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Early View Article: Online published version of an accepted article before publication in the final form. Journal Name: Journal of Case Reports and Images in Dentistry Type of Article: Case Report Title: A Case Report of Osteochondroma of the Mandibular Condyle Authors: Icoz Derya, Akgunlu Faruk, Demir Esin, Karabağlı Pınar, Ozturk Kayhan doi: To be assigned Early view version published: January 14, 2016 How to cite the article: Derya I, Faruk A, Esin D, Pınar K, Kayhan O. A Case Report of Osteochondroma of the Mandibular Condyle. Journal of Case Reports and Images in Dentistry. Forthcoming 2016. Disclaimer: This manuscript has been accepted for publication. This is a pdf file of the Early View Article. The Early View Article is an online published version of an accepted article before publication in the final form. The proof of this manuscript will be sent to the authors for corrections after which this manuscript will undergo content check, copyediting/proofreading and content formatting to conform to journal s requirements. Please note that during the above publication processes errors in content or presentation may be discovered which will be rectified during manuscript processing. These errors may affect the contents of this manuscript and final published version of this manuscript may be extensively different in content and layout than this Early View Article. Page 1 of 13

1 2 3 4 5 6 7 8 9 10 11 12 13 14 15 16 17 18 19 20 21 22 23 24 25 26 27 28 29 TYPE OF ARTICLE: Case Report TITLE: A Case Report of Osteochondroma of the Mandibular Condyle AUTHORS Icoz Derya 1, Akgunlu Faruk 1, Demir Esin 2, Karabağlı Pınar 3, Ozturk Kayhan 4 AFFILIATIONS 1 Selcuk University, Faculty of Dentistry, Department of Oral Diagnosis and Radiology, Konya, Turkey 2 phd Student, Selcuk University, Faculty of Dentistry, Department of Oral and Maxillofacial Surgery, Konya, Turkey 3 Assistant professor, Selcuk University, Faculty of Medicine, Department of Pathology, Konya, Turkey 4 Professor, Selcuk University, Faculty of Medicine, Department of Otolaryngology, Konya, Turkey CORRESPONDING AUTHOR DETAILS Icoz Derya, Selçuk University, Faculty of Dentistry Department of Oral Diagnosis and Radiology Campus Selçuklu/konya, Turkey Phone number: 0090 5398711624 Derya Icoz, Konya, Turkey, 42074 Phone number: 0090 5398711624 Email: dyilmaz.icoz@hotmail.com Short Running Title: Osteochondroma of the Mandible Guarantor of Submission: The corresponding author is the guarantor of submission. 30 31 32 Page 2 of 13

33 34 35 36 37 38 39 40 41 42 43 44 45 46 47 48 49 50 51 52 53 54 55 56 57 58 TITLE: A Case Report of Osteochondroma of the Mandibular Condyle ABSTRACT Introduction Osteochondroma is one of the most common benign bone tumors of axial skeleton but it is rarely seen in maxillofacial region. Only about 1% of these occur within the head and neck region but when it presents more than half of these appear in coronoid process. Osteochondroma of the mandibular condyle is very rare. The clinical presentation of condylar osteochondroma mostly includes malocclusion, facial asymmetry, temporomandibular joint dysfunctions and disturbances during mouth opening and radiographically, the lesion presents an enlargement of the affected structure and a slight radiopacity increase. Case Report Here, we describe a case of osteochondroma affecting the right mandibular condyle of a male patient with the chief complaints of facial asymmetry and temporomandibular joint dysfunctions. Conclusion Although osteochondroma is not a frequent lesion for maxillofacial area, it should be considered in the differential diagnosis of masses in the temporomandibular joint region. Keywords: Osteochondroma, computed tomography, panoramic radiography, mandibular condyle, temporomandibular joint 59 60 61 62 63 64 Page 3 of 13

