References
1. Zweifler RM, Drinkard R, Cunningham S, Brody ML, Rothrock J F . Implementation of a stroke code system in Mobile,
AL:diagnostic and therapeutic yield. Stroke 1997;28:981983.
2. The National Institute of Neurological Disorders and Stroke
rt-PA Stroke Study Group. Tissue plasminogen activator
for acute ischemic stroke. N Engl J Med 1995;333:15811587.
3. Adams HP, Brott TG, Furlan AJ, e t al. Guidelines for thrombolytic therapy for acute stroke: a supplement to the guidelines

Cerebral sparganosis
with intracerebral
hemorrhage:
A case report

for the management of patients with acute ischemic stroke.
Stroke 1996;27:1711-1718.
4. Quality Standards Subcommittee of the American Academy of
Neurology. Practice advisory: thrombolytic therapy for acute ischemic s t r o k e s u m m a r y statement. Neurology 1996;47:835-839.
5. Barsan WG, Brott TG, Broderick JP, Haley EC, Levy DE, Marler JR. Time of hospital presentation in patients with acute
stroke. Arch Intern Med 1993;153:2558-2561.
6. Biller J , Patrick JT, Shepard A, Adams HP. Delay time between onset of ischemic stroke and hospital arrival. J Stroke
Cerebrovasc Dis 1993;3:228-230.

Article abstract-We describe the clinical case and radiologic findings in a
woman with cerebral sparganosis in which intracerebral hemorrhage was the
presenting feature with hemiparesis and dysarthria. CT demonstrated highdensity lesions in the right frontoparietal area, suggesting a hematoma. With
conservative management, hemiparesis improved and follow-up CT revealed
what looked like a resolving hematoma. Two weeks later, she complained
once again of aggravated left hemiparesis and facial weakness. Diagnosis of
sparganosis was made on the basis of brain MRI and ELISA. Stereotactic
surgery was performed, and a live larva of sparganum was successfully
removed.
NEUROLOGY 1998;50:503-506

S.-C. Jeong, MD; J.-C. Bae, MD; S.-H. Hwang, MD, PhD; H.-C. Kim, MD, PhD; and B.-C. Lee, MD, PhD

Sparganosis is caused by a migrating tapeworm
larva of the genus Spirometra. Spargana invade subcutaneous tissue, muscle, eyeball, urogenital, and
abdominal viscera, but the CNS is not a frequent site
of invasion. Rare but increasing reports have described cerebral sparganosis from various parts of
the world, and recent developments in serologic and
radiologic studies make diagnosis of CNS infection
by spargana much easier. However, cerebral sparganosis associated with intracerebral hemorrhage is
very rare; so far, only one case has been reported in
the literature1 and another case with intraventricular hemorrhage rather than parenchymal hematoma
has been reported.2 Here we report another case of
cerebral sparganosis with hemiparesis as the main
manifestation of the disease. Apparently, this manifestation is attributable to the intracerebral hemorrhage caused by a live larva of sparganum of a n
unusual size.
Case report. A 61-year-old woman first visited the outpatient clinic of the neurology department of Hallym University Hospital because of left hemiparesis. Her history
included eating half-cooked frog and intermittently drinking water from the ponds. She had no remarkable medical
history up until 6 months ago when she suddenly developed left hemiparesis. CT of the brain was taken at the
local hospital; no specific abnormal lesion was found. She
was then treated for lacunar infarction with traditional
medicine such as acupuncture and herbs, but her left
hemiparesis improved only slightly over the following
months. Neurologic examination revealed mild left hemi-

