Ameloblastic carcinoma is a rare odontogenic tumor exhibiting not only features of ameloblastoma, but also features of carcinoma. Clinical dissemination of this lesion is more aggressive and rapid than that of ameloblastoma and it can metastasize to the lung or regional lymph node. Histologically, there are features of both ameloblastoma and carcinoma. We report a series of six cases with our treatment modalities. Ameloblastoma is a benign but locally aggressive neoplasm, which presents as a slowly growing painless swelling of the jaws. The malignant variants are exceptionally rare and may arise de-novo or from transformation of a long-standing primarily benign lesion, which has undergone several surgical treatments.

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Received: 11 March Accepted: 26 July Introduction: Ameloblastic carcinoma is an extremely rare malignant odontogenic tumor with predominantly mandibular localization. In most cases, it is treated surgically. Observation: Here, we describe a case of ameloblastic carcinoma. The patient presented a large expansive mass on the ascending branch of the left mandible, which was ulcerated and communicating with the oral cavity.

He refused the proposed surgical treatment after being informed of the risk of facial decomposition. After several years, due to progressive symptomatology, he received palliative radiotherapy of 60 Gy divided into 30 sessions. Local control of the disease was achieved. Discussion: The efficiency of radiotherapy for ameloblastic carcinoma remains controversial. Conclusion: Radiotherapy appears to be a second-line approach when surgery is not feasible for ameloblastic carcinoma treatment.

Ameloblastic carcinoma is an exceptional malignant tumor mainly occurring in the mandible. It may be primary or secondary to the transformation of a pre-existing ameloblastoma. Its prognosis is reserved because of its local aggressiveness and risk of metastatic expansion. Precise diagnosis is based on anatomopathological examination. The most common treatment is surgical via complete resection, followed by adjuvant radiotherapy.

Here, we present the case of a patient with primary ameloblastic carcinoma treated with palliative radiotherapy; therapeutic approaches to this pathology in the literature are reviewed. A year-old patient presented for consultation following discovery of a swelling of the left horizontal branch of the mandible.

His case history was type II diabetes, rhizomelic pseudo-rheumatoid arthritis, and coronary artery disease. He was surgically treated for herniated disc and triple bypass.

He had no allergies or significant family history. He was a former smoker 40 packs per year. He was previously biopsied and diagnosed with primary ameloblastic carcinoma located on the ascending left mandibular branch Fig. The surgical team recommended resection surgery: dissection with fibula free flap reconstruction.

However, the patient, in the view of the surgical risks and disfigurement, refused the surgery without follow-up. Histological section of the biopsy showing cells with elongated nuclei assuming a palissadic appearance with clarified cells having an inverted nuclear polarity. The nuclei are the site of clear-cut nuclear atypia, and mitotic figures are visible.

He was examined for increased swelling and complaint of episodes of local infections and frequent intraoral bleeding from tumors. On endobuccal examination, a lesion with mm diameter was detected behind the last lower left molar, which was bleeding on contact, painless, ulcerated, and communicating with the oral cavity.

The tongue, floor of the mouth, tonsillar lodge, and veil seemed to be unaffected. There was no restriction in mouth opening, hypoesthesia in the left labiomental area, involvement of cranial nerve pairs, or palpable lymphadenopathy.

Panoramic radiography Fig. The lesion spared the condyle, parotid, and submaxillary gland. Extension examination revealed a single adenopathy of the ipsilateral facial pedicle, which was not very specific.

Surgery with reconstruction using microanastomosed fibula flap was proposed again, but after Multidisciplinary Consultative Meeting and given his arterial condition, radiotherapy for tumor control was proposed.

The patient was again lost to follow-up. Panoramic radiography showing diffuse osteolysis of the ascending branch of the left mandible 5 years after the first consultation. Scan of facial bone window and in axial section at the mandibular level, at 5 years, showing an osteolytic and expansive lesion of the ascending branch of the left mandible. One year later, magnetic resonance imaging Fig. The patient's general condition was preserved.

Clinical examination showed a clear progression of the tumor with increased face deformation. At the intraoral level, there was a large necrotic cavity behind the 36, which bled spontaneously. At the end of irradiation, the patient reported excellent tolerance, despite known side effects, as well as disappearance of bleeding.

Six months later, his general condition was preserved and he was asymptomatic. Clinical examination revealed improvements with mucosal healing, suggesting a single arch sequella. The patient only complained of hearing loss on the left side. T1 time MRI in axial section, at 6 years, through the mandible, showing the expansive process in relation to the ascending branch of the mandible.

One year after radiotherapy, follow-up imaging Fig. A quarterly follow-up has been initiated. Facial scan bone window in axial section 1 year after radiotherapy showing a stabilization of this expansive process and communication with the oral cavity.

Ameloblastic carcinoma is an extremely rare epithelial tumor of odontogenic origin located in the mandible or maxilla, with a predilection for the mandible [ 1 ]. There are little data on this pathology because few cases have been reported.

