Rare Benign Tumors of the Mandibular Condyle: Report of 2 Cases and Literature Review

Rare Benign Tumors of the Mandibular Condyle: Report of 2 Cases and Literature Review

1830 RARE BENIGN TUMORS OF THE MANDIBULAR CONDYLE J Oral Maxillofac Surg 65:1830-1835, 2007 Rare Benign Tumors of the Mandibular Condyle: Report of...

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1830

RARE BENIGN TUMORS OF THE MANDIBULAR CONDYLE

J Oral Maxillofac Surg 65:1830-1835, 2007

Rare Benign Tumors of the Mandibular Condyle: Report of 2 Cases and Literature Review Belmiro Cavalcanti do Egito Vasconcelos, DDS, PhD,* Gabriela Granja Porto, DDS,† and Ricardo Viana Bessa-Nogueira, DDS, MSc‡ There have been few reports of tumors in the mandibular condyle, in particular pure chondromas, with 6 cases reported, and osteoid osteomas, with only 2 cases in the literature reviewed (Table 1).1-8 Chondromas are benign tumors of mature hyaline cartilage6 that are rarely seen in the facial bones.5 Osteoid osteoma is a very painful but small bone lesion with its maximum diameter rarely exceeding 2 cm and consisting of a central nidus of osteoid, atypical bone, and a discrete surrounding zone of reactive bone sclerosis.7,9,10 It is also rarely seen in the jaws.9,11 This article describes a case of a young woman with an osteoid osteoma located at the right mandibular condyle associated with the displacement of the right temporomandibular disc, which was treated by resection of the tumor and placement of the disc. A case of condylar enchondroma is also presented that was treated by condylectomy; the reshaped condyle was used for immediate articular reconstruction and to reestablish the height of the mandibular ramus. The clinical symptoms, anatomic features, histologic characteristics, and differential diagnosis of these lesions are discussed.

Received from the University of Pernambuco, Recife, Brazil. *Senior Lecturer in Oral and Maxillofacial Surgery, Director of the MSc and PhD Programs in Oral and Maxillofacial Surgery. †Postgraduate Student of the MSc Program in Oral and Maxillofacial Surgery. ‡Postgraduate Student of the PhD Program in Oral and Maxillofacial Surgery. Address correspondence and reprint requests to Dr Vasconcelos: Faculdade de Odontologia de Pernambuco, Departamento de Cirurgia e Traumatologia BMF, Av. General Newton Cavalcanti, 1650 Camaragibe—PE; e-mail: [email protected] © 2007 American Association of Oral and Maxillofacial Surgeons

0278-2391/07/6509-0027$32.00/0 doi:10.1016/j.joms.2006.06.262

Report of Cases CASE 1 A 25-year-old Brazilian woman was referred to the Division of Oral and Maxillofacial Surgery of Oswaldo Cruz Hospital, Recife-PE, in 2001, complaining of pain in the region of the left temporomandibular joint (TMJ) and limited mouth opening, which had been present for about 5 years. On examination, no facial asymmetry was clinically visible and the overlying skin in the left preauricular region appeared normal. Maximal mouth opening was 11 mm with deviation of the mandible to the left side. No malocclusion was observed. Panoramic radiograph revealed a well-circumscribed and predominantly radiopaque image at the left condylar head, but these findings were not totally clear. Computed axial tomography (CAT) scans were performed to fully assess the degree of bone loss and any soft tissue component. The images showed an irregularly shaped lesion at the left condyle. This mostly dense mass extended superiorly and medially to the left condylar head (Fig 1). The patient was hospitalized for a complete resection with bony margin of the tumor and immediate reconstruction of the left TMJ. Exposure of the left TMJ was conducted by a preauricular approach as described by Ellis and Zide.12 The tumor was resected and the intraoperative findings corresponded with a well-circumscribed lesion of a light blue color, probably of cartilaginous origin (Fig 2). An L-shaped osteotomy was performed through a retromandibular incision at the level of the sigmoid notch and the proximal stump was reshaped with a bur extracorporeally, advanced superiorly into the joint space, and fixed to the distal mandibular ramus with miniplates and screws (2 mm).13 The L-shaped osteotomy was chosen because it is a technique the surgeon was familiar with and which has proved to be appropriate for repositioning the proximal stump, thereby ensuring a good reconstruction of the TMJ and the reestablishment of the height of the mandibular ramus.13 The disc was seen to be greatly deformed and consequently removed. There was no intermaxillary fixation; in the immediate postoperative period the patient was already being followed up by a physiotherapist. The histopathologic report described a benign tumor of mature hyaline cartilage consisting of a homogeneous mass of uniform chondrocytes with small nuclei arising centrally from the medullary cavity of the condyle that was diagnosed as enchondroma (Fig 3).

