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 Table of Contents  
CASE REPORT
Year : 2017  |  Volume : 3  |  Issue : 1  |  Page : 32-35

Oral cysticercosis: Mimicking mucocele of the cheek


1 Department of Oral Pathology and Microbiology, M.A. Rangoonwala College of Dental Sciences and Research Centre, Pune, Maharashtra, India
2 Department of Oral Pathology and Microbiology, ESIC Dental College, New Delhi, India

Date of Web Publication17-Jul-2017

Correspondence Address:
Simrata Ajrawat
A3/2, Paschim Vihar, New Delhi - 110 063
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/ijohr.ijohr_1_17

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  Abstract 

Cysticercosis is a parasitic infection caused by “cysticercus.” Cysticercosis is a larval form of certain Taenia species of which cysticercus cellulosae, a larvae form of Taenia solium is the one which infects humans. The larvae form of T. solium, that is, cysticercus cellulosae resides in the muscles and other tissue in pigs that serves as intermediate host. The most frequent sites of cysticercosis are subcutaneous layers, brain, muscles, heart, liver, lungs, and peritoneum. Oral cysticercosis is rare and when it affects the mouth, it commonly occurs in the tongue, labial, or buccal mucosa, and sometimes floor of the mouth. Here, we present a case of 12-year-old female with swelling in the right cheek region as an asymptomatic nodule which clinically looked like a mucocele.

Keywords: Cysticercus cellulosae, oral cysticercosis, Taenia solium


How to cite this article:
Ajrawat S, Kharbanda J, Saxena S. Oral cysticercosis: Mimicking mucocele of the cheek. Indian J Oral Health Res 2017;3:32-5

How to cite this URL:
Ajrawat S, Kharbanda J, Saxena S. Oral cysticercosis: Mimicking mucocele of the cheek. Indian J Oral Health Res [serial online] 2017 [cited 2019 Jul 15];3:32-5. Available from: http://www.ijohr.org/text.asp?2017/3/1/32/210919


  Introduction Top


The parasitic infestation by the pork tapeworm larval stage, the cysticercus cellulosae, is called cysticercosis, and results from the ingestion of tapeworm eggs through contaminated food and water or dirty hands.

Platyhelminthes have a life cycle characterized by two stages; first as a larva, and then as an adult worm, besides an egg phase. Each of these phases requires a different host.

The adult Taenia solium, or pork tapeworm, lives in the small intestine of man, its definitive host. Infestation by T. solium is common in areas where pig breeding is not controlled and sanitation is inadequate.[1] The eggs develop into the oncospheres and penetrate the gut wall to enter the systemic circulation. Thereafter, these are filtered from circulation into the muscular tissue.[2] Oral cysticercosis is common in tongue followed by lower lip, oral mucosa, submandibular and submental region.[3] Highest incidence countries are Brazil, Chile, Ecuador, Mexico, South Africa, East Africa, and India.[1]


  Case Report Top


A 12-year-old female presented with the complaint of swelling in the right back cheek region for 1 year. Patient gave the history of painless swelling which was insidious in onset and progressed in size for 2 months and became static. Medical and dental history were noncontributory. On examination, a solitary diffuse swelling of size 3 cm in diameter was seen extending over 47–48 region. On palpation the swelling was soft to firm in consistency, nontender, and mobile. The over lying mucosa was thin and bluish. Based on these findings, the clinical diagnosis of mucocele was made and differential diagnosis of minor salivary gland tumor and fibroma was given. Excisional biopsy of the lesion was performed.

Gross specimen measuring approximately 1 cm in diameter, was soft in consistency and globular. On dividing the specimen longitudinally, a cystic cavity containing whitish material was seen which appeared caseated [Figure 1].
Figure 1: Grossing of the specimen showing a cystic cavity containing whitish material

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Sections were taken after processing the specimen and hematoxylin and eosin staining was performed. Histopathological examination revealed a cystic cavity containing a distinct component of tissue which showed papillary projections lined by amorphous eosinophilic lining. Connective tissue component had macrophages, inflammatory cells, and degenerating empty appearing cells. Host connective tissue was fibrocellular with blood vessels and inflammatory infiltrate. Based on these findings, the diagnosis of “Cysticercus Cellulosae” was given [Figure 2],[Figure 3],[Figure 4],[Figure 5].
Figure 2: Cystic cavity with papillary projections into the cavity

