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A huge ameloblastic fibro-odontoma of the maxilla
Oral Oncology EXTRA (2006) 42, 160– 162
available at www.sciencedirect.com
journal homepage: http://intl.elsevierhealth.com/journal/ooex
CASE REPORT
A huge ameloblastic fibro-odontoma of the maxilla Yan-ping Hu a, Bing Liu a, Tong Su a, Wen-feng Zhang a, Yi-fang Zhao
a,b,*
a
Department of Oral and Maxillofacial Surgery, Hospital of Stomatology, WuHan University, 237 Luoyu Road, 430079 WuHan, China b Key Laboratory for Oral Biomedical Engineering of Ministry of Education, School and Hospital of Stomatology, WuHan University, 430079 WuHan, China Received 26 October 2005; accepted 26 October 2005
KEYWORDS
Summary Ameloblastic fibro-odontoma is a benign epithelial odontogenic tumor with odontogenic mesenchyme exhibiting a histologic characteristics of ameloblastic fibroma and odontoma. It is slow-growing, and usually is small. We present a huge ameloblastic fibroodontoma in the maxilla of a 17 year-old woman. c 2005 Elsevier Ltd. All rights reserved.
Odontogenic tumor; Ameloblastic fibro-odontoma
Introduction Ameloblastic fibro-odontoma is a relatively rare, slowgrowing, expansile epithelial odontogenic tumor with odontogenic mesenchyme.1 The incidence of ameloblastic fibro-odontoma is between 1–3% in odontogenic tumor.2,3 It is a lesion similar to the ameloblastic fibroma, but also with inductive changes and formation of dentin and enamel. It may inhibit tooth eruption or displace involved teeth, although the teeth in the affected area are vital, occurs with equal frequence in the maxilla and the mandible, its usual location is in the posterior pre-molar-molar region. Radiographically, a unilocular appearance is most likely, although it is not unusual to see the suggestion of a multi* Corresponding author. Address: Department of Oral and Maxillofacial Surgery, Hospital of Stomatology, WuHan University, 237 Luoyu Road, 430079 WuHan, China. Tel.: +86 27 8764 7434; fax: +86 27 8787 3260. E-mail address: [email protected] (Y.-f. Zhao).
locular pattern, with opacities that may resemble a toothlike structure or an uneven scattering of spicule-like calcifications.4 We present a huge ameloblastic fibro-odontoma of the maxilla in a 17 year-old woman.
Case report A 17 year-old girl was referred on March 22, 2004, because of the left maxillary swelling. Five years ago, she fell down from the roof accidentally, and her left cheek was injured, one week later, the injury healed, but from then on, she found the left cheek swelling, the swelling expanded slowly, without other symptoms. One year ago, the swelling expanded rapidly than before, several teeth were displaced, and she felt pain occasionally, so she came to visit our department. Examination found that the left cheek was grossly deformed with normal overlying skin (Fig. 1) and there was no paresis in the affected area and no nasal discharge. The left maxilla was swollen on its buccal and palatal aspects from the middle incisor to the third molar,
1741-9409/$ - see front matter c 2005 Elsevier Ltd. All rights reserved. doi:10.1016/j.ooe.2005.10.012
A huge ameloblastic fibro-odontoma of the maxilla
161
Figure 3 Coronal CT showed a large soft tissue mass with irregular dense calcification.
Figure 1
The left cheek grossly deformed.
Figure 4 (HE · 20).
Figure 2 Axial CT showed a large soft tissue mass with irregular dense calcification.
Photomicrograph of a section of the specimen
the palatal vault was deformed, the oral mucosa over this area appeared healthy and asymptomatic, without evidence of infect. A computed tomography scan was performed to assess the extent of the tumor. The radiographs showed a large soft tissue mass over the left maxilla, extending into the maxillary sinus, destruction of left maxillary alveolar ridge and posterior and inferior and medial walls of the left maxillary sinus was noted, irregular areas of dense calcification were visible within the soft tissue mass (Figs. 2 and 3). The medical and family histories were unremarkable, as were the results of a review of system examination. Under
162
Figure 5 The polarized nuclei of cells resembling ameloblasts (HE · 20).
general anesthesia, the frozen biopsy indicated benign tumor, and partial maxillectomy was performed. The tumor measured 9 · 7 · 6 cm with a smooth surface. Microscopic examination of section stained with hematoxylin and eosin revealed characteristics of both ameloblastic fibroma and odontoma, the connective tissue was moderately cellular with spindle-shaped fibroblastic matrix (Figs. 4 and 5). The tumor was diagnosed as ameloblastic fibroodontoma. The soft tissue healing was uneventful, and the sign of recurrence in 18 months follow-up was absent.
