Ann Thorac Surg 1998;65:559
© 1998 The Society of Thoracic Surgeons
Case Reports
Primary Malignant Melanoma of the Trachea
Ignacio G. Duarte, MD,
Anthony A. Gal, MD,
Kamal A. Mansour, MD
Division of Cardiothoracic Surgery, Joseph P. Whitehead Department of Surgery, Emory University School of Medicine, Atlanta, Georgia, USA
Department of Pathology and Laboratory Medicine, Emory University School of Medicine, Atlanta, Georgia, USA
Accepted for publication September 29, 1997.
Dr Mansour, 1365 Clifton Rd, NE, Atlanta, GA 30322.
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Abstract
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Only three primary malignant melanomas have been identified among large series of tracheal resections. Pertinent clinical and histopathologic features of these rare malignancies, however, have rarely been presented. We present the case of a young woman who underwent tracheal resection for a primary malignant melanoma with clinical and histopathologic verification.
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Introduction
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Primary tumors of the trachea are uncommon neoplasms among large series of tracheal resections in the English-language literature. Squamous cell and adenoid cystic carcinoma are the most frequent histopathologic types, accounting for approximately 75% of the total [1] [2] [3] [4]. Of the remaining pathologic types, only three primary malignant melanomas of the trachea have been identified, but salient clinical and pathologic features were not discussed [1] [2] [3]. We present the case of a 32-year-old woman who underwent tracheal resection for primary malignant melanoma of the trachea, with histopathologic verification.
The patient, a nonsmoker, first presented to her primary care physician 3 months before operation, with complaints of a persistent cough. Despite treatment with oral antibiotics and aerosolized bronchodilators, her symptoms persisted and she had progressively worsening dyspnea on exertion and a documented 6.75-kg weight loss. She did not complain of hemoptysis or hoarseness. Preoperative bronchoscopy identified a mass on the anterior tracheal wall, and biopsy specimens proved the mass to be malignant melanoma.
The patient reported having had a skin lesion excised from her central back approximately 7 years prior, by a family practitioner, for cosmetic reasons. No formal pathologic evaluation was done, and she was told it was benign. There was no personal or family history of cutaneous neoplasms. A thorough physical examination of skin surfaces, including palms, soles, mouth, inguinal regions, and neck as well as lymph nodal basins showed all areas to be normal. Fundoscopic evaluation also had negative results. Preoperative chest radiographs and computed tomographic series revealed a large mass occluding approximately 85% of the tracheal lumen (Fig 1). It began about 6 cm below the vocal cords and extended for 1.5 cm distally. There was no evidence of local extension beyond the trachea.
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Preoperative computed tomogram of the chest demonstrating a large, obstructing intraluminal mass in the proximal trachea.
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Under isoflurane anesthesia and without muscle relaxants, rigid bronchoscopy demonstrated an irregular mass on the anterior trachea (1 x 1 cm), approximately 6 cm below the vocal cords. A median sternotomy and transverse cervical neck incision was made, and an en bloc tracheal resection was performed, encompassing the tumor and a paratracheal lymph node [5] [6]. Frozen section analysis confirmed negative margins. The trachea was primarily anastomosed using interrupted 3-0 Vicryl (Ethicon, Somerville, NJ) sutures and then a pericardial flap was elevated and interposed between the innominate artery and the suture line. Postoperatively, an episode of right upper lobe collapse resolved with aggressive chest physiotherapy and aerosolized bronchodilators, and the patient was discharged on postoperative day 9. Given the lack of proven chemotherapeutic agents in the treatment of the rare mucosal melanomas, she did not receive immediate adjuvant therapy.
The gross tracheal specimen measured 2.3 cm in length, with a luminal diameter of 1.3 cm. On the anterior luminal surface of the trachea was an irregular, raised, 1 x 0.8-cm brown-black mass. Microscopic analysis revealed an atypical intraepithelial melanocytic proliferation with definitive invasion into the submucosa extending to a depth of 0.2 cm (Fig 2). Within deeper portions of the submucosa, a highly invasive component was noted, with a prominent host lymphocytic response and considerable melanin pigment deposition (Fig 3). Immunostains for HMB-45 and S-100 protein were positive in the tumor. DNA ploidy/cell cycle analysis revealed an aneuploid, hyperdiploid cell population and a DNA index of 1.44. One paratracheal lymph node, 1.5 x 1.2 x 1.0 cm, revealed metastatic melanoma.
