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Clinical Note Pathological and Clinical Study of Japanese Ameloblastic Fibro-Odontomas

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Clinical Note Pathological and Clinical Study of Japanese Ameloblastic Fibro-Odontomas Journal of Hard Tissue Biology 26[4] (2017) 425-430 2017 The Hard Tissue Biology Network Association Printed in Japan, All rights reserved. CODEN-JHTBFF, ISSN 1341-7649 Clinical Note Pathological and Clinical Study of Japanese Ameloblastic Fibro-Odontomas Shintaro Sukegawa1), Keisuke Nakano2), Takahiro Kanno3), Hotaka Kawai2), Kenichi Matsumoto1), Yuka Sukegawa-Takahashi1), Masanori Masui1) and Yoshihiko Furuki1) 1) Division of Oral and Maxillofacial Surgery, Kagawa Prefectural Central Hospital, Takamatsu, Japan 2) Department of Oral Pathology and Medicine, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical Sciences, Okayama, Japan 3) Department of Oral and Maxillofacial Surgery, Shimane University Faculty of Medicine, Shimane, Japan (Accepted for publication, August 31, 2017) Abstract: Ameloblastic fi bro-odontoma (AFO) is defi ned as a tumor with general characteristics of an ameloblastic fi broma along with the presence of enamel and dentine. AFO is a well-encapsulated, painless, slow-growing, and expanding tumor in young patients. Histologically, it has been classifi ed as an ameloblastic fi broma or odontoma. In the 2017 new WHO classifi cation of odontogenic tumor, AFO is described that they in most cases represent developmental stages of either complex or compound odontoma and that retaining them as separate entities would therefore be illogical. However, there is still considerable confusion concerning the nature and histology of AFO and the surgical therapy for this lesion. Here we present a case of maxillary AFO and review the relevant literature regarding the clinical and surgical features of this lesion. Key words: Ameloblastic fi bro-odontoma, Odontogenic tumor, Japanese patients, Review Introduction In addition, there was a radiolucent area containing punctate calcifi ed Ameloblastic fibro-odontoma (AFO) is an uncommon mixed components (Fig. 2A). Computed tomography (CT) images revealed odontogenic tumor of the odontogenic epithelium with a mesenchyme a radiolucent lesion containing punctate calcifications scattered origin1,2). According to the World Health Organization (WHO) throughout the center of the lesion in the left maxillary area, with classification, AFO is a lesion resembling ameloblastic fibroma, constriction of the maxillary sinus (Fig. 2B). Buccopalatal expansion which also shows inductive alterations involving both enamel and and perforation of the involved cortices were not observed. Considering dentin3). Since the introduction of the WHO classification, AFO has the clinical and radiological fi ndings, the possible diff erential diagnoses been recognized as a histological and clinical entity, but it is still rare were calcifying epithelial odontogenic tumor, adenomatoid odontogenic among Japanese people. The purpose of this report was to describe a tumor, and AFO. case of AFO and review the literature on AFO among Japanese patients between 1971 and 2016. Treatment and outcomes Under local anesthesia, enucleation of the lesion with careful Materials and Methods curettage involving the fi rst and second molars was performed using Case presentation a transoral surgical approach. No bone augmentation was needed. In June 2009, a 12-year-old Japanese girl presented with a chief There was no penetration into the maxillary sinus. Wound closure was complaint of tooth eruption disturbance in the left posterior maxilla performed with a trapezoidal fl ap. There has been no recurrence during to an orthodontic clinic. Because an asymptomatic posterior left the two- year follow-up. maxillary lesion was discovered on panoramic radiography performed for routine evaluation, the patient was referred to the Division of Oral Histopathological and immunohistological studies and Maxillofacial Surgery at Kagawa Prefectural Central Hospital. The The resected specimen was examined by hematoxylin and eosin medical, social, and family histories of the patient were unremarkable. (HE) staining and immunofluorescent (IF) staining. The target An extraoral examination revealed no obvious abnormality, without proteins assessed included the following: Wnt1 (Abcam, Cambridge, swelling or facial asymmetry. An intraoral examination showed slight UK), a signal transduction protein acting on epithelial-mesenchymal tenderness at the left upper molar area. Despite eruption of the right interaction. Wnt signaling is implicated in cell proliferation and first and second upper molars, there was no eruption of the left first diff erentiation., β-catenin (E274, Abcam, Cambridge, UK), a maker of and second molars of the first dentition (Fig. 1A, B). A panoramic activation of Wnt/β-catenin signal pathway. Wnt signaling