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VASCONCELOS, PORTO, AND BESSA-NOGUEIRA

Table 1. PREVIOUSLY REPORTED CASES OF THE TUMORS (CHONDROMA AND OSTEOID OSTEOMA) IN THE MANDIBULAR CONDYLE

Study

Age (yrs)

Gender

Site

Tumor Type

Lurie, 19751 Jokinen et al, 19762 Deboise et al, 19813 Van Hemeltyck et al, 19934 Chandu et al, 19975 Lazow et al, 19986 Lind et al, 19647 Tochihara et al, 20018

28 33 25 36 66 17 48 21

Female Female Male Female Male Female Male Female

Left condyle Right condyle Right condyle Left condyle Right condyle Right condyle Right condyle Left condyle

Enchondroma Juxtacortical chondroma Juxtacortical chondroma Juxtacortical chondroma Enchondroma Enchondroma Osteoid osteoma Osteoid osteoma

Vasconcelos, Porto, and Bessa-Nogueira. Rare Benign Tumors of the Mandibular Condyle. J Oral Maxillofac Surg 2007.

Using the House and Brackmann grading system,14 the patient was classified in the immediate postoperative period as grade I, which means there were no signs of facial nerve paralysis or paresthesia. The surgeons recommended that the patient also be followed up by a physiotherapist in the immediate postoperative period. Within 6 months the maximal mouth opening was 36 mm and no occlusion alterations were observed. The patient’s follow-up continued for another 24 months, when the last panoramic radiograph was taken, which showed no alteration in the bone structures and no signs of recurrence (Fig 4). CASE 2 A 23-year-old Brazilian woman complained of severe spontaneous pain around the right TMJ, and this finding was associated with a limited maximal mouth opening (19 mm) with the mandible showing a deviation to the right and a normal occlusion was observed. On examination, a facial asymmetry was clinically visible and the overlying skin in the right preauricular region appeared to be normal. The patient reported that these signs and symptoms had been present for 5 years. During this period her complaint had

FIGURE 1. Case 1: CT showing an irregularly shaped lesion at the left condyle and 3-dimensional reconstruction showing the erosion in the left condyle. Vasconcelos, Porto, and Bessa-Nogueira. Rare Benign Tumors of the Mandibular Condyle. J Oral Maxillofac Surg 2007.

worsened gradually and she had been seen by several professionals, none of whom had made a conclusive diagnosis. A palliative treatment had been prescribed to the patient, using nonsteroidal anti-inflammatory drugs (NSAID) but with no apparent success. At the time of the first appointment, the patient already had a panoramic radiograph that revealed a well-circumscribed and predominantly radiopaque image at the right condylar head; however, this finding was not totally clear. CAT scans and a magnetic resonance imaging (MRI) were then performed to fully assess the anatomic boundaries of the lesion and to evaluate the density of the initial image. The tomographic images showed a regularly shaped 1.1 ⫻ 0.8 cm lesion at the right condyle. This mostly dense mass of apparently limited growth had a central nidus and a discrete surrounding zone of reactive bone at the right condylar head. The MRI showed that the right temporomandibular disc was displaced (Fig 5). On the basis of clinical and imaging findings, an initial diagnosis of benign condylar neoplasm was proposed. The patient was hospitalized for a complete resection of the neoplasm with bony margin and immediate plication of the displaced disc. Exposure of the left TMJ was performed by

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RARE BENIGN TUMORS OF THE MANDIBULAR CONDYLE physiotherapist. Twelve months after the operation the patient no longer reported any pain; no facial asymmetry was seen and her maximal mouth opening was 34 mm. The final panoramic radiograph at 12 months showed no major alteration of the bone structures of the TMJ or signs of recurrence (Fig 8). In both cases an intraoperative frozen section of the surrounding tissues was reviewed by a pathologist, which ruled out the hypothesis of a malignant lesion.

Discussion

FIGURE 2. Case 1: A and B, Intraoperative view of a well-circumscribed lesion.

