Oral submucous fibrosis associated oral squamous cell carcinoma: A case report with review of literature

Abstract

Oral submucous fibrosis (OSMF) is a chronic, insidious premalignant disease associated with significant functional morbidity and an increased risk for malignancy. The oral squamous cell carcinoma (OSCC) originated from OSMF are tend to be highly invasive, metastatic & well differentiated. Here, we report a case of a Bangladeshi female having a long history of areca nut chewing and was diagnosed with OSMF-associated OSCC. Histologically, the tissue biopsy showed evidence of dense fibrosis and presence of islands of atypical squamous cells in the subepithelial region. The clinical and radiological findings also support the evidence of OSMF-associated OSCC in patient.

Highlights

  • Oral submucous fibrosis (OSMF) is a chronic premalignant lesion, which has a high tendency to progress into oral squamous cell carcinoma (OSCC) if left untreated.

  • OSMF-associated OSCC is a typical case in Bangladesh and in the South Asia due to the extensive use of araca nut and betel quid, and the negligence of patients about oral health.

  • Restricting the habitual use of areca nut and betel quid could prevent the development of OSMF-related OSCC.

Introduction

Oral submucous fibrosis (OSMF) is a chronic, stealthy disease associated with increased risk for malignancy [ ]. The major risk factors for OSMF are the areca nut & betel quid chewing. The most common anatomical sites for OSMF are buccal mucosa, soft palate & inner aspects of lip. The role of areca nut as a carcinogen has been proven with a large number of animal studies demonstrating its carcinogenicity, mutagenicity and genotoxicity [ ]. Malignant transformation of OSMF to oral squamous cell carcinoma (OSCC) has been estimated to be between 2% & 8% [ ]. Most common mechanisms of malignant transformation are thought to be through collagen maturation, their interaction with myofibroblast and mast cell, and fibrosis driven vascular constriction that results in epithelial hypoxia [ ]. This fibroblastic change of lamina propria with epithelial atrophy leads to stiffness of oral mucosa, difficulties in mouth opening, trismus, and inability of eat and speak. OSMF may remain either stable or may develop severe, leaving the patient handicapped physically and psychologically. However, the increased potential for malignant transformation of OSMF should be a concern for the global oral health officials. In this case report, we present a patient having long history of areca nut chewing, developed OSF, which later progressed into oral squamous cell carcinoma.

Case report

A 60-year-old woman got herself admitted in the oral & maxillofacial surgery department, Dhaka Dental College Hospital in May 2021 with complaints of pain, burning sensation on the left side of the mouth and had restricted mouth opening for 3 months. Pain was localized, deep-seated and piercing in nature. She had history of betel quid and areca nut chewing for 15 years. The patient was diabetic and otherwise healthy.

On extra-oral examination, her face was asymmetrical with mild swelling in the left lower face ( Fig. 1 a). A palpable, mobile, non-tender, firm single lymph node swelling was found in the left submandibular region. Fibrous bands were also observed on both sides of the buccal mucosae leaving her with ‘Fish Mouth Appearance’ during mouth opening ( Fig. 1 b). The mouth opening was restricted with inter-incisal distance 30mm.

Fig. 1
a) Extraoral frontal view of the patient demonstrating an area of slight swelling on the lower left side of the mandible without overlying erythema. b) ‘Fish Mouth Appearance’ during mouth opening due to the presence of fibrous band on both sides of the buccal mucosa. c) Intraoral view showing irregular whitish indurated ulcer in the lower left alveolar ridge with missing of left mandibular canine, first and second premolars.

Intra-oral examination revealed an indurated ulcer in left lower alveolar ridge extending anterior-posteriorly from lower left lateral incisor to left mandibular first molar. In addition, loosening of left mandibular central incisor, lateral incisor and first molar with missing of left mandibular canine, first and second premolars. The lesion extended bucco-lingually from lingual sulcus to bucco-gingival sulcus ( Fig. 1 c). The ulcer was irregular in shape, measuring 5–6 cm with irregular margin, everted edge, indurated base, and the floor was filled with whitish necrotic granulation tissue. The buccal mucosa on the left side appeared blanched, extending from the commissure of the mouth to the pterygomandibular raphe. Abnormal white patches were visible on the dorsum of tongue. Loss of sensation on the left side of the lower lip was also noted. Considering patient’s history coupled with clinical examinations, our provisional diagnosis was oral submucous fibrosis associated OSCC.

The incisional biopsy taken from blanched mucosa and ulcerated area and stained with haematoxylin and eosin (H&E) staining, revealed thinness of mucosa with evidence of dense fibrosis in the subepithelial region ( Fig. 2 a). Masson’s trichrome stain stained the fibrous tissue blue in subepithelial region ( Fig. 2 b). Another tissue biopsy from the left side of the buccal mucosa and stained with H&E revealed evidence of OSCC as there were islands of atypical squamous cells in the subepithelial region ( Fig. 2 c).

Fig. 2
a) Thinness of the epithelial layer with evidance of dense fibrosis in the subepithelial region (H&E; magnification ×100). b) Masson’s trichrome staining showing blue stained fibrous tissue in the sub epithelial region (magnification ×100). c) Higher magnfication of the tissue biopsy from the left side of the buccal mucosa showing islands of atypical squamous cells in the subepithelial region (H&E; magnification ×400).

Orthopantomogram (OPG) showed marked bony erosion (radiolucent area) on the left side of the body of the mandible ( Fig. 3 ). The CT (Computed Tomography) scan revealed a plaque like lesion (46.7mm × 21.5mm) at left lower lip with deeper extension invaded mandible body & floor of the mouth ( Fig. 4 a and b). There is a superior extension of the mass over the left maxilla. Few mildly enlarged lymph nodes are noted at both sides of the4submandibular regions with the largest node reported on the left side, measuring, 10.2mm × 9.7mm ( Fig. 4 a and b). The case was evaluated as T4aN2aMx according to AJCC (The American Joint Committee on Cancer AJCC) TNM system.

Aug 14, 2022 | Posted by in Oral and Maxillofacial Surgery | Comments Off on Oral submucous fibrosis associated oral squamous cell carcinoma: A case report with review of literature
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