Unicystic Ameloblastoma of the maxilla in a 6-year-old boy: A case report
Konstantina CHATZIDIMITRIOU, Ekaterini MICHAIL-STRANTZIA, Nadia THEOLOGIE-LYGIDAKIS
University Department of Oral and Maxillofacial Surgery at the Children’s Hospital “P. and A. Kyriakou” (Head: Professor C. Alexandridis, Director: Professor I. Iatrou), Dental School, National and Kapodistrian University of Athens, Greece, Pathology Department, Children’s Hospital “A. and P. Kyriakou” (Head: E. Michail-Strantzia)
Hellenic Archives of Oral & Maxillofacial Surgery (2014) 1, 11-18
SUMMARY: Unicystic ameloblastoma originates earlier in age, is more rare and less aggressive variant then the solid ameloblastoma. Case Report: A 6-year-old male patient with non-contributory medical history was referred for evaluation due to a mildly painful swelling of the left maxilla. Clinical examination revealed an extraoral swelling and panoramic radiograph disclosed a well-corticated unilocular radiolucent lesion extending from distal of central incisor (61) to distal of second primary molar (65), which included the permanent lateral incisor and had pushed the permanent canine towards the orbit. To confirm the cystic nature of the lesion, aspiration was performed. The lesion was then enucleated along with impacted lateral incisor (22) under general anaesthesia. The specimen was examined and the final diagnosis was unicystic ameloblastoma with intraluminal proliferations. Four years later the patient presents normal maxillary development without any signs of local relapse. Conclusions: The present case is unusual due to the type of ameloblastoma, the very young age of the patient and the rare maxillary location. A more conservative treatment of unicystic ameloblastomas in children and adolescents is acceptable, while the follow-up period should be maintained for up to 10 years.
KEY WORDS: ameloblastoma in children, unicystic type, maxilla
REFERENCES
Altini M, Coleman H, Doglioni C, Favia G, Maiorano E: Calretinin expression in ameloblastomas. Histopathology 37: 27-32, 2000.
Ackermann GL, Altini M, Shear M: The unicystic ameloblastoma: A clinicopathological study of 57 cases. J Oral Pathol 17: 541–6, 1988.
Becelli R, Carboni A, Cerulli G, Perugini M, Iannetti G: Mandibular ameloblastoma: analysis of surgical treatment carried out in 60 patients between 1977 and 1998. J Craniofac Surg 13: 395-400, 2002.
Butt FM, Guthua SW, Awange DA, Dimba EA, Macigo FG: The pattern and occurrence of ameloblastoma in adolescents treated at a university teaching hospital, in Kenya: a 13-year study. J Craniomaxillofac Surg 40: 39-45, 2012.
Eckardt AM, Kokemüller H, Flemming P, Schultze A: Recurrent ameloblastoma following osseous reconstruction e a review of twenty years. J Craniomaxillofac Surg 37: 36-41, 2009.
Escande C, Chaine A, Menard P, Ernenwein D, Ghoul S, Bouattour A, Berdal A, Bertrand JC, Ruhin-Poncet B: A treatment algorithm for adult ameloblastomas according to the Pitié-Salpêtrière Hospital experience. J Craniomaxillofac Surg 37: 363-369, 2009.
Gardner DG: Some current concepts on the pathology of ameloblastomas. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 82: 660, 1996.
Gardner DG, Heikinheino K, Shear M, Philipsen HP, Coleman H: Ameloblastoma. In: Barnes L, Eveson JW, Reichart P, Sidransky D eds. World health organization classification of tumours. Pathology and genetics head and neck tumors. IARC Press, Lyon; 300, 2005.
Gerzenshtein J, Zhanq F, Caplan J, Anand V, Lineaweaver W: Immediate mandibular reconstruction with microsurgical fibula flap transfer following wide resection for ameloblastoma. J Craniofac Surg 17: 178-182, 2006.
Ghandhi D, Ayoub AF, Pogrel MA, MacDonald G, Brocklebank LM, Moos KF: Ameloblastoma: a surgeon’s dilemma. J Oral Maxillofac Surg 64: 1010-1014, 2006. Gupta N, Saxena S, Rathod VC, Aggarwal P: Unicystic ameloblastoma of the mandible. J Oral Maxillofac Pathol 15: 228-231, 2011.