65 66 67 68 69 70 71 72 73 74 75 76 77 78 79 80 81 82 83 84 85 86 87 88 89 90 91 92 93 94 95 TITLE: A Case Report of Osteochondroma of the Mandibular Condyle INTRODUCTION Osteochondroma is a benign, slow-growing osseous protuberance with cartilagenous growth potential, projects from surface of the bone usually near its growth center [1,2]. Although osteochondromas are mostly osseous, the entity is usually regarded as one of the cartilagenous lesions due to the endochondral ossification of cartilage cap [1]. The etiology of the lesion is unclear but discussions about its neoplastic, develomental and reperative nature is frequent [3]. It may occur at any age and females are more commonly affected than males [4,5]. It is one of the most common benign tumors of the axial skeleton, it constitutes approximately 20-50% of all benign tumors and 10-15% of all bone tumors, but only about 1% of these occur within the head and neck region [6,7]. The reason for the rare occurence of osteochondroma in this anatomical area is the intramembranous development of these bones, but because of the embryonic development of the temporomandibular joint by the endochondral ossification, temporomandibular joint is the most frequent facial site for osteochondroma and in this anatomical area the tumor is most often reported in relation with the coronoid process [8,9]. The other reported sites for osteochondroma in the head are cranial base, jaws, posterior maxilla, maxillary sinuses, ramus, body, coronoid process and symphyseal region of the mandible. Osteochondroma of the mandibular condyle grows slowly, and so, the symptoms may develop over a long period [1,8]. The most common clinical symptoms include malocclusion, progressive facial asymmetry, restricted mandibular movements, clicking, popping and crepitation of the affected joint and changes in the condylar morphology[1,5]. Condylectomy is usually curative for this lesion, the possibility of recurrence is a disadvantage although only one recurrence of a case has been reported for mandibular condyle which occured one year after the excision [1,10]. The current report describes the diagnostic features and treatment planning of an osteochondroma affecting the right mandibular condyle which is a rare location for osteochondroma. 96 Page 4 of 13

97 98 99 100 101 102 103 104 105 106 107 108 109 110 111 112 113 114 115 116 117 118 119 120 121 122 123 124 125 126 127 128 CASE REPORT A 34 year old male patient was referred to our department with chief complaints of asymmetry and difficulty in opening and closing of the mouth and mastication approximately for two years. He didn t seek any medical therapy until he admitted to our clinic, there was no history of facial trauma and he had no systemic diseases. On clinical examination there was a painless bony, hard swelling on the right preauricular region. The symptoms were restricted inter-incisal range, malocclusion, deviation during mouth opening and prominent facial asymmetry (Figure 1). The overlying skin was in normal color and texture. No evidence of soft tissue involvement was noted. The lesion was fixed to the underlying bone and was nonmobile. Intra-oral examination revealed contralateral cross-bite and restricted mandibular movements. Panoramic radiograph of the patient revealed mandibular asymmetry and a welldefined bone enlargement on the right condylar head with slight radiopacity increase. Slow displacement of mandible results with adaptation of bony structures for compensation. Therefore compared with the contralateral side increase in height of condylar neck and thickness of mandibular body were observed on panoramic radiography (Figure 2). For more detailed radiographic examination patient was adviced a computed tomography (CT) scan. The axial and coronal CT sections revealed a clearly distinguishable cartilaginous/bony outgrowth arising from the right condylar head and length increase of the condylar neck. The lesion was extending from anteromedial aspect of the condyle into the articular fossa (Figure 3). Patient was referred to maxillofacial surgery department with the provisional diagnosis of osteochondroma. Lesion was excised under general anesthesia and nasotracheal intubation with preauricular incision. Active jaw motion exercises including jaw opening, lateral excursion and protrusion were performed for 3 weeks. The size of the excised lesion was approximately 1.8x1.6x1.3 cm. The histopathologic examination revealed a nodular lesion with cartilaginous cap and mature bone tissue beneath, confirming the diagnosis of osteochondroma of the mandibular condyle (Figure 4). After 2 years follow-up of the patient mouth opening and TMJ functions were in normal limits but slight facial asymmetry and occlusal can t still remained. However occlusion was in Page 5 of 13

