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ORIGINAL ARTICLE

Three Cases of Imported Neurocysticercosis in Northern Italy
Maria Teresa Giordani, MD,∗ Francesca Tamarozzi, PhD,† Federico Cattaneo, MD,†
and Enrico Brunetti, MD†
∗

Infectious and Tropical Diseases Unit, San Bortolo Hospital, Vicenza, Italy; † Department of Infectious Diseases, IRCCS San
Matteo Hospital Foundation-University of Pavia, Pavia, Italy
DOI: 10.1111/jtm.12066

Background. Neurocysticercosis (NCC) is an important cause of adult-onset seizures in endemic areas, whereas it is emerging in
some nonendemic areas as well because of extensive immigration.
Method. We describe three cases of imported NCC recently admitted to San Bortolo Hospital in Vicenza, located in Northern
Italy.
Results. All patients were immigrants. One patient was human immunodeficiency virus positive with severe immunosuppression.
The diagnosis of NCC was made on the basis of magnetic resonance results; failure of anti-Toxoplasma, antitubercular, and
antifungal therapy; and regression of the cystic lesions after empiric therapy with albendazole. Serology was positive in only one
case. In one patient, NCC was diagnosed by biopsy of the brain lesion.
Conclusion. In nonendemic countries, NCC should be included in the differential diagnosis of all patients coming from endemic
areas with seizures, hydrocephalus, and compatible lesions on brain imaging. Long-term follow-up is required but may be difficult
to implement because these patients tend to move in search of employment. Screening of patient’s household contacts for Taenia
solium infection should always be carried out.

N

eurocysticercosis (NCC), caused by the larval
stage (cysticercus) of the pork tapeworm Taenia
solium, primarily involves the central nervous system and
is the most common cause of acquired epilepsy in the
developing world.1 NCC is endemic to many countries
but in Italy, as in Western Europe in general, the
few cases reported are mostly imported from endemic
areas.2 – 4
Methods and Results
We describe the complex presentation of three
cases of NCC, one of which occurred in a human
immunodeficiency virus (HIV)-positive patient. The
difficulties of the diagnostic and clinical management
Part of this material was presented as a poster at the 21st
ECCMID/27th ICC, Milan, Italy (P866, page S211).
Corresponding Author: Maria Teresa Giordani, MD,
Infectious and Tropical Diseases Unit, San Bortolo
Hospital, via Rodolfi 37, I-36100 Vicenza, Italy. E-mail:
giordanimt@gmail.com

process and the increasing relevance of this imported
infection outside the endemic areas are discussed.
Patient 1
A 34-year-old man from Senegal, who had been living in
Italy for 6 years, was admitted to San Bortolo Hospital,
Vicenza, Italy, in February 2010 because of seizures, of
which he had a history since 2008.
Magnetic resonance (MR) in T1- and T2-weighted
images (WIs) showed a brain lesion consisting of
two adjacent cysts isointense with the cerebral spinal
fluid (CSF), surrounded by edema, and showing ring
enhancement after gadolinium (Figure 1A–C). The
serology for T solium both on serum and CSF was
negative (Immunoblotting, LD Bio, Lyon, France
performed at the Parasitology Laboratory, San Matteo
Hospital Foundation, Pavia, Italy), as was ova and
parasite (O&P) fecal examination. No further lesions
compatible with parasitic infections were found, with the
possible exception of a pulmonary nodule and a single
calcification in the right hepatic lobe on ultrasound.
Fundoscopic examination was negative.
The cerebral lesion was surgically removed in
April 2010 and its pathological examination showed a
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Giordani et al.

(A)

(B)

(C)

(D)

Figure 1 Case 1. (A–C) Magnetic resonance (MR) showing a cortical–subcortical lesion in the left frontal lobe having multiple
cystic areas with content isointense with cerebrospinal fluid (CSF) (arrows) and surrounded by edema (star). (A) T1-weighted
image (WI); (B) T2-WI; and (C) ring enhancement of the cyst wall after gadolinium. (D) Hematoxylin–eosin-stained histology
section of brain lesion showing Cysticercus cellulosae scolex within degenerated material. Original magnification 20×, bar scale
500 μm.