involve in radiograph revealed a radiolucent lesion (20 × 15 mm in diameter) the nuclear localization of β-catenin. with irregular borders around the unerupted first and second molars. Search strategy of the literature Correspondence to: Dr. Sukegawa Shintaro, Division of Oral and Maxillofacial An electronic search without time or language restrictions was Surgery, Kagawa Prefectural Central Hospital, 1-2-1, Asahi-cho, Takamatsu, Kagawa 760-8557, Japan; Tel.: +81 87 811 3333; Fax: +81 87 802 1188; Email: gouwan19@ conducted in 2016 using the PubMed, Medline, and ScienceDirect gmail.com databases. The following terms were used in the search: ameloblastic 425 J.Hard Tissue Biology Vol. 26(4): 425-430, 2017 Figure 2. Panoramic radiograph and CT image. A: A panoramic radiograph Figure 1. A: Extraoral examination revealed no obvious abnormality., B: shows a radiolucent lesion (20 × 15 mm in diameter) with irregular borders Intraoral examination shows unerupted upper left first and second molars around unerupted first and second molars., B: CT images revealed a despite eruption of the upper right fi rst and second molars. radiolucent lesion containing punctate calcifi cations in the left maxillary area. Figure 3. Histological fi ndings (HE: A-D). A: Dentin like hard tissues (*) located in the tumor tissue. The background is composed of fi brous stroma and epithelial tumor nests. Scale bar= 100μm., B: Epithelial islands and mesenchymal fi brous components. Scale bar= 20μm., C: Enamel organ like epithelial tumor nests supported by dental papilla like tumor tissue. Scale bar= 20μm., D: Dentin (arrow), enamel matrix (arrowhead) and enamel (*) formation in the intermediate zone between epithelial and mesenchymal components. Scale bar= 20μm., 426 Shintaro Sukegawa et al.: Clinicopathology of Japanese Ameloblastic Fibro-Odontoma Table 1. Summary of the literature review of Japanese patients with ameloblastic fi bro-odontoma Age Mean Standard Deviation Range Median 11.5 9.0 0.5-60 9.0 Sex Male Female 31 26 % 54.4 45.6 Location Lt/ Rt Left Right 32 25 % 56.1 43.9 Mxilla/Mandible Maxilla Mandible 19 38 % 33.3 66.7 Anterior/ Posterior Anterior Posterior Anterior and Posterior 10 45 2 % 17.5 78.9 3.5 Impacted teeth Yes No 52 5 % 91.2 8.8 Treatment Exclusion Exclusion of the tumor (Retention of the involved teeth) 18 39 % 31.6 68.4 fi brodentinoma OR ameloblastic fi bro-odontoma OR ameloblastic fi bro- Immunohistochemical fi ndings (IF) odontome OR ameloblastic fi broodontoma. Moreover, Google Scholar Wnt1 was detected in almost epithelial and mesenchymal and Web Japan Medical Abstractis Society were checked. The titles and components. The peripheral columnar cells of epithelial tumor nest abstracts of all publications identifi ed through the electronic searches were strongly positive to Wnt1 (Fig. 3E). Although β-catenin was were read by the authors. The clinical aspects and the histological clearly localized on the cell membrane of tumor cells, nuclear trans- description of the lesions were thoroughly assessed to confirm the location was observed in stellate reticulm cells and in many papilla-like diagnosis of AFO. mesenchymal cells (Fig. 3F). Wnt1 and β-catenin were present in the tumor nests mainly. The both positive area were almost maerged (Fig. Ethics statement and confi rmation of patient permission 3G). The authors confirm that the patient undergoing the procedure described in this clinical note was fully informed about the condition Results of the literature review and consented to the clinical and surgical procedures, which included The results of the literature review and our case are summarized in taking photographs of the lesion and the procedure. The authors confi rm Table 18-56). that personal details of the patient included in any part of the paper and in any supplementary material have been removed before submission. Patient characteristics Age and sex Results There were 57 patients, including 31 (54.4%) male and 26 (45.6%) Histopathological fi ndings (HE) female patients (male:female ratio, 1.19). The mean age of the patients Microscopically, the lesions were composed of soft tissue was 11.5 years (standard deviation, 9.0 years; range, 0.5-60 years). components and calcified components (Fig. 3A). The soft tissue included tooth bud like nest and exhibited mainly strands and cords of Location odontogenic epithelium that resembled the dental lamina. The stromal The lesion was located in the maxilla in 19 (33.3%) patients and components were consisted of fi broblast like cell with plump nucleus mandible in 38 (66.7%). The lesion occurred on the left side in 32 which proliferating with produce dense collagen fi bers (Fig. 3B). The (56.1%) patients and on the right side in 25 (43.9%). The lesion was lesion also contained epithelial islands that consisted of a peripheral anterior in 10 (17.5%) patients, posterior in 45 (78.9%), and
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