Volume 2, Issue 1 , Pages 23-25, March 2007
Hydatid cyst of the submandibular salivary gland in a child
Article Outline
Summary
The majority of hydatid cysts are seen in the liver and lungs. In countries where Echinococcus infestation is common, high index of suspicion is required for hydatosis and although it is a rare location, a hydatid cyst should be considered in the differential diagnosis of the cervical lesions in order to avoid any dangerous complication such as untreatable multiple hydatosis or a fatal anaphylactic reaction. Surgical removal without causing any spillage of its contents is the most effective treatment. To the best of our knowledge, only one submandibular gland hydatid cyst case in a child has been reported.
A case of 9-year-old male with hydatid cyst of the submandibular salivary gland is presented. The literature was reviewed and characteristics of the disease were discussed.
Keywords: Hydatid cyst, Submandibular gland, Child
1. Introduction
Echinococcus has been called as hydatid disease or hydatidosis [1]. Hydatid disease of the head and neck region is not very common, even in countries where Echinococcus infestation is high [1].
The majority of hydatid cysts are seen in the liver and lungs [1], [2]. Hydatidosis appears less frequently in infratemporal fossa, pterygopalatine fossa, parapharyngeal space, mandible, maxillary sinus, orbit, tongue, parotid gland, and submandibular salivary gland [2], [3], [4], [5].
In this report, an example of hydatid cyst of the submandibular salivary gland in a child is described.
2. Case
A 9-year-old male high school student was admitted to our clinic in June 2005 with a 6 months history of a slow-growing, non-tender, mobile mass in the right submandibular region.
Physical examination on initial presentation revealed a diffuse soft tissue mass 5
cm in diameter involving the right submandibular region. The oral cavity floor and Wharton duct opening were normal.
Preoperative fine needle aspiration cytology (FNAC) was done. It showed only clear fluid, and no specific diagnosis was reported. Because of the clear appearance of the fluid, hydatosis was kept in mind for differential diagnosis. MRI analysis demonstrated a 20mm×20mm cystic mass in the right submandibular gland (Fig. 1). Serologic test for hydatosis was also performed. The result was reported, as negative. Chest X-rays were normal. The patient underwent surgery. There was a cystic mass adherent to the right submandibular gland. The mass was excised completely together with the right submandibular gland. No surgical difficulties were encountered and no spillage occurred in the operative field. Histopathology confirmed the diagnosis of submandibular hydatid cyst (Fig. 2).
Fig. 1.
Coronal MRI revealed a cystic mass located on the right submandibular region.
Fig. 2.
Hydatid cyst of the submandibular gland is detected by histopathological examination. Submandibular gland, germinative layer and kuticul of the hydatid cyst are seen (hematoxylin–eosin; ×200).
No evidence of a possible extraglandular site for a hydatid cyst was found. However, the patient was given postoperative 15mg/kg albendazole for 6 weeks, considering the possible presence of embryos in the circulation. The patient had an uneventful postoperative period. No sign of disease was present during the most follow-up examination performed 12 months after the operation.
3. Discussion
Hydatid disease is especially common in the sheep-raising and cattle-raising countries [2], [3]. The primary host is the dog family. The intermediate hosts are usually sheep, cattle, pigs, and human [1], [5].
Humans are accidentally infected by the orofecal route. In transferring the infection to humans, close contact between dogs and children is important. The ova enter in the human intestinal tract. The embryos pass through the intestinal wall and into portal system. The liver is the most frequent location for hydatisis. However, via the hepatic veins, inferior vena cava, heart, and pulmonary arteries, a few embryos may pass through the liver to spread throughout the body [4].
The majority of hydatid cysts appear in the liver (65%) and lungs (25%) [1]. Hydatidosis appears in infratemporal fossa, pterygopalatine fossa, parapharyngeal space, mandible, maxillary sinus, orbit, tongue, parotid gland, and submandibular salivary gland less frequently [2], [3], [4], [5].
Placitelli reported the first submandibular gland hydatid cyst case in 1938, and only a few cases have been reported in the English language literature so far [1], [4], [6], [7]. To the best of our knowledge, only one submandibular gland hydatid cyst case in a child has been reported [8].
Hydatidosis commonly appear as cystic lesions and these characteristically grow slowly (1–2cm per year) [3], [5]. The location, the size, and the pressure caused by the enlarging cyst, define the symptoms [4], [5].
The history of the patient, physical examination, diagnostic imaging, aspiration and serologic tests help diagnosis of hydatid disease. CT and MRI are the main facilities of diagnostic imaging. FNAC is preferable with minimal complications [9]. Serologic tests are controversial. Both false-positive and false-negative test results are seen frequently. The germinative layer and clear fluid is often adequate for preoperative macroscopic diagnosis. Patients with hydatid disease must undergo a thorough systemic investigation for possible multiorgan involvement (20–30%) [4], [10].
In our case, FNAC could not provide a specific diagnosis. However, because of the clear appearance of the fluid, hydatosis was kept in mind for differential diagnosis. Serologic tests revealed false-negative results for hydatosis. Location and extension of the lesion was evaluated by MRI.
Since there is no effective medical treatment, surgical removal without causing any spillage of the hydatid cyst's contents is still the most effective medical treatment [2], [4]. If it is not performed, the lesions are very likely to transform into an untreatable multiple hydatosis or anaphylactoid reaction may occur. Thus, preoperative diagnosis is significant in hydatid disease [9]. If hydatidosis can be diagnosed before the surgery, via injecting 20% hypertonic saline solution or 0.5% silver nitrate into the cyst, inactivation of daughter cysts and scolices can be achieved [1]. Pharmacologic therapy was also reported for hydatidosis in the literature. If the patient has high risk of contamination or if the surgery is not possible because of the patient's general condition, treatment with mebendazole or albendazole may be tried. But, the results are unpredictable and adverse reactions have been reported [1], [11].
In our case, the lesion was excised with the right submandibular gland and no spillage occurred in the operative field. Histopathology confirmed the diagnosis of submandibular hydatid cyst. Although no evidence of a possible extraglandular site for a hydatid cyst was found, the patient was given postoperative albendazole, considering the possible presence of embryos in the circulation.
4. Conclusion
High index of suspicion is required for hydatosis in countries where Echinococcus infestation is high, and in the differential diagnosis of lesions in the cervical region, a hydatid cyst should be considered in order to avoid any dangerous complication such as untreatable multiple hydatosis or a fatal anaphylactic reaction.
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PII: S1871-4048(06)00102-X
doi:10.1016/j.pedex.2006.11.003
© 2006 Elsevier Ireland Ltd. All rights reserved.
Volume 2, Issue 1 , Pages 23-25, March 2007
