- Email: [email protected]
Adenomatoid odontogenic tumor involving maxillary sinus in a child
Case Report
Adenomatoid odontogenic tumor involving maxillary sinus in a child Amandeep Bhullar, mds*, Raman Preet Kaur Bhullar, mds**†, Shikha Kler, mds† *Senior Lecturer, Department of Prosthodontics Crown and Bridge, **Senior Lecturer, Department of Oral and Maxillofacial Pathology, Genesis Institute of Dental Sciences and Research, Ferozepur, Punjab, †Senior Lecturer, Department of Oral and Maxillofacial Pathology, DAV (C) Dental College, Yamunanagar, Haryana, India.
Abstract Adenomatoid odontogenic tumor (AOT) is an uncommon tumor of odontogenic origin, constituting only 3% of all odontogenic tumors. Adenomatoid odontogenic tumors are frequently asymptomatic and are discovered during the course of a routine radiographic examination. The AOT is an uncommon tumor of odontogenic origin composed of odontogenic epithelium and a plethora of histoarchitectural patterns. It is a very uncommon cause of jaw swelling. We present a case of intraosseus follicular AOT involving maxillary sinus in an 8-year-old female child with the impacted tooth in the orbital floor. Keywords: Adenomatoid odontogenic tumor, children, maxillary sinus
INTRODUCTION
CASE REPORT
Adenomatoid odontogenic tumor (AOT) was first described by Dreidbladtin 1907 as adenoameloblastoma. In 1948, Stafne considered it as a distinct entity but others classified it as a variant of ameloblastoma.1,2 Philipsen and Birn proposed the widely accepted term AOT in 1969, a term that was accepted by the first edition of World Health Organization (WHO) classification of odontogenic tumors in 1971. The lesion is benign (hamartomatous) and noninvasive, with slow but progressive growth. It accounts for 2–7% of all odontogenic tumors and is less frequent than odontoma, cementoma, myxoma, and ameloblastoma.3 The majority of the cases (88%) are diagnosed in the second and third decades of life. The lesion usually presents as asymptomatic swelling and is often associated with an unerupted tooth.4 In this report, we present a description of case in an 8-year-old female child, who had an aggressive AOT located in the maxillary sinus with an impacted premolar tooth. The tumor had caused the resorption of roots of the primary canine and molars, which is quite unusual for this tumor. An impacted canine was found outside the lesion near the lateral wall of the nose. An attempt has also been made to determine the origin of the tumor.
An 8-year-old female patient reported to the Department of Oral and Maxillofacial Surgery with chief complaint of mass in the left buccal area of maxilla and the absence of left canine and first premolar in the same area. Intra-orally, the left maxillary alveolar process was prominently distal to the incisors. The oral mucosa was apparently unaffected. All teeth proved to react sensitively to adequate stimuli. The facial appearance of the patient was conspicuous and revealed asymmetry (Figure 1). The orthopantomograph (OPG) revealed a large unilocular radiolucency of 3.5–4 cm in size involving the maxillary sinus in association with the impacted left premolar in the orbital floor. The left maxillary canine was found near the lateral wall of the nose. The second left maxillary premolar was embedded in the roots of the left maxillary permanent molar. The lesion had caused the resorption of the roots of the maxillary left deciduous canine, first molar, and second molar but that has to be distinguished from the physiological process of root resorption that occurs at the same age (Figure 2). The initial differential diagnosis constituted dentigerous cyst, AOT, unicystic ameloblastoma, odontogenic keratocyst, and ameloblastic fibroma. The mass was surgically enucleated totally under general anesthesia; 63, 64, 65, 23, 24, 25, 26 were removed along with the lesion. The maxillary sinus was exposed by the fenestration of the facial aspect of the maxilla. Impacted 23 and 24 were removed surgically. Nasal antrostomy was done and paraffin socked betadine gauze was placed in the bony cavity, which was removed after 48 hours. Surgical closure was done with 3–0 vicryl. The underlying bone was thinned out.
