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CASE REPORT

Neurocysticercosis Presenting with Epilepsia
Partialis Continua: A Clinicopathologic
Report and Literature Review
Shin-Joe Yeh,1 Ruey-Meei Wu1,2*

Neurocysticercosis, a disease caused by a tapeworm larva in the central nervous system, is the leading
cause of acquired epilepsy in undeveloped regions of the world. In Taiwan, after improvements in sanitation,
tapeworm infection became very rare except in mountain areas inhibited by aborigines. However, in recent
years, immigration from other Asian countries has increased rapidly, and parasite infection of the central
nervous system may again become an important cause of adult-onset epilepsy in clinical practice. Here, we
describe a 27-year-old female Thai immigrant who presented with adult-onset epilepsia partialis continua
of the right upper extremity. Electroencephalography showed epileptiform discharge in the left central
region, while brain magnetic resonance imaging showed a small enhanced lesion in the left premotor
cortex. She underwent operation and pathology of the mass revealed a degenerated cysticercus. In this article,
we provide detailed neuroimaging findings, pathologic report, and literature review of this parasitic infection
of the central nervous system. This case calls for physicians to be aware of cysticercosis as an etiology of
adult-onset epilepsy in immigrants from endemic countries. [J Formos Med Assoc 2008;107(7):576–581]

Key Words: epilepsia partialis continua, epilepsy, neurocysticercosis, Taenia solium, tapeworm

Neurocysticercosis, the leading cause of acquired to neurocysticercosis.2 The 2003 World Health
epilepsy worldwide, is caused by Taenia solium Organization regarded T. solium as an important
infection in the central nervous system.1 There issue in global public health.3 The WHO has cal-
are an estimated 50 million cases of cysticercosis culated that over 50,000 deaths due to neuro-
worldwide.2 Most cases occur in less developed cysticercosis occur each year.5 In Taiwan, after
countries, such as parts of Asia, Latin America, improvements in public sanitation, T. solium in-
sub-Saharan Africa, and parts of Oceania. In Asia, fection has been rare in recent decades except for
it is prevalent in India, Indonesia, Vietnam, China a few reported familial cases in aborigines.6
and Nepal, and the prevalence is between 1.7% However, the immigrant population from other
and 13%.3,4 However, due to increased immigra- countries in Southeast Asia is rapidly growing, so
tion and international travel, an increasing num- awareness of Taenia infection and its neurologic
ber of cases are being reported in developed complications is important. Here, we present a
countries.1,2 For example, in New Mexico and case of neurocysticercosis with clinical manifes-
California, 10% of cases presenting as seizure in an tations of epilepsia partialis continua. The life
emergency room in a large urban hospital was due cycle of the cysticercus and the pathogenesis of

©2008 Elsevier & Formosan Medical Association
. . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . .
1
Department of Neurology, National Taiwan University Hospital and 2National Taiwan University College of Medicine,
Taipei, Taiwan.

Received: March 15, 2007 *Correspondence to: Dr Ruey-Meei Wu, Department of Neurology, National Taiwan
Revised: April 13, 2007 University Hospital, 7 Chung-Shan South Road, Taipei 100, Taiwan.
Accepted: August 7, 1007 E-mail: robinwu@ntu.edu.tw

