Clinical Neurology and Neurosurgery 115 (2013) 507511

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Clinical Neurology and Neurosurgery

journal homepage: www.elsevier.com/locate/clineuro

Review

Isolated brainstem cysticercosis: A review

Oscar H. Del Brutto a,b, , Victor J. Del Brutto b
a
b

School of Medicine, Universidad de Especialidades Espritu Santo, Guayaquil, Ecuador

Department of Neurological Sciences, Hospital Clnica Kennedy, Guayaquil, Ecuador

a r t i c l e

i n f o

Article history:
Received 2 July 2012
Received in revised form
13 December 2012
Accepted 22 December 2012
Available online 22 January 2013
Keywords:
Cysticercosis
Neurocysticercosis
Brainstem

a b s t r a c t
Objective: Review of patients with isolated brainstem cysticercus to outline the features of this form of
neurocysticercosis.
Methods: MEDLINE and manual search of patients with isolated brainstem cysticercus. Abstracted data
included: demographic prole, clinical manifestations, neuroimaging ndings, evolutive stage of parasites neurocysticercosis, therapy, and follow-up.
Results: Twenty-nine patients were reviewed. Of these, 22 (76%) came from India. Mean age was 31 years,
and 72% were men. Parasites were located in midbrain (16 patients), pons (12 patients), and medulla (one
patient). All but three lesions were less than 10 mm in diameter and most were at or near the midline.
Most common clinical forms of presentation were isolated paresis of the third cranial nerve, internuclear
ophthalmoplegia, and crossed brainstem syndromes. Neuroimaging studies showed colloidal cysticercus
in 24 patients, vesicular cysts in four, and a calcication in one. Fourteen patients received cysticidal
drugs, eight were treated with steroids alone, and three received no therapy at all. The remaining four
patients underwent surgical resection of the lesion. Twenty-seven patients recovered completely and the
remaining two were left with mild sequelae. Control neuroimaging studies showed complete or partial
resolution of the lesion in the 18 patients in whom they were performed.
Conclusions: Isolated brainstem cysticercosis is rare. Clinical and neuroimaging ndings on admission
allowed a correct differentiation of this condition from other space-occupying lesions of the brainstem
(tuberculomas, abscesses, gliomas) in most patients. The prognosis is benign provided the patients receive
prompt therapy.
2013 Elsevier B.V. All rights reserved.

Contents
1.
2.
3.
4.

Introduction . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . .
Methods . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . .
Results . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . .
Discussion . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . .
Conict of interest . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . .
Contributors . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . .
References . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . .

1. Introduction
Neurocysticercosis is highly pleomorphic due to individual
differences in the number and location of lesions within the CNS
[1]. Cysticerci may be located in brain parenchyma, subarachnoid
space, ventricular system, or spinal cord. In the former, parasites
usually lodge in the cerebral cortex or the subcortical white matter,

Corresponding author at: Air Center 3542; PO Box 522970; Miami, FL 331522970, United States. Tel.: +593 42285790.
E-mail address: oscardelbrutto@hotmail.com (O.H. Del Brutto).
0303-8467/$ see front matter 2013 Elsevier B.V. All rights reserved.
http://dx.doi.org/10.1016/j.clineuro.2012.12.026

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due to the high vascular supply of these areas [2]. So, parenchymal
brain cysticercosis usually presents as recurrent seizures which,
in up to 70% of cases, constitutes the sole manifestation of the
disease [35]. Brainstem location of parenchymal cysticerci usually
happens in the context of disseminated disease with simultaneous
involvement of cerebral hemispheres, basal ganglia, or cerebellum
[6,7]. The occurrence of isolated brainstem cysticercosis poses
diagnostic and therapeutic challenges as this form neurocysticercosis seems to be rare, may present with clinical and neuroimaging
ndings indistinguishable from those of other brainstem lesions,
and creates concern on the potential risks related to therapy. Here,
the literature on isolated brainstem cysticercosis is reviewed to

