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Keratoameloblastoma: a case report and a review of the literature on its radiologic features
Accepted Manuscript Keratoameloblastoma: a case report and a review of the literature on its radiological features Chena Lee, DDS, Byoung-Jun Park, DDS, Won-Jin Yi, PhD, Professor, Min-Suk Heo, DDS, PhD, Professor, Sam-Sun Lee, DDS, PhD, Professor, Kyung-Hoe Huh, DDS, PhD, Associate professor PII:
S2212-4403(15)01087-1
DOI:
10.1016/j.oooo.2015.07.008
Reference:
OOOO 1251
To appear in:
Oral Surgery, Oral Medicine, Oral Pathology and Oral Radiology
Received Date: 7 February 2015 Revised Date:
12 June 2015
Accepted Date: 8 July 2015
Please cite this article as: Lee C, Park B-J, Yi W-J, Heo M-S, Lee S-S, Huh K-H, Keratoameloblastoma: a case report and a review of the literature on its radiological features, Oral Surgery, Oral Medicine, Oral Pathology and Oral Radiology (2015), doi: 10.1016/j.oooo.2015.07.008. This is a PDF file of an unedited manuscript that has been accepted for publication. As a service to our customers we are providing this early version of the manuscript. The manuscript will undergo copyediting, typesetting, and review of the resulting proof before it is published in its final form. Please note that during the production process errors may be discovered which could affect the content, and all legal disclaimers that apply to the journal pertain.
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Keratoameloblastoma: a case report and a review of the<br />
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literature on its radiological features<br />
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Chena Lee, Byoung-Jun Park, Won-Jin Yi, Sam-Sun Lee, Kyung-Hoe Huh<br />
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Chena Lee<br />
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DDS, Department of Oral and Maxillofacial Radiology and Dental Research Institute, School of Dentistry, Seoul National University, Seoul, Republic of Korea Byoung-Jun Park<br />
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DDS, Department of Oral and Maxillofacial Radiology and Dental Research Institute, School of Dentistry, Seoul National University, Seoul, Republic of Korea<br />
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Won-Jin Yi<br />
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PhD, Professor, Department of Oral and Maxillofacial Radiology and Dental Research Institute, School of Dentistry, Seoul National University, Seoul, Republic of Korea<br />
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Min-Suk Heo<br />
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DDS, PhD, Professor, Department of Oral and Maxillofacial Radiology and Dental Research<br />
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Institute, School of Dentistry, Seoul National University, Seoul, Republic of Korea Sam-Sun Lee<br />
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DDS, PhD, Professor, Department of Oral and Maxillofacial Radiology and Dental Research Institute, School of Dentistry, Seoul National University, Seoul, Republic of Korea Kyung-Hoe Huh DDS, PhD, Associate professor, Department of Oral and Maxillofacial Radiology and Dental Research Institute, School of Dentistry, Seoul National University, Seoul, Republic of Korea<br />
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Corresponding author: Kyung-Hoe Huh Department of Oral and Maxillofacial Radiology, School of Dentistry, Seoul National University, 101 Daehak-ro, Jongno-gu, Seoul 110-768, Korea.<br />
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E-mail: future3@snu.ac.kr Tel) 82-2-2072-4713,<br />
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Word count for the abstract: 103 words<br />
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Fax) 82-2-744-3919<br />
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Word count for the manuscript: 2615 words Number of tables: 1<br />
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Number of figures: 6<br />
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<Abstract rel="nofollow"><br />
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Keratoameloblastoma is a rare subtype of ameloblastoma. It tends to have a poor prognosis, and therefore a careful diagnosis based on imaging is important in planning appropriate<br />
