Eur J Cardiothorac Surg 2001;19:92-95
© 2001 Elsevier Science NL
Primitive squamous-cell carcinoma after extrapleural pneumothorax for active tuberculosis
Ottavio Rena,
Caterina Casadio,
Giuliano Maggi
Thoracic Surgery Department, University of Torino, Torino, Italy
Received 19 June 2000;
received in revised form 2 November 2000;
accepted 2 November 2000.
Corresponding author. Thoracic Surgery Department, S. Giovanni Battista Hospital, v. Genova 3, I-10126 Torino, Italy Tel.: +39-11-6966951; fax: +39-11-6960170
e-mail: caterina.casadio{at}unito.it
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Abstract
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Squamous-cell carcinoma into an extrapleural pneumothorax for active tuberculosis was incorrectly diagnosed as late tubercular empyema. Right axillary thoracostomy was carried out to drain large dense effusion decompressing the brachial plexus and the sympathetic chain with symptomatic release. Surgical biopsy of the extrapleural sac allowed to identify two different tissues: normal epithelium similar to epidermis and nodular fragments composed of well-differentiated squamous carcinoma. The cause of this tumour is not clear: probably the carcinoma arose from normal epidermis carried in the extrapleural cavity during multiple air-refills to maintain the therapeutic pneumothorax.
Key Words: Pleura • Pneumothorax • Carcinoma
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1. Introduction
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During the past decades rare cases of primary squamous-cell carcinomas occurring in empyema cavities with percutaneous fistula [1,2], or after extrapleural therapeutic pneumothorax for active tuberculosis without fistula were reported [3–6]. The case of cancer which is presented below has features in common with those reported in literature.
Extrapleural pneumolysis has been performed for pulmonary tuberculosis and the consequent collapse of the lung was maintained for several years with refills of air in the extrapleural space. When the refills ceased a sterile effusion collected in the space. Even after many years epidermoid carcinoma can occur in the extrapleural space. We reported a new case.
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2. Material and methods
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In October 1998 we observed a 66-year-old non-smoker male, admitted to hospital with severe chest and shoulder pain requiring treatment with morphine (buprenorphine cloridrate). The past medical history revealed that in 1947 (51 years before) he was affected by active right-sided tuberculosis and extrapleural pneumothorax was carried out. The subsequent collapse of the lung was maintained weekly with repeated air refills. No foreign agents had been placed in the extrapleural cavity to maintain pulmonary collapse. The patient hasn't been occupationally exposed to asbestos. Air-refills has been discontinued 8 months after extrapleural pneumothorax had been performed but 2 years later (in 1950) he underwent chemo-antibiotic therapy for tubercular recurrence.
During the following 50 years the patient had no other lung diseases and he underwent occasional chest X-rays in 1986 (the roentgenogram has been lost). Only normal outcome of the right extrapleural pneumothorax has been noted at that time.
At hospital admission physical examination of the chest revealed painful swelling of the right upper hemithorax associated with right-sided Horner and Pancoast syndromes and shoulder pain.
Chest radiographs (Fig. 1) and CT-scan (Fig. 2) showed a moderate thickening of the chest wall and an abundant dense effusion occupying the upper portion of the right hemithorax, compressing the lung and extending from the anterior chest wall to the vertebral column. Mediastinal shift, compression on the trachea and the subclavian vessels were noted; there wasn't any evidence of mediastinal enlargement and lung or abdominal masses.
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Fig. 2. CT-scan showing a moderate thickening of the chest-wall and an abundant dense effusion involving the upper right hemithorax. Shift and compression of the mediastinal structures are evident.
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Thoracentesis evacuated a small volume of fluid similar to dense sterile pus. Cytological examination didn't reveal carcinoma cells. Diagnosis of late infection of the extrapleural cavity was posed and we decided to perform an axillary thoracostomy: the extrapleural sac was opened and 1 l of dense effusion contained in the sac was drained, with immediate decompression of the brachial plexus and the sympathetic cervical chain.