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O.H. Del Brutto, V.J. Del Brutto / Clinical Neurology and Neurosurgery 115 (2013) 507511

outline its clinical features, and to comment on the diagnostic and

therapeutic challenges of this overlooked form of the disease.
2. Methods
A literature search of case reports or case-series of patients
with isolated brainstem cysticercosis was performed using the
electronic database of MEDLINE (National Library of Medicine,
Bethesda, MD). The search was updated to November 5, 2012,
using the key words cysticercosis and neurocysticercosis were
combined with brainstem, midbrain, mesencephalon pons,
pontine medulla, and bulbar. Limits were not applied to the
search. Thereafter, a manual search that included the authors les
as well as the list of references of reviewed papers was undertaken.
Articles were independently reviewed by the authors, and disagreements in inclusion criteria or data abstraction were resolved
through discussion and consensus.
Selected studies included original data on patients with intrinsic
brainstem cysticercosis. Patients with subarachnoid or ventricular
cysts compressing the brainstem, as well as those with multiple
lesions involving both the brainstem and cerebral hemispheres
were excluded. To be eligible, the patient must have had isolated
cysticercosis infection of the midbrain, pons or medulla oblongata
on neuroimaging studies, and the information given in the original
publication must be clear enough to allow its inclusion as a denitive case of neurocysticercosis according to accepted diagnostic
criteria [8]. Besides clinical and neuroimaging ndings, abstracted
data of selected articles included country of origin of publications,
demographic prole of reported patients, specic location of parasites, therapy, and follow-up.
3. Results
The search detected 28 papers reporting 32 patients with isolated brainstem cysticercus [936]. Three of these papers (all
describing single case-reports) were not included in the review; in
two cases, the original publication could not be found [34,35], and
in the other, information was incomplete [36]. Some other articles
mentioned the occurrence of brainstem cysticercus among large
series of patients with specic oculomotor disturbances, but were
not included as the characteristics of patients were not detailed,
and data could not be abstracted [37,38].
Table 1 describes the characteristics of the 29 patients included
in this review. Mean age was 31.3 14.4, and 21 (72%) were men.
Twenty-two (76%) patients were reported from India, and the other
seven came from Mexico (two cases), Brazil, Korea, Nicaragua,
Nepal and Thailand (one case each). Cysticerci were located in the
midbrain in 16 patients, in the pons in 12, and in the medulla in
the remaining case. In all but three patients, cysticerci were of less
than 10 mm in diameter, and were located at or near the midline.
The most common clinical form of presentation was as an
isolated complete or partial paresis of the third cranial nerve
(unilateral or bilateral) in ten patients, followed by gaze paresis
of central origin in seven (internuclear ophthalmoplegia, oneand-a-half syndrome, lateral and upward gaze paresis, paralysis
of convergence), crossed brainstem syndromes in ve (Weber,
MillardGubler, Claude syndromes), ataxic-hemiparesis in two,
and isolated abducens palsy, facial myokymia, hemisensory decit,
bradycardia, and abnormalities of stance and gait in one patient
each. According to neuroimaging studies (MRI in 26 patients and
CT in the remaining three), 24 patients (83%) had a cysticercus in
the colloidal stage (with a discernible scolex in eight cases), four
had a vesicular (viable) cyst, and the remaining patient had a calcication surrounded by edema. Immune diagnostic tests for the
detection of anti-cysticercal antibodies (using ELISA in CSF in all

but one patient in whom serum EITB was performed) were positive in 13 of 17 patients. In no patient was investigated the source
of infection; however, all lived in cysticercosis endemic areas.
Fourteen patients received cysticidal drugs, eight were treated
with steroids alone, and three received no therapy at all. The
remaining four patients underwent surgical resection of the brain
lesion for diagnostic purposes. There were no complications related
to medical or surgical therapy in any case. Twenty-seven patients
recovered completely and the remaining two were left with
mild sequelae. In the latter, no therapy was given. Patients were
followed-up for a mean of 4.7 5.3 months. Control neuroimaging
studies showed complete or partial resolution of the lesion in the
18 patients in whom they were performed.

