Maxillary osteoblastoma in a woman: Report of a rare case and review of the literature

Maxillary osteoblastoma in a woman: Report of a rare case and review of the literature

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ARTICLE IN PRESS Journal of Oral and Maxillofacial Surgery, Medicine, and Pathology xxx (2016) xxx–xxx

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Case report

Maxillary osteoblastoma in a woman: Report of a rare case and review of the literature Yoshiro Saito, Seiji Kondo ∗ , Takaaki Kamatani, Masahiro Nagasaki, Masakatsu Itose, Hiroaki Miyazaki, Hitoshi Watanabe, Tatsuo Shirota Division of Craniomaxillofacial Surgery, Department of Oral and Maxillofacial Surgery, School of Dentistry, Showa University, 2-1-1 Kitasenzoku, Ohta-ku, Tokyo 145-8515, Japan

a r t i c l e

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Article history: Received 23 March 2016 Received in revised form 9 July 2016 Accepted 29 July 2016 Available online xxx Keywords: Osteoblastoma Bone tumors Maxilla Female patient

a b s t r a c t Osteoblastoma is a rare bone tumor showing a predilection for males, and only 15% of all osteoblastomas occur in the maxillofacial region, affecting the maxilla less than the mandible. A very rare case of osteoblastoma affecting the maxilla in a female patient is reported. The diagnosis of osteoblastoma of the maxilla was made based on clinical, radiological, and histopathological findings. The lesion was completely resected, and the excised specimen showed features that are typical of osteoblastoma. At 2-year follow-up, the patient was disease-free. This report presents this rare case of osteoblastoma of the maxilla in a female patient and discusses the importance of correct diagnosis and complete surgical approach in the treatment of this lesion. © 2016 Asian AOMS, ASOMP, JSOP, JSOMS, JSOM, and JAMI. Published by Elsevier Ltd. All rights reserved.夽

1. Introduction

2. Case report

Osteoblastoma is a rare tumor characterized by osteoid and woven bone deposition and enriched osteoblasts. It accounts for approximately 1% of all primary bone tumors [1], and it occurs most frequently in male patients, with a male-to-female incidence ratio of 2:1 [2]. The locations of the lesions primarily involve the vertebral column, long tubular bones, calvarium, and facial bones including the jaw. In the maxillofacial region, Lin et al. reported the mandibular bones (58.7%) are more commonly affected than the maxillary bones, and male patients (66.5%) are predominantly affected [3]. Jones et al. reviewed 19 cases (79.2%) arose in the mandible and 5 cases (20.8%) occurred in the maxilla [4]. Of note, very few cases of osteoblastoma have been reported affecting the maxilla of a female patient. The current study reports a very rare case of osteoblastoma that developed in the right maxilla of a 24year-old Japanese woman, along with a literature review.

A 24-year-old Japanese woman was referred to our hospital by her dental practitioner for detailed examination of the right maxilla. One year earlier, she had first noticed pain and swelling in the second premolar and right maxilla. Her dentist made a diagnosis of periodontitis, and root canal treatment was provided. However, the progressive pain and swelling did not improve. Rather, the region had grown even large and become increasingly painful. On intraoral examination, there was a diffuse bony swelling of the right hard palate extending from the right canine to the second premolar. The swelling was hard in consistency and measured approximately 2 cm in greatest dimension (Fig. 1). The second premolar in this region showed slight mobility. Mucosa over the swelling was normal, with no ulcerations. On intraoral palpation of the mass, the patient felt severe pain, although no local inflammation was observed. For the severe pain, the patient was treated with nonsteroidal anti-inflammatory drugs (NSAIDs), but she obtained no relief. There were no other secondary changes, such as paresthesia or cervical lymphadenopathy. Pantomography and computerized tomography (CT) of the right maxilla revealed an expansile, mixed radiolucent and radiopaque lesion with a radiolucent rim in the vicinity of the second premolar root. CT also showed a mass of about 10 mm × 8 mm × 8 mm (Fig. 2). A provisional diagnosis of a benign odontogenic tumor was given. Thus, the patient underwent excisional biopsy given the small lesion size, and the lesion was completely enucleated,

