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Case report
Ossifying fibroma arising from the zygomatic arch: A case report A. Uesugi a,b,*, K. Mochida a,b, H. Harada a, H. Imai a,b a Oral and Maxillofacial Surgery, Department of Oral Restitution, Division of Oral Health Sciences, Graduate School, Tokyo Medical and Dental University, 1-5-45 Yushima, Bunkyo-ku, Tokyo 113-8549 Japan b Division of Dentistry and Oral Surgery, Hitachi, Ltd. Hitachinaka General Hospital, 20-1, Ishikawa-cho, Hitachinaka-shi, Ibaraki-ken, 312-0057 Japan
A R T I C L E I N F O
A B S T R A C T
Historique de l’article : Received 13 July 2019 Accepted 25 August 2019
Introduction: Ossifying fibroma (OF) is a rare type of tumor characterized by fibrous tissue proliferation with cementum- or bone-like hard tissue formation. Since its first report by Montgomery in 1927, several cases of OF have been reported; however, no cases of OF arising from the zygomatic arch have been reported. Herein, we report a case of OF arising from the zygomatic arch. Clinical case summary: A 70-year-old female visited our department in February 2017 because of a gradually growing osseous protrusion in the right zygomatic region, which she was aware of since the previous 6 months. A 3.3 cm 3.2-cm area of swelling was observed in the region. Computed tomography confirmed the presence of a granulated lesion on the surface of the right zygomatic arch. Accordingly, benign bone tumor was diagnosed, and tumor resection was subsequently performed. Histopathological analysis revealed irregularly arranged bone trabeculae, an increased number of fibroblasts, and collagen fibers between the bone trabeculae; accordingly, OF was diagnosed. No clinical or radiographic evidence of recurrence was observed during the 1.5-year follow-up period. Discussion: A granulated lesion was present on the surface of the right zygomatic arch, and the boundary between the lesion and surrounding bone was clear. Resection of the lesion from the zygomatic arch was relatively easy. Thus, OF was diagnosed. If OF is suspected, a risk of recurrence persists; therefore, shaving the area including the bones surrounding the lesion may be necessary. Although detailed causes of OF and osteoma remain unknown, past trauma has been indicated as a common etiology. However, compared with the frequency of fractures in the zygomatic arch, the frequency of OF and osteoma is rare; thus, the etiology of OF and osteoma remains to be fully elucidated.
C 2019 Elsevier Masson SAS. All rights reserved.
Keywords: Ossifying fibroma Zygomatic arch
1. Introduction
2. Case report
Ossifying fibroma (OF) is a rare type of tumor characterized by fibrous tissue proliferation with cementum- or bone-like hard tissue formation. Since its first report by Montgomery in 1927 [1], several cases of OF have been reported. However, no cases of OF arising from the zygomatic arch have been reported. Herein, we report a case of OF arising from the zygomatic arch.
The patient was a 70-year-old female who detected an osseous protrusion in the right zygomatic arch 6 months before her initial visit to our department in February 2017. Although she presented with a 3.3 cm 3.2-cm swelling in the anterior right zygomatic region, her skin surface was intact and she did not complain of pain. She had no previous history of facial trauma and no notable medical history. Computed tomography (CT) confirmed the presence of a granulated lesion on the surface of the right zygomatic arch. Three-dimensional CT reconstruction images showed that the lesion was developing under the right zygomatic arch (Fig. 1). Accordingly, benign bone tumor was diagnosed, and subsequently, tumor resection was performed under general anesthesia in May 2017. An approximately 5-cm skin incision was made anteriorly in the right ear, following which the tumor was confirmed by performing ablation anterior to the cartilage of the external acoustic meatus and along the superficial layer of the temporal
Abbreviations: OF, Ossifying fibroma; CT, computed tomography; JTOF, Juvenile trabecular ossifying fibroma; JPOF, Juvenile psammomatoid ossifying fibroma; FD, Fibro dysplasia. * Corresponding author. E-mail addresses:
[email protected] (A. Uesugi),
[email protected] (K. Mochida),
[email protected] (H. Harada),
[email protected] (H. Imai). https://doi.org/10.1016/j.jormas.2019.08.004 C 2019 Elsevier Masson SAS. All rights reserved. 2468-7855/
Please cite this article in press as: Uesugi A, et al. Ossifying fibroma arising from the zygomatic arch: A case report. J Stomatol Oral Maxillofac Surg (2019), https://doi.org/10.1016/j.jormas.2019.08.004
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Fig. 2. Intraoperative clinical photo of the exposed lesion on the zygomatic arch. The tumor was 2.7 cm 1.8 cm in size and had no capsule (arrow).