576 J Formos Med Assoc | 2008 • Vol 107 • No 7
Neurocysticercosis presenting with epilepsia partialis continua

the infection in the central nervous system are neurologic examination showed clear conscious-
reviewed. A detailed differential diagnosis of the ness, mild weakness in the right upper limb graded
neuroimaging findings in this case is described. 4 on the Medical Research Council grading sys-
tem, and increased tendon reflex and hypesthesia
in the right upper limb. Epilepsia partialis con-
Case Report tinua was diagnosed clinically,7,8 the lesion site
probably being in the left motor cortex, near the
A 27-year-old Thai woman attended our neuro- central vertex area.
logic clinic with the chief complaint of intermit- Awake electroencephalography (EEG) was per-
tent twitching of the right elbow for 12 hours, formed. The background activity consisted of sym-
which occurred about 20 days after a 1-minute metrical alpha rhythm in the posterior head area,
episode of clonic movement in her right wrist, with an adequate alerting response to eye opening.
with clear consciousness. The second episode con- There were frequent regional semirhythmic slow
sisted of stereotyped, semirhythmic, clonic flex- waves at 1–3 Hz, 25–50 μV in the left central re-
ion movement of her right wrist that lasted for gion, with occasional lateralization or extension
1–2 minutes, which then involved her right elbow, to the right side. There was an episode of clinical
and finally her right shoulder. This was followed seizure characterized by twitching of the right
by frequent clonic flexion movements of her right upper limb, but the EEG pattern did not change
elbow, initially occurring every 10–20 minutes, during the attack (Figure 1A). In addition, hyper-
then becoming more frequent and finally per- ventilation induced an episode of generalized
sistent. She could not control the movement. sharp waves with phase reversal at C3 (Figure 1B).
Of note, the clonic movement did not disappear These EEG findings suggested a focal lesion in the
during sleep. Prior to these episodes, she had no left central region.
medical disease, headache, fever, head trauma, or Brain magnetic resonance imaging (MRI)
previous seizure. Her personal history showed showed a well-defined rim-enhanced nodule with
that she had immigrated to Taiwan with her fam- a mural nodule in the left posterior frontal lobe,
ily to work 4 years previously and returned to her with marked perifocal edema (Figure 2). There
homeland every year. Her last visit to Thailand was no signal change in the lesion on diffusion-
was 1.5 years ago; while there, her drinking water weighted image or apparent diffusion coefficient.
was obtained from a well in a field. Tumor or granuloma was considered. Blood tests,
On physical examination, she did not have including white blood cell count with a differen-
fever, neck stiffness, or lymphadenopathy. The tial count (eosinophils, 4.2%), C-reactive protein,

A B
Fp2-F4 Fp2-F4
F4-C4
F4-C4
C4-P4
C4-P4
P4-O2
Fp1-F3 P4-O2

F3-C3 Fp1-F3
C3-P3 F3-C3
P3-O1 C3-P3
Fp2-F8
P3-O1
F8-T4
T4-C4
T4-T6
100μV
100μV C4-Cz
Fp1-F7 1 sec
1 sec
F7-T3 Cz-C3

T3-T5 C3-T3 07:20

Figure 1. Electroencephalographic findings in the reported case while awake. (A) There were frequent regional semi-
rhythmic slow waves at 1–3 Hz, 25–50 μV in the left central region, with occasional lateralization or extension to the right
side. There was one episode of clinical seizure characterized by twitching of the right upper limb, not associated with elec-
trophysiologic changes. (B) Hyperventilation induced occasional generalized sharp waves with possible phase reversal at C3.

J Formos Med Assoc | 2008 • Vol 107 • No 7 577
S.J. Yeh, R.M. Wu

A B

C

Figure 2. Magnetic resonance imaging findings.
(A) T1-weighted image shows a hypointense
lesion, with (B) ring enhancement with contrast.
(C) T2-weighted image shows perifocal edema
in the left posterior frontal lobe.

tumor markers, and anti-HIV antibody titer, were focus in the left central area, which was compati-
all within normal limits. ble with the clinical impression of epilepsia par-
Her seizure was completely controlled with tialis continua. No epileptiform activity was
valproic acid (1000 mg/day) on the second day recorded during attack; this discrepancy between
of hospitalization. The lesion in the left premo- motor convulsion and electrophysiologic find-
tor area was excised by craniotomy, and pathol- ings may be due to multiple closely allied small
ogy showed a partially degenerated cysticercus groups of neurons firing asynchronously or the
(Figure 3). The stools were checked twice for discharge focus being deeply seated in the in-
parasite ova and were negative. She received praz- folded cortex.8 MRI revealed a single enhanced
iquantel for 2 weeks after the operation. Inter- mass with perifocal edema in the left premotor
mittent paresthesia on the right side of the body area. For a single brain lesion causing focal
was well controlled by increasing the dosage of seizure, the differential diagnoses include tumor,
valproic acid to 1500 mg/day. granuloma, and pyogenic abscess;8 the character-
istic brain MRI findings are summarized in the
Table.9–13 Unexpectedly, the pathologic findings
Discussion of the mass lesion showed a partially degener-
ated cysticercus (Figure 3). The cysticercus in this
A 27-year-old Thai woman had a late-onset case was probably T. solium, because there is no
seizure with continuous clonic convulsion in her report that T. saginata produces cysticercosis in
right elbow. EEG study showed an epileptogenic the human brain.14

578 J Formos Med Assoc | 2008 • Vol 107 • No 7
Neurocysticercosis presenting with epilepsia partialis continua

A B

Figure 3. (A) Pathologic findings of a partially degenerated cysticercus in the brain (hematoxylin & eosin, 10×). (B) The
partially degenerated cysticercus was surrounded by inflammatory infiltrates of lymphocytes, plasma cells and numerous
eosinophils (hematoxylin & eosin, 40×).

