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Disseminated neurocysticercosis with bilateral papilledema: a case report

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Background: Ocular cysticercosis is a disease which rarely involves cutaneous skin and the optic nerve. Patients with clinical presentation of subcutaneous nodules and papilledema should always be evaluated for cysticercosis. Case presentation: We report a rare case of ocular cysticercosis with multiple disseminated subcutaneous nodules and papilledema in both eyes. A 22-year-old Brahmin man presented with complaints of gradual loss of vision in both eyes and multiple small masses all over his body. On clinical evaluation, multiple subcutaneous nodules were seen on his face, mandibular area, elbow, arm, and abdomen. A fundus evaluation showed bilateral blurred disc margin. The case was managed with steroids and anti-parasitic drugs. Conclusion: This case report highlights the importance of ruling out neurocysticercosis in cases with multiple disseminated subcutaneous nodules and papilledema.
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C A S E R E P O R T Open Access
Disseminated neurocysticercosis with
bilateral papilledema: a case report
Ruchi Shrestha
1*
and Amin Kumar Shrestha
2
Abstract
Background: Ocular cysticercosis is a disease which rarely involves cutaneous skin and the optic nerve. Patients
with clinical presentation of subcutaneous nodules and papilledema should always be evaluated for cysticercosis.
Case presentation: We report a rare case of ocular cysticercosis with multiple disseminated subcutaneous nodules
and papilledema in both eyes. A 22-year-old Brahmin man presented with complaints of gradual loss of vision in
both eyes and multiple small masses all over his body. On clinical evaluation, multiple subcutaneous nodules were
seen on his face, mandibular area, elbow, arm, and abdomen. A fundus evaluation showed bilateral blurred disc
margin. The case was managed with steroids and anti-parasitic drugs.
Conclusion: This case report highlights the importance of ruling out neurocysticercosis in cases with multiple disseminated
subcutaneous nodules and papilledema.
Keywords: Ocular cysticercosis, Disseminated subcutaneous nodules, Papilledema
Background
Human cysticercosis is an accidental event, which results
from either ingestion of cysticercus larvae in raw or
inadequately cooked pork or ingestion of Taenia solium
in contaminated water, food, and vegetables, or autoinfec-
tion due to poor hygiene [1]. Ocular cysticercosis may
affect almost all eye tissues. The vitreous cavity, subretinal
space, and subconjunctival space are common sites while
involvement of other regions (such as extraocular muscle
and optic nerve) is relatively less common [2].
We report a rare case of ocular cysticercosis with
multiple disseminated subcutaneous nodules on the body
with bilateral papilledema with multiple calcified cysts with
scolex in brain parenchyma and cerebellum on computed
tomography (CT) scan. Ocular cysticercosis is a disease
which rarely involves cutaneous skin and optic nerve. This
case report highlights the importance of always evaluating
patients with clinical presentation of subcutaneous nodules
and papilledema for cysticercosis.
Case presentation
A 22-year-old Brahmin man presented with com-
plaints of gradual, painless, progressive loss of vision
in both eyes for 1 month. It was associated with com-
plaints of headache and dizziness for 1 month. He had a
history of multiple painless swellings over his body for the
past 1 year. Multiple swellings were present on his face,
mandibular area, elbow, arm, and abdomen. His headache
was intense on awakening and bending down position. He
had a history of fever for 2 days after intake of anti-
filariasis medication 1 month back. There was no his-
tory of vomiting, unconsciousness, seizures, or change
in behavior. He had a history of using tablet amitriptyl-
ine 75 mg before sleep for headache for the past 15
days. He was a plumber by occupation. He did not
smoke tobacco or drink alcohol; he was not a vegetarian
by diet and had a history of eating wild pork meat. There
was no significant history of similar illness in his family.
There was no significant social and environmental history.
On examination he was healthy and well oriented to time,
place, and person. His visual acuity was 6/12 in both eyes.
