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An Official Publication of the Indian Association of Oral and Maxillofacial Pathologists


 
  Table of Contents    
CASE REPORT  
Year : 2019  |  Volume : 23  |  Issue : 3  |  Page : 418-421
 

Labial hydatid cyst – A rare entity


1 Department of Oral and Maxillofacial Pathology, Awadh Dental College and Hospital, Jamshedpur, Jharkhand, India
2 Department of Oral and Maxillofacial Pathology, NSVK Dental College, Bengaluru, Karnataka, India
3 Dental Department, DVC Hospital, Durgapur, West Bengal, India
4 Department of Oral and Maxillofacial Surgery, Bardhaman Dental College and Hospital, Bardhaman, West Bengal, India

Date of Submission16-Feb-2019
Date of Acceptance27-Jun-2019
Date of Web Publication19-Dec-2019

Correspondence Address:
Elampavai Elangovan
Department of Oral and Maxillofacial Pathology, NSVK Dental College, Bengaluru, Karnataka
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/jomfp.JOMFP_322_18

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   Abstract 


Hydatid cyst is a parasitic infestation by tapeworm Echinococcus. This endemic disease is prevalent in the Middle East and Southeast Asian populations. This zoonotic disease is mainly transmitted by cattle and dogs. Few studies have been reported in the head-and-neck region. There are very few isolated cases have been reported within the oral cavity region. No case until now has been isolated from the labial mucosa. We now present one such case here.


Keywords: Hydatid cyst, labial mucosa, labial swelling


How to cite this article:
Banerjee A, Elangovan E, Mitra KS, Saha A. Labial hydatid cyst – A rare entity. J Oral Maxillofac Pathol 2019;23:418-21

How to cite this URL:
Banerjee A, Elangovan E, Mitra KS, Saha A. Labial hydatid cyst – A rare entity. J Oral Maxillofac Pathol [serial online] 2019 [cited 2020 Jan 18];23:418-21. Available from: http://www.jomfp.in/text.asp?2019/23/3/418/273503





   Introduction Top


Hydatid cyst is a parasitic zoonosis otherwise known as human cystic echinococcosis.[1] The Greek word “Hydatid” means “drop of water.”[2] This disease is endemic in the areas where dogs and cattle are found in abundance. The incidence of hydatid disease is around 1–200 per 100,000 population in India.[3] The host plays an important role in disease transmission. Humans mostly act as an accidental or end host. Echinococcus E. chinococcosis granulosus is the most common etiology behind the formation of hydatid cysts.[1]

Hydatid cyst in human is mostly encountered in the major organs such as lungs and liver (80%), followed by the other parts such as the peritoneum, spleen, kidney, skin and muscles.[4] In the maxillofacial and head-and-neck region, it is reported to as less as 1%–2%.[4],[5],[6] This disease is mostly restricted to population having specific food trends and unique lifestyles. The clinical presentation, diagnosis and management of the disease vary according to the localization, duration, shape, size and the number of cysts.

The diagnosis of the hydatid cyst in the oral and maxillofacial region creates accost among the clinical oral pathologists, oral surgeons, microbiologists and general pathologists reporting oral pathology cases due to its rare occurrence. There are very few published literature in the maxillofacial region as per the meticulous research done in various search engines such as PubMed, Cochrane Library and Semantic Scholar. There are a total of six published reports of occurrence in the oral region that involves buccal mucosa, floor of the mouth and tongue and three cases in the maxillary region, i.e., maxillary sinus.[5],[7],[8],[9],[10],[11] Our case holds a worthy citation as it was localized in the labial mucosa of the maxillary region of the oral cavity in a 62-year-old male.


   Case Report Top


A 62-year-old male reported to the department of oral pathology and diagnosis after being referred by an oral surgeon. The patient gives a very interesting history of an appearance of an asymptomatic small elevation on the labial mucosa in the maxillary region in relation to 13 and 14. The swelling is recurrent and disappears within 1–2 days. There was no noticeable extraoral swelling, lymphadenopathy or fever associated. Numerous clinical diagnoses of mucocele, epulis and salivary ductal obstruction were rendered, but since the entity appeared for short duration and used to get resolved by itself, it was never indicated for surgical intervention.

On inspection, there was a mild diffuse oval elevation of 0.5 cm × 0.7 cm having normal mucosal color and was barely noticeable [Figure 1]a. There was no sign of inflammation or discharge. On bidigital palpation, the swelling was firm to hard in consistency, nonfixed to the underlying tissue. A provisional diagnosis of minute salivary duct calculi was given and was posted for surgery after hematological examination.
Figure 1: (a) On inspection, a diffuse small swelling on the upper labial mucosa. (b) Excision of the mass

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Surgical intervention revealed no association or presence of minor salivary glands or salivary ducts along with the mass. The mass was firm in consistency resembling a small rubber ball measuring 0.5 cm × 0.6 cm, dark creamish black in color surrounded by loose tissue [Figure 1]b. The mass was not fixed to underlying deeper structures. The entire mass was sent for histopathological evaluation. The mass was slightly compressible on bidigital pressure. Grossing of the sample was done [Figure 2]. The entire mass was embedded into one block, and gradual sections of 4 μm were obtained.
Figure 2: Grossing of the sample, sample along with the peripheral tissue and hemorrhage

