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CASE REPORT
Year : 2019  |  Volume : 8  |  Issue : 1  |  Page : 62-66

Cystic echinococcus of head and neck: Anaphylactic dilemma and surgical considerations


1 Department of Oral and Maxillofacial Surgery, Sibar Institute of Dental Sciences, Guntur, Andhra Pradesh, India
2 Department of Oral and Maxillofacial Pathology, Sibar Institute of Dental Sciences, Guntur, Andhra Pradesh, India

Date of Submission21-Jun-2018
Date of Acceptance21-Aug-2018
Date of Web Publication26-Apr-2019

Correspondence Address:
Dr. Phani Kumar Kuchimanchi
Department of Oral and Maxillofacial Surgery, Sibar Institute of Dental Sciences, Takkellapadu, Guntur - 522 509, Andhra Pradesh
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/JDRNTRUHS.JDRNTRUHS_61_18

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  Abstract 


Hydatid cyst or echinococcosis is a zoonotic parasitic disease caused by dog tapeworm and occurs in humans only when they are accidental intermediate hosts for dog tapeworms. The liver is the most common organ affected (60%), followed by the lung (20%), kidneys (3%), brain (1%) and bone (1%). Previous literature recorded its occurrence in a very few cases involving head and neck region with only three cases involving cheek so far. The present case report demonstrates extreme rare occurrence of hydatid cyst in orofacial region. It is a rarity and potential risk of anaphylactic shock in susceptible individuals poses diagnostic and surgical challenge. Surgical management demands to be prepared with appropriate precautions to be taken perioperatively. Rationale for perioperative management of anaphylactic shock from cystic echinococcus is emphasized.

Keywords: Anaphylactic shock, dog tapeworm, Echinococcus granulosus, echinococcosis of cheek, hydatid cyst, IgE-mediated hypersensitivity, IgG-mediated endotoxic shock


How to cite this article:
Kuchimanchi PK, Chandrasekhar P, Manasa P L, Koneru G. Cystic echinococcus of head and neck: Anaphylactic dilemma and surgical considerations. J NTR Univ Health Sci 2019;8:62-6

How to cite this URL:
Kuchimanchi PK, Chandrasekhar P, Manasa P L, Koneru G. Cystic echinococcus of head and neck: Anaphylactic dilemma and surgical considerations. J NTR Univ Health Sci [serial online] 2019 [cited 2019 May 26];8:62-6. Available from: http://www.jdrntruhs.org/text.asp?2019/8/1/62/257170




  Introduction Top


Hydatid cyst or echinococcosis is a zoonotic parasitic disease caused by dog tapeworm that occurs primarily in sheep-grazing areas, but it is common worldwide because dog is a definitive host. Echinococcosis occurs in humans when they are accidental intermediate hosts for dog tapeworms. Infection is acquired by ingesting food contaminated with canine faeces containing parasite eggs. The principal species that infect humans are Echinococcus granulosus and Echinococcus multiloculares.[1],[2] Echinococcal larvae are sensitive to dry conditions and heat but tend to resist chemical agents and cold.[3],[4] Liver is the most common organ affected (60%), followed by the lungs (20%), kidneys (3%), brain (1%) and bone (1%).[5] Unilocular hydatid cyst is typical of E. granulosus, while multilocular or alveolar hydatid cyst in liver is caused by E. multiloculares. The present case report demonstrates extreme rare occurrence of hydatid cyst in orofacial region. Previous literature recorded its occurrence in a very few cases involving head and neck region with only three cases involving cheek so far.[6–8]


  Case Report Top


A 20-year-old female patient reported to the Department of Oral and Maxillofacial Surgery with a chief complaint of a non-tender palpable mass in right posterior cheek region, which she noticed 2 days before she reported to the hospital. Patient hails from a nomad family with agriculture as livelihood. History of present illness revealed no relevant history related to odontogenic infections or sialadenitis. The onset of the swelling was insidious with no obvious asymmetry or functional impedance. No significant cheek bite was present, no signs of infection or malignancy noted. On palpation a well-defined non-tender mass in buccal mucosa located over the anterior border of ramus was observed [Figure 1]. The mass was fixed at its posterior aspect in relation to anterior border of ramus of mandible; otherwise the lesion was free of attachment to any of the surrounding anatomical structures. It was non-compressible and firm in consistency. Cervical lymph node examination revealed no palpable lymph nodes. No signs of xerostomia were observed. On palpation, no abnormality was detected in relation to major salivary glands. Orthopantomogram revealed no abnormality. Ultrasonography revealed a thick-walled cystic lesion (1 × 0.8 cm) with hyperechoic mural focus deep to right masseter muscle at the level of angle of the mandible [Figure 2]. There is no significant vascularity within the lesion. Bilateral parotid and submandibular glands are normal in size and echotexture. A few small (5 × 5 mm sized) intraparotid nodes were present. The ultrasound picture of the lesion was suggestive of infective cysticercosis. Contrast-enhanced computed tomography (CECT) abdomen, chest and brain findings were normal.
Figure 1: Intraoral view showing the location of the mass

