurosurg 1995;
9: 465±475.
Saeki N, Rhoton AL Jr: Microsurgical anatomy of the upper basilar
artery and the posterior circle of Willis. J Neurosurg 1977; 46: 563±578.
Gibo H, Lenley C, Rhoton AL Jr. Microsurgical anatomy of the
supraclinoid portion of the internal carotid artery. J Neurosurg 1981;
55: 560±574.

Intratumoral hemorrhage
due to hemangioblastoma
arising from a cervical
nerve root ± a case report
Eiichi Ishikawa1 MD, Akira Matsumura1 MD,
Yuji Matsumaru1 MD, Izumi Anno2 MD,
Kazuhiro Nakamura1 MD, Yasukazu Shioyama3 MD,
Tsuyoshi Midorikawa4 MD, Tadao Nose1 MD
1

Departments of Neurosurgery and 2 Neuroradiology, Institute of Clinical
Medicine, University of Tsukuba, Tsukuba, Ibaraki, Japan, 3 Departments of
Diagnostic Radiology and 4 Orthopedics, Ibaraki Prefectural Central Hospital,
Tomobe, Ibaraki, Japan

Summary We describe a case of a 70 year old man suffering
from sudden weakness of the left foot. Preoperative neuroimaging
examinations showed an oval mass 2 cm in maximal diameter with
intratumoral hemorrhage at the seventh cervical vertebra. The
mass was supplied by the right lateral thoracocervical artery and
was drained to the anterior spinal vein. The intraoperative findings
showed the hard reddish tumor was not attached to the pia and
the posterior root of the fifth cervical nerve was totally encased by
the tumor. A histopathological examination revealed that hemangioblastoma encasing the posterior nerve root totally, so that the
tumor was thought to arise from it. Unusual presentation of the
neuroimaging examinations is described. & 2002 Published by
Elsevier Science Ltd.
Journal of Clinical Neuroscience (2002) 9(6), 713±716
& 2002 Published by Elsevier Science Ltd.
DOI: 10.1054/jocn.2001.1035, available online at http://www.idealibrary.com on

Keywords: nerve root tumor, hemangioblastoma, intratumoral
hemorrhage
Received 8 August 2001
Accepted 31 August 2001
Correspondence to: Akira Matsumura, MD, Department of Neurosurgery,
Institute of Clinical Medicine, University of Tsukuba, Tsukuba,
Ibaraki 305-8575, Japan. Tel.: ‡81-298-53-3220; Fax: ‡81-298-53-3214;
E-mail: matsumur@md.tsukuba.ac.jp

hemangioblastoma encasing the nerve root wholly, which is
considered to arise from the nerve root, is a very rare event.2,6,9
Here, we describe a case of intratumoral hemorrhage due to
hemangioblastoma encasing the posterior root of the fifth
cervical nerve.
CASE REPORT
A 70 year old man was admitted to a regional hospital after
sudden occurrence of pain in the neck and weakness of the left
foot on May 28, 2000. Five years before admission he had
undergone total gastrectomy due to malignant lymphoma.
There was no familial history associated with his illness.
Magnetic resonance (MR) imaging 24 hours after the development of the weakness revealed a lesion with on unclear
margin at the seventh cervical(C) vertebra. The inner part of it
was hyperintense on T1-weighted images (WI) and T2WI and
its outer part was mixed intensity on T1WI and hypointense on
T2WI (Fig. 1A). When he was referred to our hospital for
further therapy on June 19, neurological examinations revealed
mild weakness of the left arm, severe paresis of the left foot,
severe sensory disturbance below the higher level of the thoracic
(Th) vertebra, and hyperreflexia of his bilateral lower extremities. Urinary obstruction was also detected. Lumbar tapping
showed watery clear cerebral spinal fluid with no evidence of a
subarachnoid hemorrhage or xanthochromia. MR imaging 4
weeks after the development of weakness revealed that the mass
had grown and showed a clearer margin than it had at the initial
imaging (Fig. 1B). Angiographic studies showed a signal in the
manner of an arteriovenous fistula which was supplied by the
right lateral thoracocervical artery and was drained rostrally to
the anterior spinal vein (Fig. 2).
Six weeks after the patient had developed the weakness,
C6-Th1 laminoplasty and tumor removal were performed. A
hard reddish oval tumor 2 cm in maximal diameter was found
in the subarachnoidal and extramedullary region, with no
attachment to the pia. After easily blunt dissecting from the
arachnoid, a posterior nerve root of C5 totally encased with
the tumor was recognized. The tumor was supplied by an
artery through the lateral foramen of C7 and was drained to
the anterior spinal vein. Microscopic diagnosis was that the
extramedullary tumor originated from the nerve root because
the tumor had the only attachment with it.
A histopathological examination revealed that the demarcated tumor was composed of delicate vascular channels with
interstitial `stromal' cells, accompanied by massive central
hematoma formation. The diagnosis was hemangioblastoma
with organizing hematoma. Additional immunohistochemical
determination using S100 protein revealed a marked positivity
for a few cellular components, which indicated the nerve root
being totally surrounded by the tumor mass (Figs 3A,B). The
postoperative course was uneventful with no neurological
deterioration.
DISCUSSION

INTRODUCTION
Hemangioblastoma of the spine is relatively rare, representing
1.6±5.8% of all spinal tumors. Few reports have described
acute hemorrhage due to spinal hemangioblastoma, located in
either the cord parenchyma or the subarachnoid space.1±3
Although there have been some reported cases of extramedullary hemangioblastoma,2,4±10 in many of these cases the
tumor was only attached to the nerve root.5,7,10 Subdural type
& 2002 Published by Elsevier Science Ltd.

Hemangioblastoma of the spine is considered a relatively rare
tumor, representing 1.6±5.8% of all spinal tumors.1±3 Thirtytwo percent of patients with spinal hemangioblastoma have
von Hippel-Lindau syndrome, while those remaining have
sporadically occurring tumors.2 The mean age of patients with
spinal hemangioblastoma is 30 years at onset,1±3,7 with the
presented case constituting an unusual elderly example. In
usual cases of spinal hemangioblastomas, MR imaging
demonstrates hypointensity to isointensity on T1-weighted
Journal of Clinical Neuroscience (2002) 9(6)