4. Discussion
This review suggests that isolated brainstem cysticercosis is
rare. Only 29 cases were found after a thorough search of the literature, and most of them came from India. However, these ndings
must be interpreted with caution as the nature of a series based
on the literature is quite partial, since a number of cases may have
been reported in local journals or not reported at all.
That most cases came from India should not be surprising since
it is just in this country where the most common presentation
of neurocysticercosis is a single cysticercus granuloma [39]. The
benign course of patients reported in this review irrespective of
their location in the brainstem, where vital structures may be compromised goes in accordance with the overall benign prognosis
of patients with single parenchymal brain granulomas. No patient
presented with loss of consciousness or severe abnormalities in the
cardiac rhythm. Instead, all complained of focal neurological signs
pointing out to the specic brainstem area involved.
From the clinical point of view, patients with isolated brainstem cysticercosis differ from those with other space-occupying
mass lesions of the brainstem. None of the patients reviewed here
presented with intracranial hypertension, which is rather common in patients with brainstem gliomas, tuberculomas, and other
infectious diseases of the brainstem [4042]. Likewise, fever and
meningeal signs were absent in patients with brainstem cysticercosis but occur in a sizable proportion of patients with other
brainstem infections [40,41]. Neuroimaging ndings also suggested
the diagnosis in many cases, as brainstem cysticerci most often
appeared as small, well-dened, rounded enhancing lesions (Fig. 1),
whereas other space-occupying lesions are most often large and
show heterogeneous contrast enhancement or exophytic growth;
visualization of the scolex (in 8 of 24 patients with colloidal cysts)
provided further evidence favoring the diagnosis of cysticercosis.
When applying accepted diagnostic criteria for neurocysticercosis
[8], a denitive diagnosis could be done in 13 of 29 patients on
admission, as 12 patients presented with one absolute diagnostic
criterion (visualization of the scolex within the lesion) and one had
two major diagnostic criteria (suggestive lesion on neuroimaging
plus a positive serum EITB test). In the remaining 16 cases, the initial
probable diagnosis of neurocysticercosis was conrmed on followup (resolution of the lesion spontaneously or after therapy with
cysticidal drugs in 12 patients, and histological demonstration of
the parasite in the remaining four).
Only four (14%) patients with isolated brainstem cysticercus
underwent surgical resection of the lesion. This percentage of surgical procedures was mainly related to physician awareness of the
possibility of neurocysticercosis in patients presenting with a single brainstem lesion (as all came from disease-endemic areas),
and is considerably lower than the number of surgical procedures
performed in patients with a single intracranial cysticercus diagnosed in non-endemic countries, where 34% of patients detected

Table 1
Characteristics of 29 patients with isolated brainstem cysticercosis.
Age/gender

Clinical manifestations

Location

Stage of cysticercus

Immune tests

Treatment

Follow-up

Clinical evolution

Control imaging

Anand and Prasad [9]

Arruda et al. [10]
Barinagarrementeria and Del Brutto [11]
Bhatia et al. [12]
Bhattacharya and Mitra [13]
Chandran et al. [14]
Chotmongkol et al. [15]
Del Brutto and Sotelo [16]
Gupta [17]
Katz et al. [18]
Khurana et al. [19]
Lath and Rajshekar [20]

16/m
37/m
25/f
45/m
28/f
35/m
25/f
30/f
57/f
31/m
22/m
21/m
36/m
32/f
43/m
15/m
29/m
34/m
32/m
45/f
58/m
45/m
18/m
16/m
26/m
25/m
5/m
8/f
68/m

Abducens palsy
Ataxic hemiparesis
Ataxic hemiparesis
Facial myokymia
Internuclear ophthalmoplegia
Internuclear ophthalmoplegia
Third cranial nerve palsy
MillardGubler syndrome
Abnormalities of stance and gait
Third cranial nerve palsy
Third cranial nerve palsy
Paralysis of convergence
Hemisensory decit
Lateral gaze paresis
MillardGubler syndrome
Third cranial nerve palsy
One-and-a-half syndrome
Third cranial nerve palsy
Bradycardia
Bilateral ptosis
Bilateral ptosis
Weber syndrome
One-and-a-half syndrome
Third cranial nerve palsy
Paralysis of upward gaze
Weber syndrome
Third cranial nerve palsy
Third cranial nerve palsy (bilat)
Claude syndrome

