J Thorac Cardiovasc Surg 1994;108:393-395
© 1994 Mosby, Inc.
Regression of autoimmune thrombocythemia after resection of pulmonary plasma cell granuloma
Federico Venuta, MD,
Erino A. Rendina, MD,
Eugenio Pompeo, MD,
Paola Ciriaco, MD,
Tiziano de Giacomo, MD,
Alessandro Pulsoni, MD,
Costante Ricci, MD, FACP
Department of Thoracic Surgery
University of Rome "La Sapienza"
Department of Biopathology
Institute of Hematology
Rome, Italy
To the Editor:
Plasma cell granuloma (PCG), a rare, nonneoplastic lesion of the lung, first was described by Bahadori and Liebow
1 PCG accounts for fewer than 1% of all lung tumors. It occurs at all ages but predominantly in children and young adults; sexes and ethnic groups are equally affected. Clinical features, including evidence of a preceding or concurrent respiratory infection, are variable, but almost 70% of patients have no symptoms. 2 Associations with such immunologic and hematologic disorders as hypergammaglobulinemia involving immunoglobulin G, M, and A fractions, elevation of erythrocyte sedimentation rate, and thrombocytosis have been reported. 3,4 Chest radiographs usually demonstrate a single round lesion, more rarely showing double or multiple lesions. Computed tomographic scan often resembles bronchogenic carcinoma and the diagnostic accuracy of transthoracic fine-needle aspiration biopsy is also questionable. 2 The natural history of PCG of the lung is variable; it may resolve spontaneously, remain static for years, or continue to grow with possible aggressive impingement on the surrounding structures. 2 In fact, invasion of the mediastinum, the diaphragm, and the vessels has been described, as well as recurrence after surgical resection. 5 A surgical approach is therefore advocated for diagnostic purposes as well as for complete excision of the lesion and prevention of local recurrence.
We present here a case report of PCG of the lung associated with autoimmune thrombocythemia that regressed after radical resection of the pulmonary lesion. We believe that the case we describe here is the first documented observation of such an association.
The patient was a 24-year-old nonsmoking man who had been well until the age of 7 years, when he had right basal bronchopneumonia. Ten years later, a chest radiograph demonstrated a pulmonary coin lesion of the left lower lobe in the absence of any clinical symptoms. The patient refused further diagnostic procedures. When the patient was 23 years old, routine laboratory examinations were remarkable for a platelet count of 54,000 cells/ml. A few months later, laboratory and immunologic screening showed a platelet count of 48,000 cells/ml with a decreased platelet-aggregation capacity and endogen adenosine diphosphate block. Results of direct Dixon test were positive for the presence of antiplatelet autoantibodies (1000 ng immunglobulin G/10 7 platelets, normal range 0 to 100 ng immunglobulinG/10 7 platelets). A myelogram showed an increased number of megakaryocytes. Coagulation test results were within normal limits. A chest radiograph (Fig.1) integrated by computed tomographic scan of the thorax (Fig. 2) confirmed the presence of a left lower lobe opacity, the main diameter of which had increased from 2.5 to 3.0 cm, with evidence of pleural perinodular thickening. Results of bronchoscopy were negative. A left lower lobectomy was performed without difficulties. Histologic diagnosis was PCG originating in the lung with a localized proliferation of mature plasma cells, reticuloendothelial cells, and intermediate forms supported by a stroma of granulation tissue infiltrating the visceral pleura. Although only 1 U platelet concentrate was transfused before the operation and no steroids were administered, platelet count was 310,000 cells/ml on postoperative day 10. The postoperative course was uneventful, but during follow-up the laboratory tests showed a reduction of the platelet count to 140,000 cells/ ml 3 months after the operation. This value remains stable 24 months after operation. Results of direct Dixon test are actually negative for the presence of antiplatelet autoantibodies. The increased platelet count after removal of the tumor may suggest a correlation between the immunologic disorder and the presence of PCG of the lung. A secretory role of PCG has been previously supposed, 1,4 but a hyperimmune response to the lesion itself may also explain detection of high immunoglobulin levels. 4 The presence of autoantibodies in this patient and their decrease after surgical resection of the pulmonary lesion suggest an involvement of the immune system in this disorder.
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Fig. 2. Computed tomographic scan of the thorax shows a round lesion (3 cm) in the left lower lobe, with evidence of pleural perinodular thickening.
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References
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Bahadori M, Liebow AA. Plasma cell granulomas of the lung. Cancer 1973;31:191-208.
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Monzon CM, Gilchrist GS, Burgert EO, et al. Plasma cell granuloma of the lung in children. Pediatrics 1982;70:268-74.
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Mandelbaum I, Brashear RE, Hull MT. Surgical treatment and course of pulmonary pseudotumor (plasma cell granuloma). J THORAC CARDIOVASC SURG 1981;82:77-82.
