Dental Science - Case Report Adenomatoid odontogenic tumor in the maxillary antrum: A rare case entity Kavitha Krishnamurthy, Ranjith Singh Balaji, Sumana Devadiga1, Rame Gowda Rajendra Prasad2
Department of Dentistry, Adichunchanagiri Institute of Medical Sciences, B.G. Nagara, 2Department of Oral and Maxillo Facial Surgery, JSS Dental College, JSS University, Mysore, 1 Consulting Maxillo-Facial Surgeon, Mangalore, Karnataka, India
ABSTRACT Adenomatoid odontogenic tumor (AOT) is an uncommon benign tumor of odontogenic origin. It occurs in the second decade of life. Females are more commonly affected than males. AOT has a striking tendency to occur in the anterior maxilla; however, very few cases have been reported to occur in the maxillary antrum. This is a case report of a 17‑year‑old male presented with a large radiolucent lesion associated with the crown of an unerupted canine located in the right maxillary antrum, which was clinically diagnosed as dentigerous cyst. The histopathological examination revealed the presence of AOT.
Address for correspondence: Dr. Kavitha Krishnamurthy, E‑mail: k_drkavitha@yahoo. co.in Received : 18-04-14 Review completed : 18-04-14 Accepted : 23-04-14
KEY WORDS: Adenomatoid odontogenic tumor, impacted tooth, maxillary antrum
A
denomatoid odontogenic tumor (AOT), an uncommon benign epithelial lesion of odontogenic origin was first described by Dreibaldt in 1907 as a pseudo‑adenoameloblastoma.[1] It was only in 1948 when, Stafne considered it as distinct pathological entity.[2] The current WHO classification of odontogenic tumors defines AOT as being composed of odontogenic epithelium in a variety of histoarchitectural patterns, embedded in a mature connective tissue stroma and characterized by slow, but progressive growth.[3] The differential diagnosis of AOT is crucial in terms of surgical management. Local excision is adequate to free the patient from AOT.[4] This is a rare case report of an AOT in the maxillary antrum Access this article online Quick Response Code:
Website: www.jpbsonline.org DOI: 10.4103/0975-7406.137468
in association with an impacted canine, which mimicked dentigerous cyst clinically and radiographically.
Case Report A 17‑year‑old male presented with the chief complaint of swelling on the right side of the face since 6 months. He gave a history of gradual increase in size of the swelling. On extra oral examination, swelling measured approximately 4 cm × 3 cm, hard on palpation, with a smooth surface extending from ala of the nose to about 2 cm in front of the tragus of the ear and about 1 cm below the infraorbital rim to ala tragal line with the obliteration of the nasolabial fold on the right side. On intra oral examination, swelling measured approximately 3 cm × 2 cm, firm on palpation, with a smooth surface extending from left central incisor to right first molar [Figure 1]. The right upper canine, right and left lateral incisors were missing. Over retained deciduous right central incisor, right lateral incisor, right canine, and left canine were noted. Grade III mobility of all the over retained deciduous teeth and right first premolar
How to cite this article: Krishnamurthy K, Balaji RS, Devadiga S, Prasad RR. Adenomatoid odontogenic tumor in the maxillary antrum: A rare case entity. J Pharm Bioall Sci 2014;6:S196-9.
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Journal of Pharmacy and Bioallied Sciences July 2014 Vol 6 Supplement 1
Krishnamurthy, et al.: Adenomatoid odontogenic tumor in the maxillary antrum
was noted. There was no evidence of oro‑nasal and oro‑antral communication, and the palatal mucosa was intact.
The specimen was then submitted for histopathological examination [Figure 4].
Water’s view projection revealed haziness with respect to bilateral maxillary antrum.
Histopathological sections showed a cystic wall lined by epithelial cells in sheets, clusters, nests and islands and also cells in ductal, cribriform and trabecular pattern, ducts lined by columnar epithelium resembling ameloblasts, areas of rosette formation, eosinophilic hyaline material deposition and dystrophic calcifications, giving the impression of AOT [Figure 5].
