Rationale: Acantholytic squamous cell carcinoma (ASCC) is an uncommon histopathologic variant of squamous cell carcinoma (SCC), which is the most common malignancy of the oral cavity. Though ASCC showed poor prognosis, the exact diagnosis is challenging.
Patients concerns: A 59-year-old female patient with 1-month long symptoms of pain and burning sensation in the right maxilla.
Diagnoses: Incisional biopsy in the maxilla established the pathologic diagnosis of SCC.
Intervention: The patient underwent mass resection with near total maxillectomy.
Outcomes: The final diagnosis through the microscopic examination was ASCC. Palliative chemotherapy was done to relive the symptoms after the recurrence, however, the patient died of the disease at 8 months after her initial presentation.
Lessons: Special attention should be paid to this variant of SCC because most patients with ASCC have a very poor prognosis.