paresis and increased deep tendon reflexes (DTR). Diagnosis of a n old lacune was presumed. Two months later,
she came to the emergency room with more progressed
left hemiparesis and newly developed left hemihypesthesia and dysarthria. Neurologic examination again revealed a n intelligent woman of normal mentation with
dysarthria. She had mild motor weakness in the left
extremities, grade 415 in upper and lower extremity with
increased DTR, and Babinski sign was noted on the left
side. Primary sensory modalities of pain, temperature,
touch, and vibration were decreased over the left qxtremities. Physical examination revealed no signs of abnormal palpable mass or lump over the entire body.
Routine laboratory findings such as CBC (including eosinophil count), ESR, urinalysis, stool examination for
parasites, ECG, and chest radiograph were normal. CT
of the brain revealed a 2 x 1-cm high-density area in the
right frontoparietal a r e a with diffuse low-density area
around it (figure 1A). Intracerebral hemorrhage was diagnosed and she was treated conservatively. One week
later, her sensation became symmetrical, but the left
hemiparesis and dysarthria were still persistent with
only mild improvement. On the follow-up CT taken a
week later, t h e high-density lesion in the right frontoparietal lobe shrank to 1 x 0.5 cm, but the surrounding
low-density area was noticeably enlarged (figure 1B).
We recommended MRI of the brain and cerebral angiography to exclude the possibility of a n arteriovenous malformation or brain tumor. S h e refused to take the
examinations and was discharged.
Two weeks later, she returned with more aggravated leR
hemiparesis. She had left facial palsy of central type and
motor weakness with grade 015 in the left upper extremity
Copyright 0 1998 by the American Academy of Neurology 603

values > 0.22) before the operation. For cysticercus, they
were 0.04 and 0.01 (positive values > 0.18). For paragonimus, they were 0.03 and 0.01, respectively (positive values >
0.25). Micro-ELISA test was an important clue in diagnosing
this case. The absorbance value of the serum ELISA test for
sparganum was decreased to 0.76 after the operation.
Three weeks later, she was transferred to the neurosurgical department, and stereotactic surgery was performed
for cerebral sparganosis. During the surgery, the larva was
cut in half, each measuring 20 and 30 cm. A living larva of
50 cm in total length with 2 to 3 mm in thickness was
removed successfully from the lesion. Figure 3 shows part
of the larva. She is now able to walk with assistance.

Figure 1. (A) Brain CT shows a high-density lesion in the
right frontoparietal area with diffuse a low-density area
around it that appears to be a small hernatoma with surrounding edema. (Bi Follow-up CT taken 1 week later
shows a regressed high-density lesion, but surrounding
diffuse low-density area is enlarged.
and grade 315 in the left lower extremity. MRI of the brain
demonstrated multiple cystic low-signal lesions surrounded
by the area of decreased signal change on T1-weighted images with rim enhancement (figure 2, A and B) and displayed
as multiple high-signal lesions surrounded by an area of diffuse increased signal change on T2-weighted images (figure
2C). CSF examination revealed clear fluid with no cells, protein was 34 mg/mL, and glucose was 61 mg/mL (serum glucose, 60 mglmL). Micro-ELISA test for sparganum was
positive. At first it was difficult to diagnose the nature of this
particular lesion, but the micro-ELISA test proved to be a
useful tool for the evaluation of parasitic infestation. We performed the micro-ELISA test for sparganum twice, before
and after the operation. The absorbance values of serum and
CSF ELISA test for sparganum were 1.10 and 1.05 (positive
504 NEUROLOGY 50

