CASE REPORT

Fibromyxomaof the Kazuo Takaoka, Atsuko Satoh, Mikihito Matsuda*, Yasunori Fujioka** and Shoichi Inoue***

A 53-year-old male with a small cell of the lung was admitted to the Department of Respiratory Diseases, Nikko Memorial Hospital. During bronchofiberoptic examination of the , a small nodule was discovered on the anterior wall of the trachea, about 8 cmbelow the vocal cord. Histopathologically, it was diagnosed as fibromyxoma of the trachea. Primary tumors of the trachea are very rare and fibromyxomaof the trachea is extremely rare. This is only the second report of a fibromyxomaon the tracheal wall. In this report its clinical manifestations were compared with those reported in the first case. (Internal Medicine 32: 895-896, 1993) Key words: benign tumor

Case Report cartilage was located in the deeper region. A small amount of fatty tissue was also observed in the loose collagenous tissue. The patient was a 53-year-old male priest. A chest x-ray Anepithelial componentof tumor constitution wasnot recog- revealed a nodular shadow in the right upper lung and enlarge- nized, although hamartoma is composed of non-epithelial and mentof the right hilus suggesting a . Computed epithelial components. No malignant findings such as pleo- tomography of the chest also showeda tiny protrusion on the morphismor mitosis were seen. tracheal anterior wall. There was neither tumor infiltration to The final histopathologic diagnosis was made as fibromyxoma the surrounding mediastinum nor a tracheal deformity. Bronchofiberoptic examination (Fig. 1 ) revealed a small hemi- spheric nodule located on the anterior wall of the trachea, approximately 8 cm below the vocal cord. The surface of the tumor was smoothand covered with normal mucosawithout vascular engorgement. As its size was approximately only 1 mmin diameter, the tumor was removed endoscopically with biforceps in one effort. The histopathology was proved to be fibromy xoma. The lung cancer was cytologically denned as a small cell carcinoma that had originated in the right upper lobe, and was classified by UICC, TNMclassification as belonging to stage IV (T3N2M1 ). Laboratory examination provided no abnormal data except for a slightly elevated LDHlevel 242 U/l, and hyper y-globulinemia 30.1%. The tumor markers were carcinoembryonic antigen 30.4 ng/ml, neuron-specific enolase 3 1.7 ng/ml, and tissue polypeptide antigen 146.3 U/l. Histopathological Findings The tumor which was covered by a normal tracheal mucosal Fig. 1. Bronchofiberoscopic findings show smooth-surface epithelium consisted of fibrous tissue with a myxomatous hemispheric tumor with superficial vascular engorgement, arising matrix in the submucosal area (Fig. 2). A part of tracheal from anterior wall of the trachea. From the Department of Respiratory Diseases, *the Department 01 Internal Medicine, Nikko Memorial Hospital, Muroran, **tne Department ot mnoiogy, Hokkaido University School of Medicine, Sapporo and ***the Department of Environmental Medicine, Postgraduate School of Hokkaido University, Sapporo Received for publication April 19, 1993; Accepted for publication October 12, 1993 Reprint request should be addressed to Dr. Kazuo Takaoka, the Department of Respiratory Diseases, Nikko Memorial Hospital, 1 -5- 1 3 Shintomi-cho, Muroran 051

Internal Medicine Vol. 32, No. 12 (December 1993) 895 Takaoka et al

Comment Therelative incidence of tracheal tumor to lung cancer is estimated at 1 : 1 80. Eighty to ninety percent of tracheal tumors are malignant. The majority of these are either , or , or ( 1). In recent years, squamouscell carcinoma and adenoid cystic carcinoma have gradually decreased in frequency, while adenocarcinoma, small and large cell carci- nomahave gradually increased. These tumors tend to be found more frequently in the carina and the lung, i.e. from the proximal to distal (2). Benign tumors include manytypes of mesenchymal neoplasms, such as leiomyoma, mixed tumor, fibroma, neurofibroma and fibrous histiocytoma (3, 4). Leiomyomais the most frequently encountered benign tracheal tumor. Half of those benign tumors are located in the lower third of the trachea (1). They show a round and smooth surface appearance and are often pedunculated. Although they are usually clinically asymptomatic, they maysometimes cause stridDr, and are diagnosed as insidious bronchial asthma (1). Fibromyxomamayoccasionally be found in other organs such as the nasopharynx, but it is rarely found in the trachea. A review of the medical literature showed that this is only the second reported case of fibromyxoma originating in the trachea. The first case of fibromyxomaof the trachea was reported in 1985 by Pollak et al (5). The size of the tumorreported by Pollak et al (5) was large enough to restrict the air flow and to cause dyspnea and wheezing, and it required a mid-tracheal sleeve resection. Both cases showed fibroblastic proliferation with a myxoid matrix. The cause of the focal activation of fibroblasts is not yet clear. Further accumulation of case reports mayhelp to clarify the etiology of this rare fibromyxoma. Fig. 2. a) The tumor, covered by normal tracheal rrtucosa, consists of fibrous tissue with myxoidmatrix in the submucosal area. In the deeper region a part of the tracheal cartilage is seen (HE stain, x35). b) The myxoid matrix in References the loose fibrous tissue with fibroblastic proliferation is observed (HE stain, xl80). 1) Houston HE, Payne WS, Harrison EG, Olsen AM. Primary of the trachea. Arch Surg 99: 132, 1969. 2) Morita T. Cancers of the trachea and carina in comparison with lung cancer: a nation-wide autopsy case analysis in Japan from 1958 to 1987. due to the finding of fibroblastic proliferation with an extensive Lung Cancer 7: 355, 1991. myxoid matrix in loose collagenous tissue and no epithelial 3) Gilbert JG, Mazzarella LA, Feit LJ. Primary tracheal tumors in the infant component. and adult. Arch Otolaryngol 58: 1, 1953. 4) D'Aunoy R, Zoeller A. Primary tumors of the trachea: report ofacase and review of the literature. Arch Pathol Lab Med ll: 589, 1931. 5) Pollak ER, Naunheim KS, Little AG. Fibromyxoma of the trachea: a review of benign tracheal tumors. Arch Pathol Lab Med 109: 926, 1985.

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