3. Discussion
Fig. 1. Preoperative CT image. A. CT confirms the presence of a granulated lesion on the surface of the right zygomatic arch (arrow). B. Three-dimensional CT reconstruction images show that the lesion was developing under the right zygomatic arch (arrow).
fascia. The tumor was 2.7 cm 1.8 cm in size, had no capsule, and could be easily excised from the zygomatic arch; however, a part of the zygomatic arch adhered to the tumor (Fig. 2). A rough portion was identified on the resected surface and was removed, including the surrounding bone tissue such that no tumor remained. The tumor was located around the lower and inner edges of the zygomatic arch, which was consistent with the origin of the masseter muscle’s deep portion. Histopathological analysis revealed irregularly arranged bone trabeculae, an increased number of fibroblasts, and collagen fibers between the bone trabeculae. No significant nuclear atypia or appreciable mitosis was observed. Accordingly, OF was diagnosed (Fig. 3). Paralysis of the right temporal branch of the facial nerve was detected after surgery; however, it resolved after 4 months. No tumor recurrence was observed throughout the 1.5-year follow-up period (Fig. 4).
OF, first reported by Montgomery in 1927 [1], is a neoplastic disease characterized by fibrous tissue proliferation and bone-like hard tissue formation. OF development is generally slow, and its recurrence is rare following surgical removal. However, the subtypes of OF, including juvenile trabecular OF (JTOF) and juvenile psammomatoid OF (JPOF), are characterized by rapid growth, invasive deterioration of the jawbone, and frequent recurrence even after surgical removal. Their histopathological findings include dense fibroblasts and vigorous cell division; regarding JPOF, the findings additionally include sand-like appearance of hard tissue. In the present case, we observed no histopathological findings suggestive of JTOF or JPOF. With regard to the differential diagnosis of OF, osteoma and fibro dysplasia (FD) are cited. Osteoma is characterized by a bone tumor growing continuous with the surrounding cortical bone, as observed via X-ray examination, and a thick-layered bone, as observed via histopathological examination; however, the current case did not fulfill these criteria. OF and FD are histopathologically similar and commonly demonstrate the formation of irregular trabecular bone in the fibrous tissue and subsequent transition to surrounding bone tissue. On X-ray examination, OF and FD can be distinguished on the basis of the boundary between the lesion and surrounding bone, which is observed to be clear in OF and unclear in FD [2]. In this case, a granulated lesion was present on the surface of the zygomatic arch, and the boundary between the lesion and surrounding bone was clear. The resection of the lesion from the
Please cite this article in press as: Uesugi A, et al. Ossifying fibroma arising from the zygomatic arch: A case report. J Stomatol Oral Maxillofac Surg (2019), https://doi.org/10.1016/j.jormas.2019.08.004
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Fig. 3. Pathological histology. Histopathological observation reveals irregularly arranged bone trabeculae (asterisk), an increased number of fibroblasts, and collagen fibers between the bone trabeculae (arrow). A. H-E, low magnification. B. H-E, high magnification.
zygomatic arch was relatively easy. Accordingly, OF was diagnosed. Generally, resection is recommended for treating OF. However, a conservative approach with follow-up observations may be selected when the growth rate is slow and no invasive bone destruction is observed. In our case, skin swelling caused by the tumor was noticeable in the right zygomatic arch, and the patient requested tumor removal. The lesion could be easily removed from the zygomatic arch; however, a rough portion remained on the resected surface. Subsequently, it was shaved, including the surrounding bones. If OF is suspected, a risk of recurrence persists; therefore, shaving the area including the bones surrounding the lesion may be necessary. Most cases of OF arising from the maxillofacial area are reported from the maxilla and mandible. To the best of our knowledge, ours is the only report regarding OF arising from the zygomatic arch. However, osteoma often occurs in the maxillofacial region, although only four cases have been reported in the zygomatic arch [3–6]. Although detailed causes of OF and osteoma are unknown, past trauma has been indicated as a common etiology [7,8]. However, our patient had no history of trauma to the right zygomatic arch. Kaplan [9] previously reported that osteoma tends to develop at muscle attachment sites. Even with minor trauma, the periosteum is elevated by muscle traction, which promotes osteogenesis, and is maintained by sustained muscle tension. Considering that
Fig. 4. Postoperative CT image. A, B. No tumor recurrence was observed throughout the 1.5-year follow-up period.