T solium viable larva and degenerated parasitic material
within a cystic cavity (Figure 1D). After surgery the
patient was treated with albendazole 400 mg bid for
8 days, praziquantel 1.5 g once on the fourth day,
dexamethasone 4 mg bid for 8 days, and carbamazepine
200 mg tid (the latter started before neurosurgery).
Patient 2
A 24-year-old African HIV-2-positive man from
Burkina Faso, an observant Muslim, who had been
living in Italy for 8 years, was admitted to San Bortolo
Hospital in May 2010 with fever, pneumonia, and sepsis.
He had an extremely low CD4 count (24 /mm3 ).
During hospitalization the patient had a seizure.
A computed tomography (CT) scan of the brain
revealed a hypodense cortical–subcortical lesion in the
left frontal-basal lobe, whereas a previous brain CT
examination done in June 2009 after a car accident
showed a negative result. MR (Figure 2A and B)
revealed two adjacent cysts, isointense with CSF
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in T1-WI and T2-WI, surrounded by edema and
ring enhancement after gadolinium. Empirical antiToxoplasma therapy with sulfadiazine/pyrimethamine
and antifungal and antitubercular therapy were
ineffective, and after a repeat MR scan with unchanged
findings (Figure 2C), NCC was suspected. Serology for
Toxocara canis, Echinococcus granulosus, Toxoplasma gondii,
Schistosoma spp., Strongyloides stercoralis (IgG, indirect
fluorescent antibody test), Coxiella burnetii, Aspergillus
spp., and Bartonella spp., and circulating antigens for
Cryptococcus and Cytomegalovirus were negative, as were
QuantiFERON-TB test and O&P fecal examination.
Serology for T solium was also negative (immunoblotting
on serum and CSF, LD Bio, Lyon, France, performed
at the Parasitology Laboratory, San Matteo Hospital
Foundation, Pavia, Italy). The low platelet count
precluded cerebral biopsy; therefore, the patient was
started on empiric treatment with albendazole 15 mg/kg
in two divided doses plus low-dose steroidal therapy
with dexamethasone 4 mg bid. An ophthalmologic

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Imported Neurocysticercosis in Italy

(A)

(B)

(C)

(D)

Figure 2 Case 2. (A and B) Pretreatment magnetic resonance (MR) showing a frontal-basal lesion on the left hemisphere
with multiple cystic areas with content isointense with cerebrospinal fluid (CSF) (arrows) and surrounded by edema (star). (A)
T2-weighted image (WI) and (B) T1-WI. (C) T1-WI MR executed 3 weeks later showing the lesion unchanged (arrow) after
anti-Toxoplasma, broad-spectrum antibiotic and antifungal and antitubercular therapy. (D) T1-WI MR executed 48 days after start
of albendazole and steroidal therapy showing substantial reduction of the lesion size with disappearance of the cystic component
and perilesional edema (arrow).

evaluation including funduscopic examination was
performed before and after treatment started, with a
negative result. The therapy was well tolerated and
no paradoxical reactions occurred. After 48 days of
treatment, near-complete disappearance of the brain
lesion was seen on MR (Figure 2D). The serology for
T solium was repeated 6 months after treatment (ELISA
IgG Tenia Solium, DRG Instruments, Marburg,
Germany, and Immunoblotting, LD Bio, performed
at the Parasitology Laboratory, Don Calabria Hospital,
Negrar, Verona, Italy) which again returned negative.
Patient 3
A 55-year-old woman born and raised in Brazil,
previously diagnosed with NCC and treated with
ventriculo-peritoneal drainage in 1999 in her country,
was admitted to San Bortolo Hospital following
progressive loss of consciousness in February 2012.
A brain CT scan showed hydrocephalus, multiple
small calcifications in the cerebral parenchyma,

and inhomogeneous space-occupying lesions in the
cerebellum (Figure 3A and B) and in the right parietaloccipital area. MR showed a cystic lesion surrounded
by marked edema in the cerebellar vermis (Figure 3C)
and confirmed the hydrocephalus (Figure 3C) and right
parietal-occipital cystic lesion surrounded by edema
(Figure 3D). Serology for T solium returned positive
in serum and liquor in two repeated tests (ELISA Ig G
Tenia Solium, DRG Instruments, and Immunoblotting,
LD Bio, performed at the Parasitology Laboratory,
Don Calabria Hospital, Negrar, Verona, Italy). Analysis
of the CSF showed signs of disruption of bloodbrain barrier (proteins 72 mg/dL and WBC 300/field,
mainly lymphocytes). No ocular localizations were
found during the fundoscopic examination.
The patient underwent surgical replacement of the
ventricular drainage and medical therapy with intravenous dexamethasone 4 mg bid and oral albendazole 15 mg/kg/day in two divided doses for 1 month.
The neurological conditions improved quickly, no
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Giordani et al.

