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提要：外周性牙源性肿瘤又称骨外型牙源性肿瘤或软组织牙源性肿瘤,主要发生在牙龈,包括牙源性真性肿瘤及错构瘤。外周性牙源性肿瘤约占所有牙源性肿瘤的4%左右,英文文献表明其中外周性牙源性纤维瘤最多见,其次是外周性成釉细胞瘤及外周性牙源性钙化囊性瘤。外周性牙源性肿瘤临床上容易与牙龈发生的炎症性或反应性病变相混淆,明确诊断依赖组织病理学检查。外周性牙源性肿瘤不包括骨内型牙源性肿瘤穿破骨皮质侵犯牙龈。外周性牙源性肿瘤预后普遍好于相应的骨内型肿瘤,但切除不彻底仍可复发,建议长期随访。     

Odontogenic tumors: a review of 319 cases in a Nigerian teaching hospital

OBJECTIVE: This study sought to determine the relative frequency of odontogenic tumors in a Nigerian population and to compare these data with previous reports. STUDY DESIGN: Records of patients seen at the Lagos University Teaching Hospital between January 1980 and December 2003, with histologic diagnosis of odontogenic tumors (based on World Health Organisation classification, 1992), were analyzed. RESULTS: Odontogenic tumors constituted 9.6% of all the biopsies of oral and jaw lesions seen within the period under study. Three hundred and eight (96.6%) were intraosseous, and 11 (3.4%) were peripheral (peripheral odontogenic fibroma=7; peripheral myxoma=3; peripheral ameloblastoma=1). The mean age of patients was 29.9+/-15.6 years (range, 4-85 years). Among these cases, 96.6% of the tumors were benign and 3.4% were malignant. Ameloblastoma with predilection for the mandible was the most frequent odontogenic tumor (63%), followed by adenomatoid odontogenic tumor (AOT) (7.5%), myxoma (6.5%), calcifying epithelial odontogenic cyst (5.3%), and odontogenic fibroma (5.3%). More cases of malignant odontogenic tumors were seen than cases of calcifying epithelial odontogenic tumor and odontomas. The mean ages of patients with AOT, ameloblastic fibroma, and odontoma were significantly lower than those with ameloblastoma ( P<.05). No significant difference was found between the mean ages of patients with benign odontogenic tumors and those with malignant odontogenic tumors ( P=.058). CONCLUSIONS: Odontogenic tumors, especially ameloblastoma, are not considered rare among Nigerians, whereas odontoma, regarded as the most frequent odontogenic tumor in North and South America, is rare.     

Peripheral odontogenic fibroma. Report of 5 cases

The peripheral odontogenic fibroma (WHO type) is a relatively rare, benign, unencapsulated, exophytic gingival mass of fibrous connective tissue. Odontogenic epithelium is found within the gingival mass, but usually appears to play a minor role when compared to the fibrous component. According to the present concept, cases reported in the literature under the terms "odontogenic gingival epithelial harmartoma" "hamartoma of the dental lamina" and "peripheral ameloblastic fibrodentinoma" are actually examples of peripheral odontogenic fibroma. Review of the literature revealed only 30 acceptable cases that fit the present concept of peripheral odontogenic fibroma. Because of the paucity of reported cases, the histomorphological spectrum and the clinical features of this lesion have not yet been fully established. This article presents five new cases of peripheral odontogenic fibroma. The connective tissue ranged from markedly cellular to relatively acellular well collagenized. Islands and strands of epithelium were present in all five cases: in four they were scanty and in one abundant. A matrix of mineralized material was present in four cases. The peripheral odontogenic fibroma must be differentiated histologically from peripheral ossifying fibroma, which is a reactive lesion, and from the peripheral ameloblastoma and the calcifying epithelial odontogenic tumour.     