Correspondence: Dr. Raman Preet Kaur Bhullar, Senior Lecturer, Department of Oral and Maxillofacial Pathology, Genesis Institute of Dental Sciences and Research, Ferozepur, Punjab, India. E-mail: [email protected] Received: 19.10.2011 Accepted: 18.11.2011 doi: 10.1016/S0975-962X(12)60008-2 © 2012 Indian Journal of Dentistry. Published by Elsevier Ltd.
25
Bhullar, et al
Below the bone, the tumor was evident. The cyst filled the sinus completely and was consecutively excavated. There were no apparent infiltrations of the surrounding bones. The oral defect was closed by primary intention. The healing was uneventful. The radiographic follow-up examination after 2 years revealed no local recurrence. The specimen, received in formalin, consisted of one piece of gray soft-tissue measuring 6 mm × 4 mm × 3 mm in size (Figure 3). The microscopy
Figure 1 The swelling on the left side of the face.
Figure 2 The orthopantomograph revealed a large unilocular radiolucency of 3.5–4 cm in size involving the maxillary sinus in association with the impacted left premolar in the orbital floor.
Figure 3 Cut section of the tumor specimen showing well-circumscribed tumor mass.
26
revealed a well-defined cystic tumor encapsulated with a thick, regular fibrous connective tissue (Figure 4). The tumor consisted of highly cellular tissue made up of cuboidal and low columnar cells arranged in a whorl-, duct-, or ring-like pattern. Few areas of calcifications and pools of eosinophilic material were also seen (Figure 5). The ductlike structures were lined by a single row of cuboidal cells (Figure 6). All the features were diagnostic for AOT. Overall features confirmed the diagnosis of AOT.
Figure 4 A well-defined encapsulated tumor with thick fibrous connective tissue.
Figure 5 The tumor nodule composed of spindle-shaped or cuboidal epithelial cells forming rosette-, duct-, and whorl-like pattern. Area of calcification and pools of eosinophilic material are also evident (×10).
Figure 6 Characteristic tubular or duct-like structures lined by a single row of low columnar epithelial cells (×40).
© Indian Journal of Dentistry 2012/Volume 3/Issue 1
Adenomatoid odontogenic tumor involving maxillary sinus in a child
DISCUSSION Adenomatoid odontogenic tumor is a benign, slow-growing, epithelial tumor showing an intra-oral location, and in very big lesions, sometimes has an extra-oral component, which manifests as a painless mass. Patients are usually referred to dentists with complaints of impacted teeth within the lesion.5,6 It is a benign, noninvasive benign odontogenic lesion showing slow growth. It is generally intraosseous, but can occur rarely in peripheral locations. The subtyping of AOT is based on clinical and radiological findings. The follicular (intraosseous) type is by far the most frequent growth type of AOT.7,8 In the case of follicular growth type, the tumor is localized around the crown of a retained tooth and additionally may cover the upper part of the dental root. In this case, the cystlike tumor covered the tooth completely, giving access to the hard tissue after intended incision of the resection specimen. The gross macroscopic appearance of the tumor is in accordance with former definitions of the entity recommended by the WHO,3,9 the AOT may be ‘partly cystic and in some cases the solid lesion may be present only as masses in the wall of a large cyst’, as in this case. The AOT is an infrequent benign epithelial tumor, preferentially found in children and young adults and in the second decade of life. This female predilection is even more marked in Asian females: the highest being observed in Sri Lanka (3.2:1) and Japan (3:1).7 Our case presented with tumor in an 8-year-old female patient which is quite unusual for this age group. Philipsen et al observed only 2.5% of cases occurring in this age group of 1–9 years in female patients in their review of 397 cases of AOT. On radiographs, the intraosseous follicular variant of AOT shows a well-delineated, unilocular radiolucency surrounding the crown of a retained tooth, a picture indistinguishable from follicular cysts. Indeed, the radiologic findings of AOT frequently share the characteristics of other odontogenic lesions such as dentigerous cyst, calcifying odontogenic cyst or tumor, ameloblastoma, keratocystic odontogenic tumor, or periapical disease. Minute radiopacities around the retained tooth may be found in AOT and are considered a characteristic but not pathognomonic finding. The tumor is usually associated with unerupted teeth, frequently canines or lateral incisors. For cases in which the lesion appears to surround an unerupted tooth and has no radio-opaque component, dentigerous cyst may also be considered in the differential diagnosis. However, AOT often appears to envelop the crown as well as the root, whereas dentigerous cysts do not envelop the roots.10,11 This case showed well-delineated unilocular radiolucency associated with premolar till CEMENTO-ENAMEL junction in the orbital floor which is quite infrequent. Adenomatoid odontogenic tumor seldom causes resorption of roots, which was again seen in our case in association with 63, 64, and 65, but again it has to be distinguished from the physiological process that occurs at the same age. The AOT has a distinctive histopathologic appearance with the quintessential identifier being the spindle-shaped © Indian Journal of Dentistry 2012/Volume 3/Issue 1
