Ann Thorac Surg 2008;86:308-310. doi:10.1016/j.athoracsur.2008.01.056
© 2008 The Society of Thoracic Surgeons
Case Reports
Tracheal Malignant Melanoma: Successful Outcome With Tracheal Resection
Ricardo Mingarini Terra, MD*,
Helio Minamoto, MD,
Jader J.M. Junqueira, MD,
Roberto Falzoni, MD,
Paulo Manuel Pêgo-Fernandes, MD, PhD,
Fabio Biscegli Jatene, MD
Division of Thoracic Surgery, Hospital das Clínicas, São Paulo University School of Medicine, São Paulo, Brazil
Accepted for publication January 18, 2008.
* Address correspondence to Dr Terra, Al. Fernao Cardim, 161 ap. 61-Jardim Paulista, São Paulo–SP, 01403-020, Brazil (Email: rmterra{at}uol.com.br).
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Abstract
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Primary tracheal malignant melanomas are uncommon neoplasms: only five cases have been reported. Different therapeutic approaches are described, with a short life expectancy observed. We report a case of a young woman with a primary tracheal malignant melanoma who underwent complete tracheal resection and is free of disease 4 years after surgical treatment.
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Introduction
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Primary tracheal malignant melanoma is a very rare entity, and very few cases are reported [1, 2]. Most tumors diagnosed as malignant melanoma are metastatic in origin, and true primary tumors are exceptions. Because of its rarity, varied therapeutic approaches, and poor prognosis, no specific treatment is recommended for malignant melanoma [1, 3–5]. The best treatment for the most frequent tracheal tumors (squamous carcinoma and cystic adenoid carcinoma) is resection and end-to-end anastomosis [6]. A recent report documented good results with this approach in unusual tracheal tumors [7].
We report the case of a 29-year-old woman with a primary tracheal malignant melanoma who underwent complete resection and tracheal end-to-end anastomosis. She is free of disease 4 years after surgical treatment.
A 29-year-old woman, who was a nonsmoker, was admitted to our hospital in December 2003 with a recently performed tracheostomy and a diagnosis of tracheal tumor. Her previous history was progressive dyspnea that had been treated as asthma elsewhere for 9 months, but she had no improvement. A flexible bronchoscopy revealed an obstructive vegetating tracheal mass. Because of worsening symptoms leading to severe respiratory distress, she underwent a tracheostomy, but she was in a small center and no further procedures were done. She was referred to our tertiary hospital.
At admission, a rigid bronchoscopy was performed during which we identified a large vegetating mass located 3 cm distal to vocal folds just above the tracheostomy, obstructing 90% of the tracheal lumen. Several biopsies were made. Neck and chest computed tomography (CT) scans revealed a round intratracheal mass with a 2-cm extension and no evidence of invasion of adjacent structures or lymphadenopathy (Fig 1).
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Fig 1. Preoperative neck and chest computed tomography image shows a vegetating mass (arrow) obstructing the proximal trachea lumen, with no evidence of lymphadenomegaly or invasion of the adjacent structures.
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The pathologic analysis showed a malignant melanoma, which was confirmed by immunohistochemistry. The patient had no history of previous surgeries or skin biopsies. She was examined by a senior dermatologist who found no skin lesion suggestive of melanoma, and head, neck, chest, abdominal, and pelvic CT scans had not shown any tumors other than the tracheal mass.
Once the tumor was considered primary according to the 1967 criteria of Jensen and colleagues [8]—solitary, restricted to trachea, and resectable—we opted for complete resection and tracheal reconstruction.
The tracheal tumor was explored through a transversal cervical incision and a partial sternotomy. An en bloc resection was performed, including a segment of the trachea from the third to the tenth rings. The trachea was reconstructed with end-to-end anastomosis using continuous 4-0 polydioxanone (Ethicon, Somerville, NJ) suture for the posterior wall and interrupted sutures for the anterior wall. A protective tracheostomy was performed because of concern about intraoperative recurrent laryngeal nerve damage. These concerns were unfounded, and the tracheostomy was removed on the postoperative day 15.
The tracheal tumor specimen measured 2.5 x 1.7 x 0.5 cm and the total extension of the resected trachea was 3.5 cm. Microscopic analysis revealed respiratory mucosa with infiltrative spindle cell tumor with nuclear atypias and mitosis in submucosa (Fig 2). Paratracheal lymph node analysis revealed no metastatic neoplasm.
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Fig 2. Photomicrograph shows spindle cell tumor with nuclear atypias and mitosis (hematoxylin and eosin stain, original magnification x400).
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Immunohistochemistry stains were positive for S100 protein and HMB45 antigen.
She had a good postoperative course and was discharged on postoperative day 7. On postoperative day 38, she was readmitted because of wound infection and was treated for 21 days with wide-spectrum antibiotics, with good outcome.
She had a thorough clinical examination every 3 months during the first 2 postoperative years and then every 6 months. Neck CT scans and flexible bronchoscopy were performed every 6 months during the first 2 years and then yearly. After a 4-year follow-up, the patient remains asymptomatic, with no clinical, endoscopic, or radiologic evidence of disease.
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Comment
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Primary malignant melanomas of the trachea are uncommon neoplasms. As result of this rarity, specific criteria were developed to define its diagnosis: (1) no previously removed skin lesion, particularly pigmented; (2) no ocular tumors removed and no enucleation; (3) solitary tumor; (4) tumor morphologic characteristics compatible with a primary tumor; (5) no demonstrable melanoma in other organs at the time of operation; (6) and autopsy without primary melanoma being demonstrated elsewhere, more particularly in the skin or eyes [3, 8].
Only five definitive cases of primary tracheal malignant melanoma have been reported [1, 3, 4–7]. One patient received radiotherapy and chemotherapy and died 2 months later of disease progression [5]. The other 4 patients underwent tumor resection and tracheal reconstruction [1, 3, 4, 7]. One patient died in the immediate postoperative period [4]. Two patients had lymph node disease and had poor outcome: 1 died after 13 months [3] and 1 had residual disease after 1 year [1]. The last patient is cited in a large series of unusual tracheal tumors, and no specific information is offered about the case other than that resection was curative [7].
The patient described in this report had a definitive diagnosis of primary tracheal melanoma and is free of disease 4 years after surgical intervention. Radical tumor resection and tracheal reconstruction in this patient was successful.
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References
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- Mori K, Cho H, Som M. Primary "flat" melanoma of the trachea J Pathol 1977;121:101-105.[Medline]
- Grillo HC, Mathisen DJ. Primary tracheal tumors: treatment and results Ann Thorac Surg 1990;49:69-77.[Abstract]
- Duarte IG, Gal AA, Mansour KA. Primary malignant melanoma of the trachea Ann Thorac Surg 1998;65:559-560.[Abstract/Free Full Text]
- Reid JD, Mehta VT. Melanoma of the lower respiratory tract Cancer 1966;19:627-631.[Medline]
- Verweij J, Breed WP, Jansveld CA. Primary tracheo-bronchial melanoma Neth J Med 1982;25:163-166.[Medline]
- Xu LT, Sun ZF, Li ZJ, Wu LH, Wang ZZ. Tracheobronchial tumors: an eighteen-year series from Capital Hospital, Peking, China Ann Thorac Surg 1983;35:590-596.[Abstract]
- Gaissert HA, Grillo HC, Shadmehr MB, et al. Uncommon primary tracheal tumors Ann Thorac Surg 2006;82:268-273.[Abstract/Free Full Text]
- Jensen OA, Egedorf J. Primary malignant melanoma of the lung Scand J Respir Dis 1967;48:127-135.[Medline]
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Abstract
Introduction
