Case Report
Treatment of pulmonary metastases from primary intraosseous odontogenic carcinoma Domenico Galetta, Francesco Petrella, Francesco Leo, Giuseppe Pelosi, Lorenzo Spaggiari Lancet Oncol 2006; 7: 272–73 Division of Thoracic Surgery (D Galetta MD, F Petrella MD, F Leo MD, L Spaggiari MD) and Department of Pathology (G Pelosi MD), European Institute of Oncology, Milan, Italy Correspondence to: Dr Lorenzo Spaggiari, Division of Thoracic Surgery, European Institute of Oncology, Via Ripamonti 435, 20141 Milan, Italy
[email protected]
A 52-year-old man was admitted to our institution because of bilateral pulmonary nodules diagnosed by a follow-up chest radiograph. 7 years previously, the patient had had a left mandibulectomy followed by radiotherapy for primary intraosseous carcinoma. Routine laboratory test results were normal; whole-body CT scan confirmed the presence of multiple pulmonary nodules located mainly in the posterobasal region of both lungs (figure 1). No local or other distant recurrences were noted. The patient had surgery via twostep bilateral sequential thoracotomies. Functional assessment before surgery showed no cardiac disease and good spirometric results. Fibreoptic bronchoscopy was normal, and PET scan was negative. A right musclesparing lateral thoracotomy was done; lung palpation diagnosed five peripheral nodules, two in the upper lobe and three in the lower lobe. Frozen sections of one of these nodules, which was removed by conventional wedge resection, confirmed the clinical hypothesis of neoplastic metastatic disease. Thus, surgery was completed by radical wedge resection of the remaining four nodules and by lymphadenectomy. Histological analyses confirmed five metastases from a keratinising carcinoma consistent with odontogenic derivation (figure 2), ranging from 5 mm to 11 mm at the largest diameter; none of the ten lymph nodes analysed had evidence of metastases. The patient was discharged without problems 4 days after surgery, and
Figure 1: Preoperative CT scan showing bilateral pulmonary metastases
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was readmitted 3 weeks later for treatment of the other lung. A new chest CT confirmed multiple nodules in the left lung and only scar tissue on the right lung. A left lateral muscle-sparing thoracotomy showed presence of seven nodules. One wedge resection, six nodulectomies, and radical lymphadenectomy were done. Histological analyses confirmed six metastases from odontogenic intraosseous carcinoma and one parenchymal nodule without evidence of neoplastic disease; all lymph nodes were negative for metastases. The patient was discharged 6 days after surgery in good clinical health. 25 months after the second thoracotomy, the patient is alive with no evidence of disease. Odontogenic tumours are uncommon, mainly benign, neoplasms that occur as a result of interruptions in, or reactivation of, odontogenic tissues that have a role in normal odontogenesis. The nature of the tumour is determined by the stage of development at which the arrest in odontogenesis arises. Odontogenic tumours are more common in younger people than in older, but can occur at any age; these tumours account for less than 4% of all oral and maxillofacial samples sent to oral pathology services for diagnosis.1 Odontogenic tumours originate in the jaws, and the most common sites are the mandibular molar region and the maxillary cuspid region.2 Malignant odontogenic tumours are very rare, accounting for 0–6·1% of all odontogenic tumours.1 According to the WHO classification of tumours,3 malignant odontogenic tumours include ameloblastic carcinoma, primary intraosseous carcinoma, clear-cell odontogenic carcinoma, ghost-cell odontogenic carcinoma, and odontogenic sarcoma.2 Diagnosis of malignant odontogenic tumours can be complicated because of their rarity, scant clinical information, and low index of suspicion. Microscopically, malignant odontogenic tumours are characterised by hypercellularity, pleomorphism, focal necrosis, and perineural or vascular infiltration; clinically, they are characterised by distant metastatic spread.2 Because malignant odontogenic tumours are rare, it is important to rule out a malignant process of the adjacent oral or sinonasal area, or that of metastatic carcinoma, since lung cancer can metastasise to jaw bones (particularly the body of the mandible). In our patient, differentiation between primary intraosseous odontogenic carcinoma and other malignant odontogenic tumour was made on the basis of histology. Diagnosis of primary intraosseous odontogenic carcinoma was confirmed by: presence of islands of neoplastic squamous epithelium with the features of squamous-cell carcinoma; absence of clear cells with peripheral palisading of nuclei with reverse polarisation; http://oncology.thelancet.com Vol 7 March 2006
Case Report