Table. Magnetic resonance imaging (MRI) sequence findings in the differential diagnosis of a single enhanced lesion in the brain
MRI sequence Abscess9 Necrotic tumor9 CNS lymphoma*10 Tuberculoma11 Cysticercosis12,13

T1WI Hypointense Hypointense Iso- or hypointense Iso- or hyperintense Iso- or hyperintense,
hole-with-dot

T2WI Hyperintense Hyperintense Iso- or hypointense Iso- or hypointense Iso- or hyperintense,
hole-with-dot

Ring enhancement on (+) (+) Thick & (+) With little or no (+) (+)
T1WI with contrast irregular ring perifocal edema

DWI Hyperintense Hypointense Hyperintense Hyperintense Isointense

ADC Low value High value Low value
*CNS lymphoma in an immunocompromised patient. CNS = central nervous system; T1WI = T1-weighted image; T2WI = T2-weighted image; DWI =
diffusion-weighted image; ADC = apparent diffusion coefficient.

Human cysticercosis of T. solium is caused by Adult worms in human intestine
accidental ingestion of its eggs. Its lifecycle is shown (definite host)
(Taeniasis)
in Figure 4. The cysticercus (larva) hatches from
eggs in the human stomach and enters various
tissues through the blood stream. If it enters the Eggs in human feces
central nervous system, it may lodge in the paren-
chyma, ventricle, or cistern, resulting in different
Pig ingests Human ingests
clinical manifestations. In parenchymal neuro- the eggs the eggs
cysticercosis, there are four histopathologic stages
on neuroimaging, these being the viable stage,
Cysticercus in pig muscles Cysticercus in human
colloidal stage, nodular-granular stage, and calci- (intermediate host) brain, eyes, muscle, etc.
fied stage.13 Staging depends on the viability of the (cysticercosis)
cysticercus and the host inflammatory response.
In the viable stage, the larva elicits little or no Human eats Cysticercus dies
inflammatory response, so it is always asympto- undercooked pork

matic. Computed tomography (CT) or MRI shows Figure 4. The lifecycle of Taenia solium.