Computed tomography (CT) scan demonstrated expansile lytic lesion extending into the nasal cavity and right maxillary antrum with obliteration of the respective lumen in association with an impacted tooth [Figure 2]. Fine‑needle aspiration was done, which yielded about 15 ml of straw‑colored fluid. Based on the clinical and radiographic findings, the differential diagnosis of dentigerous cyst, unicystic ameloblastoma and AOT was made. The patient was operated by raising the mucoperiosteal flap intraorally. The lining of the cystic lesion was carefully separated from the mucoperiosteum and the lesion was enucleated. The impacted canine, first premolar, and all retained deciduous teeth were extracted [Figure 3].
Healing was uneventful [Figure 6].
Discussion Adenomatoid odontogenic tumor was first recognized as a distinct pathological entity by Stafne in 1948. AOT is a slow growing benign tumor, constituting only 3% of all odontogenic tumors[2] and 0.1% of tumors of jaws. The lesion is usually asymptomatic. The adjacent teeth may be slightly displaced.[5] Most common site of AOT is anterior region of maxilla. Tajima et al.[6] described an AOT located in the superior portion of the maxillary sinus. Based on the location, three variants of AOT,
Figure 1: Preoperative intraoral photograph
Figure 2: Pre operative para nasal sinus view
Figure 3: Pre-operative coronal CT scan section-maxillary sinus view
Figure 4: Pre-operative CT scan axial section-maxillary sinus view
Journal of Pharmacy and Bioallied Sciences July 2014 Vol 6 Supplement 1
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Krishnamurthy, et al.: Adenomatoid odontogenic tumor in the maxillary antrum
Figure 5: Intra-operative photograph of surgical excision
Figure 6: Excised specimen
Figure 7: Histopathological section of excisional biopsy specimen
Figure 8: Post-operative intraoral photo graph
the follicular type (73%), the extra follicular type (24%), and the peripheral type (3%) has been described.[7,8]
As previously mentioned AOT is usually solid, but is occasionally cystic. The structure of the cyst and its insertion around the crown of an unerupted tooth were typical of a dentigerous cyst. However, in this case, the multifocal cellular proliferation had the structure of an AOT [Figures 7 and 8].
The female to male ratio for all age groups and all variants is close to 2:1.[9] The tooth that is impacted commonly is the canine in 40%.[10] Conservative surgical enucleation is the treatment modality of choice. Recurrence of AOT is exceptionally rare. Therefore, the prognosis is excellent.[6] With respect to the age of the patient and the localization of the AOT in the maxillary antrum, which was extending from midline to molar region (as shown in CT) the reported case is a rare example of this tumor entity. Due to its low tendency to recur, the case here by presented was treated by conservative surgical enucleation associated with the removal of the unerupted dental element and such procedure was proposed in numerous papers.
Conservative surgical enucleation or curettage is the treatment of choice for AOT. Prognosis is good. Recurrences are rare. Very few histologically diagnosed case reports of AOT in the maxillary antrum have previously been reported. In this case, the final diagnosis of an AOT was arrived by histopathological examination of the enucleated tissue specimen. Rarity of the case is in the fact that the AOT involved the maxillary antrum, was extensive and the treatment resulted in good prognosis.
References
Conclusion
1.
Clinical, radiographic, and microscopic findings in this case are consistent with descriptions of the lesion in the dental literature.
2.
S198
John JB, John RR. Adenomatoid odontogenic tumor associated with dentigerous cyst in posterior maxilla: A case report and review of literature. J Oral Maxillofac Pathol 2010;14:59‑62. Lee JK, Lee KB, Hwang BN. Adenomatoid odontogenic tumor: A case report. J Oral Maxillofac Surg 2000;58:1161‑4.
Journal of Pharmacy and Bioallied Sciences July 2014 Vol 6 Supplement 1
Krishnamurthy, et al.: Adenomatoid odontogenic tumor in the maxillary antrum 3. 4. 5. 6.