Epulides in the dog: a review

This article is based on a review of the literature and the study of pathology sections obtained from various veterinary pathology laboratories. Epulis is a non-specific, clinical designation for a localized, exophytic growth on the gingiva. Four reactive epulides occur in human beings, namely focal fibrous hyperplasia (fibrous epulis). pyogenic granuloma. peripheral giant cell granuloma (giant cell epulis. and peripheral ossifying fibroma (calcifying fibrous epulisl). The first three also occur in dogs but only focal fibrous hyperplasia appears to be common. The peripheral ossifying fibroma has not yet been reported in dogs. Odontogenic tumors occurring on the gingiva (i.e., as epulides) are referred to as peripheral odontogenic tumors. Three types have been reported in dogs. One, the common fibromatous epulis. is equivalent to the rare peripheral odontogenic fibroma in human beings. Another, the acanthomatous epulis. appears to be a form of ameloblastoma but differs from the peripheral ameloblastoma in human beings in that it invades bone; its biological behavior is therefore that of the human intraosseous ameloblastoma. The third, a rare lesion, has been referred to in the veterinary literature as a calcifying epithelial odontogenic tumor, although it is not the canine counterpart of the human CEOT The term, amyloid-producing odontogenic tumor , has been suggested as being appropriate for this lesion.     

Peripheral odontogenic tumor: a clinicopathologic study of 30 cases. General features and hamartomatous lesions

BACKGROUND: Peripheral odontogenic tumors (POT), either neoplastic or hamartomatous, are rare. This study briefly summarizes the general features of POT and selectively reviews the histomorphologic spectrum of under-recognized hamartomatous lesions that we have designated peripheral odontogenic hamartomas (POH) in order to shed more light into the pathogenesis of POT. METHODS: Archival material accessioned at our institutions between 1970 and 2004 was systematically searched to identify examples of POT/POH. RESULTS: Among 39 660 biopsies, we retrieved 25 cases of 'classical' POT and five cases of 'unique' POH. Odontogenic fibroma and ameloblastoma were by far the most common. Of POH, two purely epithelial lesions showed multiple strands of basaloid rests [odontogenic gingival epithelial hamartoma (OGEH)] and a conglomerate of polyhedral epithelium, ghost cells and concentric calcifications (calcifying epithelial odontogenic tumor-like hamartoma), respectively. OGEH and peripheral squamous odontogenic tumor (PSOT) deserve to be a related entity. In two types of mixed POH, ectomesenchymal elements appeared juxtaposed to the squamous lining (gingival cyst-like organoid hamartoma) and ghost cells aggregated in the enamel organ of a microdont (peripheral odontoma). None of POH exhibited continuity with the surface epithelium. CONCLUSION: On the basis of this relatively limited series of cases, POH, to conceptualize a unified histogenetic source, are speculated to arise from the soft-tissue remnants of dental lamina. Gingival rests of Serres seem to retain the ability to pursue epithelial-ectomesenchymal interactions that are necessary leading to odontoma formation.     

Immunohistochemical demonstration of bone morphogenetic protein in odontogenic tumors

The aim of the present study was to describe the expression and distribution of bone morphogenetic protein (BMP) in odontogenic tumors by immunohistochemistry using monoclonal antibody against bovine BMP (BMPMcAb). Eight types of odontogenic tumors (44 cases), including ameloblastoma (20 cases), cementifying fibroma (8 cases), benign cementoblastoma (5 cases), dentinoma (3 cases), compound odontoma (2 cases), adenomatoid odontogenic tumor (2 cases), calcifying epithelial odontogenic tumor (2 cases) and odontogenic fibroma (2 cases), were studied. The results showed that, according to the immunostaining pattern of BMPMcAb, tumors could be classified into two types: all cementifying fibro-mas, benign cementoblastomas. Dentinomas, odontogenic fibromas. and compound odontomas demonstrated a positive reaction, whereas all ameloblastomas, adenomatoid odontogenic tumors, and calcifying epithelial odontogenic tumors were negative. BMPMcAb-positive odontogenic tumors were those tumors with formation of enamel, dentin, cementum or bone. Therefore. BMP might play an important role in the formation of calcified dental tissues and the development of odontogenic tumors contaning such tissues.     

Relative frequency of peripheral odontogenic tumors: a study of 45 new cases and comparison with studies from the literature

