The Laryngoscope

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Rhinological and Otological Society, Inc.

A Hydatid Cyst in an Unusual LocationThe Infratemporal Fossa

Morteza Nouroallahian, MD;1 Mehdi Bakhshaee, MD;2 Mohammad Reza Afzalzadeh, MD;1
Bahram Memar, MD3
Hydatid disease is a considerable health problem worldwide, but hydatid cysts in the infratemporal region are extremely
rare, even where the parasite is endemic. Here we report on a 17-year-old female who presented with a benign swelling in
the infratemporal fossa. During surgery a cystic mass was discovered and histopathologic examination confirmed the diagnosis of a hydatid cyst. Although rare, a hydatid cyst should be considered in a differential diagnosis of benign swellings in the
maxillofacial region. Physicians should exercise a high index of suspicion even in nonendemic locations.
Key Words: Hydatid cyst, infratemporal fossa, hydatid disease, parasite.
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INTRODUCTION

CASE REPORT

Hydatid disease, caused by larvae of the Echinococcus tapeworm, is rare in humans and affects between 1
and 220 people per 100,000 depending on the region.1
The greatest prevalence of the disease is found in the
Mediterranean and parts of central Asia, South America,
Australia, and Africa.2 The life cycle of Echinococcus
involves a definitive host (dog) and an intermediate host
(usually sheep) with humans as an accidental host following ingestion of the larvae. Once ingested, the larvae
pass into the blood stream through the intestinal mucosa, where they are most likely to infest the liver as
this is the first organ that they pass through.2 However,
in all cases of echinococcosis, a thorough systemic investigation should be performed, as 20% to 40% may have
multiorgan involvement.2 Although the occurrence of a
single hydatid cyst is common in most cases, it is unusual that a cyst will occur in the maxillofacial region
without evidence of additional hepatic or lung involvement, although the embryos must have passed through
the organs.2 Here we present the case of a young woman
with a single hydatid cyst of the infratemporal fossa as
an educational example, and a reminder to consider
hydatid disease in a differential diagnosis of benign
swellings in the maxillofacial region.

A 17-year-old woman was referred to us with a

moderately hard, nontender swelling on her right check
below the zygomatic process, which was causing asymmetry of the face and had been present for 2 years.
According to the patient, the swelling was growing very
slowly with no accompanying pain or sensation of pressure. Otherwise, the general health of the patient was
normal with no cervical lymphadenopathy. A chest radiograph, urine, and blood analysis were normal with only
a slight increase in eosinophil count. The results of fineneedle aspiration cytology were consistent with the mass
being benign and cystic. The patients personal history
revealed that she worked in agriculture and lived in
close proximity to livestock.
As the mass was a benign soft-tissue tumor with
some degeneration, an excision of the lesion was
planned. Preoperative axial computed tomography (CT)
findings revealed a well-circumscribed calcified lesion in
the infratemporal fossa measuring 41 mm  25 mm at
its greatest dimension, with a midline shift impinging
on the surrounding structures (Fig. 1). A hemicoronal
incision was made under general anesthesia, and the
frontal branch of the facial nerve was explored. A cystic
lesion was found and the cyst carefully removed in its
entirety with no difficulties encountered (Fig. 2). The
specimen consisted of soft creamy membranous material.
In histopathology, a typical hydatid hyaline ectocyst and
germinative layer with few protoscolices were seen
(Figs. 35).
Ultrasonography of the liver and an abdominal CT
were performed to rule out any visceral involvement.
The patient received albendazole 800 mg/day for 4
weeks and has been disease free for at least 12
months.
The Mashhad University of Medical Sciences institutional review board has approved this report.

From the Department of OtorhinolaryngologyHead and Neck Surgery

(M.N., M.R.A.), Imam Reza Educational Hospital, the Ear, Nose and Throat
Research Center (M.B.), and Department of Pathology (B.M.), Emmam Reza
Hospital, Faculty of Medicine, Mashad University of Medical Sciences,
Mashad, Iran.
The authors have no funding, financial relationships, or conflicts
of interest to disclose.
Send correspondence to Mehdi Bakhshaee, MD, Associate Professor of the Ear, Nose and Throat Research Center, Emmam Reza Hospital, Faculty of Medicine, Mashhad University of Medical Sciences,
Mashhad, Iran. E-mail: mehbakhsh@yahoo.com or bakhshaeem@mums.
ac.ir
DOI: 10.1002/lary.23471

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Nouroallahian et al. Hydatid Cyst in the Infratemporal Fossa

Fig. 1. Computed tomographyscan showing cystic lesion in

DISCUSSION
There have been previous case reports of hydatid
cysts in a variety of different parts of the head and neck
area including the neck,36 nasopharynx,6 skull base,6,7
maxillary region,8 pterygopalatine fossa,9 and infratemporal fossa.1012 In most cases the cysts are
asymptomatic and slow growing, with the secondary
symptoms depending on their location.3 However, even
in areas where the parasite is endemic, a hydatid cyst in
the head and neck region is very rare, and is not usually
considered in a differential diagnosis of a cystic swelling
in the head-neck and maxillofacial region.3,4,8
Although the patients medical history, family history, occupation, and place of residence may suggest the
possibility of hydatid disease, physicians are unlikely to
make a preoperative diagnosis without a high degree of
suspicion pertaining to hydatid cysts, as well as demonstrated histopathological or radiological findings.3,4 In
our case, fine-needle aspiration cytology was used to
confirm that the mass was benign and cystic, and a CT

Fig. 3. Germinative and hyalineectocyst (hematoxylin and eosin,

scan was used to confirm its location. A diagnosis of a

hydatid cyst was not considered before surgery, and a
definitive diagnosis was made only by postoperative
histopathology.
Although we used fine-needle aspiration cytology,
its use is controversial due to the risk of acute anaphylaxis or spread of daughter cysts.4 However, several
reports have indicated that it is a safe diagnostic technique if performed properly.13,14 In terms of treatment,
surgery is the most effective way to manage a hydatid
cyst in the maxillofacial region, and particular efforts
should be made to prevent breakage of the cyst during
the procedure, which could lead to anaphylaxis and/or
cyst recurrence.3,4,8,10 Inactivation of daughter cysts
before surgery can be achieved by injecting 20% saline,
formalin, or 0.5% silver nitrate into the cyst.8 The area
around the cyst can also be protected from contamination with surgical pads soaked with a scolicidal agent
(1.5% cetrimide, 1.5% chlorhexidine gluconate).4 Following surgery, imidazole derivatives such as albendazole
should be administrated to manage any potential risk of

Fig. 4. Germinative and hyalineectocyst with one protoscolex

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Nouroallahian et al. Hydatid Cyst in the Infratemporal Fossa

tial diagnosis of benign swellings of the head and neck

region.

BIBLIOGRAPHY

Fig. 5. Germinative and hyalineectocyst with one protoscolex

contamination or recurrence,3,4,8 as was done in this

case.

CONCLUSION
Although incidence in the maxillofacial region is
rare, a hydatid cyst should be considered in a differen-

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Nouroallahian et al. Hydatid Cyst in the Infratemporal Fossa