Huang IY, Lai ST, Chen CH, Chen CM, Wu CW, Shen YH: Surgical management of ameloblastoma in children. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 104: 478-485, 2007.
Iatrou I, Theologie-Lygidakis N, Leventis M: Intraosseous cystic lesions of the jaws in children: a retrospective analysis of 47 consecutive cases. Oral Surg Oral Med Oral Pathol Oral Radiol Endod. 107(4):485-92, 2009.
Isacsson G, Andersson L, Forsslund H, Bodin I, Thomsson M: Diagnosis and treatment planning of unicystic ameloblastoma. Int J Oral Maxillfac Surg 15: 759–64, 1986.
Konouchi H, Asaumi J, Yanagi Y, Hisatomi M. et al: Usefulness of contrast enhanced-MRI in the diagnosis of unicystic ameloblastoma. Oral Oncol. 42(5): 481-6, 2006.
Lee PK, Samman N, Ng IO: Unicystic ameloblastoma-Use of Carnoy's solution after enucleation. Int J Oral Maxillofac Surg 33: 263–7, 2004.
Leider AS, Eversole LR, Barkin ME: Cystic ameloblastoma. A clinicopathologic analysis. Oral Surgery Oral Medicine and Oral Pathology 60(6): 624–630, 1985. MacDonald-Jankowski DS, Yeung R, Lee KM, Li TK: Ameloblastoma in the Hong Kong Chinese, Part 2: Systematic review and radiological presentation. Dentomaxillofac Radiol. 33: 141–51, 2004.
Nakamura N, Higuchi Y, Tashiro H, Ohishi M: Marsupialization of cystic ameloblastoma: a clinical and histopathologic study of the growth characteristics before and after marsupialization. J Oral Maxillofac Surg 53: 748-54, 1995.
Nakamura N, Higuchi Y, Mitsuyasu T, Sandra F, Ohishi M: Comparison of long term result between different approaches of ameloblastoma. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 93: 13– 20, 2002.
Navarro CM, Principi SM, Massucato EM, Sposto MR: Maxillary unicystic ameloblastoma. Dentomaxillofac Radiol 331: 60–2, 2004.
Olaitain AA, Adekeye EO: Clinical features and management of ameloblastoma of the mandible in children and adolescents. Br J Oral Maxillofac Surg 34: 248, 1996.
Olaitain AA, Adekeye EO: Unicystic ameloblastoma of the mandible: a long-term follow-up. J Oral Maxillofac Surg 55: 345-348, 1997.
Oliveira-Neto HH, Spindula-Filho JV, Dallara MC, Silva CM, Mendonca EF, Batista AC: Unicystic ameloblastoma in a child: A differential diagnosis from the dentigerous cyst and the inflammatory follicular cyst. J Dent Child (Chic) 74: 245–9, 2007.
Ord RA, Blanchaert RH Jr, Nikitakis NG, Sauk JJ: Ameloblastoma in children. J Oral Maxillofac Surg 60: 762–70, 2002.
Philipsen HP, Reichart PA: Unicystic ameloblastoma. A review of 193 cases from the literature. Oral Oncol 34: 317–25, 1998.
Robinson L, Martinez MG: Unicystic Ameloblastoma: A Prognostic Distinct Entity. Cancer 40: 2278–85, 1977.
Rosentein T, Pogrel MA, Smith RA, Regezi JA: Cystic Ameloblastoma– behavior and treatment of 21 cases. J Oral Maxillofac Surg 59: 1311–1316, 2001. Takahashi K, Miyauchi K, Sato K: Treatment of ameloblastoma in children. Br J Oral Maxillofac Surg 36: 453-456, 1998.
Unlu G, Tari V, Alan H: Unicystic ameloblastoma in 8 years old child: a case report review of unicystic ameloblastoma. Int Dent Med Dis 1: 29-33, 2008.
