An unusual presentation of multiple osseous choristomas in masseter muscle

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1 CASE REPORT An unusual presentation of multiple osseous choristomas in masseter muscle Priscilla Suassuna Carneiro Lúcio 1, Eduardo Augusto Rosa 2, Gustavo Pina Godoy 3, Rivadávio Fernandes Batista de Amorim 4 1 Department of Dentistry, School of Dentistry, State University of Paraíba, Campina Grande, PB, Brazil 2 Post-Graduation Program in Medical Sciences, School of Medicine, University of Brasilia (UnB), Brasília, DF, Brazil 3 Department of Pathology, Federal University of Pernambuco, Recife, PE, Brazil 4 Laboratory of Neuromodulation & Center for Clinical Research Learning, Harvard Medical School (HMS), Charlestow-Massachusetts, USA Correspondence: Rivadávio Fernandes Batista de Amorim ramorim@neuromodulationlab.org Received: July 13, 2015 Published online: August 03, 2015 To cite this article: Priscilla Suassuna Carneiro Lúcio, et al. An unusual presentation of multiple osseous choristomas in masseter muscle. Musculoskelet Regen 2015; 2: e905. doi: /mr.905. Introduction Osseous choristoma (OC) can be defined as a tumor-like mass composed by normal lamellar bone occurring in an abnormal location. It consists a rare lesion and more likely to take place at the tongue s base, next to the foramen caecum. The occurrence of OC in other anatomical maxillofacial regions is particularly unusual [1, 2]. In most cases, no signals and clinical manifestations are not present, however symptoms such as pain, choking, dysphagia and sickness have been reported [3, 4]. Based on a careful review and a systematic search for literature (PubMed, Web of Science and Scopus) there is only one previous case of OC diagnosed at masseter muscle. Therefore, the aim of this paper is to report a very unusual case of multiples OC in the masseter muscle and to discuss the differential diagnosis for this type of lesion. Moreover, an epidemiologic profile update of OCs at the head and neck region (except cases in tongue and eyes) is also provided to highlight the main features of such uncommon lesion and how to approach it. Case Report A 32-years old white female was referred to Maxillofacial Surgery Service complaining of pain and swelling in the left side of the face. The patient was not able to describe exactly when the growth was first noticed, but the symptoms were observed one year before initial admission. The past medical history was not remarkable. Furthermore, no current medical relevant disorder or previous facial trauma at the referred area were reported. Clinical examination revealed firm, hard, well-circumscribed mass nodules, distributed from the center of the left masseter muscle and extended to the mandibular angle. No skin alteration nor functional limitations were present. Moreover, palpable lymph node were not detected at the cervical region and intra-oral physical evaluation revealed no significant alterations. Taking into account the non-specific findings of clinical analysis, radiographic and tomographic exams were performed for a better evaluation and management of the patient. Frontal facial radiography revealed a slightly shapeless radiopaque image (Figure 1A). Finally, computed tomography views and an axial image with soft tissue window showed that the lesions were located inside the left masseter muscle, laterally to the mandibular ramus (Figures 2 and 3). Page 1 of 6

2 Figure 1. Posteroanterior image and computerized tomography. Posteroanterior view of the jaws demonstrating the faint presence of a shapeless mass of radiopacity -white arrow (A) and transversal computerized tomography demonstrating small calcified spherical structures (B). According to the clinical, radiographic and tomographic features of the lesions, some diagnostic hypothesis considered, such as ossifying myositis, calcified lymph nodes, teratomas and multiple OC. Therefore, an excisional biopsy procedure was performed in order to provide a definitive diagnosis. Under general anesthesia, the lesions were surgically removed through a submandibular access. After an incision on the masseter lateral face, the calcified spherical structures were removed from the muscle fibers. Seven spherical structures were removed (Figure 4A). The largest one measured 7 x 4 mm and the lowest 5 x 4 mm. Histological sections revealed fragments of osteoid tissue showing concentric lamellar structure and lacunae with Figure 2. Three-dimensional reconstruction. Demonstrating seven spherical structures that were not attached to the mandibular bone. osteocytes (Figure 4B, C and D). In some areas, mature bone was also noted. Small amount of loose connective tissue was found in the periphery. Figure 4. Calcified spherical structures removed (A); fragments of osteoid tissue showing concentric lamellar structure and lacunae occupied by osteocytes (B, C and D) (HE, 100x and 400x). Afterwards, the definitive diagnosis was consistent with multiple OC. In the immediate postoperative period, the patient reported pain relief on the lesion area. The 3 years follow-up showed no signs of recurrence or pain. Discussion OC diagnosed at maxillofacial region is a rare condition and just few cases have been reported in the literature to date Page 2 of 6

Figure 3. Axial image. Demonstrating lesions located inside the left masseter muscle laterally to the mandibular ramus. [1, 5]. Its etiology and pathogenesis is still not totally clear [1, 5-7].