Intraocular pressure was 12 mmHg in both eyes. He had
multiple small pea-sized nodules which were soft, mobile,
non-tender, and well defined; each nodule was approxi-
mately 2 × 2 cm. There were no signs of inflammation on
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* Correspondence: drruchishrestha@gmail.com
1
Department of Ophthalmology, Reiyukai Eiko Masunaga Eye Hospital,
Banepa-1, Kavrepalanchok District, Nepal
Full list of author information is available at the end of the article
Shrestha and Shrestha Journal of Medical Case Reports (2019) 13:295
https://doi.org/10.1186/s13256-019-2227-0
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his face, mandibular area, elbow, arm, and abdomen
(Fig. 1).
His vital signs and systemic examination were normal.
He had no neurological deficit. Pupillary reaction was
sluggish in both eyes, otherwise the anterior segment
was unremarkable. Posterior segment revealed papille-
dema in both eyes in the form of blurred, elevated disc
margin and tortuous dilated vessels. Parapapillary
hemorrhage was present in his left eye. The macula was
healthy with good foveal reflex (Fig. 2).
Blood reports were hemoglobin 11.5 gm%, neutrophils
(N) 55%, lymphocytes (L) 22%, eosinophils (E) 12%, mono-
cytes (M) 1%, erythrocyte sedimentation rate (ESR) 30 mm/
first hour, total leukocyte count (TLC) 7800 cells/mm
3
,and
random blood sugar (RBS) 80 mg%. His serology was nega-
tive. His urine routine microscopy was normal. Stool rou-
tine microscopy showed absence of parasites. A CT scan
showed multiple calcified lesions with scolex in cerebellum
and brain parenchyma, with multiple sites of edema on left
side of parietal area suggestive of neurocysticercosis (Fig. 3).
A physician consultation was done and he was treated
with intravenous injection of dexamethasone and the
dose was tapered every 3 days: 4 mg dexamethasone
intravenously administered thrice daily for 3 days, 2 mg
intravenously administered bi-daily for 3 days, and 2 mg
intravenously administered once daily for 3 days. Tablet
Fig. 1 Disseminated subcutaneous nodules as indicated by arrows on submandibular area, elbow, arm, and abdomen
Fig. 2 Fundus photograph showing bilateral blurred, elevated disc margin and tortuous dilated vessels with parapapillary hemorrhage in left eye
as indicated by arrows
Shrestha and Shrestha Journal of Medical Case Reports (2019) 13:295 Page 2 of 4
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albendazole 400 mg was prescribed bi-daily for 1 month
and tablet valproic acid was prescribed 300 mg bi-daily
for 1 month. Unfortunately we lost the follow-up of our
patient.
Discussion
Ocular cysticercosis is a disease with rare involvement of
cutaneous skin and optic nerve. No cases have been
reported in Nepal with disseminated neurocysticercosis
associated with bilateral ocular involvement with papille-
dema. So our case should be the first case of disseminated
neurocysticercosis with bilateral involvement of eye which
is very rare.
Neurocysticercosis is endemic in Central and South
America, Sub-Saharan Africa, and in some regions of the
Far East including the Indian subcontinent, Indonesia, and
China. There are very limited data on the epidemiology of
cysticercosis from Nepal [3]. Soemmering reported the first
case of ocular cysticercosis in 1830 [4].
Cysticercosis is the most common parasitic disease of
the central nervous system in the world, but cysticercosis
cutis has been reported much less frequently. Approxi-
mately 54% of the patients present with subcutaneous
nodules. However, the association of neural and subcuta-
neous cysticercosis is not common. Less than 50 cases of
disseminated cysticercosis have been reported worldwide
[5]. Our patient had multiple disseminated subcutaneous
nodules over his face, mandibular area, arm, elbow, and
abdomen with multiple calcified cysts in a CT scan of his
brain, which was suggestive of neurocysticercosis but no
neurological deficit or seizure were present although
symptoms of headache and dizziness were present.
A high index of suspicion is required for the diagnosis
of ocular cysticercosis because of the endemic nature of
this infestation in this geographic location [6]. Our case
had a history of eating wild pork which is highly suspi-
cious for neurocysticercosis.
Ocular cysticercosis may affect almost all eye tissues.
The vitreous cavity, subretinal space, and subconjuncti-
val space are common sites while involvement of other
regions (such as extraocular muscle and optic nerve) is
relatively less common. Although either eye may be
affected, bilateral involvement is rare [2]. However, in
our case there was involvement of muscles as subcutaneous
nodules as well as bilateral involvement of optic nerve as
bilateral papilledema which is very rare.