Chondroma and osteoid osteoma are benign tumors that have limited growth potential.10,11,16 The former is a tumor of mature hyaline cartilage that can arise within the medullary cavity of a bone, in which case it is called an “enchondroma.”6 It can also develop adjacent to the cortical surface of the bone, but beneath the periosteum, when it is called “periosteal” or “juxtacortical chondroma.”4,6 For its part, the osteoid osteoma is an osteoblastic tumor.10,16 Chondromas are mostly encountered in the long tubular bones and in the hand, particularly in its small bones, such as the proximal phalanges. The neoplasm is usually discovered in the third and fourth decade of life and shows no sex predilection.18 Osteoid osteoma is most common in young persons,10 and is rarely

Vasconcelos, Porto, and Bessa-Nogueira. Rare Benign Tumors of the Mandibular Condyle. J Oral Maxillofac Surg 2007.

the preauricular approach described by Ellis and Zide.12 The neoplasm was resected and the intraoperative findings were those of a well-circumscribed lesion (Fig 6A). The disc was repositioned using a modified condylar disc placation, rather than the Mitek anchor described by Mehra and Wolford,15 a standard fixation screw (2 mm) was inserted 8 to 10 mm below the superior aspect of the condyle and just lateral to the midsagittal plane. After placement of the screw a polypropylene suture 3-0 was double threaded around the head of the screw. Finally, the temporomandibular disc was sutured to the screw (Fig 6B). There was a minor loss of the vertical height of the mandibular ramus of approximately 2 mm. No intermaxillary fixation was performed. In the immediate postoperative period the patient was already being followed up by a physiotherapist. The histopathologic report described a benign neoplasm of a fibrotic connective nature. The nidus was described as an interlacing, unevenly mineralized meshwork of osteoid trabeculae, without inflammatory cells. There was hypertrophia of the osteoblasts and osteoclasts that delimited the bone trabeculae, which indicated that the bone was in the process of reconstruction. The lesion did not seem to invade the periosteum or infiltrate the soft tissues (Fig 7). These features suggested a histopathologic diagnosis of osteoid osteoma. Using the House and Brackmann grading system,14 the patient was classified in the immediate postoperative period as grade I, which means there were no signs of facial nerve paralysis or paresthesia. During the immediate postoperative period the surgeons recommended that the patient also be followed up by a

FIGURE 3. Case 1: A, Photomicrograph showing a homogenous mass of chondrocytes arising from the medullary cavity of the condyle. (Magnification ⫻100.) B, Photomicrograph showing the relationship between the homogenous cartilage mass and the bone marrow of the mandibular condyle. (Magnification ⫻200.) Vasconcelos, Porto, and Bessa-Nogueira. Rare Benign Tumors of the Mandibular Condyle. J Oral Maxillofac Surg 2007.

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FIGURE 4. Case 1: Postoperative panoramic radiograph showing no alteration of the bone structures and no signs of recurrence (24 month follow-up). Vasconcelos, Porto, and Bessa-Nogueira. Rare Benign Tumors of the Mandibular Condyle. J Oral Maxillofac Surg 2007.

seen in individuals over 30 years of age.21 It is more common in men than women by a ratio of 3 to 1.18 It is generally located in the tibia or femur in approximately 50% of the reported cases, but all bones may be involved.7,18,21 Most enchondromas are asymptomatic and are detected as incidental radiographic abnormalities.5,16 Symptomatic lesions may present as a painless swelling.17 For osteoid osteoma, the major complaint is pain of gradually progressive severity,18 which at first

is mild and intermittent, but later becomes severe, constant, and more pronounced.8,19,20 This pain is not responsive to NSAIDs and is especially severe at night,8,11,21 which is an important point for the differential diagnosis from arthritis or other joint disease when the lesion occurs in the mandibular condyle.8 Radiographically, chondromas are predominantly lucent with some mineralization.3 The calcified foci vary from powder-like to dense aggregates.17 Radiographically, osteoid osteoma typically shows a central radiolucent area (the nidus) that is surrounded by a dense radiopaque border,11,21 which is a reversible change that disappears after the nidus is removed.18 The central nidus remains central and does not tend to erupt and invade the soft tissues.22 In the enchondroma, microscopically, a lobular arrangement of a neoplastic cartilage may be surrounded by bone narrow between trabeculae of normal bone.17 Enchondromas consist of normal-looking chondrocytes in lacunar spaces separated by an abundant hyaline chondroid matrix.1 Mitotic figures are

FIGURE 5. Case 2: A, MRI showing the displacement of the tempromandibular disc (white arrow). B, CT showing a regularly shaped lesion at the right condyle.