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surgical treatment for this distinctive type of ameloblastoma. A unique feature of keratoameloblastoma is atypical calcification inside of the mass, such as a ground glass appearance, internal calcification, or a mixed radiolucent and radiopaque pattern. Due to its poor prognosis and the likelihood of frequent recurrences, surgical resection is considered the<br />
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treatment of choice. This study reports a new case of keratoameloblastoma with special emphasis on its radiological features and reviews previously reported cases of<br />
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keratoameloblastoma.<br />
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<Main Text> I. Introduction Ameloblastoma occurs in various forms, and is classified into solid/multicystic, unicystic,<br />
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desmoplastic, and peripheral types. Solid/multicystic ameloblastoma is the second most common odontogenic tumor according to a 2005 World Health Organization (WHO) report.1 Solid/multicystic ameloblastoma is classified into the following histological subtypes: follicular, plexiform, basal cell, granular cell, and acanthomatous ameloblastoma.1 In addition, there is another rare subtype known as keratoameloblastoma, which<br />
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characteristically exhibits varying degrees of keratinization.2<br />
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Among these subtypes, acanthomatous ameloblastoma and keratoameloblastoma are<br />
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associated with keratinization. Acanthomatous ameloblastoma is defined by extensive squamous metaplasia of loosely arranged central cells and is often associated with keratin formation.3 In contrast, keratoameloblastoma involves the presence of keratin pearls within epithelial follicles scattered throughout fibrous connective tissue.4 We report a rare case of keratoameloblastoma and review the previously reported cases, with<br />
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a special emphasis on the radiological features and prognosis of each case.<br />
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II. Case Report A 56-year-old male presented with a complaint of recurrent swelling on his right palate. He had a history of two consecutive surgeries due to ameloblastoma over the last two years in another hospital. An expansile, corticated radiolucent lesion had been present in the right<br />
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maxilla (Figure 1A). Although calcified material within the lesion was not apparent on a panoramic radiograph, computed tomography (CT) images acquired with Genesis HiSpeed (GE Healthcare, Milwaukee, WI) revealed high attenuated material around the root apices of the right maxillary molars (Figure 1B, 1C). The lesion occupied most of the right maxillary<br />
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sinus with apparent buccal expansion but no involvement of infra-orbital foramen or pterygopalatine fossa. The patient had undergone enucleation surgery. After three months, the lesion recurred and was removed again after three more months of observation. However, 11<br />
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months after the second surgery, he recognized a recurrence of the swelling and decided to transfer to our hospital.<br />
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In our hospital, a physical examination revealed that the patient had a palatal swelling with intact overlying mucosa. A panoramic radiograph showed an ovoid, relatively well-defined radiolucent lesion with no distinct corticated rim, associated with roots of the right maxillary<br />
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molars (Figure 2A). Contrast-enhanced CT images acquired with Somatom Sensation 10 (Siemens, Erlangen, Germany) displayed a mixed low-and highly-attenuated lesion, including a large area of homogeneous calcified material. The original palatal cortex was perforated and a palatal extension of the calcified lesion was identified (Figure 2B, 2C). Based on a<br />
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preliminary diagnosis of recurrent ameloblastoma, the patient underwent enucleation surgery and the right maxillary molars were extracted. A histopathological examination revealed a<br />