Pons
Pons
Pons
Pons
Pons
Pons
Midbrain
Pons
Medulla
Midbrain
Midbrain
Midbrain
Pons
Pons
Pons
Midbrain
Midbrain
Midbrain
Pons
Midbrain
Midbrain
Midbrain
Pons
Midbrain
Midbrain
Midbrain
Midbrain
Midbrain
Midbrain

Colloidal (scolex)
Vesicular
Vesicular
Colloidal
Colloidal
Vesicular
Calcication (edema)
Colloidal
Colloidal
Colloidal
Colloidal
Colloidal
Colloidal
Colloidal (scolex)
Colloidal
Colloidal (scolex)
Colloidal (scolex)
Colloidal
Vesicular
Colloidal
Colloidal (scolex)
Colloidal (scolex)
Colloidal
Colloidal
Colloidal (scolex)
Colloidal (scolex)
Colloidal
Colloidal
Colloidal

ELISA+

ELISA+

ELISA+

ELISA+
EITB+
ELISA+

ELISA
ELISA

ELISA+

ELISA+

ELISA+

ELISA+
ELISA+

ELISA+
ELISA
ELISA
ELISA+

Albendazole
Surgical resection
Surgical resection
Steroids
Steroids
Albendazole
Albendazole
Steroids
Albendazole
Praziquantel
Steroids
Albendazole
No therapy
No therapy
Surgical resection
Steroids
Albendazole
Steroids
Surgical resection
Albendazole
Albendazole
Steroids
Albendazole
Albendazole
No therapy
Steroids
Albendazole
Albendazole
Albendazole

3 months
5 months
1 montha
7 months
6 months
2 weeks
2 months
1 year
3 weeks
1 montha
1 year
2 years
18 months
3 years
3 months
1 weeka
1 month
1 year
3 months
3 months
6 weeksa
1 month
3 months
3 weeks
3 months
2 months
3 months
3 months
3 months

Recover
Recover
Recover
Recover
Recover
Recover
Recover
Recover
Recover
Recover
Recover
Recover
Mild sequelae
Recover
Recover
Recover
Recover
Recover
Recover
Recover
Recover
Recover
Recover
Recover
Mild sequelae
Recover
Recover
Recover
Recover

Resolution

Resolution
Resolution
Resolution

Resolution
Calcication

Resolution
Resolution
Resolution
Size reduction
Size reduction
Resolution

Size reduction
Resolution

Resolution
Resolution
Size reduction
Resolution

Meena et al. [21]

Mesraoua et al. [22]
Mokta et al. [23]
Nadkarni et al. [24]
Naphade et al. [25]
Netravathi et al. [26]
Patel et al. [27]
Ranjith et al. [28]
Ranjith et al. [29]
Shukla [30]
Singh et al. [31]
Singhi et al. [32]
Song et al. [33]
a

O.H. Del Brutto, V.J. Del Brutto / Clinical Neurology and Neurosurgery 115 (2013) 507511

Authors

Precise follow-up time not specied.

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O.H. Del Brutto, V.J. Del Brutto / Clinical Neurology and Neurosurgery 115 (2013) 507511

Fig. 1. Contrast-enhanced computed tomography showing colloidal cysticercus in

the midbrain (arrow).

in a recent review were operated on for diagnostic purposes [43].

While a single parenchymal brain cysticercus (irrespective of its
location) is a diagnostic challenge, proper application of diagnostic criteria for neurocysticercosis (including a trial with cysticidal
drugs) together with increased physician awareness, allow the correct diagnosis in most cases, obviating the need of surgery.
Fourteen of the 25 non-operated patients were treated with cysticidal drugs. Their prognosis was good, with clinical improvement
and resolution of the lesion on control neuroimaging studies. However, the same was noted in patients who received steroids alone.
So, it seems that the prognosis of this form of neurocysticercosis is
benign in most patients.
Conict of interest
Nothing to disclose.
Contributors
Design of the study was done by O.H.D.; review and abstraction
of data by O.H.D. and V.J.D.; analysis of data by V.J.D.; and drafting
of the manuscript was done by O.H.D. and V.J.D.
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