The Journal of Craniofacial Surgery • Volume 26, Number 2, March 2015
TABLE 8. Treatment of 120 ORN Patients
10.
Treatment Outcome Treatment Methods
No. (%)
Sequestrectomy or debridement
18 (15.0) 32 (26.7)
Radical resection alone Radical resection + flap FOF Iliac flap PMMF ALT LDF Composite flap Other flap Ineligible operative patients Total
14 3 27 4 3 2 5 12 120
(11.7) (2.5) (22.5) (3.3) (2.5) (1.7) (4.2) (10.0)
Healed
Improved
Unhealed
15 27
2 3
1 2
12 3 24 3 3 2 4 0 96
1 0 2 1 0 0 1 0 10
1 0 1 0 0 0 0 12 14
FOF, fibular osteocutaneous flap; ALT, anterolateral thigh flap; PMMF, pectoralis major myocutaneous flap; LDF, latissimus dorsi flap; Other flap, (1 sternocleidomastoid flap, 3 forearm flaps, 1 palatal flap).
11.
12.
13.
14.
15.
16.
involving the anterior arch. Although soft flaps reconstruction of ORN defects did not achieve the functional and aesthetic outcomes like bone flap, it could control infection, solve pain, and stabilize disease. In 32 patients who underwent radical resection alone, 5 patients received second-stage reconstruction (2 fibula flaps and 3 PMMF). All in all, we agree with the view that flap reconstruction of advanced ORN defects has a high rate of complications, but it can be configured with good outcomes.12,24
CONCLUSIONS Ultimately, this study demonstrated that radical resection and immediate well-vascularized tissue flap reconstruction seem to be reliable and successful methods for a patient whose ORN does not respond to conservative nonoperative treatment. Preventing ORN occurrence is time consuming and demands teamwork, so the oral and maxillofacial surgeon should minimize the trauma for jaw as possible as he can, especially for patients who need to receive postsurgical radiotherapy.
REFERENCES 1. Nabil S, Samman N. Risk factors for osteoradionecrosis after head and neck radiation: a systematic review. Oral Surg Oral Med Oral Pathol Oral Radiol 2012;113:54–69 2. Marx RE. A new concept in the treatment of osteoradionecrosis. J Oral Maxillofac Surg 1983;41:351–357 3. Nabil S. Redefining osteoradionecrosis. Oral Surg Oral Med Oral Pathol Oral Radiol 2012;114:403–404 4. Meyer I. Infectious diseases of the jaws. J Oral Surg 1970;28:17–26 5. Delanian S, Lefaix JL. The radiation-induced fibroatropic process: therapeutic perspective via the antioxidant pathway. Radiother Oncol 2004;73:119–131 6. Marx RE, Tursun R. Suppurative osteomyelitis, bisphosphonate induced osteonecrosis, osteoradionecrosis: a blinded histopathologic comparison and its implications for the mechanism of each disease. Int J Oral Maxillofac Surg 2012;41:283–289 7. Nabil S, Samman N. Incidence and prevention of osteoradionecrosis after dental extraction in irradiated patients: a systematic review. Int J Oral Maxillofac Surg 2011;40:229–243 8. Goldwaser BR, Chuang SK, Kaban LB, et al. Conservative management of osteoradionecrosis. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 1997;84:16–21 9. Annane D, Depondt J, Aubert P, et al. Hyperbaric oxygen therapy for radionecrosis of the jaw: a randomized, placebo-controlled,
17.
18. 19.
20.
21.
22.
23.
24.
Brief Clinical Studies
double-blind trial from the ORN96 study group. J Clin Oncol 2004;22:4893–4900 McLeod NM, Pratt CA, Mellor TK, et al. Pentoxifylline and tocopherol in the management of patients with osteoradionecrosis, the Portsmouth experience. Br J Oral Maxillofac Surg 2012;50:41–44 Curi MM, Oliveira dos Santos M, Feher O, et al. Management of extensive osteoradionecrosis of the mandible with radical resection and immediate microvascular reconstruction. J Oral Maxillofac Surg 2007;65:434–438 Baumann DP, Yu P, Hanasono MM, et al. Free flap reconstruction of osteoradionecrosis of the mandible: a 10-year review and defect classification. Head Neck 2011;33:800–807 Lyons A, Ghazali N. Osteoradionecrosis of the jaws: current understanding of its pathophysiology and treatment. Br J Oral Maxillofac Surg 2008;46:653–660 Reuther T, Schuster T, Mende U, et al. Osteoradionecrosis of the jaws as a side effect of radiotherapy of head and neck tumour patients—a report of a thirty year retrospective review. Int J Oral Maxillofac Surg 2003;32:289–295 Curi MM, Dib LL. Osteoradionecrosis of the jaws: a retrospective study of the background factors and treatment in 104 cases. J Oral Maxillofac Surg 1997;55:540–544 Thorn JJ, Hansen HS, Specht L, et al. Osteoradionecrosis of the jaws: clinical characteristics and relation to the field of irradiation. J Oral Maxillofac Surg 2000;58:1088–1093 Lee IJ, Koom WS, Lee CG, et al. Risk factors and dose-effect relationship for mandibular osteoradionecrosis in oral and oropharyngeal cancer patients. Int J Radiat Oncol Biol Phys 2009;75:1084–1091 Morrish RB Jr, Chan E, Silverman S Jr, et al. Osteonecrosis in patients irradiated for head and neck carcinoma. Cancer 1981;47:1980–1983 Goldwaser BR, Chuang SK, Kaban LB, et al. Risk factor assessment for the development of osteoradionecrosis. J Oral Maxillofac Surg 2007;65:2311–2316 Wong JK, Wood RE, McLean M. Conservative management of osteoradionecrosis. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 1997;84:16–21 Celik N, Wei FC, Chen HC, et al. Osteoradionecrosis of the mandible after oromandibular cancer surgery. Plast Reconstr Surg 2002;109:1875–1881 Lubek JE, Hancock MK, Strome SE. What is the value of hyperbaric oxygen therapy in management of osteoradionecrosis of the head and neck? Laryngoscope 2013;123:555–556 Chang DW, Oh HK, Robb GL, Miller MJ. Management of advanced mandibular osteoradionecrosis with free flap reconstruction. Head Neck 2001;23:830–835 Cannady SB, Dean N, Kroeker A, et al. Free flap reconstruction for osteoradionecrosis of the jaws—outcomes and predictive factors for success. Head Neck 2011;33:424–428
