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Background: Myeloid sarcoma (MS), also referred to as granulocytic sarcoma or chloroma, is a rare type of extramedullary malignant tumor. MS comprises primitive granulocytic precursor cells that play a key role in the early stages of white blood cell development. Notably, the occurrence of this tumor in the gingiva is rare., Case Summary: The present study reported the case of MS with gingival swelling in the maxillary region, with aleukemic presentation in a 32-year-old male patient. Following two courses of chemotherapy, computed tomography of the region demonstrated complete clearance of the tumor. At the 12-month follow-up appointment, the patient was in a stable condition with the absence of progression. The etiology, clinical features, diagnosis, and relevant treatment of MS are discussed in the present study., Conclusion: Diagnosis of MS may be confirmed following histological and immunohistochemical examinations., Competing Interests: Conflict-of-interest statement: The authors have stated explicitly that there are no conflicts of interest in connection with this article., (©The Author(s) 2024. Published by Baishideng Publishing Group Inc. All rights reserved.)