BACKGROUND: Peripheral (extraosseous) odontogenic tumors are rare, and reports in the literature have mainly been single case reports or a small series of cases. The aim of this study was to determine the relative frequency of peripheral (extraosseous) odontogenic tumors relative to one another and relative to their central (intraosseous) counterparts in an oral pathology biopsy service and to compare these data with information available in the literature. METHODS: The files of the Pacific Oral and Maxillofacial Pathology Laboratory of the University of the Pacific, San Francisco, CA, USA, served as the source of material for this study. Files were systematically searched for all cases of peripheral odontogenic tumors (POTs) during a 20-year-period. RESULTS: There were 91,178 cases accessed in which central and POTs were identified in 1,133 (1.24%), central tumors in 1,088 (1.2%), and peripheral tumors in 45 (0.05%). Peripheral tumors accounted for 4% of all 1133 central and POTs. Peripheral odontogenic fibroma (PODF) was the most common of the 45 POTs accounting for 51.1% (23 cases) followed by peripheral ameloblastoma (PA) 28.9% (13 cases) and peripheral calcifying cystic odontogenic tumor (PCCOT) 13.3% (six cases). Peripheral calcifying epithelial odontogenic tumor, peripheral ameloblastic fibroma, and peripheral ameloblastic carcinoma were also identified--each comprised 2.2% (one case each). PODF was more common than its central counterpart by a 1.4:1 ratio. This was the only peripheral tumor that was more common than its central counterpart. PA accounted for 9.3% of all ameloblastomas and PCCOT for 26% of all calcifying cystic odontogenic tumors. CONCLUSION: There is only scarce information in the literature on the relative frequency of POTs. Additional studies should be conducted to determine the true relative frequency. To ensure accuracy, pathologists with experience in the field of odontogenic tumors should conduct these studies. Intraosseous tumors that perforate through the bone to the gingival tissue, clinically presenting as 'peripheral tumors' should be excluded.     

Expression of odontogenic ameloblast-associated protein, amelotin, ameloblastin, and amelogenin in odontogenic tumors: immunohistochemical analysis and pathogenetic considerations

Screening for expression of amelogenesis-related proteins represents a powerful molecular approach to characterize odontogenic tumors and investigate their pathogenesis. In this study, we have examined the presence and distribution of odontogenic ameloblast-associated protein (ODAM), amelotin (AMTN), ameloblastin (AMBN), and amelogenin (AMEL) by immunohistochemistry in samples of adenomatoid odontogenic tumor (AOT), calcifying epithelial odontogenic tumor (CEOT), developing odontoma, ameloblastoma, calcifying cystic odontogenic tumor (CCOT), ameloblastic fibroma (AF), myxoma, odontogenic fibroma (OF), and reduced enamel epithelia (REE). Positive results were obtained in those tumors with epithelial component, except for AF, OF, and ameloblastoma. ODAM was found around mineralized structures (dystrophic calcifications) and CEOT's amyloid, whereas AMTN stained the eosinophilic material of AOTs. The CCOT transitory cells to ghost cells were strongly positive with all proteins except AMEL, and the REE as well as odontomas showed immunoexpression for ODAM, AMTN, AMBN, and AMEL similar to those found in normal rat tooth germs. Based on these results, some histopathogenetic theories were formulated.     

Odontogenic tumours in children and adolescents: a study of 78 Nigerian cases.

BACKGROUND: There is paucity of literature on odontogenic tumours in children and adolescents. Available records are difficult to compare due to differences in study criteria. To contribute to the records, a 20-year study of odontogenic tumours on the basis of the WHO classification (Kramer et al., 1992) in Nigerian African children and adolescents < or =18 years of age was undertaken. MATERIAL: A retrospective survey of oral/jaw tumours and allied lesions in children and adolescents < or =18 years of age seen at the Maxillofacial Unit, Ahmadu Bello University Teaching Hospital, Kaduna, Nigeria between 1979 and 1998. Data collected were histopathologic type, age, clinical features, radiologic appearance, treatment and record of recurrence. METHOD: Odontogenic tumours selected using the WHO classification were used for further study. Data were collected from case notes, radiographs, histopathologic reports and follow-up records. Information retrieved was used to complete a questionnaire and subjected to analysis. RESULTS: Two hundred and fifty-two (252) subjects < or =18 years were recorded, from which 78 (31%) had odontogenic tumours. Among seven types of odontogenic tumours seen, ameloblastoma (54%), odontogenic myxoma (19%) and adenomatoid odontogenic tumour (9%) were predominant. All patients seen were from 6 to 18 years with more than half (53%) between 15 and 18 years of age. A patient with multiple, bilateral odontomas of the maxilla and mandible resembling Herrmann's syndrome was recorded. Seventy-three patients were treated using enucleation (37%), dentoalveolar resection with preservation of lower border (15%) and segmental resection (48%). Five patients absconded after tumour diagnosis. No tumour recurrence was recorded in 65 treated cases followed-up for between 2 months and 10 years. CONCLUSION: This report shows that while ameloblastoma was the predominant odontogenic tumour, its frequency in Nigerian African children was lower than in the adult population. A case resembling Herrmann's syndrome is also presented.     