epithelial cells that are organized in sheets, strands, or whorled masses surrounded by sparse stroma. Duct-like structures, lined by cuboidal to columnar epithelial cells with nuclei oriented away from the lumen, may be prominent or absent. The AOT is often surrounded by a thick capsule of fibrous connective tissue. Philipsen et al described small to large calcifications. These have been suggested to be abortive enamel formation, dentin, or dystrophic calcifications.11 The histogenesis of AOT is still uncertain, although recent findings strongly indicate that AOT is derived from a complex system of dental laminae or its remnants. The fact that all AOT variants show identical histology strongly points toward a common origin. In order to conceptualize a unified source of origin for the diverse locations of AOT, one has to look to odontogenic epithelium with a widespread occurrence in the jaws. Of the possible candidates, only one matches the requirements of widespread distribution namely the dental lamina complex or its remnants. Disintegration of the complex system of dental lamina gives rise to numerous epithelial remnants (HR) persisting in the jaw bones and gingiva after completion of odontogenesis that can give rise to various variants of AOT.12 The hypothesis that follicular AOTs arise from the reduced enamel epithelium (REE) that lines the follicles of unerupted teeth is fairly conclusive and is supported by evidence that is both morphologic and immunocytochemical in nature. They surround the crowns and are attached to the necks of the unerupted teeth in a true follicular relationship. Many present as cystic lesions with only mural nodules of AOT lesional tissue and, in some instances, the origin of the lesional tissue from the REE can be demonstrated histologically. Whether the origin of the follicular variant occurs before or after cystic expansion has taken place is open to conjecture. If it occurs after cystic expansion, then this effectively means origin from a dentigerous cyst, and several such case reports have been published. If it occurs before cystic expansion, then the tumor tissue will fill the follicular space and the AOT will present as a solid tumor. It is reasonable to assume that, given enough time, even those originating from a cyst may grow and fill the lumen completely. Dental lamina remnants likely represent the progenitor cells for this benign odontogenic tumor. According to this hypothesis, the lesion grows (sometimes while forming a cystic space) next to or into a nearby dental follicle, leading to the ‘envelopmental theory’. In the case reported here, the lesion surrounded a fully formed premolar, which suggests ‘envelopmental’ pathogenesis.11,12 All variants of AOT are well-encapsulated and show an identical benign behavior. Conservative surgical enucleation or curettage is the treatment of choice. Recurrence of AOT is exceptionally rare. Only three cases in Japanese patients are reported in which the recurrence of this tumor occurred. An uninvolved follicle found during surgery can be easily separated from the tumor, leading to the possible removal of the lesion while leaving the teeth in place, but in other cases, it is wise to remove both hard and soft-tissues along with the follicular tissue as done in our case.7 The patient is disease-free 27
Bhullar, et al
after 2 years of follow-up. However, further periodic routine follow-up was recommended for successful postoperative prognosis. CONCLUSION Adenomatoid odontogenic tumor is an encapsulated, nonaggressive tumor. Although AOT is not as rare as an odontogenic tumor, as had been previously thought, there have been few reports of the lesion located in the maxillary antrum involving the first premolar and in an 8-year-old female patient along root resorption. REFERENCES 1. 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. 2. Stafne EC. Epithelial tumours associated with developmental cysts of the maxilla. A report of three cases. Oral Surg Oral Pathol 1948;1: 887–94.