FIGURE 6. A, Intraoperative view of a well-circumscribed lesion. B, Trans-surgical aspect of disc plication.

Vasconcelos, Porto, and Bessa-Nogueira. Rare Benign Tumors of the Mandibular Condyle. J Oral Maxillofac Surg 2007.

Vasconcelos, Porto, and Bessa-Nogueira. Rare Benign Tumors of the Mandibular Condyle. J Oral Maxillofac Surg 2007.

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FIGURE 7. Case 2: A, Photomicrograph showing bone trabeculae with fibrotic medulla. (Magnification ⫻100.) B, Photomicrograph showing hypertrophic osteoblasts and hyperactive osteoblasts delimiting the bone trabeculae. (Magnification ⫻200.) Vasconcelos, Porto, and Bessa-Nogueira. Rare Benign Tumors of the Mandibular Condyle. J Oral Maxillofac Surg 2007.

rare.6 Fine-needle aspiration, punch biopsy, or intraoperative frozen section are not indicated because of the calcified nature of the tumor.6 In the osteoid osteoma, microscopically, the major component of the nidus is a meshwork of osteoid trabeculae of various degrees of mineralization on a background of usually vascular fibrous connective tissue.18 It originates in spongy bone and sometimes involves cortical bone

RARE BENIGN TUMORS OF THE MANDIBULAR CONDYLE

but does not penetrate the periosteum or infiltrate the soft tissues.23,24 The differential diagnosis of radiopaque TMJ tumors such as enchondromas includes osteoma, osteoblastoma, osteochondroma, condylar hyperplasia, chondroma, chondroblastoma, chondromatosis, chondrosarcoma, and osteosarcoma.6 Although the differential diagnosis lies mainly with chondrosarcoma,17 the microscopic distinction between benign chondroma and low-grade chondrosarcoma may be very difficult,3,6,17 reflecting their common radiographic and clinical findings such as the presence of pain and evidence of destruction of the bone cortex.6 On the basis of the size of the lesion, osteoid osteoma may be differentiated from osteoblastoma. Lesions with a maximum diameter of less than 1.5 to 2 cm are classified as osteoid osteoma, and those larger than this as osteoblastoma.10,11 Certain fibro-osseous lesions, such as ossifying fibroma and fibrous dysplasia, should not be confused with osteoid osteoma because they are asymptomatic, grow to a large size, cause teeth displacement, and have no nidus.18 From the pathologic standpoint the differential diagnosis of osteoid osteoma could be confused with an intracortical bone abscess (Brodie’s abscess)9,22 and sclerosing nonsuppurative osteomyelitis.22 In these inflammatory lesions, nests of inflammatory cells are to be found, which are absent from the lesions of osteoid osteoma.22 Such lesions may also be mistaken for an early osteosarcoma8; however, malignant lesions do not cause pain, but they do lead to destruction of the bone cortex. Surgical resection is the treatment of choice for both tumors.5,6,9,10,16,18,19,21 A portion of the normal tissue must be excised to prevent local recurrence.3,6,10,18,21,22 In the case of osteoid osteoma, after the nidus is removed the pain usually is relieved.9,10,21 However, incomplete removal may lead to pain. The tumors are resistant to radiation with an

FIGURE 8. Case 2: Postoperative panoramic radiograph showing no major alteration of the bone structures and no signs of recurrence (12 month follow-up). Vasconcelos, Porto, and Bessa-Nogueira. Rare Benign Tumors of the Mandibular Condyle. J Oral Maxillofac Surg 2007.

VASCONCELOS, PORTO, AND BESSA-NOGUEIRA

x-ray.6,19 Sometimes reconstruction is necessary to reestablish the TMJ, which can be achieved with either autogenous costochondral or sternoclavicular grafting or an alloplast.6 Theoretically, removal and reimplantation of the condyle as a free graft should result in a deficient vascularity, which may lead to ischemic necrosis and resorption of the condyle.25 However, experimental and clinical results13,25 have shown satisfactory revascularization and function postoperatively, but with a tendency to occasionally produce a slight flattening of the condyle, which appears to be consistent with physiologic bone loss during function.25 In the case of the enchondroma reported herein, the condyle was neither ischemic or resorbed at the 24-month follow-up. After resection via condylectomy of the tumor, in this particular case of enchondroma, a reconstruction was necessary to reestablish the height of the mandibular ramus, with the aim of recreating a functional TMJ. A reshaped condyle was used, rather than a graft from other regions of the body, limiting the surgery to just 1 site, thereby decreasing morbidity and providing satisfactory functional and cosmetic results. In the case reported of osteoid osteoma, the treatment was the complete removal of the lesion, the nidus, and the sclerotic zone, which is in agreement with Liu et al,9 Yang and Qiu,10 Unni,18 and Zulian et al.21