(A)

(C)

(B)

(D)

Figure 3 Case 3. (A) TC showing multiple calcified nodules in both hemispheres (arrows) and ventricular dilatation (star). (B)
T1-weighted image (WI) showing cystic lesion in the medulla oblongata (arrow) surrounded by edema and ventricular dilatation
(star). (C) T2-WI magnetic resonance (MR) showing the details of an extraparenchymal intraventricular cyst in the IV ventricle
(circle). (D) T2-WI MR showing a small parietal-occipital cystic lesion in the right surrounded by edema (arrow).

paradoxical reactions to the treatment were recorded,
and on discharge she did not show any neurological
deficit. A follow-up was strongly recommended on her
return to Brazil.
Discussion
Human NCC is considered a rare disease in Italy, but
the last review on Italian cases dates back to 1986,5 and
although notification of T solium infection and/or NCC
in humans is mandatory in Italy, there is no national
register for human cases. Seven autochthonous and 27
imported cases of NCC were reported in Italy from
1990 to 2011.6
The infection with the adult parasite is often longlasting and asymptomatic, and the cystic larval forms can
persist in the brain for years without causing symptoms,
which may eventually develop, most of the times as
a consequence of cyst degeneration and induction of
perilesional inflammation.7,8
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In patient 1, the long period between immigration
and development of seizures is not surprising;
particularly, in the case of a single granuloma, seizures
can be considered a late manifestation of a disease
acquired many years before.9 On the contrary, patient
2, who had a previous negative CT scan in 2009, could
have been infected in Italy. Indeed, carriers of T solium
may represent the source of the infection in nonendemic
countries, particularly for close contacts, in the absence
of infected swine in the territory.4,10,11
The diagnosis and treatment of NCC are complex
and made more difficult owing to the poor knowledge
of this disease in nonendemic countries.12 Moreover,
even when NCC is suspected, a negative serology
does not rule out the infection, as patients with only
one lesion are often seronegative.12 In a review of
case reports and case series of patients with single
cysticercus granulomas in nonendemic areas, 41% of
cases had a negative serology.9 The behavior of serology
in HIV-positive patients is also still unclear.13,14 In

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Imported Neurocysticercosis in Italy
Table 1

Summary of the epidemiological, clinical, laboratory, and instrumental data of the three patients

Age/
gender

Country
of
origin

Length
of
immigration

Case 1 34 years/M Senegal

8 years

Case 2 24 years/M Burkina
Faso

6 years

Case 3 55 years/F Brazil

1 month

Clinical
presentation
Recurrent
seizures

Classification

Neuroimaging

Single
parenchymal lesion,
active

EITB

Single viable
Negative on
parenchyserum and
mal lesion
CSF
with two
adjacent
cysts, edema
Single seizure, Single
Single viable
Negative on
delirium
parenchyparenchyserum
mal lesion,
mal lesion
(before and
active
with two
after
adjacent
therapy)
cysts, edema
Lethargy,
Multiple
Multiple
Positive on
intracranial
parenchycerebral
serum and
hypertenmal active
lesions,
CSF
sion,
and calcified
calcified
meningoenlesions,
nodules,
cephalitis
extraintraventricparenchyular cyst,
mal
ventricular
NCC
dilatation

Comorbidities

Treatment

—

Surgery and
steroids, ABZ
(8 days), and
PZQ (single)

HIV2/AIDS

Steroids and
ABZ
(60 days)

—

Ventricular
drainage,
steroids, and
ABZ
(28 days)

EITB = enzyme-linked immunoelectrotransfer blot; CSF = cerebrospinal fluid; NCC = neurocysticercosis.

these cases, the importance of repeating the serological
tests after treatment of a seronegative case to detect
seroconversion has been pointed out,15 but it was not
diagnostic in our case. To make matters worse, T solium
serology is not available in many European centers,
including our hospital; therefore, samples need to be
sent to a referral laboratory.
The imaging of brain lesions is the cornerstone
of NCC diagnosis. The epidemiology of brain lesions
varies between geographical areas, with single lesions
being mainly found in India and multiple cysticerci in
different stages being found in Asia, Africa, and Latin
America.10 In our series, this is the case in patient 3,
from Brazil, while the limited number of cysts in the
patients from Sub-Saharan Africa may result from mild
infection.10 A set of diagnostic criteria to classify NCC
cases as definitive or probable has been formalized by
a group of experts.16 All three cases were accordingly
classified as definitive NCC (Table 1).
The differential diagnosis of NCC includes
noninfectious (neoplasm, vascular malformations, and
scars) and infectious lesions [bacterial or fungal
abscess, neuro-toxoplasmosis, cerebral toxocariasis,
cryptococcosis, tuberculosis (TB), and strongyloidiasis].
NCC in HIV-infected patients poses the most difficult
problems in the differential diagnosis and in the
therapeutic approach, as different pathogens can coexist
and produce cerebral lesions having similar appearance
on CT and MR.17 In HIV-positive patients with a
CD4 count <200/mm3 , toxoplasmosis is the most
frequent cause of cerebral necrotic lesions. However,
these are more often multiple and lack a clear cystic
appearance. The lack of response to anti-Toxoplasma

