Journal of Oral and Maxillofacial Surgery, Medicine, and Pathology xxx (xxxx) xxx–xxx
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Case Report
Extrafollicular adenomatoid odontogenic tumor mimicking residual cyst Hasan Can Akguna, Damla Torulb,*, Mehtap Muglalia, Seda Gunc a b c
Department of Oral and Maxillofacial Surgery, Faculty of Dentistry, Ondokuz Mayis University, Samsun, Turkey Department of Oral and Maxillofacial Surgery, Faculty of Dentistry, Ordu University, Ordu, Turkey Department of Pathology, Faculty of Medicine, Ondokuz Mayis University, Samsun, Turkey
A R T I C LE I N FO
A B S T R A C T
Keywords: Benign tumor Hamartoma Mandible
Adenomatoid odontogenic tumor is a rarely seen nonaggressive tumor originated from the remnants of dental lamina. Majority of the cases detected in females with a peak incidence at the second decade of life. Adenomatoid odontogenic tumor is most commonly detected in the anterior region of the maxilla and occurrence of the tumor in mandible is rarely seen. The aim of this case report is to present the diagnosis and treatment of a rare case of adenomatoid odontogenic tumor located in the anterior region of the mandible of a 68-year-old edentulous man.
1. Introduction
2. Case report
Adenomatoid odontogenic tumor (AOT) is a rare benign odontogenic pathology that constitute only 0.1 % of the tumors located in head and neck [1]. The prevalence of AOT among odontogenic tumors varies from 0.6% to 38.5% with the higher prevalence being reported from the Africa [2]. The tumor originates from the dental lamina and has a slow and progressive growth pattern that does not infiltrate the bone [3–5]. It usually affects younger female patients, mostly during their second decade and rarely seen in patients above 30 years of age [4]. Maxillary anterior region is the most preferred area by the tumor in the jawbones [6,7]. AOT has three clinicopathologic variants with identical histopathological characteristics as intraosseous follicular, intraosseous extrafollicular, and peripheral [4,8]. Intraosseous extrafollicular form of the tumor accounts for nearly 25 % of reported cases and commonly radiographical appearance of the AOT mimics odontogenic cysts such as residual cyst [9]. Clinically, AOT presents as an asymptomatic lesion and may cause expansion of the bone, and displacement of neighboring teeth [4,5]. Management of AOT is usually conservative surgical excision, and the recurrences are rarely seen [7]. Because of the rarity of AOT and the characteristics of the lesion described here such as the gender, age of the patient and location of the tumor, description of this clinical case is relevant. This report presents a rare case of AOT located in the anterior mandible of a 68-year-old edentulous man.
A 68-year-old edentulous man was admitted to oral maxillofacial surgery clinic of Ondokuz Mayis University with a complaint of swelling located in the anterior region of the mandible. The medical history of the patient was unremarkable. Clinical examination revealed a painless swelling localized in anterior mandible. Radiographic examination showed a well-circumscribed radiolucency extending from the right to left premolar regions. Small amounts of radiopacity were identifiable in the radiography (Figs. 1 and 2). A preliminary diagnose of residual cyst was considered and excision under local anesthesia was planned. The lesion was removed surgically and the excised specimen submitted for histologic examination. Microscopically, the tumor is composed of spindle-shaped epithelial cells that from nodules or whorled masses of cells in scant fibrous stroma. Within nodules are variably size rosette –or duct-like spaces. These are lined by a columnar or cuboidal epithelium (Fig. 3). Histologic examination of the specimen revealed the diagnosis as AOT. The post-operative course of the patient was uneventful with a noticeable bone healing (Fig. 4). No recurrence has detected in 1-year follow-up period.
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3. Discussion AOT is a benign, slow growing entity which cause wide confusions regarding nomenclature and the pathogenesis, in the literature [10]. It is still not clear as to whether AOT is a hamartoma or a true neoplasm [4,10,11]. Various theories regarding the origin of the AOT have mentioned in the literature previously. It is reported that depending on
Corresponding author at: Ordu University, Faculty of Dentistry, Department of Oral and Maxillofacial Surgery, Ordu, 52200, Turkey. E-mail address:
[email protected] (D. Torul).
https://doi.org/10.1016/j.ajoms.2019.10.006 Received 14 August 2019; Received in revised form 11 September 2019; Accepted 7 October 2019 2212-5558/ © 2019 Asian AOMS, ASOMP, JSOP, JSOMS, JSOM, and JAMI. Published by Elsevier Ltd All rights reserved.
Please cite this article as: Hasan Can Akgun, et al., Journal of Oral and Maxillofacial Surgery, Medicine, and Pathology, https://doi.org/10.1016/j.ajoms.2019.10.006
Journal of Oral and Maxillofacial Surgery, Medicine, and Pathology xxx (xxxx) xxx–xxx
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Fig. 1. Preoperative view of the lesion on panoramic radiography.
