Objectives: Prosthodontic rehabilitation using dental implants has become a common practice in dentistry at the present time. Although severe complications related to dental osseointegrated implants are uncommon, in recent years several cases of oral squamous cell carcinoma adjacent to dental implants have been published. Study Design: A 67-year-old edentulous woman developed an oral squamous cell carcinoma around right mandibular implant about 12 months after receiving dental implants. The treatments involve chemotherapy, explantation of the implants and radiotherapy. Result and Conclusions: The use of endosseous implants has been associated with a low risk for the development of cancer. However, this case report serves as evidence to the importance of careful screening at routine dental examinations especially if the patient suffers from peri-implantitis, any mucosal erosion or discoloration, leukoplakia around implant, pain, implant loosening, bone loss around implant, mucosal erythema, and soft tissue inflammation.