Discussion The ameloblastic fibro-odontoma is a rare mixed odontogenic tumor that has been reported to have a hamartomatous behavior.5 The average age at diagnosis is nine yearold, and only one reported case in a patient beyond the age of 20 year-old, there is a 1.4:1 male:female ratio, and the most common site is the posterior mandible.6 The two most common presenting complains are swelling and failure of tooth eruption, the lesion may displace erupted teeth but other symptoms are uncommon. It is generally considered a slow-growing central jaw tumor, occasionally, the tumor exhibits marked swelling which results in facial disfigurement.7 In our case, the swelling is secondary to a trauma, we think that trauma possibly is a motivation of ameloblastic fibro-odontoma, but this is to be demonstrated. Radiography usually shows a well-defined radiolucent area containing various amounts of radiopaque material of irregular size and form, the ratio of radiopaque to radiolucent areas differs from one lesion to another.8 Ameloblastic fibro-odontoma has been confused in the earlier literature with other odontogenic mixed tumor, some investigators have suggested that ameloblastic fibroma will ultimately differentiate or mature further into an ameloblastic fibro-odontoma and then continue maturation into a completely differentiated odontoma.9 Hooker first differentiated the ameloblastic fibro-odontoma from the ameloblastic odontoma.10 If ameloblastic fibro-odontoma, ameloblastic fibroma and odontoma are simply stages in a
Y.-p. Hu et al. continuum, clinical data on each of lesions should support this, e.g. the ameloblastic fibroma should occur in younger patients, the odontoma in somewhat older patients and ameloblastic fibro-odontoma in an intermediate age group. Slootweg has investigated this problem and concluded that ameloblastic fibro-odontoma represents a separate specific neoplastic entity,11 and ameloblastic fibro-odontoma should not be considered as hamartomatous since there are cases of ameloblastic fibro-odontoma showing true neoplastic behavior and the existence of malignant variants.12 Now most investigators agree that ameloblastic fibro-odontoma is a separate entity, the revised WHO classification of odontogenic tumors defines ameloblastic fibro-odontona as ‘‘a lesion similar to ameloblastic fibroma, but also showing inductive changes that lead to the formation of both dentin and enamel’’.13 Because ameloblastic fibro-odontoma is well encapsulated and there is little tendency to local invasion, the recommended treatment is conservative surgery with enucleation, when the lesion includes an unerupted tooth, the tooth should be removed with the mass. There is very little potential for recurrence and malignant transformation of ameloblastic fibro-odontoma is rare. For our case, because of the wide involvement, partial maxillectomy is mandatory.
References 1. Kramer IRH, Pindborg JJ, Shear M. The WHO histological odontogenic tumors. Cancer 1992;70:2988–94. 2. Philipsen HP, Thosaporn W, Reichart PA, et al. Odontogenic lesions in opercula of permanent molars delayed in eruption. J Oral Pathol Med 1992;21:38–41. 3. Wu PC, Chang K. A survey of tumors of the jaw bones in HongKong Chinese 1963–1982. Br J Oral Maxillofac Surg 1985; 23:92–102. 4. Hutt PH. Ameloblastic fibro-odontoma: report of a case with documented four-year follow-up. J Oral Maxillofac Surg 1982; 40:45–8. 5. Philipsen HP, Reichart RA, Pratorius F. Mixed odontogenic tumors and odontomas:considerations on interrelationship: review of the literature and presentation of 134 new cases of odontoma. Oral Oncol 1997;33:86–90. 6. Daley TD, Wysocki GP, Pringle GA. Relative incidence of odontogenic tumors and oral and jaw cysts in a Canadian population. Oral Sueg Oral Med Oral Pathol 1994;77:276– 83. 7. Cheng H, Shimizu M, Precious D. Ameloblastic fibro-odontoma: a case report. J Can DENT Assoc 2002;68:243–6. 8. Piette EM, Tideman H, Wu PC. Massive maxillary ameloblastic fibro-odontoma: case report with surgical treatment. J Oral Maxillofac Surg 1990;48:526–30. 9. Miller AS, Lopez ACF, Pullon PA, et al. Ameloblastic fibroodontoma. Oral Surg 1976;41:354–65. 10. Hooker SP. Ameloblastic odontoma: an analysis of twenty-six cases. Oral Surg 1967;24:375–7. 11. Slootweg PJ. An analysis of the interrelationship of the mixed odontogenic tumors: ameloblastic fibroma, ameloblastic fibroodontoma and odontomas. Oral Surg 1981;51:266–76. 12. Takeda Y. Ameloblastic fibroma and related lesions: current pathological concept. Oral Oncology 1999;35:535–40. 13. Kramer IRH, Pindborg JJ, Shear M. Histological typing of odontogenic tumors. 2nd ed. Berlin: Springer-Verlag; 1992.