J Formos Med Assoc | 2008 • Vol 107 • No 7 579
S.J. Yeh, R.M. Wu

a vesicle containing a small eccentric nodule, In summary, the patient was a female Thai
which is the scolex (head). The “hole-with-dot” immigrant with neurocysticercosis who presented
imaging is pathognomonic to cysticercosis.13 There with epilepsia partialis continua. This case report
is little or no perilesional edema or enhancement highlights the importance of Taenia infection as an
due to mechanisms for successful escape from the etiology of late-onset focal seizure in immigrants
host’s immunologic system. After several years, the from endemic countries.
larva dies and an inflammatory response occurs.1
The fluid in the vesicle becomes opaque, so this
stage is called the colloidal stage. On imaging, the Acknowledgments
wall of the cyst is thickened, with perifocal edema
and contrast enhancement. Later, the cyst develops We thank Associate Professors of Parasitology Kua-
into a granuloma, which is the nodular-granular Eyre Su and Shiou-Jeng Ong for their comments
stage. On MRI, it is signal-devoid on T1- and T2- on Taenia solium infection.
weighted images and surrounded by a hyperintense
rim and perifocal edema. Finally, the granuloma
becomes a calcified nodule. The calcified stage is References
better seen on CT; on MRI, the calcified nodule
is not readily visible, but perifocal edema and 1. Garcia HH, Gonzalez AE, Evans CAW, et al. Taenia solium
contrast enhancement can be seen. In our patient, cysticercosis. Lancet 2003;362:547–56.
2. James HM. Tapeworms and seizures—treatment and
the neurocysticercosis was a parenchymal form
prevention. N Engl J Med 2004;350:215–7.
and in the colloidal stage (Figure 3).
3. Ito A, Nakao M, Wandra T. Human taeniasis and cysticer-
The symptoms of parenchymal neurocysticer- cosis in Asia. Lancet 2003;362:1918–20.
cosis are mainly due to the inflammatory response 4. Vedantam R, Durga DJ, Nguyen QD, et al. Taenia solium
induced by the degenerated or calcified cyst. Seizure taeniasis/cysticercosis in Asia: epidemiology, impact and
is the most common manifestation; others include issues. Acta Tropica 2003;87:53–60.
5. Garcia HH, Del Brutto OH. Neurocysticercosis: updated
encephalitis, meningitis, intracranial hyperten-
concepts about an old disease. Lancet Neurol 2005;4:
sion, or focal neurologic signs.15 If the cysts are 653–61.
in the ventricle or basal cistern, they may result 6. Fan PC, Chung WC. Sociocultural factors and local customs
in hydrocephalus by blocking the circulation of related to taeniasis in East Asia. Kaohsiung J Med Sci
cerebrospinal fluid, in focal signs due to direct 1997;13:647–52.
compression, or in stroke due to endarteritis. 7. Pandian JD, Thomas SV, Santoshkumar B, et al. Epilepsia
partialis continua—a clinical and electrophysiological study.
It was unclear how and where the patient
Seizure 2002;11:437–41.
ingested the eggs of T. solium. The lifespan of a 8. Juergen ET, Thomas JR, Donald WK. Epilepsia partialis
cysticercus in the human brain is estimated to be continua. A review of 32 cases. Arch Neurol 1977;34:
several years,1 and they usually do not cause symp- 266–75.
toms until they are dying. Taenia infection is rare 9. Chang SC, Lai PH, Chen WL, et al. Diffusion-weighted
MRI features of brain abscess and cystic or necrotic brain
in Taiwan6 but common in Thailand, with the
tumors. Comparison with conventional MRI. J Clin Imaging
prevalence ranging from 0% to 18.2% in a study
2002;26:227–36.
assessed by worm purging.16 In rural areas in 10. Salzman KL. Primary CNS lymphoma. In: Osborn AG,
Thailand, open-air defecation occurs and contam- Blaser SI, Salzman KL, et al, eds. Diagnostic Imaging: Brain,
inates the water supply, soil and vegetation.17 1st edition. Utah: Amirsys, 2004;I-6:122–5.
This patient did not eat uncooked vegetables, but 11. Kim TK, Chang KH, Kim CJ, et al. Intracranial tuberculoma:
comparison of MR with pathological findings. AJNR Am
her drinking water was from a well that might
J Neuroradiol 1995;16:1903–8.
have been contaminated by Taenia eggs. It might 12. Salzman KL. Neurocysticercosis. In: Osborn AG, Blaser SI,
be one of the possible sources of her parasite Salzman KL, et al, eds. Diagnostic Imaging: Brain, 1st edition.
infection. Utah: Amirsys, 2004;I-8:50–3.

580 J Formos Med Assoc | 2008 • Vol 107 • No 7
Neurocysticercosis presenting with epilepsia partialis continua

13. Garcia HH, Del Brutto OH. Imaging findings in neurocys- 16. Malinee TA. Human taeniasis in Thailand. In: Ito A, Wen H,
ticercosis. Acta Tropica 2003;87:71–8. Yamasaki H, eds. Asian Parasitology, Vol 2: Taeniasis/
14. Murray PR, Rosenthal KS, Pfaller MA. Cestodes. In: Cysticercosis and Echinococcosis in Asia. Japan: FAP
Medical Microbiology, 5th edition. St Louis: Mosby, 2005: Journal, 2005:89–98.
907–10. 17. Paron D, Jitra W. Cysticercosis in Thailand. In: Ito A, Wen H,
15. Fleury A, Dessein A, Preux PM, et al. Symptomatic human Yamasaki H, eds. Asian Parasitology, Vol 2: Taeniasis/
neurocysticercosis—age, sex and exposure factors relating Cysticercosis and Echinococcosis in Asia. Japan: FAP
with disease heterogeneity. J Neurol 2004;251:830–7. Journal, 2005:99–110.

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