February 1998

Discussion. Sparganosis is an infection caused
by larvae plerocercoid of the genus Spirometru, a
cestode tapeworm. In Asia, Spirometra erinucei is
the indicated species.3 The adult tapeworm can infect dogs, cats, and other carnivores, residing in
their gastrointestinal tract. Eggs are passed in the
feces of these primary hosts and then ingested first
by intermediate hosts, called cyclops. Cyclops, in
which eggs mature into procercoid larvae, are ingested by frogs, snakes, birds, and fishes. Procercoid larvae mature into plerocercoid in their
second intermediate hosts, and the flesh of these
animal is eaten by primary hosts, thus continuing
life cycles. Human infestation is uncommon and
seems t o occur accidentally by ingestion of polluted
water containing cyclops, by ingestion of r a w or
inadequately cooked flesh of snakes o r frogs, and
by applying the flesh of an infected intermediate
host as a poultice t o a wound. The exact transition
pathway after infection has not been clearly understood, but the larvae is known t o migrate from
primary infection focus t o a distant site, such as
skin, skeletal muscle, orbital tissue, urogenital
and abdominal viscera, and rarely t o the CNS. In
this patient, the route of infestation was thought
t o be through drinking water from ponds o r eating
poorly cooked flesh of frogs many years ago: No
other signs or symptoms of infection of organs by
sparganum could be detected other than the brain.
Since human infection was first discovered by
Patrick Manson in 1882, more than 300 cases of
human sparganosis have been reported worldwide,
but it is more common in East Asia (China,2z4Japan,
and Korea), and it is also reported in Southeast Asia
(Malaysia, India, and the Philippines). Cases have
been reported from the United States, especially in
the Southeastern ~ t a t e sOrdinarily,
.~
sparganosis involves subcutaneous soft tissue and superficial muscle fascia of the human body, and sparganosis of the
brain is rare. Cerebral sparganosis was first reported
by Takeuchi in 1918, and since then over 20 cases
have been reported in the English literatures up to
1989. However, cerebral sparganosis has increasingly been detected in recent years (over 50 cases
between 1990 and 19951, presumably due t o advances in brain imaging and introduction of ELISA
test. The cases of cerebral sparganosis too have been

Figure 2. MRI of patient at presentation. (A) Sagittal T1-weighted image shows multiple cystic low-signal lesions with
surrounding diffuse decreased signal change in the right frontoparietal area. (B) Enhanced axial T 1-weighted image
demonstrated rim enhancement of these low-signal lesions. (C) Axial T2-weighted image displays high-signal lesions with
surrounding increased signal change i n the same area.

more common in Asia than e l ~ e w h e r e ,perhaps
~,~
because of the use of unsanitary drinking water and
eating raw uncooked meat.*
The clinical manifestations of cerebral sparganosis are seizure, headache, and focal neurologic
deficits such as hemiparesis, aphasia, dysarthria,
and dizziness. Among these, seizure, headache, and
hemiparesis are the most frequent symptoms
and
Our patient complained of recurrent
weakness of the left extremities with waxing and
waning course, sensory change, dysarthria, and left
facial palsy without any seizures.
CTs of cerebral sparganosis usually demonstrate
the presence of white matter hypoattenuation with
dilatation of adjacent ventricle, irregular or nodular
enhancing lesion, small punctate calcification, and a
change in the location of the enhancing nodule on
the follow-up C T S . ~In
, ~ this particular patient, the
first CT could not be obtained. The CT performed a
month before the first admission revealed a highdensity area in the right frontoparietal region; the
lesion regressed on repeat CT taken a week later.
Brain MRI demonstrated multiple cystic areas of hypointensity on T1-weighted images with rim enhancement and areas of hyperintensity on T2weighted images. Recently, Moon et al.7 reported
that areas of hyperintensity on T1-weighted images
and areas of hypointensity areas on T2-weighted images were detected only in MRI. They believed that
these changes in signal intensity represented petechial hemorrhage and may be due to capillary or
venous injury by the migrating worm. Actually, some
cases showed cortical petechial hemorrhage at the
operation site.7 We could not certify the cause and
effect relationship between the intracerebral hemorrhage and the sparganum by the pathologic findings,
but we agree that the idea of probable vascular injury was caused by the migrating larva.
Differential diagnosis should include other infec-

tious granulomas, neoplasms, chronic infarctions,
and degenerative diseases. If an enhancing lesion
shows an irregular, amorphous, beaded, or conglomerate nodular appearance together with extensive
degeneration of the white matter with adjacent ventricular dilatation, differentiation from other granulomas or metastasis is not difficult. If the lesion has
a mass effect, it may be misdiagnosed as a neoplasm
or as other g r a n ~ l o m a s . ~ , ~
ELISA test is a very sensitive test for the detection of sparganosis when the diagnosis is in doubt.
Before the introduction of the ELISA test, human
sparganosis had to be surgically diagnosed because
its diagnosis depended almost entirely on the recovery of larvae from the lesion. In surgically proven
cerebral sparganosis, both serum and CSF ELISA
tests showed high sensitivity and concordance ,rate

