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+The patient was a 34-yr-old man who presented with complaints of fever and a chronic cough.
+He was a smoker and had a history of pulmonary tuberculosis that had been treated and cured.
+A computed tomographic (CT) scan revealed multiple tiny nodules in both lungs.
+A thoracoscopic lung biopsy was taken from the right upper lobe.
+The microscopic examination revealed a typical LCH.
+The tumor cells had vesicular and grooved nuclei, and they formed small aggregations around the bronchioles (Fig.1).
+The tumor cells were strongly positive for S-100 protein, vimentin, CD68 and CD1a.
+There were infiltrations of lymphocytes and eosinophils around the tumor cells.
+With performing additional radiologic examinations, no other organs were thought to be involved.
+He quit smoking, but he received no other specific treatment.
+He was well for the following one year.
+After this, a follow-up CT scan was performed and it showed a 4 cm-sized mass in the left lower lobe, in addition to the multiple tiny nodules in both lungs (Fig.2).
+A needle biopsy specimen revealed the possibility of a sarcoma; therefore, a lobectomy was performed.
+Grossly, a 4 cm-sized poorly-circumscribed lobulated gray-white mass was found (Fig.3), and there were a few small satellite nodules around the main mass.
+Microscopically, the tumor cells were aggregated in large sheets and they showed an infiltrative growth.
+The cytologic features of some of the tumor cells were similar to those seen in a typical LCH.
+However, many tumor cells showed overtly malignant cytologic features such as pleomorphic/hyperchromatic nuclei and prominent nucleoli (Fig.4), and multinucleated tumor giant cells were also found.
+There were numerous mitotic figures ranging from 30 to 60 per 10 high power fields, and some of them were abnormal.
+A few foci of typical LCH remained around the main tumor mass.
+Immunohistochemically, the tumor cells were strongly positive for S-100 protein (Fig.5) and vimentin; they were also positive for CD68 (Dako N1577, Clone KPI), and focally positive for CD1a (Fig.6), and they were negative for cytokeratin, epithelial membrane antigen, CD3, CD20 and HMB45.
+The ultrastructural analysis failed to demonstrate any Birbeck granules in the cytoplasm of the tumor cells.
+Now, at five months after lobectomy, the patient is doing well with no significant change in the radiologic findings.