Chondromyxoid Fibroma of the Frontal Bone Mimicking Meningioma Hao Wang, MD,* Hansheng Shu, MD,* Xuping Tian, MD,* Hui Zhang, MD,* Qiujian Zhang, MD,* Liemei Guo, MD, PhD† Abstract: Chondromyxoid fibroma (CMF) is a rare benign cartilaginous tumor that usually arises from lower-extremity long-bone metaphyses, with approximately 5.4% of all CMFs presenting in the craniofacial bones. Chondromyxoid fibroma of the frontal bone is exceedingly rare, with only a few cases reported. Herein, we report another case of CMF arising from the frontal bone mimicking meningioma. We suggest that histopathologic examination is of vital
© 2015 Mutaz B. Habal, MD
Copyright © 2015 Mutaz B. Habal, MD. Unauthorized reproduction of this article is prohibited.
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The Journal of Craniofacial Surgery • Volume 26, Number 2, March 2015
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importance for the diagnosis of CMF; complete surgical resection is the best treatment option for frontal CMF. Key Words: Benign cartilaginous tumor, chondromyxoid fibroma, frontal bone, meningioma
C
hondromyxoid fibroma (CMF), which was first reported by Jaffe and Lichtenstein1,2 in 1948 as a tumor commonly mistaken for chondrosarcoma, is a rare benign cartilaginous tumor that usually arises from lower-extremity long-bone metaphyses.1,2 Although CMF has been reported in numerous anatomic sites of the body, involvement of the head and neck is rare, with approximately 5.4% of all CMFs presenting in the craniofacial bones.2,3 Usually, these tumors arise from the mandible and maxilla, and tumors arising within the cranial vault are extraordinarily rare.2,4 To the best of our knowledge, only 14 cases of CMF of the frontal bone have been reported in the English literature.5,6 Herein, we report another case of CMF arising from the frontal bone mimicking meningioma, to emphasize its rarity, differential diagnosis, and treatment options.
CLINICAL REPORT A 64-year-old female patient was admitted with headache, decline of olfactory sensation, and blurred vision for half a year. Physical examination showed obvious decline of bilateral olfactory sensation and visual defect of bilateral outer upper region. Local examination showed a solid bony swelling of 2 2 cm over the frontal region. Cranial computed tomography (CT) scans revealed an irregular, mixeddensity mass located at bilateral frontal lobes with minute foci of calcification, and the frontal bone appeared osteolytic in bone window (Figs. 1A, B). The mass was enhanced obviously with contrast on CT scans (Fig. 1C), and CT angiography showed the anterior cerebral arteries were pushed toward the left side (Fig. 1D). Cranial magnetic resonance (MR) scans confirmed the well-defined tumor measured 5.4 5.8 4.5 cm without any local infiltration. The lesion exhibited a low-to-intermediate signal intensity on T1-weighted MR and a high inhomogeneous signal intensity on T2-weighted MR (Figs. 1E, F). Strong inhomogeneous enhancement was observed on T1-weighted MR with contrast (Figs. 1G–I). Based on the imaging findings, the differential preoperative diagnoses include meningioma, bony tumor, and Langerhans cell tumor. Then the tumor together with the dura and osteolytic bone were totally removed via a bilateral subfrontal approach (Fig. 2). Histopathologic examinations of the tumor revealed variable chondroid elements with what appeared to be immature irregular chondrocytes, containing eosinophilic cytoplasm and surrounded by fibrous myxoid intercelluar matrix. This soft tissue tumor was heterogeneous in both cell types, which consisted of stellate and spindle-shaped cells, as well as chondroid, myxoid, and fibrous elements (Figs. 3A, B). Thus, From the *Department of Neurosurgery, The Second Hospital Affiliated Bengbu Medical School, Bengbu, and †Department of Neurosurgey, Renji Hospital, Shanghai Jiao Tong University School of Medicine, Shanghai, China. Received October 6, 2014. Accepted for publication December 1, 2014. Address correspondence and reprint requests to Dr. Liemei Guo, Department of Neurosurgery, Renji Hospital, Shanghai Jiao Tong University School of Medicine, No. 160, Pujian Rd, Pudong District, Shanghai 200127, China; E-mail: [email protected] The manuscript was approved by all authors for publication. This study was not supported by any project or interest. The authors report no conflicts of interest. Copyright © 2015 by Mutaz B. Habal, MD ISSN: 1049-2275 DOI: 10.1097/SCS.0000000000001447
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FIGURE 1. Pre-operative images of the patient. Cranial CT scans showed irregular mixed density mass located at bilateral frontal lobes with minute foci of calcification (A) , and the frontal bone appeared osteolytic in bone window (B); The mass was enhanced obviously with contrast on CT scans (C), and CT angiography showed the anterior cerebral arteries were pushed toward the left side (D). The lesion exhibited a low-to-intermediate signal intensity on T1-weighted MR (E) and a high inhomogeneous signal intensity on T2-weighted MR (F). Strong inhomogeneous enhancement was observed on T1-weighted MR with contrast, and the tumor measured 5.4 cm 5.8 cm 4.5 cm without any local infiltration (G–I).