Worth HM: Benign tumors of jaw. 1st ed. Year Book Medical Publisher, Chicago; 476–546, 1963. Zhang J, Gu Z, Jiang L, Zhao J, Tian M, Zhou J, Duan Y: Ameloblastoma in children and adolescents. Br J Oral Maxillofac Surg 48: 549-554, 2010
University Department of Oral and Maxillofacial Surgery at the Children’s Hospital “P. and A. Kyriakou” (Head: Professor C. Alexandridis, Director: Professor I. Iatrou), Dental School, National and Kapodistrian University of Athens, Greece, Pathology Department, Children’s Hospital “A. and P. Kyriakou” (Head: E. Michail-Strantzia)
Hellenic Archives of Oral & Maxillofacial Surgery (2014) 1, 11-18
SUMMARY: Unicystic ameloblastoma originates earlier in age, is more rare and less aggressive variant then the solid ameloblastoma. Case Report: A 6-year-old male patient with non-contributory medical history was referred for evaluation due to a mildly painful swelling of the left maxilla. Clinical examination revealed an extraoral swelling and panoramic radiograph disclosed a well-corticated unilocular radiolucent lesion extending from distal of central incisor (61) to distal of second primary molar (65), which included the permanent lateral incisor and had pushed the permanent canine towards the orbit. To confirm the cystic nature of the lesion, aspiration was performed. The lesion was then enucleated along with impacted lateral incisor (22) under general anaesthesia. The specimen was examined and the final diagnosis was unicystic ameloblastoma with intraluminal proliferations. Four years later the patient presents normal maxillary development without any signs of local relapse. Conclusions: The present case is unusual due to the type of ameloblastoma, the very young age of the patient and the rare maxillary location. A more conservative treatment of unicystic ameloblastomas in children and adolescents is acceptable, while the follow-up period should be maintained for up to 10 years.
KEY WORDS: ameloblastoma in children, unicystic type, maxilla
REFERENCES
Altini M, Coleman H, Doglioni C, Favia G, Maiorano E: Calretinin expression in ameloblastomas. Histopathology 37: 27-32, 2000.
Ackermann GL, Altini M, Shear M: The unicystic ameloblastoma: A clinicopathological study of 57 cases. J Oral Pathol 17: 541–6, 1988.
Becelli R, Carboni A, Cerulli G, Perugini M, Iannetti G: Mandibular ameloblastoma: analysis of surgical treatment carried out in 60 patients between 1977 and 1998. J Craniofac Surg 13: 395-400, 2002.
Butt FM, Guthua SW, Awange DA, Dimba EA, Macigo FG: The pattern and occurrence of ameloblastoma in adolescents treated at a university teaching hospital, in Kenya: a 13-year study. J Craniomaxillofac Surg 40: 39-45, 2012.
Eckardt AM, Kokemüller H, Flemming P, Schultze A: Recurrent ameloblastoma following osseous reconstruction e a review of twenty years. J Craniomaxillofac Surg 37: 36-41, 2009.
Escande C, Chaine A, Menard P, Ernenwein D, Ghoul S, Bouattour A, Berdal A, Bertrand JC, Ruhin-Poncet B: A treatment algorithm for adult ameloblastomas according to the Pitié-Salpêtrière Hospital experience. J Craniomaxillofac Surg 37: 363-369, 2009.
Gardner DG: Some current concepts on the pathology of ameloblastomas. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 82: 660, 1996.
Gardner DG, Heikinheino K, Shear M, Philipsen HP, Coleman H: Ameloblastoma. In: Barnes L, Eveson JW, Reichart P, Sidransky D eds. World health organization classification of tumours. Pathology and genetics head and neck tumors. IARC Press, Lyon; 300, 2005.
Gerzenshtein J, Zhanq F, Caplan J, Anand V, Lineaweaver W: Immediate mandibular reconstruction with microsurgical fibula flap transfer following wide resection for ameloblastoma. J Craniofac Surg 17: 178-182, 2006.
Ghandhi D, Ayoub AF, Pogrel MA, MacDonald G, Brocklebank LM, Moos KF: Ameloblastoma: a surgeon’s dilemma. J Oral Maxillofac Surg 64: 1010-1014, 2006. Gupta N, Saxena S, Rathod VC, Aggarwal P: Unicystic ameloblastoma of the mandible. J Oral Maxillofac Pathol 15: 228-231, 2011.