therapy can be a valid diagnostic aid, while the
result of serology for Toxoplasma can be misleading.
Massive infections with Mycobacterium tuberculosis and
Strongyloides stercoralis are other possible causes of
cerebral lesions.18,19 In our case, these etiologies were
ruled out by serology, unsuccessful TB treatment,
and stool examination. Although S stercoralis serology
can be negative in immunocompromised patients
with hyperinfection syndrome and disseminated
strongyloidiasis, treatment with albendazole is generally
poorly effective, and gastrointestinal and cutaneous
symptoms are more frequent.19,20 Albendazole and
praziquantel are commonly used to treat active NCC
and treatment should be based on NCC staging and
presence or absence of eye involvement, taking into
account both the risk of adverse reactions after treatment
due to inflammation exacerbation and the benefit of
treatment in patients with recurrent seizures.6,21,22
Patient 1 received pharmacological therapy after
surgery, therefore a short treatment was considered
appropriate. In HIV patients, treatment of cerebral
parenchymal abscesses is recommended to avoid
the development of a calcified lesion,23,24 and the
concomitant use of steroids is mandatory in these
cases to reduce the risk of a paradoxical reaction.25
Furthermore, a long-term treatment was chosen in this
case given the presence of a viable lesion26,27 and the
patient’s general condition.
Finally, in patient 3, emergency drainage was
needed prior to any other therapy because of mental
impairment due to ventricular dilatation.28 Treatment
with dexamethasone and albendazole with a prolonged
course of 4 weeks was decided in this case of
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extraparenchymal NCC,29 which is known to have an
aggressive behavior, and to reduce the edema and avoid
the complications of arachnoiditis.24
Conclusion
NCC should be included in the differential diagnosis
of seizures, hydrocephalus, and compatible lesions on
brain imaging in nonendemic countries, especially in
patients from endemic areas, and in their household
contacts.11,30 In developed countries, the risk of
autochthonous transmission is small because of the
high hygienic standards and control of meat production;
however, household contacts of people carrying T solium
are at higher risk.11 The absence of pork consumption
should not be a sufficient epidemiological element to
exclude NCC.10 Indeed, the source of infection in
these cases is most likely a contaminated food source,
such as a contaminated handler, and in any case all
household contacts of diagnosed NCC should be tested
for infection. Unfortunately, this is not always possible
because of the high mobility of immigrants. For the
same reason, a long-term follow-up is difficult to
obtain.
NCC should always be included in the differential
diagnosis of brain lesions in HIV-positive patients
especially if empiric antitoxoplasmosis therapy fails, and
the interactions between NCC and HIV deserve further
study.
Acknowledgments
We are particularly grateful to Sam Goblirsch, MD,
for reviewing the manuscript. We also thank the
Department of Neuroradiology, Vicenza Hospital and
Daniela Danieli, MD, for providing the images and
Alessandro Segna, MD, for reviewing the neurosurgical
aspects.
Declaration of Interests
The authors state that they have no conflicts of interest.
References
1. Commission on Tropical Diseases of the International
League Against Epilepsy. Relationship between epilepsy
and tropical diseases. Epilepsia 1994; 35:89–93.
2. Chatel G, Gulletta M, Scolari C, et al. Neurocysticercosis
in an Italian traveler to Latin America. Am J Trop Med
Hyg 1999; 60:255–256.
3. Raffaldi I, Scolfaro C, Mignone F, et al. An uncommon
cause of seizures in children living in developed countries:
neurocysticercosis—a case report. Ital J Pediatr 2011;
37:9.
4. Del Brutto OH. Neurocysticercosis in Western Europe:
a re-emerging disease? Acta Neurol Belg 2012;
112:335–343.
J Travel Med 2014; 21: 17–23