References 1. Lurie R: Solitary enchondroma of the mandibular condyle. J Dent Assoc S Afr 30:589, 1975 2. Jokinen K, Stenback F, Palva A, et al: Benign cartilagenous tumour of the temporomandibular joint. J Laryngol Otol 90: 299, 1976 3. Deboise A, Compere JF, Peron, et al: Tumeurs primitives du condyle mandibulaire. Rev Stomatol Chir Maxillofac 82:93, 1981 4. Van Hemeltyck T, Richard L, Cartier S, et al: Chondrome parostéal ou juxtacortical du condyle mandibulaire. Rev Stomatol Chir Maxillofac 94:267, 1993

1835 5. Chandu A, Spencer JA, Dyson DP: Chondroma of the mandibular condyle: An example of a rare tumour. Dentomaxillofac Radiol 26:242, 1997 6. Lazow SK, Pihlstrom RT, Solomon MP, et al: Condylar chondroma: Report of a case. J Oral Maxillofac Surg 56:373, 1998 7. Lind PO, Hillerstrom K: Osteoid osteoma in the mandibular condyle: Case report and survey of the literature. Acta Otolaryngol 57:467, 1964 8. Tochihara S, Sato T, Yamamoto H, et al: Osteoid osteoma in mandibular condyle. Int J Oral Maxillofac Surg 30:455, 2001 9. Liu CJ, Chang KW, Chang KM, et al: A variant of osteoid osteoma of the mandible: Report of a case. J Oral Maxillofac Surg 60:219, 2002 10. Yang C, Qiu WL: Osteoid osteoma of the eminence of the temporomandibular joint. Br J Oral Maxillofac Surg 39:404, 2001 11. Neville BW, Damm DD, Allen CM, et al: Oral and Maxillofacial Pathology. Philadelphia, PA, Saunders, 1995, p 465 12. Ellis E, Zide MF: Surgical Approaches to the Facial Skeleton. Philadelphia, PA, Lippincott, Williams & Wilkins 1995, pp 163-185 13. Choung PH, Nam IW: An intraoral approach to treatment of condylar hyperplasia or high condylar process fractures using the intraoral vertico-sagittal ramus osteotomy. J Oral Maxillofac Surg 56:563, 1998 14. House J, Brackmann DE: Facial nerve grading system. Otolaryngol Head Neck Surg 93:146, 1985 15. Mehra P, Wolford LM: The Mitek mini-anchor for TMJ disk repositioning: Surgical technique and results. Int J Oral Maxillofac Surg 30:498, 2001 16. Berberi A, Khairallah S, Terezhalmy GT, et al: Osteoid osteoma. Quintessence Int 32:418, 2001 17. Fechner RE, Mills SE: Atlas of Tumor Pathology—Tumors of the Bones and Joints. Washington DC, Armed Forces Institute of Pathology, 1993 18. Unni KK: Dahlin’s Bone Tumor: General Aspects and Data on 11,087 Cases (ed5). Philadelphia, PA, Lippincott-Raven, 1996 19. Greene GW, Natiella JR, Spring PN: Osteoid osteoma of jaws. Oral Surg Oral Med Oral Pathol 26:342, 1968 20. Ida M, Kurabayashi T, Takahashi Y, et al: Osteoid osteoma in the mandible. Dentomaxillofac Radiol 31:385, 2002 21. Zulian MA, Vincent SK, Hiatt WR: Osteoid osteoma of the mandibular ramus. J Oral Maxillofac Surg 45:712, 1987 22. Dockerty MB, Ghormley RK, Jackson AE: Osteoid osteoma: A clinicopathologic study of 20 cases. Ann Surg 133:77, 1951 23. Jaffe HL: Osteoid osteoma. Arch Surg 31:709, 1935 24. Rushton MA: An osteoid osteoma of the mandibular alveolus. Oral Surg Oral Med Oral Pathol 4:86, 1955 25. Boyne PJ: Free grafting of traumatically displaced or resected mandibular condyles. J Oral Maxillofac Surg 47:228, 1989