夽 AsianAOMS: Asian Association of Oral and Maxillofacial Surgeons; ASOMP: Asian Society of Oral and Maxillofacial Pathology; JSOP: Japanese Society of Oral Pathology; JSOMS: Japanese Society of Oral and Maxillofacial Surgeons; JSOM: Japanese Society of Oral Medicine; JAMI: Japanese Academy of Maxillofacial Implants. ∗ Corresponding author at: Department of Oral and Maxillofacial Surgery, Faculty of Medicine, Fukuoka University 7-45-1, Nanakuma, Jonan-ku, Fukuoka, 814-0180, Japan, Phone: 81-92-801-1011, Telefax: 81-92-801-1044. E-mail address: [email protected] (S. Kondo).

http://dx.doi.org/10.1016/j.ajoms.2016.07.011 2212-5558/© 2016 Asian AOMS, ASOMP, JSOP, JSOMS, JSOM, and JAMI. Published by Elsevier Ltd. All rights reserved.夽

Please cite this article in press as: Saito Y, et al. Maxillary osteoblastoma in a woman: Report of a rare case and review of the literature. J Oral Maxillofac Surg Med Pathol (2016), http://dx.doi.org/10.1016/j.ajoms.2016.07.011

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Fig. 1. (A) Pre-operative facial appearance. (B) Pre-operative photograph showing intraoral swelling in the right hard palate. On palpation, it was bony hard in consistency with well-defined borders. Interestingly, severe pain was noted with pressure, despite the absence of findings of inflammation.

Fig. 2. Upper panel: orthopantomogram showing the lesion in the right maxilla. Lower panel: axial (A) and coronal (B) computed tomography images showing a radiolucent/radiopaque mass, 10 mm × 8 mm × 8 mm, in the right hard palate.

including the second premolar, and curettage was performed under general anesthesia (Fig. 3A). The specimens were sent for histopathological analysis, put into 10% formalin and processed for routine histopathologic examination, and then embedded in paraffin; 4-␮m-thick sections were prepared and stained with hematoxylin and eosin. Examination of the stained sections showed numerous proliferating osteoblasts and fibro-vascular connective tissue stroma forming trabeculae (Fig. 3B and C). Based on the histomorphologic characteristics of the tumor, clinical examination,

and radiological findings, the final diagnosis of an osteoblastoma of the maxilla was established. There was no sign of recurrence at 2-year follow-up in pantomography (Fig. 4). 3. Discussion In this report, a very rare case of osteoblastoma occurring in the maxilla of a female patient was described. Generally, the clinical symptoms of an osteoblastoma arising in the maxillofacial region

Please cite this article in press as: Saito Y, et al. Maxillary osteoblastoma in a woman: Report of a rare case and review of the literature. J Oral Maxillofac Surg Med Pathol (2016), http://dx.doi.org/10.1016/j.ajoms.2016.07.011

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Fig. 3. (A) Macroscopic view of the resected tumor and second premolar. (B, C) Histopathological image of the lesion. Original magnification ×40 (B), ×400 (C). Numerous proliferating osteoblasts and fibro-vascular connective tissue stroma forming small irregular bony trabeculae are shown.

Fig. 4. Orthopantomogram taken 2 years following the excision of the mass showing no signs of recurrence in the right maxilla.

are local swelling, pain, and slow bulging of bones, though most cases of osteoblastoma are asymptomatic. Its progression varies from months to 3 years [3]. Histologically, this tumor is characterized by proliferation of osteoblasts forming trabeculae set in a vascularized fibrous granulation tissue stroma [4]. The radiographic appearance has been described as radiolucent, as radiopaque, and as combined radiolucent/radiopaque lesions, although it is plausible that similar radiographic findings appear in lesions other than osteoblastoma [5].

