Dilaceration is a developmental disturbance characterized by the angulation of the crown or root of a permanent tooth, which is often related to trauma of primary dentition. We report a case of a dilacerated root in a maxillary central incisor associated with gingival hyperplasia in a patient under fixed orthodontic treatment, a combination of pathological conditions that had never been mentioned before in the scientific literature.A 10-year-old female patient presented to the Department of Odontology and Oral Pathology with tenderness to palpation and bleeding from the oral aspect of the central incisor, alerted by the proliferation of the gingiva. During clinical examination, the palpation performed with a dental probe revealed a carious lesion with dental pulp exposure on the distal aspect of right central incisor and the presence of a sessile mass of inflamed gingival tissue that proliferated inside the defect. On the preoperative radiograph a dilacerated root canal was noted, without periapical bone resorption.The main diagnosis was irreversible pulpitis and gingival hyperplasia and the treatment option was surgical removal of the inflamed tissue with histopathological examination and root canal treatment. Successful endodontic treatment with a good prognosis was recorded.The measurement of the root curvature proved to be extremely helpful in choosing the right endodontic technique and made the treatment easier than expected. An important observation was that, despite the rare clinical and radiographic aspect of this dilacerated tooth, the endodontic treatment proved to be relatively easy to perform and, therefore, the prognosis was considered favorable.