Radical Treatment of an Aggressive, Recurrent Benign Inflammatory Lesion of the Tongue: A Case Report & Review of the literature

Case Presentation

Austin J Otolaryngol. 2017; 4(1): 1089.

Radical Treatment of an Aggressive, Recurrent Benign Inflammatory Lesion of the Tongue: A Case Report & Review of the literature

Rachelle LeBlanc, Anil Sharma, Peter Spafford, Brent Wilde and Rick Jaggi*

Department of Otolaryngology-Head & Neck Surgery, College of Medicine, University of Saskatchewan, Saskatoon, Saskatchewan, Canada

*Corresponding author: Jaggi R, Department of Otolaryngology-Head & Neck Surgery and Facial Plastic Surgery, College of Medicine, University of Saskatchewan, Saskatoon, Saskatchewan, Canada

Received: April 05, 2017; Accepted: May 03, 2017; Published: May 10, 2017

Abstract

Background: There are limited publications on the management of large aggressive recurrent benign inflammatory lesions of the tongue. These lesions have a wide range of etiologies that must be explored. Many documented reports on eosinophilic ulcers/traumatic ulcers suggest surgical excision with recurrence rates relatively low.

Case Report: This article reports on an unusual case in a 48-year-old male patient with a large left anterolateral tongue ulcer with polypoid granulation tissue that has recurred after three attempts of partial glossectomy and injection of corticosteroids. A final left hemiglossectomy with a supraclavicular island flap reconstruction was performed and is currently being followed for ongoing management.

Conclusion: Aggressive, large benign inflammatory lesions of the tongue may benefit from radical treatment if all conservative management fails. Correct diagnosis, careful surgical planning, and patient preference of treatment should all be performed while maximizing success, quality of life, and improving tongue function.

Keywords: Case report; Traumatic ulcer; Eosinophilic ulcer; Pyogenic granuloma; Tongue; Hemiglossectomy

Abbreviations

CT: Computed Tomography; EU: Eosinophilic Ulcer; PG: Pyogenic Granuloma; TUG: Traumatic Ulcerative Granuloma

Introduction

Large tongue lesions in a non -smoker, non- drinker have a wide range of etiologies. The differential diagnoses include hyperplasias, papillomas, soft tissue tumor and malignancy, particularly squamous cell carcinoma. In the present case, histopathological investigations were essential to distinguish between a variety of benign inflammatory processes of the tongue. After initial investigations the differential diagnosis was narrowed to include Traumatic Ulcerative Granuloma (TUG), Eosinophilic Ulcer (EU) and Pyogenic Granuloma (PG).

TUGs/EUs are benign lesions that are usually self-limiting. They manifest as mucosal ulceration and inflammation that extends into underlying muscle, which shows a predominance of histiocyte-like mononuclear cells and eosinophils, in addition to nonspecific acute and chronic inflammatory cells [1]. The etiopathogenesis is unknown, however, trauma seems to play an important role in the development [2]. EUs are frequently located on the tongue but can also occur in other locations such as lips, buccal mucosa, palate, gingiva and floor of the mouth [3,4].

The other lesion that should be included in the differential diagnosis is PG, a hyperactive benign inflammatory lesion that most often occurs on the lips and mucosa of the oral cavity. PG lesions are exophytic, presenting with smooth or lobulated surfaces that usually bleed [5]. They typically range in size from a few millimeters to several centimeters, rarely exceeding 2.5cm [6,7]. The etiology of PGs is unknown, however, predisposing factors include chronic irritation, trauma, infections and hormonal factors [8]. PGs occur in a variety of ages, however, they are most common in the second decade of life. Women are affected more often, likely due to hormonal effects [9-11]. It is important to note that these lesions may recur if not removed completely [5].

The following case report describes an aggressive recurrent large benign inflammatory lesion treated with three attempts of partial glossectomy, intralesional corticosteroid injection and a hemiglossectomy with a supraclavicular island flap reconstruction.

Case Presentation

A 48-year old male presented to the Otolaryngology- Head and Neck Surgery department with a 6-week history of a left tongue lesion described as quite painful. He had a history of recurrent abscess ulcerations, which resolved after 2 weeks. He is a non-smoker, nondrinker with no significant risk factors for any cancer. Initial physical exam showed a 1 x 2cm ulcerative lesion along the left lateral tongue, which was firm on palpation. No other lesions noted on the entire sub sites of the oral cavity. Endoscopic laryngoscopy was performed and was completely normal with no base of tongue involvement.

Computed Tomography (CT) of the neck with contrast was performed. Findings suggested an asymmetric, ill-defined round region of hypodensity measuring 22x19mm at the left base of tongue. There was no evidence of local extension, with no crossing over the midline. A left partial glossectomy was performed and sent to surgical pathology. The specimen consisted of squamous lined mucosa with a large central ulceration. The squamous epithelium, at the margins of the ulceration, exhibited hyperkeratosis and parakeratosis. Staining with pankeratin showed no evidence of invasive carcinoma. Immunohistochemistry and special stains did not highlight infectious organisms.

Three months later the patient returned to the department with a recurrent mass on the left lateral margin of the tongue, which was painful with intermittent bleeding. Further physical examination revealed an exophytic mass along the left lateral margin of the tongue where the previous resection had taken place. There was no active bleeding and was soft on palpation. A second resection was performed and the specimen was sent to surgical pathology. The specimen consisted of several sections of polypoid mass. The mass was lined with squamous epithelium, which in some places, exhibited marked pseudo-epitheliomatous hyperplasia. In focal areas, the squamous mucosa was ulcerated with a fibropurulent cap and underlying granulation tissue. The inflammatory infiltrate consists primarily of neutrophils with a secondary population of lymphocytes, macrophages and plasma cells. Eosinophils were also noted within the areas of ulceration and granulation tissue. At this time the findings suggested a diagnosis of an ulcer with polypoid granulation tissue. It did not represent a pyogenic granuloma, as it did not have characteristic lobular architecture of the lesion.

Two months later the patient was referred for a second opinion with another recurrence of the left lateral granulomatous tongue ulcer. The lesion returned with more exuberance and pain, and was bleeding. Attempt with steroid injections into the base of the granulation tissue was made and had little effect. Further examination revealed tongue deviation significantly to the left upon protrusion, implying a left hypoglossal nerve paralysis. The patient was taken back to the operating room for an excision of the left posterior floor of mouth lesion and a partial glossectomy was performed. The surgical wound was left open to heal by secondary intention (Figure 1-3). Surgical pathology revealed left tongue ulcer with extensive inflammation.