Numerous nodules of the cavity wall were seen: pathological material was obtained from these and from the effusion. No tubercular disease or nodular tumour on the lung was detected. Grossly the specimen appeared as thick white laminar fragments or as nodules of cheese-like friable material. Microscopically the laminar fragments showed squamous epithelium covering the connective tissue: this epithelium was not neoplastic and showed the four Malpighian layers typical of the epidermis and had a verrucous appearance with hyper and paracheratotic horns and produced large amounts of keratin. The nodular lesions showed areas of infiltrating squamous carcinoma, the invasive component was well differentiated and the surrounding stroma had an extremely dense desmoplastic tissue reaction with eosinofilic matrix. Calretinin, used for differential diagnosis with mesothelioma, was positive in mesothelial cells entrapped in the stroma, while S-100 protein was positive in dendritic cells consistent with Langerhans cells of the epidermis. A diagnosis of infiltrating squamous carcinoma of the wall of the extrapleural cavity was evident.
Radical surgical excision of the neoplasm was considered not feasible because its firm adhesion at the vertebral column and axillary and mediastinal tissues. The patient was referred for exclusive radiotherapy, and survived 25 weeks after diagnosis of carcinoma. Death occurred for cardiovascular failure related to the large intrathoracic spread of the tumour.
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3. Comments
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During the fourth and fifth decade of this century extrapleural pneumothorax were performed to treat pulmonary active tuberculosis.
Collapse of the lung was maintained placing foreign agents in the artificial cavity or with air-refills for several years. When air refills stopped sterile effusion accumulated in the extrapleural space, resulting in a failure of the space to obliterate.
Onset of an empyema after extrapleural pneumothorax is well-known, while the description of a well differentiated squamous-cell carcinoma with seborroic effusion following the extra-pleural pneumothorax is very rare. In these rare cases a secondary origin of the lesion could be excluded clinically. Similar primary neoplasm has been described in patients subjected to extrapleural pneumothorax for pulmonary tuberculosis in whom the time between the induction of pneumothorax and development of cancer ranged between 3 and 22 years [3–6]. We observed a latency of 51 years.
Willen et al. [6] reported six patients submitted to surgical resection of tumour: all patients underwent exeresis of the extrapleural sac associated to upper lobectomy (two cases) or to thoracectomy (one case). In three cases the resection wasn't radical because gross disease was left behind invading the base of the neck or mediastinal organs or vertebral column: patients were referred to radiation therapy (median 30 Gy, range 25–53 Gy) after surgery. Five patients died from cancer and the median survival was 18 (range 0–36) months. One patient submitted to radical exclusive surgical treatment was referred still alive after 20 years.
Ruttner et al. [5] reported one case of tumour resected by lung decortication. Few notices about the patient reveal that she was alive 5 years after surgery. Ender et al. [4] reported one case of tumour but notices about treatment and survival are not available. The treatment of choice for these well-differentiated squamous cells appeared to be surgical extirpation with good long-term survival if the resection has been radical. The role of the radiation therapy is not clear and it seems that it could not influence the survival of the patient submitted to non-radical resection in the series referred by Willen et al. [6].
In our case the tumour local extension was too large and contraindicated surgical excision.
The well-differentiated epidermoid carcinoma produced a dense seborroic effusion which filled up the extrapleural space and compressed the brachial plexus and the sympathetic cervical chain. Surgical drainage of the cavity decompressed the nervous structures with immediate even if not complete symptomatic release.
The cause of development of this epithelial primary tumour is unknown, probably related to chronic inflammation [7–9], but others suggested pathogenic correlation with asbestos exposure [7]: in our patient occupationally asbestos exposure was excluded. It is possible that both asbestos exposure and chronic inflammation contributed to the development of the squamous carcinoma. It might be that asbestos exposure was coincidental and the tumour would have followed the extrapleural pneumothorax anyway.
It is debatable whether the neoplasm originates from metaplasia of the pleural mesothelial cells or whether it is due to the displacement in the connective tissue of the extrapleural sac of epidermal fragments after repeated needle punctures. We found pleural connective tissue covered by squamous epithelium in all respects similar to normal epidermis probably derived from epidermal fragments carried in the extrapleural cavity by needle re-fillings of the pneumothorax. Probably this epithelial layer developed the squamous-cell carcinoma invading the surrounding stroma.
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References
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Abstract
1. Introduction
