Ameloblastic carcinoma with isolated supramandibular lymph node metastasis in an adolescent: a case report and review of the literature.
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OBJECTIVES: To report a rare case of maxillary ameloblastic carcinoma (AC) with buccofacial lymph node metastasis in a 17-year-old male patient, describe its clinical, radiological, and pathological progression, and provide clinical references for routine patient management. CASE DESCRIPTION: The patient presented to our hospital with suspected recurrent peripheral ameloblastoma (PA). Cone-beam computed tomography (CBCT) revealed an osteolytic lesion in the right maxilla. Intraoperative frozen section pathology confirmed the diagnosis of ameloblastoma, extended tumorectomy, segmental maxillectomy, and submental island flap reconstruction were conducted. Histopathological examination confirmed the diagnosis of AC. Eight months after segmental maxillectomy, a soft tissue mass overlying the right mandibular buccal region rapidly enlarged. Contrast-enhanced CT revealed heterogeneous enhancement of the lesion with internal necrotic components. Intraoperative pathological examination suggested a malignant tumor. The patient subsequently underwent reoperation: extended resection of the right buccal mass, right suprahyoid lymphadenectomy, and right cervical lymph node dissection. Postoperative pathological examination confirmed AC with supramandibular lymph node metastasis. Following lymph node dissection, the patient received radiotherapy and chemotherapy for two months. At the 6-month follow-up, imaging and clinical examinations revealed no evidence of tumor recurrence or distant metastatic lesions. The patient is currently under regular long-term surveillance to monitor disease progression. CONCLUSION: This study reports a case of maxillary AC with isolated supramandibular lymph node metastasis. To our knowledge, this represents the first documented case. Clinicians should be alert to occult buccofacial lymph node metastasis in patients with AC invading the buccal soft tissues, and multimodal imaging is recommended for complete preoperative tumor staging. Individualized combined buccofacial and cervical lymph node dissection may improve locoregional control in patients complicated with buccofacial lymph node metastasis. This study can provide a reference for the identification of rare metastatic patterns and individualized diagnosis and treatment of AC.