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Journal of Clinical Oncology, Vol 25, No 15 (May 20), 2007: pp. 2133-2135
© 2007 American Society of Clinical Oncology.
DOI: 10.1200/JCO.2007.10.7086

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DIAGNOSIS IN ONCOLOGY

Pleural Mesothelioma Metastatic to Tongue

Daniel S. Higginson

Johns Hopkins University School of Medicine, Baltimore, MD

Julie Brahmer

Department of Oncology, Sidney Kimmel Comprehensive Cancer Center at Johns Hopkins Hospital, Johns Hopkins University School of Medicine, Baltimore, MD

Ralph P. Tufano

Department of Otolaryngology, Head & Neck Surgery, Johns Hopkins University School of Medicine, Baltimore, MD

Gopal K. Bajaj

Department of Radiation Oncology and Molecular Radiation Sciences, Sidney Kimmel Comprehensive Cancer Center at Johns Hopkins Hospital, Johns Hopkins University School of Medicine, Baltimore, MD

A 68-year-old man was diagnosed with malignant pleural mesothelioma (MPM) in October 2004 as a result of an incidental finding of pleural thickening on a computed tomography (CT) study conducted for a work-up of renal artery stenosis.1 An initial biopsy of the thickening indicated adenocarcinoma of the lung; however, a subsequent exploratory right thoracotomy and attempted decortication revealed a final pathology of MPM, epithelioid subtype. He did not have a known history of exposure to asbestos, but in his late teenage years worked in a shipyard.

On further imaging studies, his lesion was determined to be confined to the right lung and was deemed to be surgically resectable. Presented with the option of chemotherapy, extrapleural pneumonectomy (EPP), and adjuvant hemithoracic radiotherapy, the patient refused surgical resection and opted for chemotherapy alone. His right lung lesion progressed through six cycles of cisplatin and pemetrexed, a clinical trial involving sorafenib, and two further cycles of vinorelbine.

In April 2005, while still receiving chemotherapy, he noticed a new horizontal fissure and a firmness in his anterior oral tongue. A magnetic resonance image revealed a submucosal mass of cross sectional dimensions 2.2 x 0.9 cm in the instrinic muscles of the anterior tongue. An incisional biopsy revealed a malignant mesothelioma lesion with makers positive for calretinin, CK5/6, AE1/3, and CD15. BER-EP4 and mucicarmine stains were negative, as expected for mesothelioma lesions.

In October 2005, his tongue lesion had progressed to 2.6 x 1.4 cm and extended to the floor of the mouth. By December 2005, the mass had enlarged to 3.3 x 1.5 cm (Fig 1A). Given the rapidity of the lesion's growth, he elected to receive palliative radiotherapy to prevent the speech and swallowing symptoms detrimental to quality of life in cancers of the head and neck from continued progression.


Figure 1
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Fig 1.
 
The patient received a total dose of 50 Gy in 20 fractions, applied in two lateral fields and completed in February 2006 (Fig 2B). He tolerated radiotherapy well, experiencing only moderate xerostomia, relieved by drinking copious amounts of water. He did not report odynophagia, dysphagia, or otalgia. After 2 months, a follow-up CT indicated stable disease (Fig 1B). In June 2006, another mass suspicious for metastasis was discovered in his right gluteus muscle (Fig 3). By December 2006, his pleural and gluteal masses had progressed, but his tongue mass remained stable.


Figure 2
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Fig 2.
 

Figure 3
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Fig 3.
 
Once considered primarily a localized pleural disease, perhaps owing to the predominance of respiratory failure as cause of death, extrapleural MPM metastases are now understood to be common. Recent interest in the use of combined CT and positron emission tomography has found a prevalence of extrathoracic metastases as high as 10% to 25% at the time of presentation.2-4 A postmortem analysis of 63 cases revealed distant metastases in 63% of cases, most commonly in contralateral lung, liver, peritoneum, and adrenal glands.5 Other unusual sites include the brain, bone, retroperitoneum, skeletal muscle, and skin.

Four other cases of tongue metastases have been reported, and to our knowledge, this is the first use of radiotherapy for such a lesion.6-9 There are isolated reports of the successful use of palliative radiotherapy in treating distant metastases in muscle, spine, and skin.10-12 For thoracic lesions, palliative radiotherapy is approximately 70% successful in providing pain relief and nearly 50% successful in alleviating Pancoast syndrome and superior vena cava syndrome.13

Although local disease progression has historically been the cause of death in most mesothelioma patients, new advances in the use of hemithoracic adjuvant radiotherapy after EPP seem to demonstrate improved local control rates and have uncovered the relative importance of distant metastases.14,15 In a phase II trial conducted at the Memorial Sloan-Kettering Cancer Center (New York, NY), 54 patients received EPP and conformal radiotherapy to 54 Gy in 30 fractions.16 In these patients, 64% failed distantly compared with only 13% who failed locoregionally. At the M.D. Anderson Cancer Center (Houston, TX), 28 patients received intensity modulated radiotherapy to 45 to 50 Gy after EPP. In-field local control was 100% at 9 months follow-up compared with an 18% distant metastasis rate.14 Extended to 62 patients, this trial now demonstrates only one in-field failure and three marginal failures while more than 50% of patients failed distantly.15 Systemic agents and selected use of palliative radiotherapy may be increasingly important in this disease.