7.
Philipsen HP, Reichart PA, Zhang KH, Nikai H, Yu QX. Adenomatoid odontogenic tumor: Biologic profile based on 499 cases. J Oral Pathol Med 1991;20:149‑58. Friedrich RE, Scheuer HA, Zustin J. Adenomatoid odontogenic tumor (AOT) of maxillary sinus: Case report with respect to immunohistochemical findings. In Vivo 2009;23:111‑6. Geist SM, Mallon HL. Adenomatoid odontogenic tumor: Report of an unusually large lesion in the mandible. J Oral Maxillofac Surg 1995;53:714‑7. Handschel JG, Depprich RA, Zimmermann AC, Braunstein S, Kübler NR., Adenomatoid odontogenic tumor of the mandible: Review of the literature and report of a rare case. Head Face Med 2005;1:3. Tajima Y, Sakamoto E, Yamamoto Y. Odontogenic cyst giving rise to
Journal of Pharmacy and Bioallied Sciences July 2014 Vol 6 Supplement 1
an adenomatoid odontogenic tumor: Report of a case with peculiar features. J Oral Maxillofac Surg 1992;50:190‑3. 8. Regezi JA, Kerr DA, Courtney RM. Odontogenic tumors: Analysis of 706 cases. J Oral Surg 1978;36:771‑8. 9. Courtney RM, Kerr DA. The odontogenic adenomatoid tumor. A comprehensive study of twenty new cases. Oral Surg Oral Med Oral Pathol 1975;39:424‑35. 10. Philipsen HP, Srisuwan T, Reichart PA. Adenomatoid odontogenic tumor mimicking a periapical (radicular) cyst: A case report. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2002;94:246‑8.
Source of Support: Nil, Conflict of Interest: None declared.
S199
Copyright of Journal of Pharmacy & Bioallied Sciences is the property of Medknow Publications & Media Pvt. Ltd. and its content may not be copied or emailed to multiple sites or posted to a listserv without the copyright holder's express written permission. However, users may print, download, or email articles for individual use.
Department of Dentistry, Adichunchanagiri Institute of Medical Sciences, B.G. Nagara, 2Department of Oral and Maxillo Facial Surgery, JSS Dental College, JSS University, Mysore, 1 Consulting Maxillo-Facial Surgeon, Mangalore, Karnataka, India
ABSTRACT Adenomatoid odontogenic tumor (AOT) is an uncommon benign tumor of odontogenic origin. It occurs in the second decade of life. Females are more commonly affected than males. AOT has a striking tendency to occur in the anterior maxilla; however, very few cases have been reported to occur in the maxillary antrum. This is a case report of a 17‑year‑old male presented with a large radiolucent lesion associated with the crown of an unerupted canine located in the right maxillary antrum, which was clinically diagnosed as dentigerous cyst. The histopathological examination revealed the presence of AOT.
Address for correspondence: Dr. Kavitha Krishnamurthy, E‑mail: k_drkavitha@yahoo. co.in Received : 18-04-14 Review completed : 18-04-14 Accepted : 23-04-14
KEY WORDS: Adenomatoid odontogenic tumor, impacted tooth, maxillary antrum
A
denomatoid odontogenic tumor (AOT), an uncommon benign epithelial lesion of odontogenic origin was first described by Dreibaldt in 1907 as a pseudo‑adenoameloblastoma.[1] It was only in 1948 when, Stafne considered it as distinct pathological entity.[2] The current WHO classification of odontogenic tumors defines AOT as being composed of odontogenic epithelium in a variety of histoarchitectural patterns, embedded in a mature connective tissue stroma and characterized by slow, but progressive growth.[3] The differential diagnosis of AOT is crucial in terms of surgical management. Local excision is adequate to free the patient from AOT.[4] This is a rare case report of an AOT in the maxillary antrum Access this article online Quick Response Code:
Website: www.jpbsonline.org DOI: 10.4103/0975-7406.137468
in association with an impacted canine, which mimicked dentigerous cyst clinically and radiographically.