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solid unencapsulated lesion composed of numerous follicles of odontogenic epithelium filled with central keratin plugs (Figure 3A). The basal epithelium displayed cuboidal to columnar cells with hyperchromatic nuclei (Figure 3B). The final histopathological diagnosis was keratoameloblastoma, with a varying level of keratinization. It was later confirmed that this pattern of massive keratin pearls was also found on the histopathology of the initial lesion in the previous hospital. Follow-up CT images taken four months after the preceding operation revealed a small ovoid lesion composed of calcified material on the anterolateral wall of the right maxillary sinus. This lesion of highly attenuated foci gradually increased in size during the 10-month followup period (Figure 4). The patient therefore underwent enucleation surgery again.<br />
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ACCEPTED MANUSCRIPT Two months after the second surgery in our hospital, another lesion of homogeneous calcified material, showing a ground glass appearance, was recognized on his right maxillary tuberosity on a follow-up CT image. This recurrent mass, close to the posterior surgical margin of the prior tumor mass, kept growing during nine months of observation. The mass with a safety margin of surrounding bone was resected.<br />
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However, four months after the third surgery in our hospital, a follow-up CT image revealed multifocal recurrences on the resection margins of the right maxilla. A magnetic resonance imaging (MRI) examination performed eight months after the third surgery in our hospital showed that these lesions of fine calcified materials had increased in size. These lesions had a<br />
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hypointense T2 signal intensity with minimal enhancement (Figure 5). Mass resection with a right partial maxillectomy was conducted. Nevertheless, 19 months after the fourth surgery in<br />
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our hospital, multifocal recurrent lesions were found on the surgical resection margins of the right residual maxilla, zygoma, orbital floor, and pterygoid plate (Figure 6). These masses were removed with a sufficient safety margin, and the right orbital floor was reconstructed with titanium mesh. The clinicoradiological follow-up has so far been uneventful for 10<br />
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months.<br />
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III. Discussion<br />
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Keratoameloblastoma is a rare head and neck tumor that can be distinguished from other subtypes of ameloblastoma by its distinctive imaging features. However, few cases of this<br />
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pathosis have been reported. Reviews and reports in the extant English-language literature discuss only 11 cases.4-12 Prior reports have focused on the histological features of keratoameloblastoma, while little attention has so far been paid to its radiological features. This unique lesion was first recognized in 1958.7,<br />
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Subsequently, controversy emerged<br />
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regarding the classification of keratoameloblastoma as a subtype of ameloblastoma rather<br />
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than as a subtype of acanthomatous ameloblastoma. Norval et al. suggested the term variant of acanthomatous ameloblastoma instead of keratoameloblastoma.13 According to Kramer et al., keratoameloblastoma and papilliferous keratoameloblastoma are unique in that they show massive keratinization.14 Most subtypes of ameloblastoma show either minimal or no keratinization. Even though the acanthomatous type is often associated with keratinization, this is not the pathognomonic feature of this type of ameloblastoma.<br />
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Rather, acanthomatous ameloblastoma is characterized by extensive squamous metaplasia.14 Siar et al. also pointed out that keratinization and acanthomatous changes in ameloblastoma occur with different frequencies; keratinization is rare, whereas acanthomatous changes are<br />
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relatively common.10<br />
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Papilliferous keratoameloblastoma and keratoameloblastoma were defined as separate entities in 1970 by Pindborg et al.2 The histological features of papilliferous<br />
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keratoameloblastoma, characterized by papillary keratin lining, are different from those of keratoameloblastoma.15 Therefore,<br />