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Figure 2: Ultrasonography revealed a thick-walled cystic lesion (1 × 0.8 cm) with hyperechoic mural focus deep to right masseter muscle at the level of angle of the mandible

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Under local anaesthesia, the lesion was approached intraorally through an incision overlying the lesion measuring about 2 cm in length was given on right buccal mucosa along the third molar region at the level of occlusion and the entire mass was carefully dissected out separating it from buccal mucosa anteriomedially, masseter laterally and periosteum of the anterior ramus of the mandible posteriorly delivering the mass in toto. The surgical site was irrigated with hydrogen peroxide and betadine before closure of the surgical site using 3.0 vicryl. The histopathologic examination of the specimen [Figure 3] exhibited fibrous capsule with dense diffuse chronic inflammatory cells and few multinucleated giant cells. The intermediate fibrous capsule showed absence of nucleus and consists of numerous laminations. The inner layer showed brood capsule with daughter cysts that is suggestive of a hydatid cyst [Figure 4] and [Figure 5].
Figure 3: Showing the excised specimen

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Figure 4: Histopathology image (10x using pentahead microscope) showing fibrous capsule with dense diffuse chronic inflammatory cells and few multinucleated giant cells

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Figure 5: Histopathology image (20x zoom respectively using pentahead microscope) showing intermediate fibrous capsule showed absence of nucleus and consists of numerous laminations and the inner layer showing brood capsule with daughter cysts

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  Discussion Top


Hydatid disease occurs as a result of infestation by the larval stage of the tapeworm. The definite host, commonly dog, is infected by eating the viscera of sheep containing hydatid cyst. The infected faeces of the dog contaminate grass and farmlands from where the ova are ingested by the intermediate hosts like sheep and man. Thus man is at potential risk of acquiring the infection by handling dogs as well as by eating contaminated vegetables. The ova ingested by the man are liberated from the chitinous wall by the action of gastric juice and pass through the intestinal mucosa from where they are carried to the liver by portal venous system. Further development of majority of ova into hydatid cyst occurs in hepatic sinusoids, which act as first filter. The ova, which pass through liver enter the right heart and are caught in pulmonary capillary bed forming pulmonary hydatid cyst. Further, ova that enter the systemic circulation establishes into hydatid cyst in brain, spleen, bone and muscles. The patient's family history and occupation being agriculture background strongly favours the current diagnosis.

Hydatid cyst in the oral cavity is extremely a rare occurrence. The literature review (Science Direct Search) revealed that the occurrence of hydatid cyst in cheek mucosa was reported only once in the year 2002, over the past 25 years in head and neck region.[6] The other areas of head and neck reported were parotid gland, submandibular gland, mandible, pterygopalatine fossa, neck and orbit [Table 1].
Table 1: Incidence Of Hydatid Cyst In Head And Neck Region Reported So Far In The Literature

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In this particular case the CECT of abdomen and brain showed no evidence of hydatid disease involving liver, lungs or brain, suggestive of primary (solitary) hydatid cyst in cheek, which is extremely rare. The dietary habits, cultural background and occupation of the patient presenting with a mass in head and neck region owing to endemic tendency of the disease in developing countries like India, supports the differential diagnosis of hydatid disease. Ultrasonography is the preferred tool as it is non-invasive and safer over fine-needle aspiration cytology, since the fluid content of hydatid cyst is known to cause anaphylactic reaction when it is breached.

Hydatid disease requires both surgical and medical therapy. Surgically the cystic lesions are excised carefully in toto as the fluid spill from cystic cavity may cause anaphylactic shock[14] in susceptible individuals. Medical therapy includes oral administration of antihelminthic drugs (e.g. albendazole) like imidazole derivatives post-operatively.[14]

Anaphylaxis and hydatid cyst

The cause of anaphylactic shock in cystic echinococcus is still unclear. But, it has been thought that the antigens in the cyst and cyst fluid may have a role in inducing shock. However, anaphylactic shock as a result of surgery on an echinococcal cyst remains unpredictable and its pathogenesis remains elusive. Differences in immune reactions to different allergens, with possible different amounts of IgG and IgE produced, resulting in different likelihoods of shock. An alternative hypothesis is that in crossing the intestinal wall after ingestion, the larvae set up an immune reaction resembling that of a food allergy, which could account for the similar incidences with age. Another possibility is that children have lower immunity, and the complex of antigen and antibody caused by cyst leakage is relatively in excess in children, resulting in a stronger hypersensitivity reaction leading to shock.