The preoperative clinical diagnoses of osteoblastoma in the maxillofacial region have varied markedly, because it has been confused with other osteogenic lesions. The differential diagnosis of this tumor should include osteogenic tumors, cementoblastomas, osteoid osteoma, fibrous dysplasia, osteosarcoma, and so on [5,6]. Imaging modalities including CT and magnetic resonance imaging (MRI) are valuable to distinguish these lesions to make a provisional diagnosis [3,6]. However, in this case, a provisional diagnosis was not provided by CT, since the radiographic findings were not specific to osteoblastoma. Furthermore, radiographic lesions larger than 1.5 cm in diameter or greater have been termed osteoblastomas, because osteoblastomas have a greater growth potential than osteoid osteomas [7,8]. Based on these points, the present case with a diameter of less than 1 cm was atypical. The distinction between osteoblastoma and osteoid osteoma could be difficult based on the size of the lesion. Other than radiological findings, the suspicion of osteoblastoma should be established based on histopathological findings. The typical osteoblastoma appears to be composed of small irregular bony trabeculae and solid osteoids separated by a vascular stroma [4,5]. This solid osteoid has histological similarities to osteoid osteoma [9]. Therefore, the differential diagnosis is sometimes considered to be difficult even with a biopsy. In the present case, since a biopsy

Table 1 Previously reported cases of maxillary osteoblastoma in female patients. Age (y)

Treatment

Size (cm)

Author

Year

13 22 30 20 19 34 14 24 25 24

Dissection and curettage Curettage Removed Shelled out Curettage – – – – Enucleation and curettage

3 1.5 × 2 × 2 7×5 1 1.8 × 1.5 × 1.3 – – – – 1.0 × 0.8 × 0.8

Kent et al. [10] Yip and Lee [11] Chatterji et al. [12] Van Der Waal et al. [13] Gordon et al. [14] Jones et al. [4] Jones et al. [4] Jones et al. [4] Jones et al. [4] Saito et al.

1969 1974 1978 1983 2001 2006 2006 2006 2006 2016

Please cite this article in press as: Saito Y, et al. Maxillary osteoblastoma in a woman: Report of a rare case and review of the literature. J Oral Maxillofac Surg Med Pathol (2016), http://dx.doi.org/10.1016/j.ajoms.2016.07.011

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was not performed given the lesion’s small size, surgery with a preoperative diagnosis of osteoblastoma could not be performed. As described above, the differential diagnosis may be complicated due to the tumor’s rarity and its ambiguous radiological and histopathological findings. Although most patients with osteoblastoma are asymptomatic, swelling and pain are the typical presenting symptoms of osteoblastoma [4,10–14]. In particular, some patients have significant pain that is not relieved with NSAIDs [4]. In fact, this conspicuous clinical feature was seen in the present case. On the other hand, the pain associated with most osteoid osteomas is relieved with NSAIDs. The relief of pain with NSAIDs is an important clinical characteristic of osteoid osteoma [15]. This unique presenting sign may be useful for the differential diagnosis of osteoblastoma when histologic and/or radiographic parameters are characteristic of osteoblastoma. According to previous reports, the treatment of this tumor is considered to be surgical, that is, local conservative excision and curettage [16]. Since the border line between the lesion and normal bones is indistinct because of intraosseous osteoplastic infiltration, excision depending on the tumor size and site, followed by curettage has been generally considered appropriate for the treatment of this tumor [17]. In the maxilla and in female patients, conventional surgical approaches similar to those used for other sites were selected for treatment of this tumor (Table 1) [4,10–14]. Despite tumor removal, however, about 10% of these tumors from all sites have been found to recur [18]. In the mandible and in male patients, all recurrences were seen within 1 year after surgery [17]. Recurrence was attributed to inadequate initial treatment, such as incomplete local curettage or partial resection of the tumor [18,19]. Furthermore, this tumor is classified in the WHO classification of 2013 as having an intermediate character between benign and malignant tumors, that is, having the potential for locally aggressive growth. In the present case, an excisional biopsy following enucleation and curettage were performed without an extensive safety margin, including peripheral maxillectomy from the first premolar to the first molar. However, fortunately, this patient’s postoperative course was uneventful over the 2-year follow-up period. Adequate initial treatment, such as total resection, is important to avoid recurrence even in maxillary osteoblastoma. In conclusion, a rare case of osteoblastoma that occurred in the maxilla of a female patient was described. Correct diagnosis and complete surgical excision of the tumor are essential to decrease the risk of recurrence.