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current<br />
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review,<br />
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cases<br />
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of<br />
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keratoameloblastoma were excluded. Since keratoameloblastoma is so rare, there is no specific description in the WHO classification. It is only mentioned briefly as one of the differential diagnoses of primary intraosseous squamous cell carcinoma derived from a keratocystic odontogenic tumour.<br />
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However, in 2007, Whitt et al. pointed out that keratoameloblastoma was only omitted by WHO in their 2005 report because of the limited information available about this condition.10 Even though it is rare, cases of keratoameloblastoma have been consistently reported since its<br />
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ACCEPTED MANUSCRIPT initial recognition. The current study is significant in that it provides a detailed description of the radiological features of this subtype of ameloblastoma. A clinical overview of previous cases revealed that keratoameloblastoma has been diagnosed in patients between 26 and 76 years of age, with a mean age of 42.25 years. Keratoameloblastoma occurs in both males and females without any significant difference in<br />
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frequency. The lesion occurred in the mandible (eight cases) more frequently than in the maxilla (four cases). Of the 12 cases, five cases, including the present case, were tracked to assess the prognosis. Of the cases in which follow-up results were reported, one case recurred after seven months, while the present case recurred six times over seven years and four<br />
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months. Leaving aside three reports that did not discuss the surgical method employed, resection was performed in five cases and four cases involved enucleation. Two of the four<br />
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enucleated lesions showed recurrence. The clinical and radiological findings are summarized in Table 1.<br />
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In the English-language literature on keratoameloblastoma, only four studies presented radiographic images, and one of these studies provided advanced CT images showing a threedimensional analysis of the lesion. Most authors focused on a histological description rather than radiological features. However, the precise diagnosis of keratoameloblastoma using<br />
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radiological findings may be important since keratoameloblastoma has a tendency to recur frequently. Thus, the current study presents detailed radiographic findings in order to facilitate the differential diagnosis of keratoameloblastoma. In general, the solid type of ameloblastoma may present as a unilocular or multilocular<br />
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radiolucent lesion on radiographic images. It may also show scalloping borders. Displacement of the involved tooth can be observed and root resorption of the adjacent tooth<br />
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is a common feature.1 Similarly, when reviewing previous cases, keratoameloblastoma typically presented as a multilocular radiolucency with septa and a scalloping margin. The lesion commonly showed expansion and perforation of the cortical plates.1, 10-12 The most characteristic feature of keratoameloblastoma was scattered hard tissue attenuation within the mass. Four cases, including the present case, had radiopaque materials in the lesion.10-12 In one study, the whole mass was described as having a ground glass appearance, while other studies showed irregular calcifications on the background of soft tissue attenuation. The present case showed heterogeneous high attenuation foci intermixed with areas of a ground glass appearance. This radiopaque material was consistently observed from the initial to all the recurrent lesions.<br />
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ACCEPTED MANUSCRIPT This pattern of radiopacity is a characteristic radiological feature of keratoameloblastoma. It is not common for other types of ameloblastoma to show calcified materials within the lesion. Moreover, the origin and composition of these hard tissue materials are different from those found in conventional ameloblastoma. The high-density materials in other subtypes of conventional ameloblastoma are thought to be dentin or dentinoid material.16, 17<br />