the tumor was classified as CMF. Immuohistochemical examinations showed glial fibrillary acidic protein staining was negative, and vimentin staining was positive (Figs. 3C, D). The postoperative course was uneventful, and the patient discharged 7 days later. With a 1-year follow-up, the patient recovered well without tumor recurrence.
DISCUSSION Chondromyxoid fibroma is an unusual benign cartilaginous tumor, accounting for less than 1% of all primary neoplasms of the bone and less than 0.5% of all benign tumors of the bone.7 The sites of predilection for CMFs are long bones, flat bones, and small bones of the hands and feet, with a incidence of 5.4% in the craniofacial bones.3,7 Skull bones develop from endochondral tissue, and it is believed that CMF arises from cartilage or embryonic cartilage residue.5 Chondromyxoid fibroma occurs in all age groups, but mostly in the second and third decades, with a slight predilection in males.5 Regarding intracranial CMFs, the range of mean age of patients with this condition is 37 to 39 years, and it is 32.7 years (range, 15–50 years) for patients with frontal CMF.5 To date, including the preset case, there are 15 cases of CMF of the frontal bone in the English literature.5,6 The clinical presentations of CMF vary according to the site of origin. The common clinical presentation in cases of frontal CMF involvement is headache with bony swelling as in the present case; others include neuralgia, facial pain, exophthalmos, convulsions, and diplopia.5 The radiographic features of CMF are characteristic. X-ray shows radiolucent lesion with well-defined margins, and CT scan shows osteolytic lesion with a sclerotic margin with foci of calcification.5,6,8 It is reported that microscopic calcification can be observed in roughly 34% of cases but can only be documented in 12.5% of plain films or CT images, and the calcification occurs more frequently in older patients.6 Magnetic resonance imaging scans show a hypointense lesion on T1-weighted and hyperintense on T2weighted images with intense contrast enhancement.5,6,8 However, to some extent, the radiographic features of CMF are similar to meningioma, and sometimes, it might be misdiagnosed as meningioma preoperatively, such as in the present case.
FIGURE 2. Postoperative images of the patient. Postoperative images of cranial MR revealed the tumor was completely removed.
© 2015 Mutaz B. Habal, MD
Copyright © 2015 Mutaz B. Habal, MD. Unauthorized reproduction of this article is prohibited.
The Journal of Craniofacial Surgery • Volume 26, Number 2, March 2015
Brief Clinical Studies
REFERENCES
FIGURE 3. Histopathologic and immunohistochemical examinations of the tumor. A, Histopathologic study with hematoxylin-eosin staining demonstrated variable chondroid elements with what appeared to be immature irregular chondrocytes, containing eosinophilic cytoplasm and surrounded by fibrous myxoid intercelluar matrix (A). This soft tissue tumor was heterogeneous in both cell types, which consisted of stellate and spindle-shaped cells, as well as chondroid, myxoid, and fibrous elements (B). Immunohistochemical examinations showed the tumor cells were negative staining with glial fibrillary acidic protein (GFAP) (C) and positive staining with vimentin (D).