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Figure 3: Papillary projections lined by amorphous eosinophilic lining; indicating the scolex (S)

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Figure 4: Connective tissue component showing degenerating empty appearing cells

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Figure 5: Connective tissue component showing macrophages and inflammatory cells

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  Discussion Top


Aristophanes and Aristotle first described cysticercosis in the 3rd century BC in pigs. In 1550, it was observed in humans by Parunoli.[4]T. solium (tape worm) infection is endemic in many parts of the world including Latin America, India, Eastern Europe, Asia, Africa, Russia, Indonesia, Philippines, and Mexico.[5]

The tape worm is composed of scolex (head) and proglottids (caudal end). Every proglottid contains 40,000 to 60,000 eggs and are released with the feces. Pigs being the intermediate host, are infected with it following ingestion of eggs from the ground contaminated with human feces, when it develops into the larval form. The eggs develop into the oncospheres and penetrate the gut wall to enter the systemic circulation [Figure 6]. Thereafter, these are filtered from circulation into the muscular tissue. Humans are infected with the consumption of raw or undercooked pork.[2]T. solium eggs could be ingested through consumption of contaminated water and vegetables by humans and autoinfection due to improper hygiene may also be a cause.[2]
Figure 6: Life cycle of Taenia solium[4]

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In the present case, no proper history could be elicited from the patient or her guardian and it may be assumed that compromised hygiene or contaminated consumption of food or water may be the source of such infection. Humans being a definitive host for cysticercosis, it can manifest in various tissues and organs of the individual. The symptomatic presentation of cysticercosis is seen only in case of involvement of central nervous system (CNS) and ocular system.[6]

Generalized symptoms include headache, fever, and myalgia. Involvement of CNS may result in episodes of seizures, increased intracranial pressure, meningitis, and mental disorders.[7],[8] Other commonly affected sites in the body include heart, liver, lung, and peritoneum.[5]

Oral and maxillofacial region has abundance of musculature in spite of which the incidence of oral cysticercosis is rare. The most frequently involved site of the oral cavity are the tongue (42.15%) followed by lips (26.15%) with the lower lip being affected in 64.71% and buccal mucosa (18.9%).[2]

Patients infected with cysticercosis usually present with the chief complaint of swelling rather than pain unless secondarily infected.[2] In the present case also the patient complained of an asymptomatic swelling which was progressive in size.

According to Delgado-Azañero et al., oral cysticercosis presents as firm nodules on palpation because of their high intraluminal pressure.[9]

According to Lee et al., two similar cases were reported in which the most common complaint by patients was swelling. Pain is not a frequent feature unless secondarily infected. Lesions on the tongue interfered with the movement, causing discomfort during speaking and eating. According to him, most oral presentations are in the form of painless, well-circumscribed, soft swellings that may mimic fluctuant lesions like mucocele.[10]

In the present case, the presentation was similar to mucocele of the buccal mucosa and clinicians did not consider any other pathology in their differential diagnosis and cysticercosis was a histologic revelation.

Histopathological examination helps in the diagnosis of cysticercosis by the detection of a cystic space containing the cysticercus cellulosae. The scolex has four suckers and a double crown of rostellar hooklets. A duct-like invaginated segment, lined by a homogeneous anhistic membrane, composes the caudal end. The eosinophilic membrane that lines the capsule is double-layered, consisting of an outer acellular and an inner sparsely cellular layer. After 3–5 years, the larva dies and the cyst undergoes calcification.[2]

Although an excisional biopsy is usually considered the only definitive diagnostic procedure, there are some other diagnostic tools that may be used. Computerized tomography or magnetic resonance imaging is valuable for diag-nosing cerebral cysticercosis.[11] Serological investigations, such as enzyme-linked immunosorbent assay or enzyme-linked immunoelectrotransfer blot, used for detecting antibodies to T. solium in the serum and cerebrospinal fluid can confirm the diagnosis, although they are not 100% sensitive.[12]

The treatment for oral cysticercosis is surgical enucleation. Drugs such as praziquantel and albendazole are potent anthelminthic used in the treatment of cysticercosis, replacing niclosamide, which was the drug of choice for the treatment of the disease for a long time. Drugs should be used, especially in symptomatic patients, disseminated cysticercosis, and cases where surgical treatment is risky or not possible, as in neurocysticercosis.[13]

Because the patient in the present case had no other manifestation of cysticercosis and did not have any other symptoms, no additional treatment was prescribed other than excisional biopsy and a course of anthelminthic. Periodic follow-ups did not reveal any kind of recurrence or untoward symptoms.


