Aim and objective: Aim of this case report is to illustrate a rare subcutaneous emphysema during third molar extraction. Background: Third molar extraction is a common procedure in oral surgery. Common complications that may occur after the surgical procedure include bleeding, pain, swelling, and trismus. Subcutaneous emphysema in an unusual complication during dental extraction and it could lead to life-threatening situations. Case description: In this report, a 26-year-old female patient experienced a facial subcutaneous emphysema, occurred in a few seconds during the surgical extraction of an impacted lower left third molar performed by the use of a high-speed turbine. The patient showed a normal mouth opening but an impossibility to fully open the left eye; there was no pain or breathing difficulties. A manual reduction of the emphysematous zone was performed. Amoxicillin 875 mg with clavulanic acid 125 mg in tablets every 12 h for 6 days was prescribed; 4 mg of intramuscular betamethasone (Bentelan, Defiante Farm Unipessoal LDA, Funchal, Portugal) were injected locally at the intervention site, and 80 mg of ketoprofen every 8 hours in case of pain, were administered after surgery; the patient was observed for 4 hours then allowed to leave the hospital. Follow-up visits were scheduled at 1, 3, and 6 days after surgery. The healing was obtained in 6 days. Conclusion: The prevention of subcutaneous emphysema requires minimal invasive surgery avoiding the use of high-speed air-turbine and flaps should be minimal elevated and properly retracted by an expert operator. Even if subcutaneous emphysema is self-limiting and usually heals in 7–10 days, its prevention and early and accurate diagnosis are essential to avoid complications or life-threatening conditions. Clinical significance: The procedures described in this case report may be of clinical significance in the knowledge, prevention, and treatment of subcutaneous emphysema, a potentially life-threatening condition.