available at www.sciencedirect.com
journal homepage: http://intl.elsevierhealth.com/journal/ooex
CASE REPORT
A huge ameloblastic fibro-odontoma of the maxilla Yan-ping Hu a, Bing Liu a, Tong Su a, Wen-feng Zhang a, Yi-fang Zhao
a,b,*
a
Department of Oral and Maxillofacial Surgery, Hospital of Stomatology, WuHan University, 237 Luoyu Road, 430079 WuHan, China b Key Laboratory for Oral Biomedical Engineering of Ministry of Education, School and Hospital of Stomatology, WuHan University, 430079 WuHan, China Received 26 October 2005; accepted 26 October 2005
KEYWORDS
Summary Ameloblastic fibro-odontoma is a benign epithelial odontogenic tumor with odontogenic mesenchyme exhibiting a histologic characteristics of ameloblastic fibroma and odontoma. It is slow-growing, and usually is small. We present a huge ameloblastic fibroodontoma in the maxilla of a 17 year-old woman. c 2005 Elsevier Ltd. All rights reserved.
Odontogenic tumor; Ameloblastic fibro-odontoma
Introduction Ameloblastic fibro-odontoma is a relatively rare, slowgrowing, expansile epithelial odontogenic tumor with odontogenic mesenchyme.1 The incidence of ameloblastic fibro-odontoma is between 1–3% in odontogenic tumor.2,3 It is a lesion similar to the ameloblastic fibroma, but also with inductive changes and formation of dentin and enamel. It may inhibit tooth eruption or displace involved teeth, although the teeth in the affected area are vital, occurs with equal frequence in the maxilla and the mandible, its usual location is in the posterior pre-molar-molar region. Radiographically, a unilocular appearance is most likely, although it is not unusual to see the suggestion of a multi* Corresponding author. Address: Department of Oral and Maxillofacial Surgery, Hospital of Stomatology, WuHan University, 237 Luoyu Road, 430079 WuHan, China. Tel.: +86 27 8764 7434; fax: +86 27 8787 3260. E-mail address: [email protected] (Y.-f. Zhao).
locular pattern, with opacities that may resemble a toothlike structure or an uneven scattering of spicule-like calcifications.4 We present a huge ameloblastic fibro-odontoma of the maxilla in a 17 year-old woman.
Case report A 17 year-old girl was referred on March 22, 2004, because of the left maxillary swelling. Five years ago, she fell down from the roof accidentally, and her left cheek was injured, one week later, the injury healed, but from then on, she found the left cheek swelling, the swelling expanded slowly, without other symptoms. One year ago, the swelling expanded rapidly than before, several teeth were displaced, and she felt pain occasionally, so she came to visit our department. Examination found that the left cheek was grossly deformed with normal overlying skin (Fig. 1) and there was no paresis in the affected area and no nasal discharge. The left maxilla was swollen on its buccal and palatal aspects from the middle incisor to the third molar,
1741-9409/$ - see front matter c 2005 Elsevier Ltd. All rights reserved. doi:10.1016/j.ooe.2005.10.012
A huge ameloblastic fibro-odontoma of the maxilla
161
Figure 3 Coronal CT showed a large soft tissue mass with irregular dense calcification.
Figure 1
The left cheek grossly deformed.
Figure 4 (HE · 20).
Figure 2 Axial CT showed a large soft tissue mass with irregular dense calcification.
Photomicrograph of a section of the specimen
the palatal vault was deformed, the oral mucosa over this area appeared healthy and asymptomatic, without evidence of infect. A computed tomography scan was performed to assess the extent of the tumor. The radiographs showed a large soft tissue mass over the left maxilla, extending into the maxillary sinus, destruction of left maxillary alveolar ridge and posterior and inferior and medial walls of the left maxillary sinus was noted, irregular areas of dense calcification were visible within the soft tissue mass (Figs. 2 and 3). The medical and family histories were unremarkable, as were the results of a review of system examination. Under
162
Figure 5 The polarized nuclei of cells resembling ameloblasts (HE · 20).
general anesthesia, the frozen biopsy indicated benign tumor, and partial maxillectomy was performed. The tumor measured 9 · 7 · 6 cm with a smooth surface. Microscopic examination of section stained with hematoxylin and eosin revealed characteristics of both ameloblastic fibroma and odontoma, the connective tissue was moderately cellular with spindle-shaped fibroblastic matrix (Figs. 4 and 5). The tumor was diagnosed as ameloblastic fibroodontoma. The soft tissue healing was uneventful, and the sign of recurrence in 18 months follow-up was absent.