Huang IY, Lai ST, Chen CH, Chen CM, Wu CW, Shen YH: Surgical management of ameloblastoma in children. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 104: 478-485, 2007.
Iatrou I, Theologie-Lygidakis N, Leventis M: Intraosseous cystic lesions of the jaws in children: a retrospective analysis of 47 consecutive cases. Oral Surg Oral Med Oral Pathol Oral Radiol Endod. 107(4):485-92, 2009.
Isacsson G, Andersson L, Forsslund H, Bodin I, Thomsson M: Diagnosis and treatment planning of unicystic ameloblastoma. Int J Oral Maxillfac Surg 15: 759–64, 1986.
Konouchi H, Asaumi J, Yanagi Y, Hisatomi M. et al: Usefulness of contrast enhanced-MRI in the diagnosis of unicystic ameloblastoma. Oral Oncol. 42(5): 481-6, 2006.
Lee PK, Samman N, Ng IO: Unicystic ameloblastoma-Use of Carnoy's solution after enucleation. Int J Oral Maxillofac Surg 33: 263–7, 2004.
Leider AS, Eversole LR, Barkin ME: Cystic ameloblastoma. A clinicopathologic analysis. Oral Surgery Oral Medicine and Oral Pathology 60(6): 624–630, 1985. MacDonald-Jankowski DS, Yeung R, Lee KM, Li TK: Ameloblastoma in the Hong Kong Chinese, Part 2: Systematic review and radiological presentation. Dentomaxillofac Radiol. 33: 141–51, 2004.
Nakamura N, Higuchi Y, Tashiro H, Ohishi M: Marsupialization of cystic ameloblastoma: a clinical and histopathologic study of the growth characteristics before and after marsupialization. J Oral Maxillofac Surg 53: 748-54, 1995.
Nakamura N, Higuchi Y, Mitsuyasu T, Sandra F, Ohishi M: Comparison of long term result between different approaches of ameloblastoma. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 93: 13– 20, 2002.
Navarro CM, Principi SM, Massucato EM, Sposto MR: Maxillary unicystic ameloblastoma. Dentomaxillofac Radiol 331: 60–2, 2004.
Olaitain AA, Adekeye EO: Clinical features and management of ameloblastoma of the mandible in children and adolescents. Br J Oral Maxillofac Surg 34: 248, 1996.
Olaitain AA, Adekeye EO: Unicystic ameloblastoma of the mandible: a long-term follow-up. J Oral Maxillofac Surg 55: 345-348, 1997.
Oliveira-Neto HH, Spindula-Filho JV, Dallara MC, Silva CM, Mendonca EF, Batista AC: Unicystic ameloblastoma in a child: A differential diagnosis from the dentigerous cyst and the inflammatory follicular cyst. J Dent Child (Chic) 74: 245–9, 2007.
Ord RA, Blanchaert RH Jr, Nikitakis NG, Sauk JJ: Ameloblastoma in children. J Oral Maxillofac Surg 60: 762–70, 2002.
Philipsen HP, Reichart PA: Unicystic ameloblastoma. A review of 193 cases from the literature. Oral Oncol 34: 317–25, 1998.
Robinson L, Martinez MG: Unicystic Ameloblastoma: A Prognostic Distinct Entity. Cancer 40: 2278–85, 1977.
Rosentein T, Pogrel MA, Smith RA, Regezi JA: Cystic Ameloblastoma– behavior and treatment of 21 cases. J Oral Maxillofac Surg 59: 1311–1316, 2001. Takahashi K, Miyauchi K, Sato K: Treatment of ameloblastoma in children. Br J Oral Maxillofac Surg 36: 453-456, 1998.
Unlu G, Tari V, Alan H: Unicystic ameloblastoma in 8 years old child: a case report review of unicystic ameloblastoma. Int Dent Med Dis 1: 29-33, 2008.
Worth HM: Benign tumors of jaw. 1st ed. Year Book Medical Publisher, Chicago; 476–546, 1963. Zhang J, Gu Z, Jiang L, Zhao J, Tian M, Zhou J, Duan Y: Ameloblastoma in children and adolescents. Br J Oral Maxillofac Surg 48: 549-554, 2010
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