3 Figure 3. Axial image. Demonstrating lesions located inside the left masseter muscle laterally to the mandibular ramus. [1, 5]. Its etiology and pathogenesis is still not totally clear [1, 5-7]. Some authors suggest that this disturbance is derived from developmental malformations. Others consider a neoplastic or reparative origin, like a reactive process or calcification of lymphatic or fibrous connective tissues. Moreover, congenital abnormality, degenerating fibroma suffering ossification or a post-traumatic mesenchymal metaplasia have also been considered as possible causes for OC development [6]. Sookasam and Philipsen [8] proposed that OC can be divided into two pathogenic groups: lesions of the posterior part of the tongue near the foramen caecum possibly due to developmental malformation, and lesions of other sites including the anterior and lateral part of the tongue, buccal and palatal mucosa may arise from post-traumatic ossification. In the present case, the patient did not report any previous trauma at the region. However, the possibility of previous trauma cannot be totally ruled out since minors or non-significant traumas would be easily not remembered by the patient. Moreover, OC usually shows a slow growth pattern which may difficult to detect the lesion and perform early diagnosis. Page 3 of 6

4 Figure 4. Anatomical and histopathological image injury. Calcified spherical structures removed (A); fragments of osteoid tissue showing concentric lamellar structure and lacunae occupied by osteocytes (B, C and D) (HE, 100x and 400x). Salivary gland or neural tumors, phlebolithiasis, granular cell tumor, thyroid nodule and hyperplastic lingual tonsillitis must be considered as differential diagnosis when the OC occurs in the tongue. If the gingiva is involved, the other possible diagnosis are and giant cell granulomas, odontogenic fibroma in a pheriferical location and thyroglossal cyst. Either salivary gland calculi or tumors, fibroma and lymph node ossification should be ruled out if the OC location is the buccal mucosa. Considering the submentum region, other differential diagnosis are also possible such as lymph node ossification and progressive ossifying myositis [4, 9-11]. Therefore, a correct diagnosis is still challenging due to the several possible differential diagnosis. Analyzing the most relevant scientific databases for medical and health sciences (PubMed, Web of Science and Scopus) we searched for papers in English language using the key words "osseous choristoma" only in title/abstract, in humans, during the period between 1960 and The search returned 83 papers meeting these criteria. Titles and abstracts were carefully read, and OC from tongue and ocular region were excluded, taking into account that these anatomical regions represent the most common affected sites. Therefore, a review of the OS diagnosed at the head and neck region (excluding tongue and eye) would be of great importance in order to reach a good overview of its profile. We have included in our collecting data cases that have matched not only appropriate clinical documentation but also histological identification of tumor-like masses presenting bone formation without neoplastic characheristics. Thus, a final number of 17 papers matched these criteria and were considered. The reviewed papers described 18 patients with OC in head and neck region with ages ranging from 2 to 75 years (mean age of 40.8 years), without a peculiar age distribution. There were 9 males and 9 females. The duration of the lesions ranged from three months to more than 40 years (Table 1). The majority of the cases were asymptomatic. A case that occurred in the masseter muscle presented as a non- swelling mass, slowly expanding, located at the left facial region [1]. In this way, our present case shared almost these same basic features. In another case there was an interference in the mouth opening and a previous episode of trauma [5, 6]. One patient presented some developmental malformations, like plagiocephaly, bifid uvula with a submucosal cleft, minor facial asymmetry and speech problems [12]. Page 4 of 6