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Two possible explanations exist for the dense materials that occur in keratoameloblastoma. One possibility is pathological ossification, which is the conversion of accumulated keratin and necrotic cells from dystrophic calcifications into a hard tissue substance.4,13 The other possibility, demonstrated in the present case, is that the radiopacity is best described as<br />
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extensive keratinization based on microscopic examination. Whether the radiopacity reflects extensive keratin or hard tissue material developed from accumulated keratin, it is a<br />
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distinctive radiological feature of keratoameloblastoma. The present report suggests that keratoameloblastoma should be considered in the differential diagnosis of benign tumors containing radiopaque materials.<br />
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However, radiopacity may be confusing for clinicians and radiologists, because it radiographically resembles the normal woven bone pattern found in the postoperative healing process. During the follow-up period after the surgery, special care should be taken to ensure<br />
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that a recurrent lesion is not confused with the postoperative bone healing process. Five cases were treated with resection of the tumor mass. Of the four cases treated with enucleation surgery, there was information about the prognosis in three cases. Recurrence occurred in two patients, including the present case, and no recurrence occurred in one case.<br />
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The first recurrence occurred seven and three months post-operatively, respectively. The present case recurred six times over seven years and four months. Due to the frequent<br />
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recurrence of the lesion, the patient underwent CT and/or MR images at intervals of two to four months during the entire follow-up period. The patient underwent four enucleation and three resection surgeries. The recurrent sites were mostly close to the margins of the previous lesions. In microscopic imaging, the lesions were not encapsulated well; thus, infiltration into surrounding bone can be assumed. The micro-invasiveness of keratin material and tumor cells into surrounding tissues may cause recurrent lesions. Although there have not been enough clinically described cases of keratoameloblastoma to suggest a generalized proper treatment protocol, we recommend resection with a sufficient safety margin in order to ensure the best prognosis possible. Further studies are needed on the micro-infiltrative character and the prognosis of keratoameloblastoma.<br />
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IV. References<br />
<br />
[1]<br />
<br />
Barnes L, Eveson JW, Reichart P, Sidransky D. Pathology and Genetics of Head and<br />
<br />
Neck Tumours. Lyon: IARCPress: 2005: 290-300. Pindborg JJ. Pathology of the dental hard tissues. Philadelphia: Saunders: 1970.<br />
<br />
[3]<br />
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Neville BW, Waldron CA, Herscharaft EA. Oral and Maxillofacial Pathology. Third<br />
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[2]<br />
<br />
ed. Philadelphia; Saunders: 1995:702-710. [4]<br />
<br />
Takeda Y, Satoh M, Nakamura S, Ohya T. Keratoameloblastoma with unique<br />
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<br />
histological architecture: an undescribed variation of ameloblastoma. Virchows Archiv. 2001;439:593-596.<br />
<br />
Adeyemi B, Adisa A, Fasola A, Akang E. Keratoameloblastoma of the mandible. J<br />
<br />
Oral Maxillofac Pathol. 2010;14:77-79. [6]<br />
<br />
Kaku<br />
<br />
T,<br />
<br />
Ohuchi<br />
<br />
T,<br />
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<br />
[5]<br />
<br />
Hattori<br />
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Y,<br />
<br />
Nishimura<br />
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M,<br />
<br />
Nakade<br />
<br />
O,<br />
<br />
Abiko<br />
<br />
Y.<br />
<br />
Keratoameloblastoma of the mandible. J Oral Patho Med. 2000;29:350. [7]<br />
<br />
Pindborg JJ, Weinmann JP. Squamous cell metaplasia with calcification in<br />
<br />