Fig. 3. (A) Nodular swirls of epithelial cells containing duct like spaces of various sizes (HEX100), (B) Duct like spaces lined by cuboidal to columnar epithelium (HEX200).
common occurrence within the tooth-bearing areas AOT originate from the remnants of the dental lamina. Recently, it is reported that MAPK / ERK signaling pathway is activated accompanied with the mutation at codon 12 most of KRAS which is the most common mutated oncogene, and these are considered to play role in the pathogenesis of AOT [12–14]. Extrafollicular variant of the AOT has reported to occur as a response to an unknown stimulus that triggers the remnants of the dental lamina outside the eruptive path. Depending on the spatial relationship between the tumor and the adjacent teeth, extrafollicular AOT appear as an intra-bony radiolucent lesion localized laterally or apically to adjacent teeth, without impairing the eruption process [8,9,11]. Thus,
Fig. 2. Preoperative view of the lesion on CBCT.
its origin, AOT may observed in both solid or cystic forms. Thakur et al. [10] hypothesized that AOT arises from the residual enamel epithelium and grows as a cystic pathology. Also, they added that if the AOT arises from the epithelial remnants in the gubernacular dentis then a solid neoplastic pathology occurs. According to Philipsen et al. [11] due its
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Fig. 4. Postoperative panoramic view.
radiographs more effective than panoramic radiographs [17,19,24]. Also, to facilitate diagnosis computed tomography (CT) may be used as an alternative imaging modality compared to conventional radiographs [19]. The extrafollicular variant of AOT cause confusion regarding diagnosis and needs to be differentially diagnosed from several lesions of odontogenic or non-odontogenic origin [17,25]. Conservative excision is the preferred treatment modality for AOTs because the tumor is benign and generally well encapsulated. However, recurrences have been also reported. Hence, rigorous follow-up constitutes an important part of the treatment [7,10,19].
it may radiographically appear as a residual, radicular or lateral periodontal cyst and cause confusions in terms of the initial diagnosis [8,11]. Mosavat et al. [15] and Curran et al. [9] reported an extrafollicular AOT mimicking a periapical cyst. In another case report Saito et al. [16] reported an unusual extrafollicular AOT presenting as a residual cyst in anterior mandible of an edentulous man. Similarly, in our case extrafollicular AOT detected in anterior mandible of an edentulous patient. Among the odontogenic tumors AOT constitute the fourth most common neoplasm [15]. It commonly detected between of 5–50 years of age and nearly 69 % of cases seen in the second decade of life [7,15]. Becker et al. [17] analyzed 272 cases of AOT and reported that the AOT can be diagnosed in a wide age range between 3 to 82 years with a mean of 18.4 years. Extrafollicular variant of AOT usually observed in older patients than the follicular variant. The probable cause of this considered as the lack of impacted tooth associated with follicular AOT which leads the patient to see a dentist [4,17]. AOT has a marked female preponderance and the tumor more commonly observed in black patients. Majority of the lesions occur in the anterior regions of the jaws, especially in maxilla [5,7,15,17]. Swasdison et al. [4] reported 67 cases of AOT majority of the cases located in maxilla. However, Arotiba et al. [18] mentioned that extrafollicular AOTs were more commonly detected in the mandible. Different from the norms, our patient is an edentulous, 68-year-old male and there were very limited cases of extrafollicular AOT in the literature with the characteristic of our case especially in terms of the age of the patient. Also, in concordance with literature extrafollicular AOT in our case detected in anterior mandible. Clinically, AOTs have slow growth pattern with usually a symptom-free behavior [8,17]. Because of the asymptomatic and slow growing nature of AOT patients don’t need dental treatment until an obvious deformity occurs [5]. Particularly in the extrafollicular type, the displacement of neighboring teeth is also reported as a common finding observed in central variants of the tumor [8]. Regarding size the AOT generally about 1−3 cm in diameter. Unusually large lesions that show aggressive behavior rarely seen [19,20]. Shaikh et al. [1] reported an aggressive AOT 6.5 cm × 3.5 cm in size and has shown a rapid progression more than 6 cm within 1 month with unilateral paresthesia. Oliviera et al. [7] reported an unusual AOT extending between the teeth 44 to 36. WHO defined the AOT as a tumor originated from the odontogenic epithelium with duct-like structures and with varying degrees of inductive change in the connective tissue [21]. Rosette-like configuration created by columnar or cuboidal epithelial cells and tumor droplets of eosinophilic and uncalcified material can be seen as unique features of AOT [3,22]. Radiographically, AOT usually appear as a unilocular radiolucency which may contain fine calcifications [20]. Toida et al. [23] reported that in approximately 78 % of the AOTs, calcified deposits were seen and detection of these deposits by intraoral
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