Departments of ¹Plastic and Reconstructive Surgery and ²Otolaryngology, Derriford Hospital, Derriford Road, Plymouth, PL6 8DH, England.

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Tumors of the minor salivary glands of the upper aero-digestive tract are rare, accounting for up to 20% of tumors of salivary glands [1]. These tumors are more likely to be malignant as compared to tumors of the major salivary glands, although benign tumors are still common place [2]. The most frequently encountered benign tumor is the pleomorphic adenoma which usually occurs in the palate at the junction between the hard and soft palate [3,4], although other intraoral sites have been reported [1,4-7]. We present here a rare occurrence of a pleomorphic adenoma arising from right glosso-tonsillar sulcus.

A 41 year old female patient presented to the ear, nose and throat (ENT) clinic with a four month history of a progressive feeling of globus, the sensation of food sticking in her throat. and persistent sore throat. She did not report any history of trauma, dysphonia or odynophagia. In addition there was no history of weight loss. The patient’s past medical history was unremarkable aside from a tonsillectomy at the age of 15. The patient was a non-smoker and consumed moderate amounts of alcohol. On examination there was a large pedunculated cystic lesion on a stalk arising from the lower pole of the right tonsillar fossa and tongue base. A provisional diagnosis of a benign lingual tonsil cyst was made. Clinically the lesion had benign appearances therefore imaging was deemed unnecessary and a decision was taken to proceed for complete surgical excision for histological analysis. Surgical excision under general anaesthesia plus pharyngo-laryngoscopy took place approximately one month following initial assessment in clinic on a routine basis. Intra-operatively, the patient had a superficial lesion at the right lingual tonsil/tongue base area that was dissected fully without being ruptured with bipolar diathermy [Fig.1-3]. Otherwise, the larynx, pharynx and hypo-pharynx were normal in appearance.

The excised operative sample measured 20 × 12 × 10 mm and was sent for a routine histological analysis. The microscopic examination revealed a minor salivary gland containing a circumscribed encapsulated tumour composed of ductal epithelial cells arranged in small islands, with a trabeculated and corded pattern mixed with chondromyxoid stroma [Fig.4]. This was reported as being consistent with a cellular pleomorphic salivary gland adenoma in a minor salivary gland of the glosso-tonsillar sulcus. 

Surgical excision as day case surgery was successful and uncomplicated. Ten days post-operatively the patient had a minor secondary hemorrhage that resolved with conservative measures. Given the clinical, radiological and histopathological findings, the patient has been listed for regular outpatient follow-ups with periodic MRI scans of the head and neck to rule out recurrences. No recurrence of the tumor has been reported to date.

Minor salivary glands are small mucous glands found throughout the upper aero-digestive tract. Common locations of PSA of the minor salivary glands are the palate, buccal mucosa, lips and retro-molar area [4,6]. Rare cases have been reported in the nasopharynx [1] and para-pharyngeal space [7]. Clinically these tumors tend to be slow growing and usually present as an enlarging mass; other symptoms include location-specific symptoms such as odynophagia (such as our case), or compression symptoms such as cranial nerve palsies [1,3,5,6,7]. The differential diagnosis should include other benign growths (mucocele, myoepithelioma, monomorphic adenoma), as well as malignant tumors of which the most common minor salivary gland tumour is an adenoid cystic carcinoma. Symptoms that are suspicious of malignancy include pain, bleeding, loosening of teeth and ulceration (unless as a consequence of trauma) [3]. Although the majority are benign, there are malignant varieties of PSA (carcinoma ex pleomorphic adenoma, carcinoma sarcoma and metastasising PSA) which should be kept in mind and can be accurately differentiated on histological analysis [1].

The diagnosis of PSA is carried out through careful clinical assessment, histological analysis, and complementary imaging, usually in the form of MRI [1,3,4,6]. Surgical excision is the mainstay of treatment with no role for radiotherapy or chemotherapy initially, although it can be considered in recurrent disease [1]. PSA’s classically have a thin incomplete capsule; therefore, complete surgical excision with a cuff of surrounding healthy tissue should be carried out while every effort should be made to preserve vital adjacent structures [4]. For tumors such as the one described here an intraoral approach is appropriate and advisable. It is advised that long term follow-up should occur due to the high risk of recurrence and malignant potential [8,9]. 

We have presented here a rare case of this tumor in the right glossotonsillar sulcus. In the light of literature review and the details of this case we recommend that all similar cases should be treated with a high index of suspicion, as most minor salivary glands tumors are malignant.

Contributors: HW: manuscript writing, literature review, and patient management; MA: manuscript editing, literature review, and patient management; MM: critical inputs into the manuscript, and patient management. HW will act as guarantor. All authors approved the final version of this manuscript.

Funding: None; Competing interests: None stated.