28
3. Barnes L, Eveson JW, Reichrt P, Sidransky D, eds. World Health Organization Classification of Tumours, Pathology and Genetics of Head and Neck Tumours. Lyon: IARC Press 2005:304–5. 4. Chattopadhyay A. Adenomatoid odontogenic tumour: review of literature and report of 30 cases from India. Ind J Dent Res 1994;5:89–95. 5. Ghosh LS. Adamantinoma of the upper jaw: report of a case. Am J Pathol 1934;10:773–90. 6. Takahashi K, Yoshino T, Hashimoto S. Unusually large cystic adenomatoid odontogenic tumour of the maxilla: case report. Int J Oral Maxillofac Surg 2001;30:173–5. 7. Toida M, Hyodo I, Okuda T, Tatematsu N. Adenomatoid odontogenic tumor: report of two cases and survey of 126 cases in Japan. J Oral Maxillofac Surg 1990;48:404–8. 8. Olgac V, Koseolu BG, Kasapolu C. Adenomatoidodontogenic tumor: a report of an unusual maxillary lesion. Quintessence Int 2003;34: 686–8. 9. Philipsen HP, Reichart PA. Adenomatoid odontogenic tumour: facts and figures. Oral Oncol 1998;35:125–31. 10. 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. 11. Philipsen HP, Reichart PA. Odontogenic Tumors and Allied Lesions 2nd ed. Surrey, UK: Quintessence Publishing Company 2004:105–15. 12. Philipsen HP, Samman N, Ormiston IW, Wu PC, Reichart PA. Variants of the adenomatoid odontogenic tumor with a note on tumor origin. J Oral Pathol Med 1992;21:348–52.
© Indian Journal of Dentistry 2012/Volume 3/Issue 1
Adenomatoid odontogenic tumor involving maxillary sinus in a child Amandeep Bhullar, mds*, Raman Preet Kaur Bhullar, mds**†, Shikha Kler, mds† *Senior Lecturer, Department of Prosthodontics Crown and Bridge, **Senior Lecturer, Department of Oral and Maxillofacial Pathology, Genesis Institute of Dental Sciences and Research, Ferozepur, Punjab, †Senior Lecturer, Department of Oral and Maxillofacial Pathology, DAV (C) Dental College, Yamunanagar, Haryana, India.
Abstract Adenomatoid odontogenic tumor (AOT) is an uncommon tumor of odontogenic origin, constituting only 3% of all odontogenic tumors. Adenomatoid odontogenic tumors are frequently asymptomatic and are discovered during the course of a routine radiographic examination. The AOT is an uncommon tumor of odontogenic origin composed of odontogenic epithelium and a plethora of histoarchitectural patterns. It is a very uncommon cause of jaw swelling. We present a case of intraosseus follicular AOT involving maxillary sinus in an 8-year-old female child with the impacted tooth in the orbital floor. Keywords: Adenomatoid odontogenic tumor, children, maxillary sinus
INTRODUCTION
CASE REPORT
Adenomatoid odontogenic tumor (AOT) was first described by Dreidbladtin 1907 as adenoameloblastoma. In 1948, Stafne considered it as a distinct entity but others classified it as a variant of ameloblastoma.1,2 Philipsen and Birn proposed the widely accepted term AOT in 1969, a term that was accepted by the first edition of World Health Organization (WHO) classification of odontogenic tumors in 1971. The lesion is benign (hamartomatous) and noninvasive, with slow but progressive growth. It accounts for 2–7% of all odontogenic tumors and is less frequent than odontoma, cementoma, myxoma, and ameloblastoma.3 The majority of the cases (88%) are diagnosed in the second and third decades of life. The lesion usually presents as asymptomatic swelling and is often associated with an unerupted tooth.4 In this report, we present a description of case in an 8-year-old female child, who had an aggressive AOT located in the maxillary sinus with an impacted premolar tooth. The tumor had caused the resorption of roots of the primary canine and molars, which is quite unusual for this tumor. An impacted canine was found outside the lesion near the lateral wall of the nose. An attempt has also been made to determine the origin of the tumor.