Discussion The ameloblastic fibro-odontoma is a rare mixed odontogenic tumor that has been reported to have a hamartomatous behavior.5 The average age at diagnosis is nine yearold, and only one reported case in a patient beyond the age of 20 year-old, there is a 1.4:1 male:female ratio, and the most common site is the posterior mandible.6 The two most common presenting complains are swelling and failure of tooth eruption, the lesion may displace erupted teeth but other symptoms are uncommon. It is generally considered a slow-growing central jaw tumor, occasionally, the tumor exhibits marked swelling which results in facial disfigurement.7 In our case, the swelling is secondary to a trauma, we think that trauma possibly is a motivation of ameloblastic fibro-odontoma, but this is to be demonstrated. Radiography usually shows a well-defined radiolucent area containing various amounts of radiopaque material of irregular size and form, the ratio of radiopaque to radiolucent areas differs from one lesion to another.8 Ameloblastic fibro-odontoma has been confused in the earlier literature with other odontogenic mixed tumor, some investigators have suggested that ameloblastic fibroma will ultimately differentiate or mature further into an ameloblastic fibro-odontoma and then continue maturation into a completely differentiated odontoma.9 Hooker first differentiated the ameloblastic fibro-odontoma from the ameloblastic odontoma.10 If ameloblastic fibro-odontoma, ameloblastic fibroma and odontoma are simply stages in a
Y.-p. Hu et al. continuum, clinical data on each of lesions should support this, e.g. the ameloblastic fibroma should occur in younger patients, the odontoma in somewhat older patients and ameloblastic fibro-odontoma in an intermediate age group. Slootweg has investigated this problem and concluded that ameloblastic fibro-odontoma represents a separate specific neoplastic entity,11 and ameloblastic fibro-odontoma should not be considered as hamartomatous since there are cases of ameloblastic fibro-odontoma showing true neoplastic behavior and the existence of malignant variants.12 Now most investigators agree that ameloblastic fibro-odontoma is a separate entity, the revised WHO classification of odontogenic tumors defines ameloblastic fibro-odontona as ‘‘a lesion similar to ameloblastic fibroma, but also showing inductive changes that lead to the formation of both dentin and enamel’’.13 Because ameloblastic fibro-odontoma is well encapsulated and there is little tendency to local invasion, the recommended treatment is conservative surgery with enucleation, when the lesion includes an unerupted tooth, the tooth should be removed with the mass. There is very little potential for recurrence and malignant transformation of ameloblastic fibro-odontoma is rare. For our case, because of the wide involvement, partial maxillectomy is mandatory.
References 1. Kramer IRH, Pindborg JJ, Shear M. The WHO histological odontogenic tumors. Cancer 1992;70:2988–94. 2. Philipsen HP, Thosaporn W, Reichart PA, et al. Odontogenic lesions in opercula of permanent molars delayed in eruption. J Oral Pathol Med 1992;21:38–41. 3. Wu PC, Chang K. A survey of tumors of the jaw bones in HongKong Chinese 1963–1982. Br J Oral Maxillofac Surg 1985; 23:92–102. 4. Hutt PH. Ameloblastic fibro-odontoma: report of a case with documented four-year follow-up. J Oral Maxillofac Surg 1982; 40:45–8. 5. Philipsen HP, Reichart RA, Pratorius F. Mixed odontogenic tumors and odontomas:considerations on interrelationship: review of the literature and presentation of 134 new cases of odontoma. Oral Oncol 1997;33:86–90. 6. Daley TD, Wysocki GP, Pringle GA. Relative incidence of odontogenic tumors and oral and jaw cysts in a Canadian population. Oral Sueg Oral Med Oral Pathol 1994;77:276– 83. 7. Cheng H, Shimizu M, Precious D. Ameloblastic fibro-odontoma: a case report. J Can DENT Assoc 2002;68:243–6. 8. Piette EM, Tideman H, Wu PC. Massive maxillary ameloblastic fibro-odontoma: case report with surgical treatment. J Oral Maxillofac Surg 1990;48:526–30. 9. Miller AS, Lopez ACF, Pullon PA, et al. Ameloblastic fibroodontoma. Oral Surg 1976;41:354–65. 10. Hooker SP. Ameloblastic odontoma: an analysis of twenty-six cases. Oral Surg 1967;24:375–7. 11. Slootweg PJ. An analysis of the interrelationship of the mixed odontogenic tumors: ameloblastic fibroma, ameloblastic fibroodontoma and odontomas. Oral Surg 1981;51:266–76. 12. Takeda Y. Ameloblastic fibroma and related lesions: current pathological concept. Oral Oncology 1999;35:535–40. 13. Kramer IRH, Pindborg JJ, Shear M. Histological typing of odontogenic tumors. 2nd ed. Berlin: Springer-Verlag; 1992.