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Microphotograph of the tracheal mass depicting the atypical intraepithelial melanocytic proliferation with submucosal invasion characteristic of a primary malignant melanoma. (Hematoxylin and eosin; x100 before 28% reduction.)
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Representative section of the malignant melanoma in the wall of the trachea demonstrating prominent intracytoplasmic melanocytic pigmentation. (Hematoxylin and eosin; x50 before 28% reduction.)
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Subsequently, advanced metastatic disease developed with local spread to the neck (jugular nodes) and distant multiorgan metastases. The patient was evaluated at an outside institution, where she received high-dose 
-interferon and interleukin-2 as part of a clinical trial. She died of metastatic melanoma approximately 13 months after operation; no autopsy was performed. Despite complete resection of the tumor, the patient’s early demise was not unexpected given the extent and aggressive nature of the tumor.
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Comment
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As a result of the extreme rarity of primary respiratory tract malignant melanomas, strict criteria for its diagnosis have been proposed: a solitary tumor, absence of demonstrable tumor outside the trachea, junctional changes (atypical melanocytic hyperplasia and mitoses) in the mucosa with "dropping off" of melanoma cells, invasion beyond the epithelium into submucosa, and melanin histologically present [7] [8] [9] [10]. Although definitive histologic examination of the previously removed skin lesion was not available, complete physical examination for evidence of metastatic disease before tracheal resection had negative results, and there was no family history of cutaneous disease. Most significantly, however, the tumor displayed features of the pathologic criteria of a primary tracheal malignant melanoma. In particular, the junctional changes with invasion into the submucosa present in this case are perhaps the most convincing feature of a primary melanoma.
Various theories have been postulated as to the histogenesis of lower respiratory tract malignant melanomas [9] [10]. Some have suggested that during embryogenesis, melanocytes migrate to the developing lower respiratory tract. Alternatively, the transformation of respiratory epithelial cells into melanocytes (ie, "melanogenic metaplasia") has been proposed by others. Finally, a more modern concept is that some unknown precursor cell, possibly related to a neuroendocrine cell (ie, Kulchitsky cell), could show evidence of melanocytic differentiation.
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References
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- Grillo HC, Mathisen DJ Primary tracheal tumors: treatment and results. Ann Thorac Surg 1990;49:69-77.[Abstract]
- Regnard JF, Fourquier P, Levasseur P, The French Society of Cardiovascular Surgery. Results and prognostic factors in resections of primary tracheal tumors: a multicenter retrospective study. J Thorac Cardiovasc Surg 1996;111:808-814.[Abstract/Free Full Text]
- Pearson FG, Todd TRJ, Cooper JD Experience with primary neoplasms of the trachea and carina. J Thorac Cardiovasc Surg 1984;88:511-518.[Abstract]
- Li W, Ellerbroek NA, Libshitz HI Primary malignant tumors of the trachea. Cancer 1990;66:894-899.[Medline]
- Randleman CD, Unger ER, Mansour KA Malignant fibrous histiocytoma of the trachea. Ann Thorac Surg 1990;50:458-459.[Abstract]
- Mansour KA, Lee RB, Miller JI, Jr Tracheal resections: lessons learned. Ann Thorac Surg 1994;57:1120-1125.[Abstract]
- Gal AA, Marchevsky AM, Koss MN Unusual tumors of the lung. In: Marchevsky AM, ed. Surgical pathology of lung neoplasms. New York: Marcel Dekker, 1990:325-388.
- Miller DL, Allen MS Rare pulmonary neoplasms. Mayo Clin Proc 1993;68:492-498.[Medline]
- Colby TV, Koss MN, Travis WD Tumors of the lower respiratory tract. Armed Forces Institute of Pathology. Atlas of tumor pathology, third series, fascicle 13. Armed Forces Institute of Pathology, 1995:483-487.
- Jennings TA, Axiotis CA, Kress Y, Carter D Primary malignant melanoma of the lower respiratory tract. Am J Clin Pathol 1990;94:649-655.[Medline]
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Abstract
Introduction