ameloblastomas. Acta Pathol Microbiol Scand. 1958;44:247-252.<br />
<br />
Raubenheimer EJ, van Heerden WF, Noffke CE. Infrequent clinicopathological<br />
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[8]<br />
<br />
findings in 108 ameloblastomas. J Oral Pathol Med. 1995;24:227-232. [9]<br />
<br />
Said-al-Naief NA, Lumerman H, Ramer M, Kopp W, Kringstein GJ, Persenchino F.<br />
<br />
Keratoameloblastoma of the maxilla: A case report and review of the literature. Oral Surg<br />
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[10]<br />
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Oral Med Oral Pathol Oral Radiol Endod. 1997;84:535-539. Siar CH, Ng KH. 'Combined ameloblastoma and odontogenic keratocyst' or<br />
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[11]<br />
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'keratinising ameloblastoma'. Br J Oral Maxillofac Surg. 1993;31:183-6. Sisto JM, Olsen GG. Keratoameloblastoma: complex histologic variant of<br />
<br />
ameloblastoma. J Oral Maxillofac Surg. 2012;70:860-864. [12]<br />
<br />
Whitt JC, Dunlap CL, Sheets JL, Thompson ML. Keratoameloblastoma: a tumor sui<br />
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generis or a chimera? Oral Surg Oral Med Oral Pathol Oral Radiol Endod. 2007;104:368-76. [13]<br />
<br />
Norval EJ, Thompson IO, van Wyk CW. An unusual variant of keratoameloblastoma.<br />
<br />
J Oral Pathol Med. 1994;23:465-467. [14]<br />
<br />
Kramer IR, Pindborg JJ, Shear M. Histological typing of odontogenic tumours.<br />
<br />
Second ed. Berlin; New Yor; Springer: 1992. [15]<br />
<br />
Mohanty N, Rastogi V, Misra SR, Mohanty S. Papilliferous keratoameloblastoma: an<br />
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ACCEPTED MANUSCRIPT extremely rare case report. Case Rep Dent. 2013;2013:1-4. [16]<br />
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Slabbert H, Altini M, Crooks J, Uys P. Ameloblastoma with Dentinoid Induction -<br />
<br />
Dentinoameloblastoma. J Oral Pathol Med. 1992;21:46-8. [17]<br />
<br />
Hazarey KJ, Daftary DK. An unusual epithelial odontogenic tu. J Oral Maxillofac<br />
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Pathol. 2002;1:16-21.<br />
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<Figure Legends><br />
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Figure 1. (A) The first panoramic radiograph taken at the previous hospital shows a corticated<br />
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radiolucent lesion surrounding the impacted third molar and occupying the right maxillary sinus. (B) Axial and coronal bone window CT images (120 kV, 210 mAs, pixel spacing = 0.33 mm, slice<br />
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thickness = 5 mm) of the initial lesion reveal an expansion of the buccal cortex of the right maxilla (white arrows). Note amorphous high-attenuated material around the root apices of the right<br />
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maxillary molars (black arrow). The size of the initial lesion at its largest area was 45x41 mm2 and<br />
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attenuated portion of the lesion.<br />
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the mean CT numbers were 663 ± 83.49 for the calcified portion and 37.12 ± 13.35 for the low<br />
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Figure 2. (A) A panoramic radiograph taken at the first visit to our hospital shows an ovoid, relatively well-defined radiolucency with no distinct corticated margin on the right posterior<br />
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maxilla (black arrows). An axial CT image (120 kV, 150 mAs, pixel spacing = 0.39 mm, slice thickness = 2 mm) with bone window (B) and soft tissue window (C) reveals fine<br />
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calcifications perforating the palatal cortex (white arrows).<br />
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Figure3. (A) A solid unencapsulated mass with extensive follicles of odontogenic epithelium filled with a central keratin plug (hematoxylin and eosin stain [H&E], original magnification 12x). (B) Scattered microcysts filled with keratin material within a dense collagenous connective tissue stroma (H&E, original magnification 200x).<br />
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Figure 4. Axial CT images (120 kV, 150 mAs, pixel spacing = 0.39 mm, slice thickness= 2 mm) with bone window (A) and soft tissue window (B), taken 10 months after the first surgery in our hospital, demonstrate the recurrent lesion in the anterolateral wall of the right<br />