Based on the radiographic features, there were multiple differential diagnostic considerations, including chondrosarcomas, chondroblastoma, enchondroma, and chordoma. Predominantly, the histopathologic features of CMF as defined by the World Health Organization are that CMF is “a benign tumor characterized by lobules of spindle-shaped or stellate cells with abundant myxoid or chondroid intercellular material separated by zones of more cellular tissue rich in spindle-shaped or round cells with varying numbers of multinucleated giant cells of different sizes.”5,7 Chondrosarcoma can be differentiated from CMF by its invasive pattern of growth and presence of mitosis. Furthermore, presence of a well-formed hyaline matrix and absence of fibrous component are other features distinguishing chondrosarcomas from CMFs.7,8 Chondroblastoma is another benign tumor with cartilaginous differentiation, but it generally lacks a lobular pattern and perilobular fibrous component.3,7 Enchondromas show lobular architecture as in CMFs, but the lack of welldifferentiated hyaline cartilage helps to exclude the diagnosis of CMF.7 Similarly, chordoma, although it bears a superficial resemblance, lacks the degree of cellularity and other architectural features of CMFs.6,7 In addition, the routine use of immunohistochemistry plays a very limited role in resolving the differential diagnosis because the above tumors mentioned may express vimentin and S100 protein.7 Complete surgical resection is considered to be the optimal treatment for intracranial CMF.2 Curettage is recommended when complete resection is not possible.9 Despite the benign behavior of CMF, recurrence rate has been reported to be as high as 11% even in case of complete surgical resection.3 Nevertheless, the rate could increase up to 80% when surgical excision is incomplete.10 Recurrences tend to show identical morphology of the original tumor without histological evidence of dedifferentiation or progression.11 The severe functional and cosmetic morbidities after wide resection of the tumor should be kept in mind, and cranioplasty should be performed for prevention of cosmetic bone defect in the frontal area.5 The administration of radiotherapy is controversial, because it has been associated with an increased risk of malignant transformation and a potential long-term sequelae.2 Given the benign nature of CMF and its excellent response to surgical resection, radiotherapy should be reserved only for recurrences or primary tumors that are surgically difficult to reach.2 In conclusion, CMF is a rare, benign neoplasm occasionally occurring in the skull bone, with very rare occurrence in the frontal bone. Histopathologic diagnosis is essential to provide the patient with an accurate management of the pathology. Complete surgical resection remains the main and best treatment modality for patients with CMF of the frontal bone.
1. Jaffe HL, Lichtenstein L. Chondromyxoid fibroma of bone; a distinctive benign tumor likely to be mistaken especially for chondrosarcoma. Arch Pathol 1948;45:541–551 2. McClurg SW, Leon M, Teknos TN, et al. Chondromyxoid fibroma of the nasal septum: case report and review of literature. Head Neck 2013;35:E1–E5 3. Wu CT, Inwards CY, O’Laughlin S, et al. Chondromyxoid fibroma of bone: a clinicopathologic review of 278 cases. Hum Pathol 1998;29:438–446 4. Hammad HM, Hammond HL, Kurago ZB, et al. Chondromyxoid fibroma of the jaws. Case report and review of the literature. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 1998;85:293–300 5. Hakan T, Vardar Aker F. Chondromyxoid fibroma of frontal bone: a case report and review of the literature. Turk Neurosurg 2008;18:249–253 6. Kadom N, Rushing EJ, Yaun A, et al. Chondromyxoid fibroma of the frontal bone in a teenager. Pediatr Radiol 2009;39:53–56 7. Karkuzhali P, Chithralekha S, Muthuvel E, et al. Chondromyxoid fibroma of the parietal bone. Neuropathology 2005;25:84–88 8. Sharma M, Velho V, Binayake R, et al. Chondromyxoid fibroma of the temporal bone: a rare entity. J Pediatr Neurosci 2012;7:211–214 9. Schajowicz F, Gallardo H. Chondromyxoid fibroma (fibromyxoid chondroma) of bone. A clinico-pathological study of thirty-two cases. J Bone Joint Surg Br 1971;53:198–216 10. Gherlinzoni F, Rock M, Picci P. Chondromyxoid fibroma. The experience at the Istituto Ortopedico Rizzoli. J Bone Joint Surg Am 1983;65:198–204 11. Shek TW, Peh WC, Leung G. Chondromyxoid fibroma of skull base: a tumour prone to local recurrence. J Laryngol Otol 1999;113:380–385
Treatment of Mandibular Symphyseal Fracture Combined With Dislocated Intracapsular Condylar Fractures Xiaofeng Xu, MDS, Jun Shi, MD, Bing Xu, DDS, Jiewen Dai, MD, Shilei Zhang, MD Objectives: To evaluate the treatment methods of mandibular symphyseal fracture combined with dislocated intracapsular condylar fractures (MSF&DICF) and to compare the effect of different treatment methods of condylar fractures. From the Department of Oral and Cranio-maxillofacial Science, College of Stomatology, Shanghai Ninth People's Hospital, Shanghai Jiaotong University, School of Medicine, Shanghai, China. Received October 19, 2014. Accepted for publication December 1, 2014. Address correspondence and reprint requests to Bing Xu, DDS, Department of Craniomaxillofacial Science, Shanghai Ninth People's Hospital, School of Dentistry, Shanghai Jiaotong University, 639 Zhizaoju Rd, Shanghai, China 200011; E-mail: [email protected]; or Jun Shi, MD, Department of Craniomaxillofacial Science, Shanghai Ninth People's Hospital, School of Dentistry, Shanghai Jiaotong University, 639 Zhizaoju Rd, Shanghai, China 200011; E-mail: [email protected] The authors report no conflicts of interest. The authors declare that there are no competing interests and that they have no support or funding to report. Dr Bing Xu and Dr Jun Shi contributed equally to this work and they should be considered co–corresponding authors. Copyright © 2014 by Mutaz B. Habal. MD ISSN: 1049-2275 DOI: 10.1097/SCS.0000000000001451
© 2015 Mutaz B. Habal, MD
Copyright © 2015 Mutaz B. Habal, MD. Unauthorized reproduction of this article is prohibited.