Odontogenic tumours, a collaborative retrospective study of 75 cases covering more than 25 years from Estonia.

INTRODUCTION: The aim of the present collaborative study was to analyse retrospectively the character of odontogenic tumours in Estonia, involving the entire Estonian population (1.4 million), and to compare their prevalence with the figures presented in similar reports from other countries. MATERIAL AND METHODS: All material for the retrospective study was retrieved from the files of the Departments of Maxillofacial Surgery in Tartu and Tallinn, Estonia, where all in/out-patients are treated from the whole country. The final diagnosis in each case of odontogenic tumour was based on the 1992 WHO histological criteria. RESULTS: A total of 75 odontogenic tumours was found, 74 (98.6%) of which were benign, and 1 (1.3%) was malignant. The frequency of odontogenic tumours in this study was the lowest ever reported. The most common tumours were odontoma (34.3%), followed by ameloblastoma with different subtypes (25.3%), ameloblastic fibroma (16%), odontogenic myxoma (12%) and benign cementoblastoma (8%). CONCLUSION: Odontogenic tumours are relatively rare in Estonia compared with the data from other countries.     

The peripheral odontogenic fibroma: an attempt at clarification

Two different lesions of the gingiva that have been referred to previously as peripheral odontogenic fibromas are discussed. The first of these is the rare extraosseous counterpart of the central odontogenic fibroma (WHO type)1 and is therefore referred to in this article as the peripheral odontogenic fibroma (WHO type). It is probably treated adequately by simple excision, but a study of its biologic behavior is lacking. The second lesion is reactive, is common, and has a marked tendency to recur. It has been known by numerous synonyms, including calcifying fibrous epulis and peripheral ossifying fibroma, as well as peripheral odontogenic fibroma. The term peripheral ossifying fibroma should be retained for this lesion to avoid confusion with the peripheral odontogenic fibroma (WHO type).     

同源盒基因HOXC13在牙源性肿瘤中的表达

目的:检测同源盒基因HOXC13 mRNA在牙源性肿瘤中的表达,探讨其发生的意义。方法:采用原位杂交法检测47例成釉细胞瘤(ameloblastoma,AB)(原发29例,复发14例,恶变4例)、3例牙源性钙化囊性瘤(CCOT)、3例成釉细胞纤维瘤(AF)、2例牙源性钙化上皮瘤(CEOT)、10例牙源性角化囊性瘤(KCOT)的HOXC13 mRNA水平,同时选取7例正常口腔黏膜上皮作为对照。采用SPSS10.0软件包对数据进行χ2检验。结果:HOXC13 mRNA在AB中的阳性率为97.9%(46/47),CCOT中为100%(3/3),CEOT中为100%(2/2),KCOT上皮中为70.0%(7/10),正常口腔黏膜细胞中为42.9%(3/7),AB、KCOT、正常黏膜3组间差异显著(Ρ=0.001),但角化及颗粒样变退化细胞却为阴性。3例AF均为阴性。结论:牙源性肿瘤的发生、发展与HOXC13的高表达有关,且受其调控。HOXC13 mRNA在牙源性病损上皮中表达有异质性,该基因可促进上皮增殖,阻抑成釉细胞的终末分化,其丢失可导致上皮细胞角化和退变。     

Rare peripheral odontogenic tumors: report of 5 cases and comprehensive review of the literature

Peripheral odontogenic tumor (POT) is a rarely encountered lesion. We report 5 cases of POT including adenomatoid odontogenic tumor (AOT), keratocystic odontogenic tumor (KCOT), ameloblastic fibroma (AF), developing odontoma (DO), and calcifying cystic odontogenic tumor (CCOT), and also provide a review of relevant literature to define the tumor profile. Except for PCCOT with enough frequency (>100 cases), PAOT (n = 14), PKCOT (n = 15), PAF (n = 5), and PDO (n = 7) were scarce in the literature. As to the age distribution, PAOT, PAF, and PDO fell within the first 2 decades, whereas PKCOT arose in middle-aged adults. A marked female predominance was apparent in PAOT, PKCOT, and PAF. Approximately 90% of PAOT occurred in the maxilla. PAOT and PDO arose primarily in the incisor area, and PKCOT and PAF were typically located in permanent canine/premolar and deciduous molar regions, respectively. Although most PAOT and all PKCOT affected the buccal gingiva, PDO showed a strong predilection for the lingual aspect. With the exception of PKCOT, there was no propensity for recurrence in the above POT. At this time, it remains to be determined whether the biologic behavior of PKCOT is the same as for KCOT. In view of the reported cases, a true extraosseous origin of PAOT and PAF, for the most part, is challenging.     