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ACCEPTED MANUSCRIPT maxillary sinus. The fine, homogeneous calcifications, indicating keratin materials, occupy the whole mass. The size of the recurrent lesion at its largest area was 15x11 mm2.<br />
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Figure 5. MRI taken eight months after the third surgery in our hospital shows a recurrent lesion with a hypointense signal in a fat suppressed T2-weighted image (fast spin echo; TR, 3167 ms; TE, 88 ms) (A) and minimal enhancement on fat suppressed post-gadolinium T1weighted image (spin echo; TR, 367 ms; TE, 10 ms) (B), corresponding to fine calcified<br />
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materials on CT image. The size of the recurrent lesion at its largest area was 25x11 mm2.<br />
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Figure 6. Axial CT images (120 kV, 150 mAs, pixel spacing = 0.43 mm, slice thickness = 2 mm), taken 19 months after the fourth surgery in our hospital, demonstrate multiple recurrent lesions on the previous surgical resection margins, zygoma (A), anterior maxilla (B), and<br />
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ACCEPTED MANUSCRIPT Table 1. Summary of clinical and radiological features of previously reported cases of keratoameloblastoma, including our case. Age Author<br />
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Location<br />
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Clinical findings<br />
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Radiological findings<br />
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Treatment<br />
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Recurrence<br />
<br />
/Sex Asian<br />
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Anterior mandible<br />
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Expansile swelling without pain<br />
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Multilocular radiolucent lesion<br />
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35/M<br />
<br />
Asian<br />
<br />
Left mandible<br />
<br />
Hard swelling<br />
<br />
35/F<br />
<br />
Asian<br />
<br />
Right maxilla<br />
<br />
Massive swelling with buccopalatal expansion<br />
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39/F<br />
<br />
Asian<br />
<br />
Left anterior<br />
<br />
No specific findings<br />
<br />
Resection<br />
<br />
-<br />
<br />
Left hemimandibulectomy<br />
<br />
-<br />
<br />
Lesion with ground-glass appearance and indistinct border<br />
<br />
-<br />
<br />
-<br />
<br />
Radiolucent cystic lesion<br />
<br />
Enucleation<br />
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-<br />
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30/M<br />
<br />
-<br />
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SC<br />
<br />
Siar et al.10<br />
<br />
Raubenheimer 57/F<br />
<br />
-<br />
<br />
Mandible<br />
<br />
-<br />
<br />
26/M<br />
<br />
African<br />
<br />
Right maxillary<br />
<br />
Initial: no specific findings<br />
<br />
Second molar area<br />
<br />
Recurrent:<br />
<br />
et al.8 Said-al-Naief et al.9<br />
<br />
mass<br />
<br />
protruding<br />
<br />
from<br />
<br />
35/M<br />
<br />
Asian<br />
<br />
Right posterior<br />
<br />
Pain<br />
<br />
mandible<br />
<br />
Whitt et al.12<br />
<br />
76/M<br />
<br />
45/M<br />
<br />
Asian<br />
<br />
Caucasian<br />
<br />
Initial: well-defined radiolucent lesion<br />
<br />
Primary and recurrent lesion:<br />
<br />
Recurrent: large mass filling most of right maxillary sinus<br />
<br />
curettage<br />
<br />
-<br />
<br />
Recurrence (seven months)<br />
<br />
Second recurrent lesion: mass excision<br />
<br />
with<br />
<br />
partial<br />
<br />
maxillectomy<br />
<br />
swelling<br />
<br />
on<br />
<br />
the<br />
<br />
left<br />
<br />
posterior<br />
<br />
-<br />
<br />
-<br />
<br />
second molar roots<br />
<br />
Left posterior<br />
<br />
Diffuse<br />
<br />
mandible<br />
<br />
mandibular alveolar ridge without pain<br />
<br />
canine to the second molar of left mandible<br />
<br />
Anterior maxilla<br />
<br />
Both facial and palatal expansion of alveolar<br />
<br />
2-cm radiolucent lesion displacing nearby roots with<br />
<br />
process of anterior maxilla<br />
<br />
internal calcifications<br />
<br />
AC C<br />
<br />
Takeda et al.4<br />
<br />
-<br />
<br />
