FREDERIC B.ASKINMDWILLIAM H.WESTRAMD
IN SEPTEMBER 2000, a 38-year-old man presented with a 2-month history of a slow-growing, indurated, nontender, fixed mass, measuring 2 × 3 cm, in the right submandibular area. He reported a decline in body weight but denied fever, dentalgia, and odynophagia. A physical examination of his oral cavity demonstrated some buccal leukoplakia located near the right lower molar, but the dentition appeared intact, without carious teeth. Computed tomographic scans of the patient's neck revealed abnormal enhancement in the inferior aspect of the right submandibular gland, with involvement of the platysma muscle and the presence of a "dirty fat" sign in adjacent fat planes (Figure 1), but no associated abnormalities were observed in the mandible. A malignancy was highly suspected, followed by a chronic granulomatous process, such as tuberculosis or nocardiosis. With the patient under general anesthesia, surgical exploration revealed excessive fibrosis, adhesion, and necrotic tissue over the right submandibular area, with some encasing of the marginal mandibular branch of the facial nerve. A frozen section reflected chronic inflammation without malignancy. After careful dissection and preservation of the marginal mandibular branch of the facial nerve, the submandibular gland with surrounding necrotic and fibrotic soft tissue was then removed en bloc. Histopathological examination revealed no remarkable change to the submandibular gland, although periglandular lymph nodes did demonstrate a characteristic picture of basophilic sulfur granules (Figure 1, asterisk) with radiating bacterial filaments on a hematoxylin-eosin–stained section (Figure 2). On gram (Figure 3A) and Gomori methenamine silver (Figure 3B) stains, numerous gram-positive branching and filamentous bacilli within the granules were clearly demonstrated. Cultures were negative for aerobic and anaerobic bacteria, fungi, and mycobacteria. The patient had an excellent postoperative recovery, without facial palsy, and was discharged on the sixth day after surgery. Intravenous penicillin was administered postoperatively for 5 days, and then the regimen was changed to oral amoxicillin (500 mg 4 times daily) and intramuscular penicillin G benzathine (2.4 million U/wk for 8 weeks). Twelve months after surgery, the patient remained well, with no evidence of recurrence at follow-up.
Guo Y, Lan M, Tai S, Chang S, Chen W. Pathology Quiz Case 1. Arch Otolaryngol Head Neck Surg. 2002;128(9):1099. doi:10.1001/archotol.128.9.1099
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