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The H&E-stained section revealed a trilaminar cystic structure surrounded by the loose fibrous tissue [Figure 3]a and [Figure 3]b. The outermost layer or pericyst, intermediate layer was avascular dark eosinophilic refractile layer or exocyst and the inner most layer or endocyst projecting toward the lumen can be appreciated [Figure 4]a and [Figure 4]b. The lumen was filled by amorphous, light eosinophilic substance showing branching spaces with small vesicles or brood capsules [Figure 4]c. These brood capsules represent developing daughter cysts filled by clear fluid. However, in this case, hooklets were not appreciated much. The surrounding tissue was loose and fibrous and showed the presence of chronic inflammatory cells. Histopathological interpretation of the hydatid cyst of the oral cavity was rendered.
Figure 3: (a) The scanning histological view of the entire specimen showing trilaminar structure. (b) The attached surrounding tissue is loose well-arranged fibrous tissue with chronic inflammatory component

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Figure 4: The higher magnification views (a) the pericyst attached to the outer fibrous layer, intermediate layer or exocyst and the innermost layer toward the lumen is endocyst. (b) The intermediate layer depicting refractile eosinophilic substance sandwiched between the outermost layer and the innermost layer and is well demarcated. (c) The lumen depicting small vesicles or brood capsules along with branched lumen space

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The patient was sent for further investigations such as ultrasonography (USG) of the upper lip and lower abdomen, chest radiographs and blood investigations which showed no abnormality. The recommended dosage of albendazole was prescribed for 1 month, and the patient was kept under thorough follow-up. There was no such entity found elsewhere in the body and neither any recurrence happened in the same site.


   Discussion Top


Only a few research efforts have been put forth in studying the parasitic cysts of the oral cavity as these entities pose themselves as a dilemma to the diagnosticians.

Echinococcosis is a parasitic infection occurring in humans and other animals such as dogs, sheep, cattle and fox. This parasitic disease is caused by cestodes of genus Echinococcus.[12] It is found to occur mostly in the Mediterranean countries, South America, Australia, central parts of Asia and East African countries.[13] The parasitic infestations result in hydatid cyst which is formed by the larval form Echinococcus granulosus.[14] The life cycle of this parasite occurs in two hosts, wherein the adult tapeworms reside in the intestine of carnivores, i.e., dogs and foxes, which are the definitive hosts, and the larval forms reside in the intermediate hosts, i.e., man, sheep and cattle. Humans acquire this disease if he/she accidentally ingests the ova passed in the stool of dog or the food contaminated by the ova or by contact with the infected dogs.[12],[14] The larva from the ova penetrates the intestinal mucosa and reaches the liver through the portal vein and develops into a cyst. Majority of the larvae affect the liver, followed by lungs, abdominal cavity, kidneys and bone. These cysts are very rarely positioned in the head-and-neck region,[15] and few reported cases in literature showed site predilections such as the right side submandibular region,[16] infratemporal region,[17] tongue [15] and floor of the mouth.[18]

The disease often starts without symptoms and this may last for a year. The signs and symptoms depend on the cyst's location and size. The diagnosis of this condition can be made by histopathology, various imaging techniques (USG, computed tomography and magnetic resonance imaging) and serological tests and antigen assays.[18]

The presence of an antigen specific for E. granulosus that appeared immunoelectrophoretically as a band of characteristic morphology and location when tested against sera from human patients has been reported.[19]

In a study, they reported a case of intraoral hydatid cyst of the buccal mucosa which was previously diagnosed as calcified buccal lymph node based on history and clinical examination. Later, only after the surgical removal of the lesion was done, histopathology confirmed it to be a hydatid cyst. No recurrence was noted.[7]

In another study, again, a case of hydatid cyst occurring in the buccal mucosa was reported. A provisional diagnosis of mucocele was made based on history and clinical examination. A histopathological diagnosis of hydatid cyst of the buccal mucosa was given later. Routine hematological examinations revealed no abnormalities. The patient was then started on albendazole at 15 mg/kg/day for a month. A chest radiograph and abdominal sonography scans were requested. No hydatid cysts were identified anywhere else in the body. The patient is on follow-up till date with no recurrences.[5]

The occurrence of the cyst intraorally has been well-documented. Involvement of the buccal mucosa is rare, and only two other cases have been reported in the literature. The latter occurred in a 6-year-old male and was asymptomatic with no organ involvement.[20] In all of these cases, a history of association with sheep or animals could not be elicited.

Case reports localized from the antrum reveal aspiration tests which are positive for the demonstration of protoscolices. However, they do carry the danger of dissemination or anaphylaxis. Again, histopathology remains the gold standard for diagnosis in such cases.[21]

In the present study, based on history and clinical examination, multiple provisional diagnoses of mucocele, epulis and salivary duct obstruction were given. Only after histopathology, a conclusive diagnosis of intraoral hydatid cyst of the labial mucosa was made.