Alves and Sheikh,[21] in their study, correlated age and occurrence of anaphylactic shock in different scenarios and reported that younger individuals are more susceptible. In a retrospective study on 446 patients surgically treated for hydatid cyst, Li et al., reported that younger age and presence of a pulmonary cyst are risk factors for anaphylactic shock.[22] Li et al.,[22] in their study found that the hypersensitivity reaction in case of hydatid cyst differed from that of a typical type 1 immediate hypersensitivity shock. It was suggested that the anaphylactic shock seen in hydatid cyst cases was rather a combination of type 1 immediate hypersensitivity and endotoxic shock. Of the symptoms and signs listed in the standards (Diagnostic Criteria of National Institute of Allergy and Infectious Diseases), the most pertinent in perioperative setting were considered in the study like hypotension; dysrhythmia; bronchospasm; flushing; urticaria; angioedema; morbilliform rash; piloerection; oedema of lips, tongue and uvula; periorbital erythema; conjunctival erythema; excessive tear formation; cough; stridor and rhinorrhoea. The antibodies were assessed per operatively using a rapid test by the Dot Immuno Gold Filtration Assay (DIGFA).

A rationale for perioperative management of anaphylactic shock from cystic echinococcus is as follows:[22]

  1. The reactions usually occur in the first 1–15 min after contact with hydatid fluid, but mostly within 3–5 min
  2. Minimizing exposure of tissues and blood to cystic fluids may be crucial in avoiding anaphylactic shock and sudden death
  3. The mechanism of anaphylactic shock caused by cystic echinococcus might be different from that of type I hypersensitivity. IgG was found in increased amount than IgE in case of anaphylactic reaction from hydatid cyst, unlike typical type 1 hypersensitivity reaction
  4. Large amounts of vasoactive materials that block vasoconstriction and cause failure of peripheral circulation are released as a result of leakage from an echinococcal cyst similar to what happens with type I hypersensitivity shock. Therefore, besides the general measures of anti-shock treatment, epinephrine should be used immediately in small doses: up to 50 μg each time for adults, and pediatric dosing for children, with a dosing interval of 5 min
  5. Use of vasoactive agents should be considered if severe hypotension persists even after three consecutive doses and should be treated to have cytotoxic and synergic effects until the condition stabilizes
  6. Dopamine infusion pump, 2 mg/kg/min continuously for 35–125 min, might help in stabilization of patient if there is persistent hypotension after the use of epinephrine
  7. Intravenous dexamethasone can be advantageous as it helps in inhibition of differentiation and proliferation of immune cells, consequently reducing the production of antibodies, decreasing vessel permeability, reducing inflammation, stabilizing lysosomal membranes and preventing release of a variety of enzymes
  8. Immediate fluid replacement with appropriate fluids prevents circulatory failure from third space loss, which might occur as a result of endotoxic shock in cystic echinococcus
  9. Early intervention is the key. Perioperative rupture of an echinococcal cyst with signs of anaphylactic shock requires immediate radical surgical intervention which helps in reduction of mortality rate.



  Conclusion Top


Over a period of time, with ever changing lifestyles and food habits, this extremely rare disease could become endemic irrespective of the socio-economic status. Solitary hydatid cyst in head and neck area, due to its extreme rarity is a potential diagnostic challenge. When a patient with positive history is encountered, the surgeon should carefully evaluate the condition and follow recommended surgical guidelines to prevent anaphylactic shock perioperatively.

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

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2.
Georgopoulos S, Korres S, Riga M, Kouvidou Ch, Balatsouras D, Ferekidis E, et al. Hydatid cyst in the duct of the submandibular gland. Int J Oral Maxillofac Surg 2007;36:177-9.  Back to cited text no. 2
    
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Yumusakhuylu AC, Yilmaz AS, Devecioglu I, Oysu C. Management of a pterygopalatine fossa hydatid cyst. Auris Nasus Laryn×2012;39:537-9.  Back to cited text no. 16
    
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Hilmani S, Bertal A, Sami A, Ouboukhlik A, El-Kamar A, El-Azhari A. Primary hydatid cyst of neck. J Neuroradiol 2008;359:188-9.  Back to cited text no. 18
    
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Katilmis H, Ozturkcan S, Ozdemir I, Guvenc IA, Ozturan S. Primary hydatid cyst of the neck. Am J Otolaryngol Head Neck Med Surg 2007;28:205-7.  Back to cited text no. 19
    
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21.
Alves B, Sheikh A. Age specific aetiology of anaphylaxis. Arch Dis Child 2001;85:348.  Back to cited text no. 21
    
22.
Li Y, Zheng H, Cao X, Liu Z, Chen L. Demographic and clinical characteristics of patients with anaphylactic shock after surgery for cystic echinococcosis. Am J Trop Med Hyg 2011;85:452-5.  Back to cited text no. 22
    


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