Background: Actinomycosis is a rare, chronic granulomatous infection caused by Actinomyces species, often affecting the cervicofacial region, particularly the submandibular gland.

Case Presentation: We report a unique case of actinomycosis in a 47-year-old male with a history of recurrent sialolithiasis. The patient presented with swelling, tenderness, and discharge from the submandibular region. Despite prior surgical interventions and antibiotic treatments, symptoms persisted, leading to a thorough evaluation. Imaging studies indicated significant sialolithiasis. A biopsy of the affected gland revealed actinomycotic granulomas, confirming the diagnosis.

Management: The patient was treated with prolonged high-dose penicillin therapy and underwent surgical resection of the affected gland.

Conclusion: This case underscores the importance of considering actinomycosis in differential diagnoses of chronic submandibular infections, especially in patients with recurrent sialolithiasis. Early recognition and appropriate management, including long-term antibiotic therapy and surgical intervention, are crucial for effective treatment.