Adenomatoid odontogenic tumour: A case report

Adenomatoid odontogenic tumour (AOT) is a rare benign odontogenic tumour characterized by a progressively slow growing pattern and symptomless behavior. The differential diagnosis between AOT and other odontogenic tumours, such as ameloblastoma, should be well conducted in order to avoid extensive ablative surgery. This report presents an unusual case of an 11-year-old male patient who referred to the oral surgeon due to a significant painless gingival swelling in the anterior mandible. A panoramic X-ray revealed a round radiolucid image of an intraosseous lesion with well defined boards and related to the left lateral incisor and left canine. The Computerized Tomography was performed and the sagittal sections revealed a tooth image in contact with the inferior board of the tumour. Additionally, the coronal sections showed the presence of a tooth inside the lesion. Several calcifying nodules could be distinguished within the cystic area. The clinical diagnostic hypothesis was of calcifying epithelium odontogenic tumour but the histological sections were consistent with AOT. The tumour was enucleated under local anesthesia. After one year follow-up there were no signs of reoccurrence. With respect to the distinguishing tumour enlargement and localization in the lower jaw, the reported case is an uncommon example of AOT.     

Relative frequency of central odontogenic tumors: a study of 1,088 cases from Northern California and comparison to studies from other parts of the world.

PURPOSE: To determine the relative frequency of central odontogenic tumors in relation to all biopsy specimens and to one another in an oral pathology biopsy service and to compare the data with previous studies from different parts of the world. METHODS: Files from the Pacific Oral Pathology Laboratory of the University of the Pacific, San Francisco, CA served as a source of material for this study. Files were systematically searched for all cases of central (intraosseous) odontogenic tumors during a 20-year period. RESULTS: Central odontogenic tumors were identified in 1,088 (1.2%) cases out of the 91,178 accessed. Individually, of all odontogenic tumors, 75.9% were odontomas. The prevalence of the remaining tumors appears to be a rare occurrence. The second most common was ameloblastoma (11.7%), followed by odontogenic myxoma (2.2%). Odontomas are considered hamartomas or developmental anomalies. When excluded from the list of individual odontogenic tumors, ameloblastoma is the most common (48.5%), followed by odontogenic myxoma (9.2%), adenomatoid odontogenic tumor (7.3%), ameloblastic fibro-odontoma (7.3%), ameloblastic fibroma (6.5%), calcifying odontogenic cyst (6.5%), and odontogenic fibroma (6.1%). Each remaining tumor comprises less than 4%. CONCLUSIONS: Studies related to the relative frequency of individual odontogenic tumors from different parts of the world are difficult to compare because most studies are outdated, the list of tumors is limited, and new entities are not included. To determine the real relative frequency, further studies should be conducted, especially in Western societies, by experienced pathologists in the field of odontogenic tumors.     

Peripheral epithelial odontogenic tumors: a review

Peripheral (extraosseous or soft tissue) odontogenic tumors are rare lesions that occur in the soft tissue overlying the tooth-bearing areas of the mandible and the maxilla. A review of the English-language literature revealed only 48 well-documented cases of peripheral epithelial odontogenic tumors. Thirty-two were peripheral ameloblastomas; six were peripheral adenomatoid odontogenic tumors; nine were peripheral calcifying epithelial odontogenic tumors; and one was a peripheral squamous odontogenic tumor. An additional four cases were reported as peripheral ameloblastomas in extragingival locations, but their odontogenic origin is debatable. Although the peripheral ameloblastoma is histologically similar to its central counterpart, it differs in its clinical features and biologic behavior. It does not exhibit an aggressive, destructive behavior and does not invade the underlying bone. Conservative excision of the tumor with minimal but adequate margins is the treatment of choice and recurrences are uncommon. This benign biologic behavior appears to be true also for lesions diagnosed as peripheral calcifying epithelial odontogenic tumors and undoubtedly is true for the peripheral adenomatoid odontogenic tumors.     