5 Author Year Patient age Table 1. Summary of published cases of OC of head and neck (PubMed Database). Gender Location Lesion Size Number of lesions Page 5 of 6 Symptoms Symptoms duration Krolls et al [2] a M Buccal mucosa NR 1 tumor 1 years (cheek) Herd [14] F Buccal Mucosa 10 x 7 mm 1 40 years Davis [9] M Buccal Mucosa 5 mm 1 1 years Mesa et al [16] M Buccal Mucosa 18 x 17 x 10 mm 1 asymptomatic 1 years Sheridan [7] b M Alveolus 15 mm 1 asymptomatic 15 years Sookasam and Philipsen [8] F Buccal Mucosa Almond size 1 2 years Sookasam and Philipsen [8] F Buccal Mucosa Halzenut size 1 NR Tohill et al [4] c F Retromolar pad 15 x 12 x 5 mm 2 problems with 2 years denture Hodder and MacDonald [12] F Buccal Mucosa 20 x 11 x 6 mm 1 6 months Long and Koutnik [5] F Buccal Mucosa 12 x 11 x 6 mm 1 interference in NR the mouth opening Psimopoulou and Antoniades F Submental region 7 x 5 x 3 mm 2 swelling, not 8 months [11] 20 x 15 mm Lin et al [15] M Buccal Mucosa 20 x 20 x 15 mm 1 5 years Gaitan-Cepeda et al [13] F Buccal Mucosa 20 x 10 x 10 mm 1 4 years Dalkiz et al [1] M Masseter muscle 3-20 mm 8 tenderness, not 5 years Johan et al [10] M Submandibular region 20 mm 1 asymptomatic swelling 3 months Kamburoglu et al [6] F Submandibular 15 x 20 x 10 mm 1 swelling, not 1 years region Noroozi and Arora [17] M Buccal mucosa 25 x 20 mm 1 swelling, not 6 months (cheek) Goswamy [3] M Buccal Mucosa 10 x 8 x 8 mm 1 asymptomatic 1 year NR- not reported; mm- milimeter. a This study presented 9 patients, 8 with OC in the tongue submucosal, and one patient with lesion in the cheek. This analysis refers just to this patient, since the lingual choristomas are excluded from this review. b This study showed 2 cases, but just one included in the criteria. c This study presented 3 patients, just one met the inclusion criteria. [2, 3, 5, Twelve cases presented the OC in the buccal mucosa 8, 9, 12-17]. There was only a single case of an OC occurring in the masseter muscle [1]. The lesions size varied from significantly across the cases report (3 mm to 20 mm). In such circumstance, the osseous process could be associated with ossification of a branchial arc remains, development of ectopic mesenchymal cells and osseous differentiation. Furthermore, metaplastic ossification changes occurring in a degenerating fibroma, lymphatic or fibrous stromal tissue calcification must also be taken into account [1]. It is important to mention that the clinical, histopathological and radiographic features of the unique case found in masseter are very similar to our report. Surgical excision is the main treatment modality of OC [10]. Usually the lesions do not recur [2] with only two cases of recurrence being reported in the literature [1, 5]. Histologically the OC demonstrates mature lamellar pattern with trabecular appearance. The intertrabecular spaces show enclose haemopoietic marrow showing scarce fibroblastic spindle cells and blood vessels within the connective tissue [10]. To sum up, it is important that the scientific community recognizes variations in the location of the OC and can make a correct diagnosis followed by effective treatment. In addition, a close clinical and radiographic follow-up is of utmost importance after surgical excision of an OC, despite its benign nature. Conflicting Interests The author(s) declare that they have no Conflicting interests. Author contributions PSCL has been involved in drafting the manuscript; EAR and GPG have made substantial contributions to conception and design; RFBA has made substantial contributions to conception and design and has been revising it critically for important intellectual content. References 1. Dalkiz M, Hakan Yurdakul R, Pakdemirli E, Beydemir B. Recurrent osseous choristoma of the masseter muscle: case report. J Oral Maxillofac Surg 2001; 59: Krolls SO, Jacoway JR, Alexander WN. Osseous choristomas (osteomas) of intraoral soft tissues. Oral Surg Oral Med Oral Pathol 1971; 32: Goswamy M, Tabasum S, Kudva P, Gupta S. Osseous choristoma of the periodontium. J Indian Soc Periodontol 2012; 16: Tohill MJ, Green JG, Cohen DM. Intraoral osseous and cartilaginous choristomas: report of three cases and review of the literature. Oral Surg Oral Med Oral Pathol 1987; 63: Long DE, Koutnik AW. Recurrent intraoral osseous choristoma. Report of a case. Oral Surg Oral Med Oral Pathol 1991; 72:

6 Kamburoglu K, Ozen T, Sencimen M, Ortakoglu K, Günhan O. Osseous choristoma of the submandibular region: case report. Dentomaxillofac Radiol 2009; 38: Sheridan SM. Osseous choristoma: a report of two cases. Br J Oral Maxillofac Surg 1984; 22: Sookasam M, Philipsen HP. The intra-oral soft tissue osteoma: report of two cases. J Dent Assoc Thai 1986; 36: Davis GB. Intraoral osseous choristoma: report of case. J Oral Surg 1980; 38: Johann AC, Garcia BG, Nacif TR, de Freitas JB, do Carmo MA, Mesquita RA. Submandibular osseous choristoma. J Craniomaxillofac Surg 2006; 34: Psimopoulou M, Antoniades K. Submental osseous choristoma: a case report. J Oral Maxillofac Surg 1998; 56: Hodder SC, MacDonald DG. Osseous choristoma of buccal mucosa: report of a case. Br J Oral Maxillofac Surg 1988; 26: Gaitan-Cepeda LA, Quezada-Rivera D, Ruiz-Rodriguez R. Osseous choristoma of the oral soft tissue. Case report. Med Oral 2003; 8: Herd JR. Extra-osseous osteoma. Aust Dent J 1976; 21: Lin CC, Chen CH, Chen YK, Shen YH, Lin LM. Osseous choristoma of oral cavity--report of two cases and review of the literature. Kaohsiung J Med Sci 1998; 14: Mesa ML, Schneider LC, Northington L. Osteoma of the buccal mucosa. J Oral Maxillofac Surg 1982; 40: Noroozi AR, Arora E. Odontogenic choristoma: report of a case. J Oral Maxillofac Surg 2011; 69: Page 6 of 6

Article in press. Lingual Osseous Choristoma: Report of Three Cases. Case Report

Article in press. Lingual Osseous Choristoma: Report of Three Cases. Case Report Case Report Lingual Osseous Choristoma: Report of Three Cases Sompid Kintarak 1, Saipin Cheunchoksant 2, Thongchart Siripan 3, Patcharee Kumplanont 3 1 Department of Stomatology, Faculty of Dentistry,