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TABLE 8. Treatment of 120 ORN Patients
10.
Treatment Outcome Treatment Methods
No. (%)
Sequestrectomy or debridement
18 (15.0) 32 (26.7)
Radical resection alone Radical resection + flap FOF Iliac flap PMMF ALT LDF Composite flap Other flap Ineligible operative patients Total
14 3 27 4 3 2 5 12 120
(11.7) (2.5) (22.5) (3.3) (2.5) (1.7) (4.2) (10.0)
Healed
Improved
Unhealed
15 27
2 3
1 2
12 3 24 3 3 2 4 0 96
1 0 2 1 0 0 1 0 10
1 0 1 0 0 0 0 12 14
FOF, fibular osteocutaneous flap; ALT, anterolateral thigh flap; PMMF, pectoralis major myocutaneous flap; LDF, latissimus dorsi flap; Other flap, (1 sternocleidomastoid flap, 3 forearm flaps, 1 palatal flap).
11.
12.
13.
14.
15.
16.
involving the anterior arch. Although soft flaps reconstruction of ORN defects did not achieve the functional and aesthetic outcomes like bone flap, it could control infection, solve pain, and stabilize disease. In 32 patients who underwent radical resection alone, 5 patients received second-stage reconstruction (2 fibula flaps and 3 PMMF). All in all, we agree with the view that flap reconstruction of advanced ORN defects has a high rate of complications, but it can be configured with good outcomes.12,24
CONCLUSIONS Ultimately, this study demonstrated that radical resection and immediate well-vascularized tissue flap reconstruction seem to be reliable and successful methods for a patient whose ORN does not respond to conservative nonoperative treatment. Preventing ORN occurrence is time consuming and demands teamwork, so the oral and maxillofacial surgeon should minimize the trauma for jaw as possible as he can, especially for patients who need to receive postsurgical radiotherapy.
REFERENCES 1. Nabil S, Samman N. Risk factors for osteoradionecrosis after head and neck radiation: a systematic review. Oral Surg Oral Med Oral Pathol Oral Radiol 2012;113:54–69 2. Marx RE. A new concept in the treatment of osteoradionecrosis. J Oral Maxillofac Surg 1983;41:351–357 3. Nabil S. Redefining osteoradionecrosis. Oral Surg Oral Med Oral Pathol Oral Radiol 2012;114:403–404 4. Meyer I. Infectious diseases of the jaws. J Oral Surg 1970;28:17–26 5. Delanian S, Lefaix JL. The radiation-induced fibroatropic process: therapeutic perspective via the antioxidant pathway. Radiother Oncol 2004;73:119–131 6. Marx RE, Tursun R. Suppurative osteomyelitis, bisphosphonate induced osteonecrosis, osteoradionecrosis: a blinded histopathologic comparison and its implications for the mechanism of each disease. Int J Oral Maxillofac Surg 2012;41:283–289 7. Nabil S, Samman N. Incidence and prevention of osteoradionecrosis after dental extraction in irradiated patients: a systematic review. Int J Oral Maxillofac Surg 2011;40:229–243 8. Goldwaser BR, Chuang SK, Kaban LB, et al. Conservative management of osteoradionecrosis. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 1997;84:16–21 9. Annane D, Depondt J, Aubert P, et al. Hyperbaric oxygen therapy for radionecrosis of the jaw: a randomized, placebo-controlled,
17.
18. 19.
20.
21.
22.
23.
24.
Brief Clinical Studies
double-blind trial from the ORN96 study group. J Clin Oncol 2004;22:4893–4900 McLeod NM, Pratt CA, Mellor TK, et al. Pentoxifylline and tocopherol in the management of patients with osteoradionecrosis, the Portsmouth experience. Br J Oral Maxillofac Surg 2012;50:41–44 Curi MM, Oliveira dos Santos M, Feher O, et al. Management of extensive osteoradionecrosis of the mandible with radical resection and immediate microvascular reconstruction. J Oral Maxillofac Surg 2007;65:434–438 Baumann DP, Yu P, Hanasono MM, et al. Free flap reconstruction of osteoradionecrosis of the mandible: a 10-year review and defect classification. Head Neck 2011;33:800–807 Lyons A, Ghazali N. Osteoradionecrosis of the jaws: current understanding of its pathophysiology and treatment. Br J Oral Maxillofac Surg 2008;46:653–660 Reuther T, Schuster T, Mende U, et al. Osteoradionecrosis of the jaws as a side effect of radiotherapy of head and neck tumour patients—a report of a thirty year retrospective review. Int J Oral Maxillofac Surg 2003;32:289–295 Curi MM, Dib LL. Osteoradionecrosis of the jaws: a retrospective study of the background factors and treatment in 104 cases. J Oral Maxillofac Surg 1997;55:540–544 Thorn JJ, Hansen HS, Specht L, et al. Osteoradionecrosis of the jaws: clinical characteristics and relation to the field of irradiation. J Oral Maxillofac Surg 2000;58:1088–1093 Lee IJ, Koom WS, Lee CG, et al. Risk factors and dose-effect relationship for mandibular osteoradionecrosis in oral and oropharyngeal cancer patients. Int J Radiat Oncol Biol Phys 2009;75:1084–1091 Morrish RB Jr, Chan E, Silverman S Jr, et al. Osteonecrosis in patients irradiated for head and neck carcinoma. Cancer 1981;47:1980–1983 Goldwaser BR, Chuang SK, Kaban LB, et al. Risk factor assessment for the development of osteoradionecrosis. J Oral Maxillofac Surg 2007;65:2311–2316 Wong JK, Wood RE, McLean M. Conservative management of osteoradionecrosis. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 1997;84:16–21 Celik N, Wei FC, Chen HC, et al. Osteoradionecrosis of the mandible after oromandibular cancer surgery. Plast Reconstr Surg 2002;109:1875–1881 Lubek JE, Hancock MK, Strome SE. What is the value of hyperbaric oxygen therapy in management of osteoradionecrosis of the head and neck? Laryngoscope 2013;123:555–556 Chang DW, Oh HK, Robb GL, Miller MJ. Management of advanced mandibular osteoradionecrosis with free flap reconstruction. Head Neck 2001;23:830–835 Cannady SB, Dean N, Kroeker A, et al. Free flap reconstruction for osteoradionecrosis of the jaws—outcomes and predictive factors for success. Head Neck 2011;33:424–428