AUTHORS' DISCLOSURES OF POTENTIAL CONFLICTS OF INTEREST

Although all authors completed the disclosure declaration, the following authors or their immediate family members indicated a financial interest. No conflict exists for drugs or devices used in a study if they are not being evaluated as part of the investigation. For a detailed description of the disclosure categories, or for more information about ASCO's conflict of interest policy, please refer to the Author Disclosure Declaration and the Disclosures of Potential Conflicts of Interest section in Information for Contributors.

Employment: N/A Leadership: N/A Consultant: Julie Brahmer, Genentech Stock: N/A Honoraria: N/A Research Funds: Julie Brahmer, Wyeth, Astra Zeneca, Pfizer, Mederex Testimony: N/A Other: N/A

REFERENCES

1. Glazer CA, Waldman EH, Ansari-Lari AM, et al: Pathology quiz case 1: Metastatic mesothelioma involving the tongue. Arch Otolaryngol Head Neck Surg 132: 10121014-1015, 2006

2. Flores RM, Akhurst T, Gonen M, et al: Positron emission tomography defines metastatic disease but not locoregional disease in patients with malignant pleural mesothelioma. J Thorac Cardiovasc Surg 126: 11-16, 2003[Abstract/Free Full Text]

3. Erasmus JJ, Truong MT, Smythe WR, et al: Integrated computed tomography-positron emission tomography in patients with potentially resectable malignant pleural mesothelioma: Staging implications. J Thorac Cardiovasc Surg 129: 1364-1370, 2005[Abstract/Free Full Text]

4. Truong MT, Marom EM, Erasmus JJ: Preoperative evaluation of patients with malignant pleural mesothelioma: Role of integrated CT-PET imaging. J Thorac Imaging 21: 146-153, 2006[CrossRef][Medline]

5. Scharmach M, Neumann V, Muller KM, et al: Comparison of patterns of metastasis between malignant pleural mesotheliomas and pulmonary carcinomas. Pneumologie 60: 277-283, 2006[CrossRef][Medline]

6. Zanconati F, DelConte A, Bonifacio-Gori D, et al: Metastatic pleural mesothelioma presenting with solitary involvement of the tongue: Report of a new case and review of the literature. Int J Surg Pathol 11: 51-55, 2003[Abstract/Free Full Text]

7. Tho LM, O'Rourke NP: Unusual metastases from malignant pleural mesothelioma. Clin Oncol (R Coll Radiol) 17: 293, 2005[Medline]

8. Piattelli A, Fioroni M, Rubini C: Tongue metastasis from a malignant diffuse mesothelioma of the pleura: Report of a case. J Oral Maxillofac Surg 57: 861-863, 1999[CrossRef][Medline]

9. Kerpel SM, Freedman PD: Metastatic mesothelioma of the oral cavity: Report of two cases. Oral Surg Oral Med Oral Pathol 76: 746-751, 1993[CrossRef][Medline]

10. Akyurek S, Nalca Andrieu M, Hicsonmez A, et al: Skeletal muscle metastasis from malignant pleural mesothelioma. Clin Oncol (R Coll Radiol) 16: 585, 2004

11. Cheng WF, Berkman AW: Malignant mesothelioma with bone metastases. Med Pediatr Oncol 18: 165-168, 1990[CrossRef][Medline]

12. Cassarino DS, Xue W, Shannon KJ: Widespread cutaneous and perioral metastases of mesothelioma. J Cutan Pathol 30: 582-585, 2003[CrossRef][Medline]

13. Ball DL, Cruickshank DG: The treatment of malignant mesothelioma of the pleura: Review of a 5-year experience, with special reference to radiotherapy. Am J Clin Oncol 13: 4-9, 1990[Medline]

14. Ahamad A, Stevens CW, Smythe WR, et al: Promising early local control of malignant pleural mesothelioma following postoperative intensity modulated radiotherapy (IMRT) to the chest. Cancer J 9: 476-484, 2003[Medline]

15. Stevens CW, Forster KM, Smythe WR, et al: Radiotherapy for mesothelioma. Hematol Oncol Clin North Am 19: 1099-1115, vii, 2005[CrossRef][Medline]

16. Rusch VW, Rosenzweig K, Venkatraman E, et al: A phase II trial of surgical resection and adjuvant high-dose hemithoracic radiation for malignant pleural mesothelioma. J Thorac Cardiovasc Surg 122: 788-795, 2001[Abstract/Free Full Text]





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