  Conclusion Top


It is of utmost importance to submit all lesions for histopathological diagnosis irrespective of its innocuous and bland clinical appearance. The findings of cysticercosis cellulosae in the oral tissues are quite often a histopathological revelation and unexpected diagnosis for the clinician. However, timely treatment and intervention will save the patient from serious systemic and CNS manifestations caused by this parasitic infection.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.

 
  References Top

1.
Prabhu SR. Oral Diseases in the Tropics. Oxford: Oxford University Press; 1992. p. 126-9.  Back to cited text no. 1
    
2.
Ribeiro AC, Luvizotto MC, Soubhia AM, de Castro AL. Oral cysticercosis: Case report. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2007;104:e56-8.  Back to cited text no. 2
    
3.
Nigam S, Singh T, Mishra A, Chaturvedi KU. Oral cysticercosis – Report of six cases. Head Neck 2001;23:497-9.  Back to cited text no. 3
[PUBMED]    
4.
Kulkarni PG, Palakurthy P, Muddana K, Nandan RK. Oral cysticercosis – A diagnostic dilemma. J Clin Diagn Res 2015;9:ZD01-2.  Back to cited text no. 4
    
5.
Elias FM, Martins MT, Foronda R, Jorge WA, Araújo NS. Oral cysticercosis: Case report and review of the literature. Rev Inst Med Trop Sao Paulo 2005;47:95-8.  Back to cited text no. 5
    
6.
Flisser A, Plancarte A, Correa D, Rodriguez-Del-Rosal E, Feldman M, Sandoval M, et al. New approaches in the diagnosis of Taenia solium cysticercosis and taeniasis. Ann Parasitol Hum Comp 1990;65 Suppl 1:95-8.  Back to cited text no. 6
    
7.
Lustmann J, Copelyn M. Oral cysticercosis. Review of the literature and report of 2 cases. Int J Oral Surg 1981;10:371-5.  Back to cited text no. 7
    
8.
Webb DJ, Seidel J, Correll RW. Multiple nodules on the tongue of a patient with seizures. J Am Dent Assoc 1986;112:701-2.  Back to cited text no. 8
    
9.
Delgado-Azañero WA, Mosqueda-Taylor A, Carlos-Bregni R, Del Muro-Delgado R, Díaz-Franco MA, Contreras-Vidaurre E. Oral cysticercosis: A collaborative study of 16 cases. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2007;103:528-33.  Back to cited text no. 9
    
10.
Lee KH, Cepeda L, Miller M, Siegel DM. Mucoceles not – Oral cysticercosis and minor salivary gland adenocarcinoma: Two case reports. Dermatol Online J 2009;15:8.  Back to cited text no. 10
    
11.
Rajshekhar V. Etiology and management of single small CT lesions in patients with seizures: Understanding a controversy. Acta Neurol Scand 1991;84:465-70.  Back to cited text no. 11
    
12.
Diaz JF, Verastegui M, Gilman RH, Tsang VC, Pilcher JB, Gallo C, et al. Immunodiagnosis of human cysticercosis (Taenia solium): A field comparison of an antibody-enzyme-linked immunosorbent assay (ELISA), an antigen-ELISA, and an enzyme-linked immunoelectrotransfer blot (EITB) assay in Peru. The Cysticercosis Working Group in Peru (CWG). Am J Trop Med Hyg 1992;46:610-5.  Back to cited text no. 12
    
13.
Jay A, Dhanda J, Chiodini PL, Woodrow CJ, Farthing PM, Evans J, et al. Oral cysticercosis. Br J Oral Maxillofac Surg 2007;45:331-4.  Back to cited text no. 13
    


    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5], [Figure 6]



 

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Introduction
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