Chondromyxoid Fibroma of the Frontal Bone Mimicking Meningioma Hao Wang, MD,* Hansheng Shu, MD,* Xuping Tian, MD,* Hui Zhang, MD,* Qiujian Zhang, MD,* Liemei Guo, MD, PhD† Abstract: Chondromyxoid fibroma (CMF) is a rare benign cartilaginous tumor that usually arises from lower-extremity long-bone metaphyses, with approximately 5.4% of all CMFs presenting in the craniofacial bones. Chondromyxoid fibroma of the frontal bone is exceedingly rare, with only a few cases reported. Herein, we report another case of CMF arising from the frontal bone mimicking meningioma. We suggest that histopathologic examination is of vital
© 2015 Mutaz B. Habal, MD
Copyright © 2015 Mutaz B. Habal, MD. Unauthorized reproduction of this article is prohibited.
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The Journal of Craniofacial Surgery • Volume 26, Number 2, March 2015
Brief Clinical Studies
importance for the diagnosis of CMF; complete surgical resection is the best treatment option for frontal CMF. Key Words: Benign cartilaginous tumor, chondromyxoid fibroma, frontal bone, meningioma
C
hondromyxoid fibroma (CMF), which was first reported by Jaffe and Lichtenstein1,2 in 1948 as a tumor commonly mistaken for chondrosarcoma, is a rare benign cartilaginous tumor that usually arises from lower-extremity long-bone metaphyses.1,2 Although CMF has been reported in numerous anatomic sites of the body, involvement of the head and neck is rare, with approximately 5.4% of all CMFs presenting in the craniofacial bones.2,3 Usually, these tumors arise from the mandible and maxilla, and tumors arising within the cranial vault are extraordinarily rare.2,4 To the best of our knowledge, only 14 cases of CMF of the frontal bone have been reported in the English literature.5,6 Herein, we report another case of CMF arising from the frontal bone mimicking meningioma, to emphasize its rarity, differential diagnosis, and treatment options.
CLINICAL REPORT A 64-year-old female patient was admitted with headache, decline of olfactory sensation, and blurred vision for half a year. Physical examination showed obvious decline of bilateral olfactory sensation and visual defect of bilateral outer upper region. Local examination showed a solid bony swelling of 2 2 cm over the frontal region. Cranial computed tomography (CT) scans revealed an irregular, mixeddensity mass located at bilateral frontal lobes with minute foci of calcification, and the frontal bone appeared osteolytic in bone window (Figs. 1A, B). The mass was enhanced obviously with contrast on CT scans (Fig. 1C), and CT angiography showed the anterior cerebral arteries were pushed toward the left side (Fig. 1D). Cranial magnetic resonance (MR) scans confirmed the well-defined tumor measured 5.4 5.8 4.5 cm without any local infiltration. The lesion exhibited a low-to-intermediate signal intensity on T1-weighted MR and a high inhomogeneous signal intensity on T2-weighted MR (Figs. 1E, F). Strong inhomogeneous enhancement was observed on T1-weighted MR with contrast (Figs. 1G–I). Based on the imaging findings, the differential preoperative diagnoses include meningioma, bony tumor, and Langerhans cell tumor. Then the tumor together with the dura and osteolytic bone were totally removed via a bilateral subfrontal approach (Fig. 2). Histopathologic examinations of the tumor revealed variable chondroid elements with what appeared to be immature irregular chondrocytes, containing eosinophilic cytoplasm and surrounded by fibrous myxoid intercelluar matrix. This soft tissue tumor was heterogeneous in both cell types, which consisted of stellate and spindle-shaped cells, as well as chondroid, myxoid, and fibrous elements (Figs. 3A, B). Thus, From the *Department of Neurosurgery, The Second Hospital Affiliated Bengbu Medical School, Bengbu, and †Department of Neurosurgey, Renji Hospital, Shanghai Jiao Tong University School of Medicine, Shanghai, China. Received October 6, 2014. Accepted for publication December 1, 2014. Address correspondence and reprint requests to Dr. Liemei Guo, Department of Neurosurgery, Renji Hospital, Shanghai Jiao Tong University School of Medicine, No. 160, Pujian Rd, Pudong District, Shanghai 200127, China; E-mail: [email protected] The manuscript was approved by all authors for publication. This study was not supported by any project or interest. The authors report no conflicts of interest. Copyright © 2015 by Mutaz B. Habal, MD ISSN: 1049-2275 DOI: 10.1097/SCS.0000000000001447
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FIGURE 1. Pre-operative images of the patient. Cranial CT scans showed irregular mixed density mass located at bilateral frontal lobes with minute foci of calcification (A) , and the frontal bone appeared osteolytic in bone window (B); The mass was enhanced obviously with contrast on CT scans (C), and CT angiography showed the anterior cerebral arteries were pushed toward the left side (D). The lesion exhibited a low-to-intermediate signal intensity on T1-weighted MR (E) and a high inhomogeneous signal intensity on T2-weighted MR (F). Strong inhomogeneous enhancement was observed on T1-weighted MR with contrast, and the tumor measured 5.4 cm 5.8 cm 4.5 cm without any local infiltration (G–I).