Figure 3. Part of live larva removed during the stereotactic operation with comparable scale under it. It was measured to be about 20 c m long.
February 1998 NEUROLOGY 50 505

in diagnosing cerebral sparganosis, and negative
conversion was noted in patients with successful surgical
However, it is difficult to say whether
the decrement in the serum absorbance value was
due to the treatment or to a natural course of human
immune response to the spargnum. Further studies
might be necessary to evaluate human immune response to the sparganum. The serologic diagnosis by
means of ELISA could be a useful tool in the epidemiologic study of human sparganosis in a susceptible
population, as well as in individual diagnosis. As for
the management of cerebral sparganosis, surgical removal seems to be the most effective way of treatment.g It is a commonly held opinion that there is no
known antihelminthic effective in sparganosis.
Niclosamides are effective antihelminthic agents
while the tapeworm is still within the intestine of
the primary host. Once the tapeworm extends beyond the intestine, there is no evidence t o support
the therapeutic value of Praziquantel (Bayer Korea
Pharm. Co., Korea) and Albendazole (Handok
Pharm. Co., Korea), which have been effective agents
for neurocysticercosis.lO
From the Department of Neurology, Hallym University College of
Medicine, Korea.
Received August 19, 1996. Accepted in final form August 13, 1997.

506

NEUROLOGY 50 February 1998

Address correspondence and reprint requests to Br. Byung-Chul
Lee, Department of Neurology, Hallym University College of Medicine, 94-200 Yungdungpo-dong, Yungdungpo-ku, Seoul 150-020,
Korea.

References
1. Chamadol W, Tangdumrongkul S , Thanaphaisal C, et al. Intracerebral hematoma caused by sparganum. A case report.
J Med Assoc Thai 1992;75:602-605.
2. Wong CW, Ho YS. Intraventricular hemorrhage and hydrocephalus caused by intraventricular parasitic granuloma suggesting cerebral sparganosis. Acta Neurochir (Wien) 1994;129:
205-208.
3. Lee SH, Chai JY,Seo BS. Cho SY. Two cases of human infeetion by adult of spirometra erinacei. Korean J Parasitol 1984;
22~66-69.
4. Cho SY, Bae J, Seo BS. Some aspects of human sparganosis in
Korea. Korean J Parasitol 1975;13:60-77.
5. Norman SH, Kreutner A J r . Sparganosis: clinical and pathologic observation in ten cases. South Med J 1980;73:297-300.
6. Chang KH, Chi JG, Cho SY, e t al. Cerebral sparganosis. Analysis of 34 cases with emphasis on CT features. Neuroradiology
1992;34:1- 8.
7. Moon WK, Chang KH, Cho SY, et al. Cerebral sparganosis.
MR imaging versus CT features. Radiology 1993;188:751-757.
8. Kim H, Kim SI, Cho SY. Serological diagnosis of human sparganosis by means of micro-ELISA. Korean J Parasitol 1984;
22:222-228.
9. Anders K, Foley K, Stern WE, Brown WJ. Intracranial sparganosis: a n uncommon infection. J Neurosurg 1984;60:12821286.
10. American Medical Association. Drug evaluation annual 1992.
Chicago: American Medical Association, 1992:1632-1645.

Cerebral sparganosis with intracerebral hemorrhage: A case report
S.-C. Jeong, J.-C. Bae, S.-H. Hwang, et al.
Neurology 1998;50;503-506
DOI 10.1212/WNL.50.2.503
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