Case Report A 17‑year‑old male presented with the chief complaint of swelling on the right side of the face since 6 months. He gave a history of gradual increase in size of the swelling. On extra oral examination, swelling measured approximately 4 cm × 3 cm, hard on palpation, with a smooth surface extending from ala of the nose to about 2 cm in front of the tragus of the ear and about 1 cm below the infraorbital rim to ala tragal line with the obliteration of the nasolabial fold on the right side. On intra oral examination, swelling measured approximately 3 cm × 2 cm, firm on palpation, with a smooth surface extending from left central incisor to right first molar [Figure 1]. The right upper canine, right and left lateral incisors were missing. Over retained deciduous right central incisor, right lateral incisor, right canine, and left canine were noted. Grade III mobility of all the over retained deciduous teeth and right first premolar
How to cite this article: Krishnamurthy K, Balaji RS, Devadiga S, Prasad RR. Adenomatoid odontogenic tumor in the maxillary antrum: A rare case entity. J Pharm Bioall Sci 2014;6:S196-9.
S196
Journal of Pharmacy and Bioallied Sciences July 2014 Vol 6 Supplement 1
Krishnamurthy, et al.: Adenomatoid odontogenic tumor in the maxillary antrum
was noted. There was no evidence of oro‑nasal and oro‑antral communication, and the palatal mucosa was intact.
The specimen was then submitted for histopathological examination [Figure 4].
Water’s view projection revealed haziness with respect to bilateral maxillary antrum.
Histopathological sections showed a cystic wall lined by epithelial cells in sheets, clusters, nests and islands and also cells in ductal, cribriform and trabecular pattern, ducts lined by columnar epithelium resembling ameloblasts, areas of rosette formation, eosinophilic hyaline material deposition and dystrophic calcifications, giving the impression of AOT [Figure 5].
Computed tomography (CT) scan demonstrated expansile lytic lesion extending into the nasal cavity and right maxillary antrum with obliteration of the respective lumen in association with an impacted tooth [Figure 2]. Fine‑needle aspiration was done, which yielded about 15 ml of straw‑colored fluid. Based on the clinical and radiographic findings, the differential diagnosis of dentigerous cyst, unicystic ameloblastoma and AOT was made. The patient was operated by raising the mucoperiosteal flap intraorally. The lining of the cystic lesion was carefully separated from the mucoperiosteum and the lesion was enucleated. The impacted canine, first premolar, and all retained deciduous teeth were extracted [Figure 3].
Healing was uneventful [Figure 6].
Discussion Adenomatoid odontogenic tumor was first recognized as a distinct pathological entity by Stafne in 1948. AOT is a slow growing benign tumor, constituting only 3% of all odontogenic tumors[2] and 0.1% of tumors of jaws. The lesion is usually asymptomatic. The adjacent teeth may be slightly displaced.[5] Most common site of AOT is anterior region of maxilla. Tajima et al.[6] described an AOT located in the superior portion of the maxillary sinus. Based on the location, three variants of AOT,
Figure 1: Preoperative intraoral photograph
Figure 2: Pre operative para nasal sinus view
Figure 3: Pre-operative coronal CT scan section-maxillary sinus view
Figure 4: Pre-operative CT scan axial section-maxillary sinus view
Journal of Pharmacy and Bioallied Sciences July 2014 Vol 6 Supplement 1
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Krishnamurthy, et al.: Adenomatoid odontogenic tumor in the maxillary antrum
Figure 5: Intra-operative photograph of surgical excision
Figure 6: Excised specimen
Figure 7: Histopathological section of excisional biopsy specimen
Figure 8: Post-operative intraoral photo graph
the follicular type (73%), the extra follicular type (24%), and the peripheral type (3%) has been described.[7,8]
As previously mentioned AOT is usually solid, but is occasionally cystic. The structure of the cyst and its insertion around the crown of an unerupted tooth were typical of a dentigerous cyst. However, in this case, the multifocal cellular proliferation had the structure of an AOT [Figures 7 and 8].
