We report very rare and first case of infected submandibular sialocele following surgery for sialadenitis in a 45 years old man. He had persistent pain and discharge from the submandibular region after surgery. Sialocele was diagnosed on CECT neck and was confirmed on surgical exploration.

Bakshi J, Kaur N, Paudel S (2016) Sialocele – A Rare Entity Following Submandibular Gland Excision. J Ear Nose Throat Disord 1(1): 1015.

• Submandibular

Sialoceles are characterised by extravasation of saliva into the surrounding soft tissue, and may be idiopathic, posttraumatic, or iatrogenic [1-3]. Sialocele is most commonly seen in parotid gland and rarely in submandibular gland and sublingual gland. Sialoceles of the submandibular gland are rare, and only five cases of submandibular sialoceles have been published so far. Common causes of injuries include penetrating wounds (sharp instruments), perforating wounds (firearms) and injuries secondary to surgical procedures4 .Infected sialocele is even more rare with only 1 case published so far [5]. Diagnosis depends on a proper history taking with complete physical examination and imaging in order to discard other lesions of a similar appearance. This paper describes an infected sialocele of the submandibular gland 4 months post surgical excision of submandibular gland for chronic submandibular sialadenitis with history of 2 years.

A 45 years healthy male presented with complaints of swelling in the left submandibular region since 2years insidious onset and gradually progressive which was associated with pain while chewing food. 19 months later patient underwent surgical excision of the left submandibular gland with preoperative findings of 3.5 X 2 cm left submandibular gland swelling with histological features of chronic non specific sialadenitis. 4 months post surgery patient noticed a swelling in left submandibular region gradually progressive and only associated with pain on chewing food. No associated history of fever was present. Total leucocyte count was increased at 14,300 cells per cubic millimetre. 

On examination there was a 3X2 cm firm, non tender, mobile swelling in left submandibular region and was bimanually palpable with 2X2 cm mucosal bulge present in left floor of mouth just beneath the ventral aspect of tongue. CECT Neck was done which showed a cystic lesion along the course of left submandibular duct.

Patient underwent surgical excision of left sialocele with findings of 5 X 3 cm cystic swelling in the left submandibular region with ~7 ml of purulent fluid coming out of the swelling. Fluid analysis showed Pus cells present with aerobic bacterial culture sterile and no AFB seen in entire specimen.

Sialoceles are characterised by extravasation of saliva into the surrounding soft tissue, and may be idiopathic, posttraumatic or iatrogenic [1-3]. Most commonly sialoceles are encountered in parotid gland with only few published sialoceles being found in submandibular gland. Sialoceles in parotid gland occur following superficial parotidectomy rather than near total or total parotidectomy[6]. Only 3 cases of idiopathic submandibular sialocele have been published so far [1]. However, no infected submandibular sialocele post surgery has been published.

Infected sialoceles are rare in the salivary glands with rarer being in the submandibular gland due to less likely chances of sialocele itself as parotid gland is the most frequently involved gland.

We think that ours is the first case of an iatrogenic, postoperative infected submandibular sialocele.

Diagnosis can be made with proper history taking along with past history of surgery or other trauma along with the imaging modalities like Ultrasonography or CECT neck. Patients will have increased total leukocyte count with presence of fever or not with tender swelling. Post operative fluid analysis of the aspirated fluid with gram staining, culture sensitivity and AFB staining needs to be done when infected sialocele is suspected.

1. Ang AH, Goh YH. Idiopathic submandibular sialoceles in the neck. OtolaryngolHead Neck Surg 2005; 132: 517–519.

2. Marchese-Ragona R, Blotta P, Pastore A, TugnoliV, Elopra R, De Grandis D. Management of parotid sialocele with botulinum toxin. Laryngoscope 1999; 109: 1344–1346.

3. Capaccio P, Cuccarini V, Benicchio V, Minorati D, Spadari F, Ottaviani F. Treatment of iatrogenic submandibular sialocele with botulinum toxin. Case report. Br J Oral Maxillofac Surg. 2007; 45: 415-417.

4. Medeiros Junior R, Rocha Neto AM, Queiroz IV, Cauby Ade F, Gueiros LA, Leao JC. Giant sialocele following facial trauma. Braz Dent J. 2012; 23: 82-86.

5. Williams JB, Orr SC. Images in emergency medicine. Acute submandibular sialadenitis with infected sialocele. Ann Emerg Med. 2007; 50: 24.

6. Witt RL. The incidence and management of siaolocele after parotidectomy. Otolaryngol Head Neck Surg. 2009; 140: 871-874.