Background: Lymphangiomas are rare benign malformations of the lymphatic system, most frequently affecting the head and neck region. Oral involvement is uncommon, and when the tongue is affected, particularly its anterior two-thirds, it can cause major functional and esthetic disturbances. Case Presentation: We report the case of a 13-year-old boy who had undergone total glossectomy at the age of 4 for cystic lymphangioma. During follow-up, the patient presented marked dental and craniofacial alterations, including severe maxillary and mandibular crowding and a skeletal Class III pattern. This case highlights the significant morphological and functional consequences resulting from the absence of the tongue during growth. Conclusions: This case underscores the crucial role of the tongue in craniofacial growth and occlusal development. Early total glossectomy can result in long-term dental and skeletal disturbances, emphasizing the importance of early multidisciplinary follow-up combining surgical, orthodontic, and functional rehabilitation. Further studies are needed to establish appropriate management and rehabilitation protocols for such rare cases.
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