Similarly, another study by Vaidya et al. also pointed
out that the clinical features of disseminated cysticerco-
sis depend on the localization of the cysts in the organs,
parasitic burden, and host parasitic interaction. The
central nervous system is the most commonly involved
location in disseminated cysticercosis followed by stri-
ated muscles, subcutaneous tissues, and orbits [7].
Poudel et al. reported probably the first case of dissemi-
nated cysticercosis in Nepal. The patient had disseminated
subcutaneous nodules on bilateral temporal right medial
lower arm and right chest (axillary regions). Magnetic
resonance imaging revealed multiple diffuse (parenchymal,
intraventricular, calcified) cysts with starry sky appearance
[8]. Similarly, in our study, our patient had disseminated
subcutaneous nodules on his face, mandibular area, arm,
elbow, and abdomen with multiple calcified cysts in a CT
scan of his brain suggestive of neurocysticercosis. How-
ever, there was an absence of papilledema unlike our
study. Sune et al. reported a case of bipolar disorder and
bilateral papilledema with diffuse parenchymatous cysti-
cercosis demonstrated on CT scan as in our study [9].
However, in contrast, our patient had no neurological or
psychological symptoms.
The diagnosis of neurocysticercosis in our study was
confirmed by the presence of: one absolute criterion,
Fig. 3 Computed tomography scan of the brain showing multiple calcified lesions with scolex as indicated by arrows in cerebellum and brain
parenchyma, with multiple sites of edema on left side of parietal area
Shrestha and Shrestha Journal of Medical Case Reports (2019) 13:295 Page 3 of 4
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that is, calcified cysts with scolex on neuroimaging; one
epidemiologic criterion, that is, ingestion of pork; and
one minor criterion, that is, clinical manifestations.
These three criteria fulfill the diagnostic criteria of
neurocysticercosis as proposed by Del Brutto et al.[10].
A single therapeutic approach is not expected to be
useful in every patient with neurocysticercosis. Therapy
usually includes a combination of symptomatic and cysti-
cidal drugs. Surgery also has a role in the management of
some patients. Albendazole has been superior to prazi-
quantel in trials comparing the efficacy of these drugs.
Cysticidal drugs must be used with caution in patients
with giant subarachnoid cysticerci because the inflamma-
tory reaction developed by the host in response to the
acute destruction of the parasite may occlude leptomenin-
geal vessels surrounding the cyst; concomitant steroid
administration is mandatory to avoid the hazard of a cere-
bral infarct [11]. Antiepileptic therapy may also be appro-
priate for patients who do not present with seizures but
who are at high risk for seizures [12]. Our patient also
received a combination of steroids, cysticidal drugs, and
antiepileptic drugs as mentioned in the literature.
Conclusion
Cysticercosis is an endemic disease with a diversity of clin-
ical presentations, disseminated lesions, and unusual ocular
presentations. Neurocysticercosis may be diagnosed as a
cause of bilateral papilledema in endemic zones of Nepal.
This case report highlights the importance of ruling out
cysticercosis in cases with multiple disseminated subcuta-
neous nodules and bilateral papilledema.
Acknowledgements
Not applicable.
Authorscontributions
Drafting and submission of manuscript was done by RS. AKS edited the final
manuscript and sequence alignment. Both authors read and approved the
final manuscript.
Funding
None.
Availability of data and materials
Not applicable.
Ethics approval and consent to participate
Ethical approval was taken from the Institutional Review Board.
Consent for publication
Written informed consent was obtained from the patient for publication
of this case report and any accompanying images. A copy of the written
consent is available for review by the Editor-in-Chief of this journal.
Competing interests
The authors declare that they have no competing interests.
Author details
1
Department of Ophthalmology, Reiyukai Eiko Masunaga Eye Hospital,
Banepa-1, Kavrepalanchok District, Nepal.
2
Department of Orthopedics, Nepal
Police Hospital, Panipokhari, Kathmandu, Nepal.
Received: 2 June 2019 Accepted: 9 August 2019
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