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Short communication

Calcied onchocercal nodule in the infratemporal fossa:

a rare case
C. Mauprivez a,b,c, , P. Harlay a , F. Gruffaz d , P. Goudot e
a

Service dOdontologie, Assistance Publique-Hpitaux de Paris, Groupe Hospitalier Piti-Salptrire, Paris, France
EA 2496, Universit Paris Descartes
c Facult de Chirurgie Dentaire, Universit Paris Diderot
d Laboratoire dAnatomie et Cytologie Pathologiques, Assistance Publique-Hpitaux de Paris, Groupe Hospitalier Piti-Salptrire, Paris, France
e Service de Stomatologie et de Chirurgie Maxillo-Faciale, Assistance Publique-Hpitaux de Paris, Groupe Hospitalier Piti-Salptrire, Paris, France
b

Accepted 14 September 2015

Abstract
Onchocerciasis is a parasitic tropical disease that often presents as multiple, superficial nodules. When it is unique and deeply situated, its
clinical diagnosis is difficult. We report a case of a calcified onchocercoma that was localised to the infratemporal fossa.
2015 The British Association of Oral and Maxillofacial Surgeons. Published by Elsevier Ltd. All rights reserved.

Keywords: Parasitic disease; Onchocerciasis; Onchocercoma; Infratemporal fossa

Introduction
Onchocerciasis, or river blindness, is a parasitic infection
caused by the filarial nematode, Onchocerca volvulus. It
infects at least 25 million people worldwide, is rarely seen
in Western countries,13 but is currently endemic in tropical
Africa, Latin America, and Yemen.13 The infection is transmitted to humans by blackflies of the genus Simulium that
transfer infective larvae during a meal of blood. The larvae
develop into adult filariae in subcutaneous tissues, and these
reside in fibrous nodules known as onchocercomas where
they can live for up to 15 years. A fertilised female larva
can produce millions of microfilariae during her lifetime.3
Older nodules often contain necrotic material with calcified

remnants of filariae. Classic manifestations include itchy skin

rashes, subcutaneous nodules, and sclerosing keratitis.
The diagnosis of onchocerciasis may be suspected if the
patient has eosinophilia. It is confirmed by the presence of
microfilariae in a bloodless skin biopsy specimen, in the
anterior chamber of the eye, and by adult worms in an
onchocercoma.4 The microfilaricidal agent ivermectin is the
main means of controlling the disease. Endosymbiotic Wolbachia bacteria in the worms are targets for a new therapeutic
approach, which comprises doxycycline 100 mg/day for six
weeks, and can be used to sterilise adult worms in the long
term. 13 We describe what we think is the first case of calcified onchocercoma in the infratemporal fossa.
Case report

Corresponding author at: Facult de Chirurgie Dentaire, Universit ParisDiderot, 5 rue Garancire 75006 Paris, Tel.: +33157278712;
fax: +33157278701.
E-mail address: cedric.mauprivez@univ-paris-diderot.fr
(C. Mauprivez).

A 33-year-old African woman presented with a painless mass

in the right maxillary vestibule. Her past medical history was
unremarkable. On examination her oral mucosa looked normal, and there was no variation in colour. The lesion was

http://dx.doi.org/10.1016/j.bjoms.2015.09.012
0266-4356/ 2015 The British Association of Oral and Maxillofacial Surgeons. Published by Elsevier Ltd. All rights reserved.

Please cite this article in press as: Mauprivez C, et al. Calcified onchocercal nodule in the infratemporal fossa: a rare case. Br J Oral
Maxillofac Surg (2015), http://dx.doi.org/10.1016/j.bjoms.2015.09.012

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Fig. 1. Orthopantomogram showing a lesion composed of many calcifications above the right third maxillary molar.

Fig. 2. Surgical specimen of onchocerma. A macroscopic examination showed a 3.0 x 1.5 x 1.5 cm tumour that was hard, white, and oblong. The appearance
of the upper pole of the tumour suggested parasitic origin.

well-defined, firm, mobile, and slightly tender on palpation.

There was no cervical lymphadenopathy, and we could find
no abnormalities of the head and neck.
An orthopantomogram showed a radiopaque, heterogeneous, ovoid lesion with clear limits from the apex of the
maxillary molar to the lower fissure of the orbit, which
contained what looked like calcifications (Fig. 1). Axial
computed tomographic imaging with the soft-tissue window
setting showed that the lesion was localised inside the buccal
fat pad in the infratemporal fossa. There were no signs of
bony destruction or of a mass. Magnetic resonance imaging
showed a well-defined lesion with low signal intensity on T1
and T2-weighted images, which suggested either calcinosis
or a calcified phlebolith. Further investigations included estimation of full blood count and electrolytes, which were all
within the reference ranges.
The lesion was removed by intraoral excision under local
anaesthesia, and histological examination showed a calcified

onchocercal nodule (Fig. 2 and 3). She was then referred for
medical evaluation. No other nodules were found on palpation. A skin-snip biopsy specimen was taken, and she had an
ophthalmological examination with a slit lamp to eliminate
systemic onchocerciasis. No microfilariae were found, and
no chemotherapy was necessary.

Discussion
Numerous parasitoses such as cysticercosis, hydatid cysts,
echinococcosis, trichinellosis, dracunculiasis, and onchocerciasis might evolve into calcified tumours or dystrophic
calcifications. Living larvae are not visible radiographically,
but death and calcification of a parasite in subcutaneous
and muscular sites allow the identification of larvae by
radiographic means. Many cases of oral cysticercosis in the
masticatory muscles, tongue, oral mucosa, and lips have been

Please cite this article in press as: Mauprivez C, et al. Calcified onchocercal nodule in the infratemporal fossa: a rare case. Br J Oral
Maxillofac Surg (2015), http://dx.doi.org/10.1016/j.bjoms.2015.09.012

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Fig. 3. Histological section of the onchocercoma. Microscopic examination showed a section of worms, with striated cuticles and the uterine cavity filled with
microfilariae, encysted around fibrous connective tissue (haematoxylin and eosin, original magnification x 40).

reported, including a calcified cysticercosis nodule in the

cheek.5 However, we know of few cases of filariasis in the oral
cavity,68 though there have been three reports of onchocerciasis of the face and mouth, and one case of an onchocercoma
inside the temporalis muscle.9,10 We know of no cases of
calcified onchocercal nodules in the oral and maxillofacial
regions. Calcified nodules are typically seen in the scrotal
region and around the hip and elbow.3
Excision of the nodule with macroscopic and microscopic
examination can diagnose infestation, but do not necessarily
indicate the potential to pass on infection (infectivity), which
requires microfilaridermia.4
Our patient had most likely been infected in childhood
when she lived in an area where the disease was endemic.
There are no effective medical treatments against adult worms
encapsulated in nodules, and the first line treatment for suppression of adult worms or onchocercal nodules is excision.
Onchocercoma should be included in the differential
diagnosis of intraoral nodules or calcified tumours in the maxillofacial region in African populations or in expatriates from
areas where it is endemic.

Conict of Interest
We have no conflict of interest.

Ethics statement/conrmation of patients permission

Not applicable.
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Please cite this article in press as: Mauprivez C, et al. Calcified onchocercal nodule in the infratemporal fossa: a rare case. Br J Oral
Maxillofac Surg (2015), http://dx.doi.org/10.1016/j.bjoms.2015.09.012