An 8-year-old female patient reported to the Department of Oral and Maxillofacial Surgery with chief complaint of mass in the left buccal area of maxilla and the absence of left canine and first premolar in the same area. Intra-orally, the left maxillary alveolar process was prominently distal to the incisors. The oral mucosa was apparently unaffected. All teeth proved to react sensitively to adequate stimuli. The facial appearance of the patient was conspicuous and revealed asymmetry (Figure 1). The orthopantomograph (OPG) revealed a large unilocular radiolucency of 3.5–4 cm in size involving the maxillary sinus in association with the impacted left premolar in the orbital floor. The left maxillary canine was found near the lateral wall of the nose. The second left maxillary premolar was embedded in the roots of the left maxillary permanent molar. The lesion had caused the resorption of the roots of the maxillary left deciduous canine, first molar, and second molar but that has to be distinguished from the physiological process of root resorption that occurs at the same age (Figure 2). The initial differential diagnosis constituted dentigerous cyst, AOT, unicystic ameloblastoma, odontogenic keratocyst, and ameloblastic fibroma. The mass was surgically enucleated totally under general anesthesia; 63, 64, 65, 23, 24, 25, 26 were removed along with the lesion. The maxillary sinus was exposed by the fenestration of the facial aspect of the maxilla. Impacted 23 and 24 were removed surgically. Nasal antrostomy was done and paraffin socked betadine gauze was placed in the bony cavity, which was removed after 48 hours. Surgical closure was done with 3–0 vicryl. The underlying bone was thinned out.
Correspondence: Dr. Raman Preet Kaur Bhullar, Senior Lecturer, Department of Oral and Maxillofacial Pathology, Genesis Institute of Dental Sciences and Research, Ferozepur, Punjab, India. E-mail: [email protected] Received: 19.10.2011 Accepted: 18.11.2011 doi: 10.1016/S0975-962X(12)60008-2 © 2012 Indian Journal of Dentistry. Published by Elsevier Ltd.
25
Bhullar, et al
Below the bone, the tumor was evident. The cyst filled the sinus completely and was consecutively excavated. There were no apparent infiltrations of the surrounding bones. The oral defect was closed by primary intention. The healing was uneventful. The radiographic follow-up examination after 2 years revealed no local recurrence. The specimen, received in formalin, consisted of one piece of gray soft-tissue measuring 6 mm × 4 mm × 3 mm in size (Figure 3). The microscopy
Figure 1 The swelling on the left side of the face.
Figure 2 The orthopantomograph revealed a large unilocular radiolucency of 3.5–4 cm in size involving the maxillary sinus in association with the impacted left premolar in the orbital floor.
Figure 3 Cut section of the tumor specimen showing well-circumscribed tumor mass.
26
revealed a well-defined cystic tumor encapsulated with a thick, regular fibrous connective tissue (Figure 4). The tumor consisted of highly cellular tissue made up of cuboidal and low columnar cells arranged in a whorl-, duct-, or ring-like pattern. Few areas of calcifications and pools of eosinophilic material were also seen (Figure 5). The ductlike structures were lined by a single row of cuboidal cells (Figure 6). All the features were diagnostic for AOT. Overall features confirmed the diagnosis of AOT.
Figure 4 A well-defined encapsulated tumor with thick fibrous connective tissue.
Figure 5 The tumor nodule composed of spindle-shaped or cuboidal epithelial cells forming rosette-, duct-, and whorl-like pattern. Area of calcification and pools of eosinophilic material are also evident (×10).
Figure 6 Characteristic tubular or duct-like structures lined by a single row of low columnar epithelial cells (×40).
