Volume 4, Issue 2 , Pages 53-55, March 2009
Submental primary hydatid cyst: A rare differential diagnosis of midline neck swelling in children
Article Outline
Summary
Hydatid cysts of the cervical region are extremely rare especially in children and the diagnosis is quite challenging. We report herein a child with a hydatid cyst that was primarily located in the submental region without any pulmonary or hepatic involvement. A hydatid cyst of the neck should be considered as a possible diagnosis while evaluating any slow growing cyst in the cervical region especially in a patient living in endemic areas so as to keep surgical precautions in mind while operating to avoid any dangerous complications such as contamination and a fatal anaphylactic reaction.
Keywords: Hydatid cyst, Submental cyst, Echinococcus
Introduction
Hydatid disease is a cyclozoonotic infestation caused most commonly by Echinococcus granulosus. The dog and other canine species are primary hosts while sheep, cattle, horses and occasionally humans are intermediate hosts. Humans usually contract the disease in childhood or adolescence by ingesting ova shed in faeces of infected dogs [1], [2]. In humans, there is a localized cystic disease found primarily in the liver and lung (70–85%) [2], [3], [4] with unusual locations being peritoneum, heart, spleen, kidney, spine, bones, chest wall and other organs [3], [4], [5]. Although, some cases of primary hydatid cysts located in cervical region in adults have been reported in the literature [1], [6], [7], [8] none were present in the submental region.
We therefore thought it would be of interest to report a child with primary hydatid cyst of the submental region for its rarity.
Case report
An 8-year-old girl presented with a slow growing painless swelling over the submental region of neck for last 2 years. She had no other associated complain. On physical examination, a non-tender, cystic swelling 5
cm×4cm size was present over the submental region of neck (Fig. 1).
Figure 1.
Nontender, firm swelling 5
cm×4cm size present over submental region of neck.
On ultrasound examination of the submental region of neck, there was a cyst 5cm×4cm size with anechoic, clear fluid content and trilaminar wall, highly suggestive of a hydatid cyst (Fig. 2). The abdominal ultrasonography and chest X-ray were normal.
Figure 2.
Ultrasound examination of the submental region of neck shows a unilocular cyst 5
cm×4cm size with anechoic, clear fluid content and trilaminar wall, highly suggestive of a hydatid cyst.
Albendazole chemotherapy (10mg/(kgday)) was given for a week then surgery was planned.
Under general anaesthesia, neck exploration was done. The swelling was found to be located in the submental triangle beneath the mylohyoid muscle (Fig. 3). Following the protection of surrounding structures by packing all around with sponges soaked in povidone iodine, cyst decompression was done using syringe-needle which revealed crystal clear fluid. Daughter cysts along with the germinative membrane were evacuated from the cyst cavity following cystotomy. Postoperatively, albendazole chemotherapy (10mg/(kgday)) was continued for 3 weeks. She showed an uneventful recovery and is doing well with a follow up duration of 2 years.
Figure 3.
Intraoperative picture showing the hydatid cyst beneath the retracted mylohyoid muscle.
Discussion
Hydatid cysts develop most frequently in the liver and lungs and rarely involve the brain, eye, heart, bone or other internal organs [9]. Hydatid cysts located in the neck are extremely rare even in the endemic areas and to date, very few cases have been reported. Prousalidis et al. [3] in a review of 49 cases with hydatid cysts especially those located in the various organs other than the liver and lungs, found none in the cervical region. Few cases of cervical region primary hydatid cyst involving posterior cervical triangle [10], submandibular region [6], parotid region [7] and thyroid gland [11] have been reported in adults.
In our case the hydatid cyst was located in the submental region of the neck without involvement of any other organ which has not been reported.
The diagnosis of hydatid disease may be established with clinical presentation, plain radiography, ultrasonography and CT scan. Unless suspected or demonstrative radiological findings are available, preoperative diagnosis may be missed. Hence, the cystic fluid may spurt out during the operation giving rise to early or late complications. USG and CT scan is useful in visualizing cystic masses by demonstrating internal septae and daughter cysts [1], [7], [8]. Intraoperatively, the presence of whitish, clear cyst fluid and the germinative layer is often adequate for diagnosis of hydatid disease. The optimal treatment of choice is surgical removal of cyst content including the germinative layer.
In our case, although we found no signs of any other organ involvement but considering the possible presence of embryos in the circulation, we gave supplementary albendazole medication.
References
- . Primary hydatid cyst of the neck. Eur. Arch. Otorhinolaryngol. 1999;256:202–204
- Hydatid disease of lungs, study of 386 cases. Am. Rev. Respir. Dis. 1992;146:185–189
- Uncommon sites of hydatid disease. World J. Surg. 1998;22:17–22
- A multivesicular cardiac hydatid cyst with hepatic involvement. Eur. J. Cardiothorac. Surg. 1998;14:335–337
- . Chest wall echinococcosis. Chest. 1994;105:1277–1279
- . Submandibular hydatid cyst. J. Craniomaxillofac. Surg. 1991;19:359–361
- Cervical echinococcal hydatid cyst. J. Laryngol. Otol. 1989;103:435–437
- Hydatid cyst in the head & neck area. Am. J. Otolaryngal. 1995;16:123–125
- . Infectious Diseases and Medical Microbiology. 2nd ed.. W.B. Saunders Company; 1986;p. 945
- . Primary hydatid cyst of the posterior cervical triangle. J. Laryngol. Otol. 2002;116:153–155
- . Echinococcosis of the thyroid gland. Report of two cases. JAMA. 1967;200:178–179
PII: S1871-4048(08)00045-2
doi:10.1016/j.pedex.2008.06.001
© 2008 Elsevier Ireland Ltd. All rights reserved.
Volume 4, Issue 2 , Pages 53-55, March 2009