Till date, no intraoral hydatid cyst localized to labial mucosa has been reported. This would be the first case to be reported. We would also like to mention that proper eliciting of history and making a habit of including hydatid cyst as a differential diagnosis in any cystic swellings of the head-and-neck region will help the clinicians to report such cases in future.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
   References Top

1.
Eckert J, Gemmell MA, François-Xavier M, Pawlowski ZS, World Health Organization. WHO/OIE manual on echinococcosis in humans and animals: A public health problem of global concern. In: Eckert J, editor. 1st ed. Paris, France: World Organisation for Animal Health. 2002. p. 1-5.  Back to cited text no. 1
    
2.
Saez J, Pinto P, Apt W, Zulantay I. Cystic echinococcosis of the tongue leading to diagnosis of multiple localizations. Am J Trop Med Hyg 2001;65:338-40.  Back to cited text no. 2
    
3.
Parikh F. Echinococcosis – Cut to cure but what about control? J Assoc Physicians India 2012;60:9-10.  Back to cited text no. 3
    
4.
Onerci M, Turan E, Ruacan S. Submandibular hydatid cyst. A case report. J Craniomaxillofac Surg 1991;19:359-61.  Back to cited text no. 4
    
5.
Lavanya RM, Kamath VV, Komali Y, Krishnamurthy S. Hydatid cyst of the buccal mucosa: An unusual presentation. Indian J Dent 2015;6:157-60.  Back to cited text no. 5
[PUBMED]  [Full text]  
6.
Geramizadeh B. Unusual locations of the hydatid cyst: A review from Iran. Iran J Med Sci 2013;38:2-14.  Back to cited text no. 6
    
7.
Alaparthi RK, Yelamanchili S, Nunsavathu PN, Sode U. Intraoral hydatid cyst: A rare case report. J Indian Acad Oral Med Radiol 2015;27:457-60.  Back to cited text no. 7
  [Full text]  
8.
Aggarwal N, Panja T, Biswas K, Kar T. Hydatid cyst of tongue: A diagnostic challenge. Bengal J Otolaryngol Head Neck Surg 2017;25:170-2.  Back to cited text no. 8
    
9.
Raghunatha BG, Parasuram G, Muralidhara RS, Ramesh Chandra P, Kanchumurthy A. Hydatid cyst in the soft palate: A rare case report. J Evol Med Dent Sci 2015;4:4371-4.  Back to cited text no. 9
    
10.
Cemil B, Tun K, Gurcay AG, Uygur A, Kaptanoglu E. Cranial epidural hydatid cysts: Clinical report and review of the literature. Acta Neurochir (Wien) 2009;151:659-62.  Back to cited text no. 10
    
11.
Singh DB, Kumar S, Bhattacharya AB. Hydatid cyst in parotid gland- A rare case. IOSR J Dent Med Sci 2013;7:1-4.  Back to cited text no. 11
    
12.
Sultana N, Hashim TK, Jan SY, Khan Z, Malik T, Shah W. Primary cervical hydatid cyst: A rare occurrence. Diagn Pathol 2012;7:157.  Back to cited text no. 12
    
13.
Rochidi Y, Raji A, Elhattab Y. A rare localization of hydatidosis: A cervical hydatid cyst. Fr ORL 2007;92:315-7.  Back to cited text no. 13
    
14.
Khanna S, Mishra SP, Gupta D, Kumar S, Khanna AK, Gupta SK. Unusual presentation of hydatid cyst: A case report. Int J Res Appl Nat Soc Sci 2013;1:25-8.  Back to cited text no. 14
    
15.
Kirmani MA, Sajad M, Patigaroo AR, Khan AR. Hydatid cyst of tongue. JK Pract 2007;14:107.  Back to cited text no. 15
    
16.
Berkiten G, Topaloglu I. Submandibular hydatid cyst fistulized into the oral cavity. B-ENT 2013;9:251-3.  Back to cited text no. 16
    
17.
Umesh K, Sulabha AN, Sameer AC, Neelakant MW, Sangamesh NC, Ali RP. Hydatid cyst of infratemporal region – A rare case report. J Med Sci 2010;3:94-8.  Back to cited text no. 17
    
18.
Nayar RC, Rajwanshi A, Menn SB, Arora MM. Hydatid cyst in the floor of the mouth. Indian J Otolaryngol 1982;34:78-9.  Back to cited text no. 18
    
19.
Rasheed K, Zargar SA, Telwani AA. Hydatid cyst of spleen: A diagnostic challenge. N Am J Med Sci 2013;5:10-20.  Back to cited text no. 19
    
20.
Bouckaert MM, Raubenheimer EJ, Jacobs FJ. Maxillofacial hydatid cysts. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2000;89:338-42.  Back to cited text no. 20
    
21.
Megalamani SB, Balasubramanium D, Gopalakrishnan S, Saxena SK. Hydatid cyst of maxillary sinus. Clin Rhinol 2011;4:107-9.  Back to cited text no. 21
    


    Figures

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



 

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