the tumor was classified as CMF. Immuohistochemical examinations showed glial fibrillary acidic protein staining was negative, and vimentin staining was positive (Figs. 3C, D). The postoperative course was uneventful, and the patient discharged 7 days later. With a 1-year follow-up, the patient recovered well without tumor recurrence.
DISCUSSION Chondromyxoid fibroma is an unusual benign cartilaginous tumor, accounting for less than 1% of all primary neoplasms of the bone and less than 0.5% of all benign tumors of the bone.7 The sites of predilection for CMFs are long bones, flat bones, and small bones of the hands and feet, with a incidence of 5.4% in the craniofacial bones.3,7 Skull bones develop from endochondral tissue, and it is believed that CMF arises from cartilage or embryonic cartilage residue.5 Chondromyxoid fibroma occurs in all age groups, but mostly in the second and third decades, with a slight predilection in males.5 Regarding intracranial CMFs, the range of mean age of patients with this condition is 37 to 39 years, and it is 32.7 years (range, 15–50 years) for patients with frontal CMF.5 To date, including the preset case, there are 15 cases of CMF of the frontal bone in the English literature.5,6 The clinical presentations of CMF vary according to the site of origin. The common clinical presentation in cases of frontal CMF involvement is headache with bony swelling as in the present case; others include neuralgia, facial pain, exophthalmos, convulsions, and diplopia.5 The radiographic features of CMF are characteristic. X-ray shows radiolucent lesion with well-defined margins, and CT scan shows osteolytic lesion with a sclerotic margin with foci of calcification.5,6,8 It is reported that microscopic calcification can be observed in roughly 34% of cases but can only be documented in 12.5% of plain films or CT images, and the calcification occurs more frequently in older patients.6 Magnetic resonance imaging scans show a hypointense lesion on T1-weighted and hyperintense on T2weighted images with intense contrast enhancement.5,6,8 However, to some extent, the radiographic features of CMF are similar to meningioma, and sometimes, it might be misdiagnosed as meningioma preoperatively, such as in the present case.
FIGURE 2. Postoperative images of the patient. Postoperative images of cranial MR revealed the tumor was completely removed.
© 2015 Mutaz B. Habal, MD
Copyright © 2015 Mutaz B. Habal, MD. Unauthorized reproduction of this article is prohibited.
The Journal of Craniofacial Surgery • Volume 26, Number 2, March 2015
Brief Clinical Studies
REFERENCES
FIGURE 3. Histopathologic and immunohistochemical examinations of the tumor. A, Histopathologic study with hematoxylin-eosin staining demonstrated variable chondroid elements with what appeared to be immature irregular chondrocytes, containing eosinophilic cytoplasm and surrounded by fibrous myxoid intercelluar matrix (A). This soft tissue tumor was heterogeneous in both cell types, which consisted of stellate and spindle-shaped cells, as well as chondroid, myxoid, and fibrous elements (B). Immunohistochemical examinations showed the tumor cells were negative staining with glial fibrillary acidic protein (GFAP) (C) and positive staining with vimentin (D).