The female to male ratio for all age groups and all variants is close to 2:1.[9] The tooth that is impacted commonly is the canine in 40%.[10] Conservative surgical enucleation is the treatment modality of choice. Recurrence of AOT is exceptionally rare. Therefore, the prognosis is excellent.[6] With respect to the age of the patient and the localization of the AOT in the maxillary antrum, which was extending from midline to molar region (as shown in CT) the reported case is a rare example of this tumor entity. Due to its low tendency to recur, the case here by presented was treated by conservative surgical enucleation associated with the removal of the unerupted dental element and such procedure was proposed in numerous papers.
Conservative surgical enucleation or curettage is the treatment of choice for AOT. Prognosis is good. Recurrences are rare. Very few histologically diagnosed case reports of AOT in the maxillary antrum have previously been reported. In this case, the final diagnosis of an AOT was arrived by histopathological examination of the enucleated tissue specimen. Rarity of the case is in the fact that the AOT involved the maxillary antrum, was extensive and the treatment resulted in good prognosis.
References
Conclusion
1.
Clinical, radiographic, and microscopic findings in this case are consistent with descriptions of the lesion in the dental literature.
2.
S198
John JB, John RR. Adenomatoid odontogenic tumor associated with dentigerous cyst in posterior maxilla: A case report and review of literature. J Oral Maxillofac Pathol 2010;14:59‑62. Lee JK, Lee KB, Hwang BN. Adenomatoid odontogenic tumor: A case report. J Oral Maxillofac Surg 2000;58:1161‑4.
Journal of Pharmacy and Bioallied Sciences July 2014 Vol 6 Supplement 1
Krishnamurthy, et al.: Adenomatoid odontogenic tumor in the maxillary antrum 3. 4. 5. 6.
7.
Philipsen HP, Reichart PA, Zhang KH, Nikai H, Yu QX. Adenomatoid odontogenic tumor: Biologic profile based on 499 cases. J Oral Pathol Med 1991;20:149‑58. Friedrich RE, Scheuer HA, Zustin J. Adenomatoid odontogenic tumor (AOT) of maxillary sinus: Case report with respect to immunohistochemical findings. In Vivo 2009;23:111‑6. Geist SM, Mallon HL. Adenomatoid odontogenic tumor: Report of an unusually large lesion in the mandible. J Oral Maxillofac Surg 1995;53:714‑7. Handschel JG, Depprich RA, Zimmermann AC, Braunstein S, Kübler NR., Adenomatoid odontogenic tumor of the mandible: Review of the literature and report of a rare case. Head Face Med 2005;1:3. Tajima Y, Sakamoto E, Yamamoto Y. Odontogenic cyst giving rise to
Journal of Pharmacy and Bioallied Sciences July 2014 Vol 6 Supplement 1
an adenomatoid odontogenic tumor: Report of a case with peculiar features. J Oral Maxillofac Surg 1992;50:190‑3. 8. Regezi JA, Kerr DA, Courtney RM. Odontogenic tumors: Analysis of 706 cases. J Oral Surg 1978;36:771‑8. 9. Courtney RM, Kerr DA. The odontogenic adenomatoid tumor. A comprehensive study of twenty new cases. Oral Surg Oral Med Oral Pathol 1975;39:424‑35. 10. Philipsen HP, Srisuwan T, Reichart PA. Adenomatoid odontogenic tumor mimicking a periapical (radicular) cyst: A case report. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2002;94:246‑8.
Source of Support: Nil, Conflict of Interest: None declared.
S199
Copyright of Journal of Pharmacy & Bioallied Sciences is the property of Medknow Publications & Media Pvt. Ltd. and its content may not be copied or emailed to multiple sites or posted to a listserv without the copyright holder's express written permission. However, users may print, download, or email articles for individual use.