negative staining of ameloblastic components of laminin in the basement membrane; and absence of ghost cells and sarcomatous components. Primary intraosseous odontogenic carcinoma arises from residues of odontogenic epithelium, and is located within bone without demonstrable evidence of a primary carcinoma in other sites or from oral or sinusal mucosa.4 Histologically, these tumours are squamous-cell carcinomas that range from well differentiated to poorly differentiated lesions. They invade and destroy medullary bone, leading to tooth mobility and invasion or to compression of the alveolar nerve (thus explaining the presence of pain and paresthesia).1 Defined by Shear in 1969,5 primary intraosseous odontogenic carcinoma usually occurs in the body and posterior region of the mandible, affects more men than women, and arises in those aged 4–76 years (mean 51 years). Primary intraosseous odontogenic carcinoma is an aggressive disease that tends to produce local and distant metastases. However, bilateral pulmonary metastases from primary intraosseous odontogenic carcinoma after a disease-free interval of more than 1 year are very rare. We could find no data in a search of articles published in English for surgical or multimodality treatment for recurrence. Surgical resection is a widely accepted treatment for pulmonary metastases when a complete resection can be obtained in the absence of disseminated disease in patients with sufficient pulmonary reserve.6 Neither the number of lesions nor the disease-free interval excludes a patient from curative resection provided that the primary tumour has been treated and they can tolerate pulmonary resection. However, the number of metastases amenable to complete resection, the effect of disease-free interval, and the role of chemotherapy remain controversial.7,8 No data were available from a search for papers done at the time of our diagnosis on pulmonary metastases from malignant odontogenic tumour and specifically from primary intraosseous odontogenic carcinoma. We decided that this patient should receive surgery, based on the epithelial histology of the lesion, its peripheral localisation and small size, the patient’s age and good performance status, and absence of local recurrence or other metastases. Because of the posterobasal localisation of the metastases in both lungs, we avoided a one-step operation with longitudinal sternotomy (which is associated with absence of complete exposure) or clam-shell incision (which is invasive). We suggest that a bilateral two-step operation offers the best surgical fields for metastatic disease, allowing thorough investigation of the hemithorax and adequate palpation of the lung. This procedure allows for a more comfortable postoperative period, avoiding potential concomitant bilateral pulmonary complications.
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Figure 2: Primary intraosseous carcinoma Note keratinising carcinoma within a desmoplastic stroma. Healthy pulmonary parenchyma (lower right) surrounds nests of tumour cells (haematoxylin and eosin stain, magnification 100).
In summary, we report a case of bilateral pulmonary metastases from primary intraosseous odontogenic carcinoma. To our knowledge, this is the first report of recurrent tumours in both lungs with a long disease-free interval after primary intraosseous odontogenic carcinoma. Imaging studies did not identify the indeterminate clinical nature of the lesions. Complete surgical resection gave a definitive diagnosis and longterm prognosis. Protracted follow-up is necessary to ascertain the real aggressiveness of the tumours. Conflict of interest We declare no conflicts of interest. References 1 Mosqueda Taylor A, Meneses Garcia A, Ruiz Gody Rivera LM, et al. Malignant odontogenic tumors. A retrospective and collaborative study of seven cases. Med Oral 2003; 8: 110–21. 2 Goldenberg D, Sciubba J, Koch W, Tufano RP. Malignant odontogenic tumors: a 22-year experience. Laryngoscope 2004; 114: 1770–74. 3 Barnes L, Eveson JW, Reichart P, Sidransky D. Pathology and genetics of head and neck tumours. Lyon: International Agency for Research on Cancer, 2005. 4 Kramer IRH, Pindborg JJ, Shear M. Histological typing of odontogenic tumours. 2nd edn. Berlin: Springer-Verlag, 1992. 5 Shear M. Primary intra-alveolar epidermoid carcinoma of the jaw. J Pathol 1969; 97: 645–51. 6 Burt ME, Liu D, Abolhoda A, et al. Isolated lung perfusion for patients with unresectable metastases from sarcoma: a phase I trial. Ann Thorac Surg 2000; 69: 1542–49. 7 Martini N, McCormack PM. Evolution of the surgical management of pulmonary metastases. Chest Surg Clin N Am 1998; 8: 13–27. 8 Ratto GB, Toma S, Civalleri D, et al. Isolated lung perfusion with platinum in the treatment of pulmonary metastases from soft tissue sarcoma. J Thorac Cardiovasc Surg 1996; 112: 614–22.
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