129 130 131 132 133 134 135 136 137 138 139 140 141 142 143 144 145 146 147 148 149 150 151 152 153 154 155 156 157 158 159 160 acceptable range (Figure 5). Postoperative panoramic radiography of the patient proved condylar remodeling and a more acceptable appearance in terms of symmetry (Figure 6). DISCUSSION Although osteochondromas of the mandibular condyle is rare, early identification is essential to provide timely treatment and to prevent dramatic impacts on TMJ such as severe pain, hypomobility, clicking and locking of the TMJ, as well as headaches and cervical pain [8,9]. Trauma and inflammation have been implicated as predisposing or initiating factors but this is not valid for all cases of osteochondroma including the present case, the patient had no history of trauma to related region. Review of the literature reveals osteochondromas are frequently seen in second and third decades of the life and males are more commonly affected than males, however osteochondroma of the mandibular condyle is mainly seen in fourth decade of life with a mean age of 39.7 years and females are more commonly affected than males [1,9]. The symptoms of the tumor vary depending on the location and the most common locations of the condylar osteochondroma is the medial aspect of the condyle (52%), followed by anterior location (20%) and rarely in the lateral or superior positions (<1%). The growth of an osteochondroma is usually slow, causing gradual displacement and elongation of the mandible [8,9]. Meng et al reviewed that hyperplasia of ramus and body of mandible on the affected side may be seen in condylar osteochondroma cases. Horizontal mandibular deviation caused by tumor is compensated by maxillary vertical overdevelopment and/or mandibular enlargement [4]. In our case hemimandibular hypertrophy with increased vertical height of entire mandible and slight compensatory vertical overdevelopment of maxilla was seen. The careful assessment of the patient s history and radiographic evaluation provide valuable information for diagnosis and treatment planning of the TMJ lesions. Differential diagnosis of the TMJ lesions is not always very easy. Osteochondroma should be distinguished from condylar hyperplasia, osteoma, chondroma, chondroblastoma, benign osteoblastoma, giant cell tumor, myxoma, fibro-osteoma, fibrous dysplasia, fibrosarcoma and chondrosarcoma [8]. The radiographic Page 6 of 13

161 162 163 164 165 166 167 168 169 170 171 172 173 174 175 176 177 178 179 180 181 182 183 184 185 186 187 188 189 190 191 192 appearance of the condylar osteochondroma is an enlarged sclerotic mass of the condyle and CT is an invaluable imaging tool for treatment planning and differential diagnosis of the tumor especially with the unilateral condylar hyperplasia. CT clearly depicts the continuation of the cortex and medulla of the parent bone with the tumor and is the best imaging option for calcified cartilage cap [9]. Common clinical manifestations of the condylar osteochondroma were compatible with the present case [4]. Despite the integration of the clinical signs with osteochondroma the diagnosis of the present case report was done based on the combination of clinical, radiological and histopathological findings for definite diagnosis. The aim of the treatment of a condylar osteochondroma should be achieving the acceptable mouth opening, recover facial asymmetry, establish facial harmony and occlusion [8]. In the present case the treatment was curative and acceptable as functional but facial asymmetry could not be provided completely due to the enlargement of the hemimandible but patient refused corrective surgery for facial asymmetry. CONCLUSION Although osteochondroma is not a frequent lesion for maxillofacial area, it should be considered in the differential diagnosis of masses in the temporomandibular joint region, mandibular symphysis, maxillar sinuses, mandibular corpus and ramus. For more appropriate treatment method, a reliable diagnosis of the osteochondroma is necessary. Panoramic radiography and CT evaluation should be performed for suspected condylar osteochondroma cases and for the final decision the diagnosis should be supported with histopathologic examination. CONFLICT OF INTEREST None AUTHOR S CONTRIBUTIONS Derya Icoz Group 1: Substantial contributions to conception and design, acquisition of data Group 2: Drafting the article, revising it critically for important intellectual content Page 7 of 13

193 194 195 196 197 198 199 200 201 202 203 204 205 206 207 208 209 210 211 212 213 214 215 216 217 218 219 220 221 222 223 224 Group 3: Final approval of the version to be published Faruk Akgunlu Group 1: Substantial contributions to conception and design, acquisition of data Group 2: Drafting the article, revising it critically for important intellectual content Group 3: Final approval of the version to be published Esin Demir Group 1: Substantial contributions to conception and design, acquisition of data Group 2: Drafting the article, revising it critically for important intellectual content Group 3: Final approval of the version to be published Pınar Karabaglı Group 1: Substantial contributions to conception and design, acquisition of data Group 2: Drafting the article, revising it critically for important intellectual content Group 3: Final approval of the version to be published Kayhan Ozturk Group 1: Substantial contributions to conception and design, acquisition of data Group 2: Drafting the article, revising it critically for important intellectual content Group 3: Final approval of the version to be published REFERENCES 1. Ongole R, Pillai RS, Ahsan AK, Pai KM. Osteochondroma of the mandibular condyle. Saudi Med J 2003;24(3):213-16. 2. Kishore DN, Kumar HRS, Umashankara KV, Rai KK. Osteochondroma of the Mandible: A Rare Case Report. Case Reports in Pathology 2013;Article ID:167862 doi:10.1155/2013/167862. 3. Yamamoto FP, Silva BSF, Modes RW, Fonseca FP, Pontes FSC, Sousa SC. Computed tomography and scintigraphy for diagnosis and treatment planning of the condylar osteochondroma: A case report. Rev Odonto Cienc 2010;25(4):422-26. Page 8 of 13