Giordani et al.
5. Bussone G, La Mantia L, Frediani F, et al. Neurocysticercosis: clinical and therapeutic considerations. Review of
Italian literature. Ital J Neurol Sci 1986; 7:525–529.
6. Del Brutto OH, Garcia HH. Neurocysticercosis
in nonendemic countries: time for a reappraisal.
Neuroepidemiology 2012; 39:145–146.
7. Garcia HH, Gonzalez AE, Evans CA, Gilman RH. Taenia
solium cysticercosis. Lancet 2003; 362:547–556.
8. de Souza A, Nalini A, Kovoor JM, et al. Natural history of
solitary cerebral cysticercosis on serial magnetic resonance
imaging and the effect of albendazole therapy on its
evolution. J Neurol Sci 2010; 288(1-2):135–141.
9. Del Brutto OH, Nash TE, Garcia HH. Cysticerci-related
single parenchymal brain enhancing lesions in nonendemic countries. J Neurol Sci 2012; 319(1-2):32–36.
10. Winkler AS. Neurocysticercosis in sub-Saharan Africa: a
review of prevalence, clinical characteristics, diagnosis, and
management. Pathog Glob Health 2012; 106:261–274.
11. Moore AC, Lutwick LI, Schantz PM, et al. Seroprevalence
of cysticercosis in an orthodox Jewish community. Am J
Trop Med Hyg 1995; 53:439–442.
12. Garcia HH. Neurocysticercosis in immigrant populations.
J Travel Med 2012; 19:73–75.
13. Garcia HH, Rodriguez S, Gilman RH, et al.
Neurocysticercosis: is serology useful in the absence
of brain imaging? Trop Med Int Health 2012;
17:1014–1018.
14. Foyaca-Sibat H, Cowan LD, Carabin H, et al. Accuracy
of serological testing for the diagnosis of prevalent
neurocysticercosis in outpatients with epilepsy, Eastern
Cape province, South Africa. PLoS Negl Trop Dis 2009;
3:e562.
15. Dournon N, Epelboin L, Brion MC, et al. Seroconversion
of neurocysticercosis occurring after anti-helminthic
treatment. J Travel Med 2012; 19:383–386.
16. Del Brutto OH, Rajshekhar V, White AC Jr, et al.
Proposed diagnostic criteria for neurocysticercosis.
Neurology 2001; 57:177–183.
17. Lattuada E, Lanzafame M, Concia E, Vento S. Intracranial
mass lesions in HIV/AIDS patients from developing
countries endemic for neurocysticercosis. Int J STD AIDS
2007; 18:144–145.
18. Masdeu JC, Tantulavanich S, Gorelick PP, et al. Brain
abscess caused by strongyloides stercoralis. Arch Neurol
1982; 39:62–63.
19. Buonfrate D, Requena-Mendez A, Angheben A, et al.
Severe strongyloidiasis: a systematic review of case reports.
BMC Infect Dis 2013; 13:78.
20. Mascarello M, Gobbi F, Angheben A, et al. Prevalence
of strongyloides stercoralis infection among HIVpositive immigrants attending two Italian hospitals, from
2000 to 2009. Ann Trop Med Parasitol 2011; 105:
617–623.
21. Garcia HH, Evans CA, Nash TE, et al. Current
consensus guidelines for treatment of neurocysticercosis.
Clin Microbiol Rev 2002; 15:747–756.
22. Coyle CM, Tanowitz HB. Diagnosis and treatment of
neurocysticercosis. Interdiscip Perspect Infect Dis 2009;
2009:180742.
23. Bhigjee AI, Rosemberg S. Optimizing therapy of seizures
in patients with HIV and cysticercosis. Neurology 2006;
67(12 Suppl 4):S19–S22.
24. Nash TE, Garcia HH. Diagnosis and treatment of
neurocysticercosis. Nat Rev Neurol 2011; 7:584–594.

Imported Neurocysticercosis in Italy
25. Prasad S, MacGregor RR, Tebas P, et al. Management
of potential neurocysticercosis in patients with HIV
infection. Clin Infect Dis 2006; 42:e30–e34.
26. Garcia HH, Pretell EJ, Gilman RH, et al. A trial of
antiparasitic treatment to reduce the rate of seizures
due to cerebral cysticercosis. N Engl J Med 2004; 350:
249–258.
27. Del Brutto OH, Roos KL, Coffeyn C, et al. Meta-analysis:
cysticidal drugs for neurocysticercosis: albendazole and
praziquantel. Ann Intern Med 2006; 145:43–51.

23
28. Nash TE, Pretell EJ, Lescano AG, et al. Perilesional
brain oedema and seizure activity in patients with calcified
neurocysticercosis: a prospective cohort and nested casecontrol study. Lancet Neurol 2008; 7:1099–1105.
29. Figueroa JJ, Davis LE, Magalhaes A. Extraparenchymal
neurocysticercosis in Albuquerque, New Mexico. J
Neuroimaging 2011; 21:38–43.
30. Brunetti E, White AC Jr. Cestode infestations: hydatid
disease and cysticercosis. Infect Dis Clin North Am 2012;
26:421–435.

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