osteoma stops growing at a certain size, it may grow to be reactive rather than tumorous. In our case, we considered that some stimulus was applied to the origin of the masseter muscle, following which OF occurred. However, compared with the frequency of fractures in the zygomatic arch, the frequency of OF and osteoma is rare; thus, the etiology of OF and osteoma remains to be fully elucidated. Recent reports have considered the possibility that overexpression of the RHAMM gene, the protein of which is a hyaluronan receptor and plays an important role in cell proliferation, is associated with OF [10]. However, the specificity of gene expression in the entire body, including the zygomatic arch, remains unclear. In conclusion, we described a case of OF arising from the zygomatic arch. To clarify the etiology of this relatively rare condition, more cases should be accumulated for further analysis.
Ethics approval and consent to participate The presented case report is in accordance with the ethical standards of the institutional and/or national research committee
Please cite this article in press as: Uesugi A, et al. Ossifying fibroma arising from the zygomatic arch: A case report. J Stomatol Oral Maxillofac Surg (2019), https://doi.org/10.1016/j.jormas.2019.08.004
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and with the 1964 Helsinki declaration and its later amendments or comparable ethical standards. Informed consent for participation was obtained from the patient presented. Since no experimentation with human subjects was performed, no review/ approval by an ethics board was required.
Consent for publication Informed consent for publication of this article and its contents was obtained from the patient.
Availability of data and material The data and material supporting the conclusions of this article are included within the article. Funding This report did not receive any specific grant from funding agencies in the public, commercial, or not-for-profit sectors.
Authors’ contributions AU conceived this report, drafted the article, and revised it critically for important intellectual content. KM, HH, and HI helped to draft the manuscript. All authors read and approved the final manuscript.
Disclosure of interest The authors report no financial or other conflict of interest relevant to this article, which is the intellectual property of the authors.
Acknowledgements The authors would like to thank Dr. Takashi Horiguchi from the Department of Pathology, Hitachinaka General Hospital, for his kind assistance in revising the pathological diagnosis of this case. References [1] Montgomery AH. Ossifying fibroma of the jaw. Arch Surg 1927;15:30–44. [2] Boland T, Ross Beirne O. Zygomatic exostosis. Int J Oral Surg 1983;12:124–6. [3] Furlaneto EC, Rocha JR, Heitz C. Osteoma of the zygomatic arch report of a case. Int J Oral Maxillofac Surg 2004;33:310–1. [4] Dura˜o AR, Chilvarquer I, Hayek JE, Provenzano M, Kendall MR. Osteoma or the zygomatic arch and mandible: Report or two cases. Rev Port Estomatol Med Dent e Cir Maxilofac 2012;53:103–7. [5] Quintans AD, Castro JA, Machado PE, Costa FW. Solitary osteoma in the zygomatic arch. J Craniofac Surg 2013;24:2209–10. [6] Starch-Jensen T. Peripheral solitary osteoma of the zygomatic arch: a case report and literature review. Open Dent J 2017;11:120–5. [7] Smith AG, Zavaleta A. Osteoma, ossifying fibroma, and fibrous dysplasia of facial and cranial bone. Arch Path 1952;54:507–27. [8] Thoma KH. Oral Pathology (ed 4). St Louis, MO: Mosby; 1954. p. 1251–9. [9] Kaplan I, Calderon S, Buchner A. Peripheral osteoma of the mandible: a study of 10 new cases and analysis of the literature. J Oral Maxillofac Surg 1994;52:467–70. [10] Hatano H, Ogawa I, Shigeishi H, Kudo Y, Ohta K, Higashikawa K, et al. Expression of receptor for hyaluronan-mediated motility (RHAMM) in ossifying fibromas. Histol Histopathol 2013;28:473–80.
Please cite this article in press as: Uesugi A, et al. Ossifying fibroma arising from the zygomatic arch: A case report. J Stomatol Oral Maxillofac Surg (2019), https://doi.org/10.1016/j.jormas.2019.08.004