Round radiolucent lesion between the right first and<br />
<br />
EP<br />
<br />
Kaku et al.6<br />
<br />
the<br />
<br />
-<br />
<br />
TE D<br />
<br />
extraction socket<br />
<br />
M AN U<br />
<br />
mandible<br />
<br />
Multilocular radiolucent lesion extending from the left<br />
<br />
Mandibular resection<br />
<br />
No recurrence<br />
<br />
Enucleation<br />
<br />
No recurrence<br />
<br />
ACCEPTED MANUSCRIPT Adeyemi et<br />
<br />
38/M<br />
<br />
-<br />
<br />
Right mandible<br />
<br />
al.5<br />
<br />
Initial: firm swelling on right mandible with<br />
<br />
Initial: no information<br />
<br />
Mandibular resection<br />
<br />
No recurrence<br />
<br />
buccolingual expansion<br />
<br />
Revisitation: multilocular radiolucent lesion extending<br />
<br />
Iliac bone reconstruction<br />
<br />
Revisitation: enlarged mass compared to the<br />
<br />
from lateral incisor to the angle of mandible<br />
<br />
initial visit with erythematous overlying<br />
<br />
RI PT<br />
<br />
mucosa and bleeding on palpation without tenderness 35/F<br />
<br />
African<br />
<br />
Right posterior<br />
<br />
A red nodular lesion penetrated into the right<br />
<br />
Multilocular mixed radiolucent and radiopaque lesion<br />
<br />
Mandibular resection<br />
<br />
mandible<br />
<br />
retromolar<br />
<br />
extending from right second molar to the middle third of<br />
<br />
Plate reconstruction<br />
<br />
pad<br />
<br />
with<br />
<br />
firm,<br />
<br />
compressive<br />
<br />
-<br />
<br />
SC<br />
<br />
Sisto11<br />
<br />
swelling buccolingually.<br />
<br />
the ramus with destruction of inferior mandibular cortex<br />
<br />
M AN U<br />
<br />
on panoramic radiography<br />
<br />
The volume of the mass was measured as 4×3× 2 cm with buccal and lingual cortical bone perforation on CT images<br />
<br />
Right maxilla<br />
<br />
Swelling on the right palate with intact overlying<br />
<br />
Initial: mixed radiolucent and radiopaque lesion with<br />
<br />
First through fourth operations:<br />
<br />
mucosa<br />
<br />
scalloping margin and incomplete septa<br />
<br />
enucleation<br />
<br />
Recurrent: CT revealed homogeneous high-attenuated<br />
<br />
TE D<br />
<br />
Asian<br />
<br />
materials.<br />
<br />
EP<br />
<br />
56/M<br />
<br />
AC C<br />
<br />
Present case<br />
<br />
Fifth<br />
<br />
through<br />
<br />
operation: resection<br />
<br />
seventh<br />
<br />
First recurrence(three months) Second recurrence(eleven months) Third recurrence (four months) Fourth recurrence (two months) Fifth recurrence(four months) Sixth recurrence(19 months)<br />
<br />
AC C<br />
<br />
EP<br />
<br />
TE D<br />
<br />
M AN U<br />
<br />
SC<br />
<br />
RI PT<br />
<br />
ACCEPTED MANUSCRIPT<br />
<br />
AC C<br />
<br />
EP<br />
<br />
TE D<br />
<br />
M AN U<br />
<br />
SC<br />
<br />
RI PT<br />
<br />
ACCEPTED MANUSCRIPT<br />
<br />
AC C<br />
<br />
EP<br />
<br />
TE D<br />
<br />
M AN U<br />
<br />
SC<br />
<br />
RI PT<br />
<br />
ACCEPTED MANUSCRIPT<br />
<br />
AC C<br />
<br />
EP<br />
<br />
TE D<br />
<br />
M AN U<br />
<br />
SC<br />
<br />
RI PT<br />
<br />
ACCEPTED MANUSCRIPT<br />
<br />
AC C<br />
<br />
EP<br />
<br />
TE D<br />
<br />
M AN U<br />
<br />
SC<br />
<br />
RI PT<br />
<br />
ACCEPTED MANUSCRIPT<br />
<br />
AC C<br />
<br />
EP<br />
<br />
TE D<br />
<br />
M AN U<br />
<br />
SC<br />
<br />
RI PT<br />
<br />
ACCEPTED MANUSCRIPT<br />
<br />
</div>
</div>
</div>
</div>
</div>
</div>
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S2212-4403(15)01087-1
DOI:
10.1016/j.oooo.2015.07.008
Reference:
OOOO 1251
To appear in:
Oral Surgery, Oral Medicine, Oral Pathology and Oral Radiology
Received Date: 7 February 2015 Revised Date:
12 June 2015
Accepted Date: 8 July 2015
Please cite this article as: Lee C, Park B-J, Yi W-J, Heo M-S, Lee S-S, Huh K-H, Keratoameloblastoma: a case report and a review of the literature on its radiological features, Oral Surgery, Oral Medicine, Oral Pathology and Oral Radiology (2015), doi: 10.1016/j.oooo.2015.07.008. This is a PDF file of an unedited manuscript that has been accepted for publication. As a service to our customers we are providing this early version of the manuscript. The manuscript will undergo copyediting, typesetting, and review of the resulting proof before it is published in its final form. Please note that during the production process errors may be discovered which could affect the content, and all legal disclaimers that apply to the journal pertain.
ACCEPTED MANUSCRIPT