225 226 227 228 229 230 231 232 233 234 235 236 237 238 239 240 241 242 243 244 245 246 247 248 249 250 251 252 253 254 4. Meng Q, Chen S, Long X, Cheng Y, Deng M, Cai H. The clinical and radiographic characteristics of condylar osteochondroma. Oral Surg Oral Med Oral Pathol Oral Radiol 2012;114(1):66-74. 5. Kumar A, Omprakash TL, Goudar GN, Kumar R. Osteochondroma of the mandibular condyle. Journal of Dental Sciences and Research 2010;1(2):57-66. 6. Murphey MD, Choi JJ, Kransdorf MJ, Fleming DJ, Gannon HF. Imaging of osteochondroma: variants and complications with radiographic-pathologic correlation. Radiographics 2000;20:1407-34 7. Samir K and El-Mofty, Diagnostic Surgical Pathology of the Head and Neck, Saunders, Philadelphia, Pa, USA, 2nd edition, 2009. 8. More CB, Gupta S. Osteochondroma of mandibular condyle: A clinicoradiographic correlation. Journal of Natural Science, Biology and Medicine 2013; 4(2):465-68. 9. Avinash KR, Rajagapol KV, Ramakrishnaiah, Carnelio S, Mahmood NS. Computed tomographic features of mandibular osteochondroma. Dentomaxillofacial Radiology 2007; 20:434-36. 10. Otero SG, Cuellar CN, Teigeiro ME, Luenga JFA, Vila CN. Osteochondroma of the mandibular condyle: resection and reconstruction using vertical sliding osteotomy of the mandibular ramus. Med Oral Pathol Cir Bucal 2009;14(4):194-97. FIGURE LEGENDS Figure 1. Pre-operative extraoral view of the patient showing prominent facial asymmetry. Figure 2. Panoramic radiography of the patient reveals asymmetry on the right side, well defined bone enlargement on the condylar head, length increase on condylar neck and hemimandibular hypertrophy. 255 Page 9 of 13

256 257 258 259 260 261 262 263 264 265 266 267 268 Figure 3. Coronal and axial CT sections show the bony/cartilagenous enlargement of the condyle. Figure4. The histopathologic image is showing the osteochondroma with cartilaginous cap, mature bone tissue and marrow tissue. (HEX 50) Figure 5: Post-operative images of the patient show acceptable mouth opening and occlusion although a slight facial asymmetry and occlusal cant remained. Figure 6: After 2 years follow-up post-operative panoramic radiography showing the excised condyle and condylar remodelling. FIGURES 269 270 271 272 Figure 1. asymmetry. Pre-operative extraoral view of the patient showing prominent facial 273 Page 10 of 13

274 275 276 277 278 Figure 2. Panoramic radiography of the patient reveals asymmetry on the right side, well defined bone enlargement on the condylar head, length increase on condylar neck and hemimandibular hypertrophy. 279 280 281 282 283 Figure 3. Coronal and axial CT sections show the bony/cartilagenous enlargement of the condyle. 284 Page 11 of 13

285 286 287 288 Figure 4: The histopathologic image is showing the osteochondroma with cartilaginous cap, mature bone tissue and marrow tissue. (HEX 50) 289 290 291 Page 12 of 13

292 293 294 295 Figure 5: Post-operative images of the patient show acceptable mouth opening and occlusion although a slight facial asymmetry and occlusal cant remained. 296 297 298 299 300 Figure 6: After 2 years follow-up post-operative panoramic radiography showing the excised condyle and condylar remodelling. Page 13 of 13