Document g26jGwrLr5Q9JpeOE0oDa0Re9

Case Report Respiration 200;67:456-458 Received: July 16, 1999 Accepted after revision: November 16, 1999 Deciduoid Pleural Mesothelioma Affecting a Young Female without Prior Asbestos Exposure Katharina Gloeckner-Hofmanna Xiong-Zeng Zhua Heinrich Bartelsb Alfred C. Fellera Hartmut Merza institute of Pathology, Medical University of Lubeck, and bDepartment of Hematology and Oncology, Stadtisches Krankenhaus Sud, Lubeck, Germany Key Words Pleural mesothelioma Deciduoid mesothelioma Adenocarcinoma Asbestos Abstract Pleural mesothelioma is commonly associated to asbes tos exposure. A 40-year-old woman is described who presented with shortness of breath. She had a smoking history but no history of asbestos exposure. Chest radi ography and computed tomography showed a large tumour on the right lower lung. An open pleural biopsy was performed. A metastatic adenocarcinoma of the pleura was primarily diagnosed. The tumour progressed and after surgical excision an accurate histological and immunohistochemical examination was performed. It revealed a pleural mesothelioma with a deciduoid differ entiation that has not been described before. Copyright 2000 S. KargerAG, Basel Malignant mesothelioma of the thorax is commonly associated in patients with long-term exposure to asbes tos. Asbestos-related mesothelioma develops over a peri od of 14-72 years [1]. The presenting complaints are chest pain, dyspnoea, and recurrent pleural effusions. Fifty per cent ofaffected patients die within 12 months of diagnosis [2]. Mesothelioma has to be differentiated from metastat ic adenocarcinoma of the pleura [3]. The symptoms may be similar and a biopsy has to be performed. Recently, a variant of peritoneal mesothelioma has been described. It affected young females without a history of exposure to asbestos [4-6]. Moreover, histology revealed a deciduoid differentiation and prognosis was poor. We describe a case of deciduoid mesothelioma of the pleura, a localisa tion that has not been reported before. Case Report A 40-year-old woman was admitted to the hospital complaining of dyspnoea. On admission, she was free from fever, cough, chest pain or weight loss. She had been smoking 12 cigarettes a day for the past 15 years. She was the mother of five healthy children; she was not pregnant and had no history of exposure to asbestos. On physical examination, the vesicular sound of the right lung was decreased. Chest radiography showed markedly reduced radiolucency of the right lower lung. Computed tomography (CT) of the chest showed a large tumour of 7 cm diameter in the posterior mediastinum paraver tebrally, close to the heart. Laboratory tests were within normal lim its except for a CA-15.3 of 67.24 U/ml. Exploratory thoracotomy showed infiltration of the pleura, pericardium and diaphragm but KARRE RR Fax + 41 61 306 12 34 E-Mail karger@karger.ch www.karger.com 2000 S. Karger AG, Basel 0025-7931/00/0674-0456$17.50/0 Accessible online at: www.karger.com/journals/res Hartmut Merz, MD Institute of Pathology, Medical University of Lubeck Ratzeburger Allee 160 D-23538 Lubeck (Germany) Tel. +49 451 500 2714, Fax +49 451 500 3328, E-Mail Merz@patho.uni-Luebeck.de sparing primarily the lung tissue. An open pleural biopsy was per formed and primarily diagnosed as metastatic adenocarcinoma. Postoperatively, the woman was thoroughly examined in order to find the primary tumour site, but none was detected in the lung, thy roid gland, breast, kidney, adrenals, ovaries, pancreas or elsewhere. Following operation, chemotherapy was started with 3 cycles of carboplatin, etoposide (VP-16) and vindesine followed by combined chemotherapy 5-fluorouracil (5-FU) and radiotherapy. At that time, the patient presented with sinus tachycardia. A CT scan revealed enlarging tumour masses compressing the left atrium of the heart. The main part of the tumour was surgically removed half a year later. Histological examination revealed a variant of malignant pleural mesothelioma with deciduoid differentiation (fig. 1). Immunohistochemical investigations showed that the tumour expressed pancytokeratin (KL-1, Dianova, Germany; dilution: 1:100), vimentin (DAKO, Denmark; dilution: 1:50), EMA (epithelial membrane anti gen, DAKO; dilution 1:50) and calretinin (Zymed, USA; dilution: 1:50), a marker suggested to be specific for mesothelial cells. No reac tivity was detected by P-HCG (DAKO); dilution: 1:3,000) and the progesterone/oestrogen receptors (both DAKO; dilution: 1.25). Thereafter, the patient took a downhill course; no further aggressive treatment was given and she died shortly thereafter. a Discussion The pleura may be involved in primary or secondary tumours. Secondary metastatic involvement is far more common than are primary tumours. The most frequent metastatic malignancies arise from primary neoplasms of the lung and breast. Malignant mesothelioma usually occurs in older patients with a history of asbestos expo sure. Because ofthe young age of our patient and a history of smoking with no history of asbestos exposure, a diagno sis of metastatic adenocarcinoma was primarily consid ered. Histologically, it is not easy to differentiate adeno carcinoma from mesothelioma [3]. A panel of antibodies has to be used to diagnose a mesothelioma [7]. Recently, a new antibody, calretinin, was developed that is specific for mesothelial cells and therefore makes the diagnosis of mesothelioma more reliable [8]. In the presented case, calretinin was positive. Another special feature ofour case was a deciduoid dif ferentiation. Deciduoid mesothelioma is an extremely rare variant of malignant mesothelioma. So far, only 4 cases have been reported [4-6]. The tumour was located in the peritoneum of young women (13-24 years). The aetiology of this lesion is still unknown. All reported cases of deciduoid mesothelioma in young women raise the pos sibility that the neoplastic lesion may be induced or stim ulated by endogenous hormones. Two cases of malignant pleural mesothelioma that produced human chorionic gonadotropin were reported by Okamoto et al. [9]. In the present case, tumour cells neither stained for P-HCG nor b Fig. 1. a Malignant mesothelioma of the pleura. A difficult differ ential diagnosis from metastatic adenocarcinoma. HE. x 45. b Tu mour cells are large with round nuclei, prominent nucleoli with deciduoid differentiation. HE. x 190. for progesterone/oestrogen receptors. Thus, we and others [4] did not find any evidence to relate the pathogenesis of mesothelioma to endogenous hormones. This case is the first case of deciduoid pleural mesothe lioma and points out the problems in diagnosing a meso thelioma versus an adenocarcinoma metastatic to the pleura. 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