It may be primary or secondary to pre-existing benign ameloblastoma, as described by Lin et al. Primary ameloblastic carcinoma is the most common. Clinically, it can manifest as pain, rapidly growing swelling, bleeding, ulceration, tooth mobility, trismus, labiomental paresthesia, dysphonia, or dysphagia [ 3 ].

Histological examination is pivotal to confirm diagnosis. It is a malignant epithelial tumor that retains the characteristics of ameloblastic differentiation but also shows cytological characteristics of malignancy [ 3 ].

For diagnosis, most studies use immunohistochemical analysis of Ki67—a frequently elevated marker in ameloblastic carcinoma—which is an index of tumor proliferation [ 4 — 7 ].

This is an additional aid for the differential diagnosis of ameloblastomas. Bello et al. In the present case, histological examination was conducted outside the hospital monitoring facility and did not report assessments of any of these markers.

There is no histological distinction between primary and secondary ameloblastic carcinoma. In , the World Health Organization classification included ameloblastic carcinoma in ameloblastic tumors and described three subcategories: primary, secondary, and peripheral types. The new classification of odontogenic lesions in 4th edition [ 8 ] Fig.

Imaging is non-specific and only has diagnostic value in locoregional extension examination. Management remains controversial, although surgery with healthy margins of exeresis offers the best prognosis and remains the treatment of choice.

Most studies are in favor of surgery, followed by radiotherapy. Giridhar et al. The effectiveness of radiotherapy remains controversial. Jensen et al. According to retrospective study of radiotherapy alone for recurrence old surgery and adjuvant radiotherapy after surgery [ 10 ] in , radiotherapy offered the potential for disease control when exeresis margins were affected or when recurrence was noted.

In other studies [ 6 — 11 ], this approach was reportedly ineffective. In our case, radiotherapy seemed to have stabilized the course of the disease, as seen on the last scan. However, decreased hearing acuity seemed to be a complication of radiation therapy and not tumor progression given the absence of radiological evolution of the tumor.

The tumor evolves via either a rapid and aggressive local extension or a distant metastatic lesion, particularly pulmonary, appearing in the 4th to 12th month after the surgery [ 6 ]. The bone, liver, and brain are other metastatic sites. The prognosis is reserved, with a high recurrence rate [ 7 ] and overall survival rates of Careful long-term follow-up is, therefore, essential in the management of this tumor [ 11 ].

Table of the new classification of odontogenic tumors [ 8 ]. For ameloblastic carcinoma, surgery remains the treatment of choice to date, sometimes combined with adjuvant radiotherapy, because it offers the best survival potential and prognosis.

The effectiveness of radiation therapy remains controversial across various studies; however, this could be an alternative to surgery when it is not feasible and could provide good local control of the disease, as presented in this clinical case.

Data correspond to usage on the plateform after The current usage metrics is available hours after online publication and is updated daily on week days. Open Access. Pattern of care and impact of prognostic factors in the outcome of ameloblastic carcinoma: a systematic review and individual patient data analysis of cases.

Eur Arch Otorhinolaryngol ;— The variability and complexity of ameloblastoma: carcinoma ex ameloblastoma or primary ameloblastic carcinoma. J Cranio-Maxillofac Surg ;— Ameloblastic carcinoma: an analysis of 6 cases with review of the literature.

Alpha-smooth muscle actin within epithelial islands is predictive of ameloblastic carcinoma. Oral Oncol ;— Ameloblastic carcinoma of mandible: facts and dilemmas.

Tumori ;e—e Ameloblastic carcinoma: a Brazilian collaborative study of 17 cases. Histopathology ;— The World Health Organization classification of odontogenic lesions: a summary of the changes of the 4th edition.

Head Neck Pathol ; 68— Carbon ion therapy for ameloblastic carcinoma. Radiat Oncol ; Treatment of ameloblastoma and ameloblastic carcinoma with radiotherapy.

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Ameloblastic carcinoma: An analysis of 12 cases with a review of the literature

Ameloblastic carcinoma: A clinicopathologic dilemma - Report of two cases with total review of literature from to Indu Bhusan Kar 1 , R. Ameloblastic carcinoma AC is a rare primary odontogenic tumor that has histological features of both ameloblastoma and carcinoma. A total number of 92 case reports speak about its rare incidence, affecting mostly the mandible as a locally destructive lesion. The maxilla is affected even more rarely as only 35 cases have been reported until in scientific literature.


Ameloblastic carcinoma

Ameloblastic carcinoma is a rare form of odontogenic tumor , one that develops in the jawbones from the epithelial cells that generate the tooth enamel. Symptoms include swelling in the jaw and pain, both of which get worse as the cancer grows. It is usually treated with surgery; chemotherapy has not been proven to be effective. The majority of cases appear sporadically in patients without a past history of cancer.

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