© Indian Journal of Dentistry 2012/Volume 3/Issue 1
Adenomatoid odontogenic tumor involving maxillary sinus in a child
DISCUSSION Adenomatoid odontogenic tumor is a benign, slow-growing, epithelial tumor showing an intra-oral location, and in very big lesions, sometimes has an extra-oral component, which manifests as a painless mass. Patients are usually referred to dentists with complaints of impacted teeth within the lesion.5,6 It is a benign, noninvasive benign odontogenic lesion showing slow growth. It is generally intraosseous, but can occur rarely in peripheral locations. The subtyping of AOT is based on clinical and radiological findings. The follicular (intraosseous) type is by far the most frequent growth type of AOT.7,8 In the case of follicular growth type, the tumor is localized around the crown of a retained tooth and additionally may cover the upper part of the dental root. In this case, the cystlike tumor covered the tooth completely, giving access to the hard tissue after intended incision of the resection specimen. The gross macroscopic appearance of the tumor is in accordance with former definitions of the entity recommended by the WHO,3,9 the AOT may be ‘partly cystic and in some cases the solid lesion may be present only as masses in the wall of a large cyst’, as in this case. The AOT is an infrequent benign epithelial tumor, preferentially found in children and young adults and in the second decade of life. This female predilection is even more marked in Asian females: the highest being observed in Sri Lanka (3.2:1) and Japan (3:1).7 Our case presented with tumor in an 8-year-old female patient which is quite unusual for this age group. Philipsen et al observed only 2.5% of cases occurring in this age group of 1–9 years in female patients in their review of 397 cases of AOT. On radiographs, the intraosseous follicular variant of AOT shows a well-delineated, unilocular radiolucency surrounding the crown of a retained tooth, a picture indistinguishable from follicular cysts. Indeed, the radiologic findings of AOT frequently share the characteristics of other odontogenic lesions such as dentigerous cyst, calcifying odontogenic cyst or tumor, ameloblastoma, keratocystic odontogenic tumor, or periapical disease. Minute radiopacities around the retained tooth may be found in AOT and are considered a characteristic but not pathognomonic finding. The tumor is usually associated with unerupted teeth, frequently canines or lateral incisors. For cases in which the lesion appears to surround an unerupted tooth and has no radio-opaque component, dentigerous cyst may also be considered in the differential diagnosis. However, AOT often appears to envelop the crown as well as the root, whereas dentigerous cysts do not envelop the roots.10,11 This case showed well-delineated unilocular radiolucency associated with premolar till CEMENTO-ENAMEL junction in the orbital floor which is quite infrequent. Adenomatoid odontogenic tumor seldom causes resorption of roots, which was again seen in our case in association with 63, 64, and 65, but again it has to be distinguished from the physiological process that occurs at the same age. The AOT has a distinctive histopathologic appearance with the quintessential identifier being the spindle-shaped © Indian Journal of Dentistry 2012/Volume 3/Issue 1
epithelial cells that are organized in sheets, strands, or whorled masses surrounded by sparse stroma. Duct-like structures, lined by cuboidal to columnar epithelial cells with nuclei oriented away from the lumen, may be prominent or absent. The AOT is often surrounded by a thick capsule of fibrous connective tissue. Philipsen et al described small to large calcifications. These have been suggested to be abortive enamel formation, dentin, or dystrophic calcifications.11 The histogenesis of AOT is still uncertain, although recent findings strongly indicate that AOT is derived from a complex system of dental laminae or its remnants. The fact that all AOT variants show identical histology strongly points toward a common origin. In order to conceptualize a unified source of origin for the diverse locations of AOT, one has to look to odontogenic epithelium with a widespread occurrence in the jaws. Of the possible candidates, only one matches the requirements of widespread distribution namely the dental lamina complex or its