Ameloblastic fibroma: growth potentiality of odontogenic epithelium and coexpression of intermediate filament proteins in fibromatous cells

Four cases of ameloblastic fibroma are described immunohistochemically in terms of intermediate-sized proteins in both epithelial and mesodermal components. Keratin proteins were demonstrated by polyclonal anti-keratin antiserum (TK: detecting 41-65 kDa keratins) and 2 monoclonal antibodies to keratin (KL1: 55-57 kDa, PKK1: 44, 46, 52 and 54 kDa), and monoclonal antibodies to vimentin and desmin. Two types of odontogenic epithelial tumour cells were discriminated: undifferentiated odontogenic cells and common ameloblastoma cells. Keratin expression was found to be stronger in undifferentiated cells than in the ameloblastoma cells. Undifferentiated cells were PAS-positive, while ameloblastoma cells were negative. Fibroma cells were strongly positive for vimentin, and negative for desmin. Keratin proteins were also expressed slightly. Thus, coexpression of keratin and vimentin was seen in fibroma cells. Histogenesis is discussed from the standpoint of the distribution patterns of keratin and vimentin, as well as with respect to the histopathology.     

颌骨肿瘤中骨形成蛋白-2基因扩增的RT-PCR研究

目的探讨颌骨肿瘤中骨形成蛋白(BMP)-2基因的扩增与颌骨肿瘤的发生、发展及生物学行为之间的关系。方法采用反转录-聚合酶链式反应(RT-PCR)技术检测9类87例新鲜颌骨肿瘤标本中BMP-2基因扩增结果。结果含肿瘤性硬组织的全部42例颌骨肿瘤(骨肉瘤、软骨肉瘤、牙源性纤维瘤、骨化性纤维瘤、化牙骨质纤维瘤)和部分颌骨成釉细胞瘤(27/31)均显示人BMP-2特异性扩增条带,而不含肿瘤性硬组织的其他颌骨肿瘤(牙源性钙化上皮瘤、纤维肉瘤、脂肪肉瘤)均未见特异性扩增条带。结论颌骨肿瘤中BMP-2基因扩增存在差异,BMP-2基因扩增与肿瘤性硬组织的形成有关,与某些颌骨肿瘤的发生、发展及生物学行为之间存在一定关系。     

Ameloblastic fibroma: growth potentiality of odontogenic epithelium and coexpression of intermediate filament proteins in fibromatous cells

Four cases of ameloblastic fibroma are described immunohistochemically in terms of intermediate-sized proteins in both epithelial and mesodermal components. Keratin proteins were demonstrated by polyclonal anti-keratin antiserum (TK: detecting 41–65 kDa keratins) and 2 monoclonal antibodies to keratin (KL1: 55–57 kDa, PKK1: 44, 46, 52 and 54 kDa), and monoclonal antibodies to vimentin and desmin. Two types of odontogenic epithelial tumour cells were discriminated: undifferentiated odontogenic cells and common ameloblastoma cells. Keratin expression was found to be stronger in undifferentiated cells than in the ameloblastoma cells. Undifferentiated cells were PAS–positive, while ameloblastoma cells were negative. Fibroma cells were strongly positive for vimentin, and negative for desmin. Keratin proteins were also expressed slightly. Thus, coexpression of keratin and vimentin was seen in fibroma cells. Histogenesis is discussed from the standpoint of the distribution patterns of keratin and vimentin, as well as with respect to the histopathology.     

Calcifying odontogenic cyst with ameloblastic fibroma: report of three cases

Although it is a rare event, odontogenic tumors such as ameloblastoma, ameloblastic fibroma (AF), ameloblastic fibro-odontoma, and odontoma have been reported associated with calcifying odontogenic cyst (COC). There are only four cases of COC with AF cited in the English literature. However, three of these four cases were either included in a review of a series of cases or reported as an abstract, and limited clinical and histological information was provided. We present three additional cases of COC with AF and discuss the management for this combined lesion. Because COC is known for its histologic diversity and variable clinical behavior, and the clinical significance of an association of COC with AF is still unknown, we think it is valuable to report COC with AF with detailed clinical and pathological documentation.