This case report presents an analysis of the clinical, radiographic, and histological features of a peri-implant lesion around an implant placed immediately after extraction of a tooth with a periapical lesion. A 52-year-old man received an immediate implant (3.75 × 11.5 mm2) placed in the anterior region of the maxilla. Three years after implant placement, the patient presented with swelling in the anterior portion of the maxilla. Radiographic examination showed a well-circumscribed radiolucency around the implant. The implant and the lesion were removed and fixed in 10% buffered formalin and processed. Histological analysis showed 3 types of epithelium: respiratory, cuboidal, and non-keratinized stratified squamous. In the cyst wall peripheral nerves, arteries, veins, and chronic inflammation were present. The diagnosis was nasopalatine duct cyst. We concluded that the nasopalatine duct cyst can develop in association with dental implants. Clinically, the lesion is similar to the classical nasopalatine duct cyst. Histological analysis should be mandatory in all cases of peri-implant lesions and in all dental periapical lesions before immediate implant placement.