Septic thrombophlebitis of the internal jugular vein (IJV), known as Lemierre's syndrome, is a rare complication secondary to infections in the head and neck. Cholesteatoma of the middle ear complicated with Lemierre's syndrome is rarely observed in clinical practice. Currently, the treatment controversy centers on whether anticoagulation therapy is necessary for IJV and distant metastatic emboli induced by Lemierre's syndrome. A 46-year-old female patient with middle ear cholesteatoma underwent modified radical mastoidectomy and tympanoplasty surgery and complicated with Lemierre's syndrome, presenting with intermittent high fever, chills, headache, and left lateral neck pain. Computed tomography (CT) revealed thrombosis and internal gas in the left IJV, while blood culture and blood pathogenic microorganism metagenomic detection were negative. We administered sodium ceftriaxone (1 g every 12 hours) for 3 days. According to the secretion culture results showing 90% Actinomyces europaeus and 10% Corynebacterium without mycolic acid, penicillin (2.4 million IU) was added intravenously every 6 hours. The patient's infection worsened on the first day after surgery. We adjusted to upgrade anti-infection vancomycin 1 g every 12 hours, combined with meropenem (1 g every 8 hours) and metronidazole (0.5 g) every 8 hours for 4 weeks, and subcutaneous injection of enoxaparin 0.4 mL every 12 hours for 1 week, then adjusted to rivaroxaban tablets (15 mg bid). Amoxicillin-clavulanate for 2 weeks and rivaroxaban 10 mg were administered orally for 3 months after discharge. A follow-up neck CT scan with intravenous contrast suggested that the gas in the left IJV had disappeared, but the thrombus persisted. During the 3 month follow-up, the patient's vital signs, blood routine, and D-dimer levels were within the normal range. The surgical area healed well, and the patient reported no discomfort. Lemierre's syndrome represents a potentially-fatal complication that results in considerable mortality and must be identified early and aggressively treated.

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