Ethics Informed consent for including the patient’s photographs was obtained from the patient. Acknowledgment No sources of funding. References [1] Lichtenstein L, Sawyer WR. Benign osteoblastoma; further observations and report of twenty additional cases. J Bone Joint Surg Am 1964;46:755–65. [2] Bilkay U, Erdem O, Ozek C, Helvaci E, Kilic K, Ertan Y, et al. A rare location of benign osteoblastoma: review of the literature and report of a case. J Craniofac Surg 2004;15:222–5. [3] Lin B, Cai ZG, Yu GY, Jia LF. Osteoblastoma of the maxilla and mandibule: a report of 2 cases and literature review. Chin J Dent Res 2012;15:153–8. [4] Jones AC, Prihoda TJ, Kacher JE, Odingo NA, Freedman PD. Osteoblastoma of the maxillaa and mandibulae: a report of 24 cases, review of the literature, and discussion of its relationship to osteoid osteoma of the jaws. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2006;102:639–50. [5] Capodiferro S, Maiorano E, Giardina C, Lacaita MG, Muzio LL, Favia G. Ostaoblastoma of the mandible: clinicathologic study of four cases and literature review. Head Neck 2005;27:616–21. [6] Arantes M, Resende M, Honavar M, Pires MM, Pereira JP, Vaz AR. Benign osteoblastoma of the sphenoid bone. Skull Base 2009;19:437–41. [7] Dorfman HD, Czerniak B. Bone tumors. St Louis: Mosby; 1998. p. 85–114. [8] Unni KK. Dahin’s bone tumors. General aspects and data on 11,087 cases. Lippincott-Raven; 1996. p. 121–42. [9] Ribera MJ. Osteoblastoma in the anterior maxilla mimicking periapical pathosis of odontogenic origin. J Endod 1996;22:142–6. [10] Kent JN, Castro HF, Girotti WR. Benign osteoblastoma of the maxillaa. Case report and review of the literature. Oral Surg Oral Med Oral Pathol 1969;27:209–19. [11] Yip WK, Lee HT. Benign osteoblastoma of the maxillaa. Oral Surg Oral Med Oral Pathol 1974;38:259–63. [12] Chatterji P, Purohit GN, Ramdeo Bikaner IN. Benign osteoblastoma of the maxilla (periosteal). J Laryngol Otol 1978;92:337–45. [13] van der Waal I, Greebe RB, Elaas EA. Benign osteoblastoma or osteoid osteoma of the maxilla. Report of a case. Int J Oral Surg 1983;12:355–8. [14] Gordon SC, Maclntosh RB, Wesley RK. A review of osteoblastoma and case report of metachronous osteoblastoma and unicystic ameroblastoma. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2001;91:570–5. [15] Healy JH, Ghelman B. Osteoid osteoma and osteoblastoma: current concepts and recent advances. Clin Orthop 1996;204:76–85. [16] Mahajan A, Kumar P, Desai K, Kaul RP. Osteoblastoma in the retromolar region—report of an unusual case and review of literature. J Maxillofac Oral Surg 2013;12:338–40. [17] Smith RA, Hansen LS, Resnick D, Chan W. Comparison of the osteoblastoma in gnathic and extragnathic sites. Oral Surg Oral Med Oral Pathol 1982;54:285–98. [18] Jackson RP. Recurrent osteoblastoma: a review. Clin Orthop Relat Res 1978;131:229–33. [19] Remagen W, Prein J. Bnign osteoblastoma. Oral Surg 1975;39:279–83.

Conflict of interest The authors declare that they have no competing interests.

Please cite this article in press as: Saito Y, et al. Maxillary osteoblastoma in a woman: Report of a rare case and review of the literature. J Oral Maxillofac Surg Med Pathol (2016), http://dx.doi.org/10.1016/j.ajoms.2016.07.011