Based on the radiographic features, there were multiple differential diagnostic considerations, including chondrosarcomas, chondroblastoma, enchondroma, and chordoma. Predominantly, the histopathologic features of CMF as defined by the World Health Organization are that CMF is “a benign tumor characterized by lobules of spindle-shaped or stellate cells with abundant myxoid or chondroid intercellular material separated by zones of more cellular tissue rich in spindle-shaped or round cells with varying numbers of multinucleated giant cells of different sizes.”5,7 Chondrosarcoma can be differentiated from CMF by its invasive pattern of growth and presence of mitosis. Furthermore, presence of a well-formed hyaline matrix and absence of fibrous component are other features distinguishing chondrosarcomas from CMFs.7,8 Chondroblastoma is another benign tumor with cartilaginous differentiation, but it generally lacks a lobular pattern and perilobular fibrous component.3,7 Enchondromas show lobular architecture as in CMFs, but the lack of welldifferentiated hyaline cartilage helps to exclude the diagnosis of CMF.7 Similarly, chordoma, although it bears a superficial resemblance, lacks the degree of cellularity and other architectural features of CMFs.6,7 In addition, the routine use of immunohistochemistry plays a very limited role in resolving the differential diagnosis because the above tumors mentioned may express vimentin and S100 protein.7 Complete surgical resection is considered to be the optimal treatment for intracranial CMF.2 Curettage is recommended when complete resection is not possible.9 Despite the benign behavior of CMF, recurrence rate has been reported to be as high as 11% even in case of complete surgical resection.3 Nevertheless, the rate could increase up to 80% when surgical excision is incomplete.10 Recurrences tend to show identical morphology of the original tumor without histological evidence of dedifferentiation or progression.11 The severe functional and cosmetic morbidities after wide resection of the tumor should be kept in mind, and cranioplasty should be performed for prevention of cosmetic bone defect in the frontal area.5 The administration of radiotherapy is controversial, because it has been associated with an increased risk of malignant transformation and a potential long-term sequelae.2 Given the benign nature of CMF and its excellent response to surgical resection, radiotherapy should be reserved only for recurrences or primary tumors that are surgically difficult to reach.2 In conclusion, CMF is a rare, benign neoplasm occasionally occurring in the skull bone, with very rare occurrence in the frontal bone. Histopathologic diagnosis is essential to provide the patient with an accurate management of the pathology. Complete surgical resection remains the main and best treatment modality for patients with CMF of the frontal bone.
1. Jaffe HL, Lichtenstein L. Chondromyxoid fibroma of bone; a distinctive benign tumor likely to be mistaken especially for chondrosarcoma. Arch Pathol 1948;45:541–551 2. McClurg SW, Leon M, Teknos TN, et al. Chondromyxoid fibroma of the nasal septum: case report and review of literature. Head Neck 2013;35:E1–E5 3. Wu CT, Inwards CY, O’Laughlin S, et al. Chondromyxoid fibroma of bone: a clinicopathologic review of 278 cases. Hum Pathol 1998;29:438–446 4. Hammad HM, Hammond HL, Kurago ZB, et al. Chondromyxoid fibroma of the jaws. Case report and review of the literature. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 1998;85:293–300 5. Hakan T, Vardar Aker F. Chondromyxoid fibroma of frontal bone: a case report and review of the literature. Turk Neurosurg 2008;18:249–253 6. Kadom N, Rushing EJ, Yaun A, et al. Chondromyxoid fibroma of the frontal bone in a teenager. Pediatr Radiol 2009;39:53–56 7. Karkuzhali P, Chithralekha S, Muthuvel E, et al. Chondromyxoid fibroma of the parietal bone. Neuropathology 2005;25:84–88 8. Sharma M, Velho V, Binayake R, et al. Chondromyxoid fibroma of the temporal bone: a rare entity. J Pediatr Neurosci 2012;7:211–214 9. Schajowicz F, Gallardo H. Chondromyxoid fibroma (fibromyxoid chondroma) of bone. A clinico-pathological study of thirty-two cases. J Bone Joint Surg Br 1971;53:198–216 10. Gherlinzoni F, Rock M, Picci P. Chondromyxoid fibroma. The experience at the Istituto Ortopedico Rizzoli. J Bone Joint Surg Am 1983;65:198–204 11. Shek TW, Peh WC, Leung G. Chondromyxoid fibroma of skull base: a tumour prone to local recurrence. J Laryngol Otol 1999;113:380–385
Treatment of Mandibular Symphyseal Fracture Combined With Dislocated Intracapsular Condylar Fractures Xiaofeng Xu, MDS, Jun Shi, MD, Bing Xu, DDS, Jiewen Dai, MD, Shilei Zhang, MD Objectives: To evaluate the treatment methods of mandibular symphyseal fracture combined with dislocated intracapsular condylar fractures (MSF&DICF) and to compare the effect of different treatment methods of condylar fractures. From the Department of Oral and Cranio-maxillofacial Science, College of Stomatology, Shanghai Ninth People's Hospital, Shanghai Jiaotong University, School of Medicine, Shanghai, China. Received October 19, 2014. Accepted for publication December 1, 2014. Address correspondence and reprint requests to Bing Xu, DDS, Department of Craniomaxillofacial Science, Shanghai Ninth People's Hospital, School of Dentistry, Shanghai Jiaotong University, 639 Zhizaoju Rd, Shanghai, China 200011; E-mail: [email protected]; or Jun Shi, MD, Department of Craniomaxillofacial Science, Shanghai Ninth People's Hospital, School of Dentistry, Shanghai Jiaotong University, 639 Zhizaoju Rd, Shanghai, China 200011; E-mail: [email protected] The authors report no conflicts of interest. The authors declare that there are no competing interests and that they have no support or funding to report. Dr Bing Xu and Dr Jun Shi contributed equally to this work and they should be considered co–corresponding authors. Copyright © 2014 by Mutaz B. Habal. MD ISSN: 1049-2275 DOI: 10.1097/SCS.0000000000001451
© 2015 Mutaz B. Habal, MD
Copyright © 2015 Mutaz B. Habal, MD. Unauthorized reproduction of this article is prohibited.
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