Ann Thorac Surg 1998;66:948-950
© 1998 The Society of Thoracic Surgeons
Case Reports
Inflammatory pseudotumors of the lung
Christos Alexiou, FRCSa,
Zbigniev Obuszko, MDa,
David Beggs, FRCSa,
William E. Morgan, FRCSa
a Department of Cardiothoracic Surgery, City Hospital, Nottingham, United Kingdom
Accepted for publication April 3, 1998.
Address reprint requests to Mr Alexiou, Department of Cardiothoracic Surgery, City Hospital, Hucknall Rd, Nottingham NG5 1PB, UK
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Abstract
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Two unusual cases of inflammatory pseudotumors in young female patients are described. One presented with massive hemoptysis and a solitary circumscribed mass treated with urgent lobectomy. The second presented initially with cough and a small right lower lobe mass. She presented again, 8 years later, with a lung mass so expanded as to necessitate a pneumonectomy with partial resection of surrounding structures. Both cases indicate the need for early and complete removal of the inflammatory pseudotumors.
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Introduction
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Inflammatory pseudotumors (IPTs) (or plasma cell granulomas) are benign, essentially nonneoplastic, tumorlike masses, inflammatory in origin. Although they have been widely reported under different names (eg, postinflammatory tumors, histiocytoma, xanthoma, fibroxanthoma, xanthogranuloma, plasma cell tumor), their first detailed description was made in 1973 [1]. We present 2 cases that illustrate the diversity of clinical presentations and unpredictability of the natural course of IPTs.
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Case reports
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Patient 1
A 16-year-old girl, who was a nonsmoker, presented with a 4-day history of severe hemoptysis of 50 to 100 mL, occurring several times per day. On examination she was distressed and was coughing up blood every few minutes. Chest radiographs and an urgent computed tomographic scan (Fig 1) showed a 5-cm right lower lobe mass. At bronchoscopy the right lower lobe bronchus was filled with blood clot. In view of the distress and the uncertain diagnosis and prognosis, right thoracotomy and right lower lobectomy were performed. Histologic examination revealed a fibrohistiocytic subtype of IPT of the lung. Two years later she is well with no tumor recurrence.
Patient 2
A 17-year-old girl first presented with a chronic cough. A chest radiograph showed a small right lower lobe rounded mass with collapse. Bronchoscopy revealed stenosis of the right lower bronchus. She was then lost to follow-up but presented again 8 years later coughing up purulent sputum. Chest radiographs and computed tomographic scan (Fig 2) on this occasion showed right lower lobe collapse and a large rounded mass in the midzone posteriorly. Fine needle aspiration biopsy was unhelpful. At bronchoscopy, the right main bronchus had a slitlike orifice but no endobronchial lesion. At thoracotomy there was a large, lobulated, hard tumor in the right lower lobe of the lung, extending to the paravertebral gutter and invading chest wall, diaphragm, mediastinum, and pericardium around the inferior pulmonary vein. Frozen section was inconclusive. Complete removal of the tumor necessitated a right pneumonectomy with resection of parts of the chest wall, right hemidiaphragm, and pericardium. Histologic examination demonstrated the presence of plasma cell granuloma. At follow-up 8 years later she remains well without clinical or radiologic evidence of tumor recurrence.
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Comment
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Inflammatory pseudotumors of the lung are rare, with no predisposition to sex or race. More than half of the patients are less than 40 years of age, and 15% are between the ages of 1 and 10 years. Their etiology and pathogenesis remain uncertain, although prior lung infection was recorded in one third of the patients [1, 2]. Suggestions include immune mediated response and organizing intraalveolar pneumonia with progression to other histopathologic types [2].
Although IPTs are regarded as inflammatory or reactive lesions rather than neoplasms, they may have features such as angioinvasion, local recurrence, distant metastases, and cytogenetic clonal changes [3–5].
Macroscopically IPTs are well-circumscribed, nonencapsulated, firm, white-yellow masses (Fig 3). Most are parenchymal but some are endobronchial and may cause obstruction. Less than 5% invade the mediastinum and chest wall [1, 2, 6]. Microscopically, the lesions consist of variable mixtures of fibroblasts and granulation tissue, fibrous tissue, and inflammatory cells including lymphocytes, histiocytes, giant cells, macrophages, neutrophils, eosinophils, and typically large numbers of plasma cells [1, 2]. Immunohistochemistry has demonstrated the polyclonal nature of the plasma cells, with immunoglobulin G predominance [5].
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Fig 3. Cuts through the excised masses, with the right lower lobe (A) and right lung (B) demonstrating the typical pale yellow appearance of the inflammatory pseudotumor.
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Many patients are asymptomatic, and the IPTs are discovered by incidental findings on examination of radiographs; if the patients are symptomatic, cough, hemoptysis, shortness of breath, clubbing, chest pain, and arthralgia may be noted [1]. Our first patient presented with massive hemoptysis necessitating urgent lobectomy. Radiologically IPTs typically present as solitary circumscribed masses. Multiple lung masses, pneumonic consolidation, atelectasis, hilar masses, and cavitation are unusual [1, 2, 6]. Percutaneous fine needle aspiration biopsy is considered insufficient for diagnosis, and frozen section is also subject to errors [2].
Early and complete surgical resection of the IPTs remains the best treatment option to exclude malignancy and to achieve cure. Delay in diagnosis and treatment may increase considerably the magnitude of the surgical intervention required, as shown in our second patient.
Nonsurgical treatment modalities including radiotherapy, chemotherapy, and steroids may have a place in the setting of incomplete surgical resection, multifocal disease, postoperative tumor recurrence, or contraindications to lung resection [7, 8]. The natural history of IPTs is unpredictable. Although spontaneous regression may occur, local expansion may cause significant morbidity and occasionally death [1].
In conclusion, although the most common picture of the IPTs is one of an asymptomatic, well-circumscribed lung mass that may mimic cancer, clinicians need to bear in mind their diverse clinical presentations. Surgical excision is usually indicated to reach a firm diagnosis and to attempt cure.
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References
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- Bahadori M., Liebow A.A. Plasma cell granulomas of the lung. Cancer 1973;31:191-208.[Medline]
- Matsubara O., Tan-Liu N.S., Kenney R.M., Mark E.J. Inflammatory pseudotumors of the lung: progression from organizing pneumonia to fibrous histiocytoma or to plasma cell granuloma in 32 cases. Hum Pathol 1988;19:807-814.[Medline]
- Warter A., Satge D., Roeslin N. Angioinvasive plasma cell granulomas of the lung. Cancer 1987;59:435-443.[Medline]
- Maier H.C., Sommers S.C. Recurrent and metastatic pulmonary fibrous histiocytoma/plasma cell granuloma in a child. Cancer 1987;60:1073-1076.[Medline]
- Snyder C.S., Dell’Aquila M., Haghighi P., Baergen R.N., Suh Y.K., Yi E.S. Clonal changes in inflammatory pseudotumour of the lung. Cancer 1995;76:1545-1549.[Medline]
- McCall I.W., Woo-Ming M. The radiological appearances of plasma cell granuloma of the lung. Clin Radiol 1978;29:145-150.[Medline]
- Imperato J.P., Folkman J., Sagerman R.H., Cassdy J.R. Treatment of plasma cell granuloma of the lung with radiation therapy. A report of two cases and a review of the literature. Cancer 1986;57:2127-2129.[Medline]
- Shirakusa T., Kusano T., Motonaga R., Eimoto T. Plasma cell granuloma of the lung-resection and steroid therapy. Thorac Cardiovasc Surg 1987;35:185-188.[Medline]
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Abstract
Introduction