remnants. Disintegration of the complex system of dental lamina gives rise to numerous epithelial remnants (HR) persisting in the jaw bones and gingiva after completion of odontogenesis that can give rise to various variants of AOT.12 The hypothesis that follicular AOTs arise from the reduced enamel epithelium (REE) that lines the follicles of unerupted teeth is fairly conclusive and is supported by evidence that is both morphologic and immunocytochemical in nature. They surround the crowns and are attached to the necks of the unerupted teeth in a true follicular relationship. Many present as cystic lesions with only mural nodules of AOT lesional tissue and, in some instances, the origin of the lesional tissue from the REE can be demonstrated histologically. Whether the origin of the follicular variant occurs before or after cystic expansion has taken place is open to conjecture. If it occurs after cystic expansion, then this effectively means origin from a dentigerous cyst, and several such case reports have been published. If it occurs before cystic expansion, then the tumor tissue will fill the follicular space and the AOT will present as a solid tumor. It is reasonable to assume that, given enough time, even those originating from a cyst may grow and fill the lumen completely. Dental lamina remnants likely represent the progenitor cells for this benign odontogenic tumor. According to this hypothesis, the lesion grows (sometimes while forming a cystic space) next to or into a nearby dental follicle, leading to the ‘envelopmental theory’. In the case reported here, the lesion surrounded a fully formed premolar, which suggests ‘envelopmental’ pathogenesis.11,12 All variants of AOT are well-encapsulated and show an identical benign behavior. Conservative surgical enucleation or curettage is the treatment of choice. Recurrence of AOT is exceptionally rare. Only three cases in Japanese patients are reported in which the recurrence of this tumor occurred. An uninvolved follicle found during surgery can be easily separated from the tumor, leading to the possible removal of the lesion while leaving the teeth in place, but in other cases, it is wise to remove both hard and soft-tissues along with the follicular tissue as done in our case.7 The patient is disease-free 27
Bhullar, et al
after 2 years of follow-up. However, further periodic routine follow-up was recommended for successful postoperative prognosis. CONCLUSION Adenomatoid odontogenic tumor is an encapsulated, nonaggressive tumor. Although AOT is not as rare as an odontogenic tumor, as had been previously thought, there have been few reports of the lesion located in the maxillary antrum involving the first premolar and in an 8-year-old female patient along root resorption. REFERENCES 1. 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. 2. Stafne EC. Epithelial tumours associated with developmental cysts of the maxilla. A report of three cases. Oral Surg Oral Pathol 1948;1: 887–94.
28
3. Barnes L, Eveson JW, Reichrt P, Sidransky D, eds. World Health Organization Classification of Tumours, Pathology and Genetics of Head and Neck Tumours. Lyon: IARC Press 2005:304–5. 4. Chattopadhyay A. Adenomatoid odontogenic tumour: review of literature and report of 30 cases from India. Ind J Dent Res 1994;5:89–95. 5. Ghosh LS. Adamantinoma of the upper jaw: report of a case. Am J Pathol 1934;10:773–90. 6. Takahashi K, Yoshino T, Hashimoto S. Unusually large cystic adenomatoid odontogenic tumour of the maxilla: case report. Int J Oral Maxillofac Surg 2001;30:173–5. 7. Toida M, Hyodo I, Okuda T, Tatematsu N. Adenomatoid odontogenic tumor: report of two cases and survey of 126 cases in Japan. J Oral Maxillofac Surg 1990;48:404–8. 8. Olgac V, Koseolu BG, Kasapolu C. Adenomatoidodontogenic tumor: a report of an unusual maxillary lesion. Quintessence Int 2003;34: 686–8. 9. Philipsen HP, Reichart PA. Adenomatoid odontogenic tumour: facts and figures. Oral Oncol 1998;35:125–31. 10. 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. 11. Philipsen HP, Reichart PA. Odontogenic Tumors and Allied Lesions 2nd ed. Surrey, UK: Quintessence Publishing Company 2004:105–15. 12. Philipsen HP, Samman N, Ormiston IW, Wu PC, Reichart PA. Variants of the adenomatoid odontogenic tumor with a note on tumor origin. J Oral Pathol Med 1992;21:348–52.
© Indian Journal of Dentistry 2012/Volume 3/Issue 1