KMID : 0371619990150020249
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Journal of Wonkwang Medical Science 1999 Volume.15 No. 2 p.249 ~ p.254
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A Case of Pulmonary Histiocytosis-X Associated with Bilatera, Recurrent, and Spontaneous Pneumothorax
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Song Woo-Kern
Yang Sei-Hoon
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Abstract
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Pulmonary histiocytosis X can be associated with disseminated histiocytosis X, but isolated case in the lung parenchyme is rare. Pulmonary histiocytosis X is a diffuse, smoking-related, chronic interstitial lung disease characterized by proliferation and infiltration by langerhans cell and eosinophils. The hallmark of this disorder is the electron microscopic finding of X-body or Birbeck granule that present in the lung of patient. Pulmonary histiocytosis X is common in young adult and shows variable clinical characteristics. It caused by unknown etiology. A 18 year-old man was reffered to our hospital because of pneumothorax. Plain chest X-ray and chest CT revealed thin-walled cysts of various sizes in the both lungs. Video-associated thoracoscopic lung biopsy was performed. Light microscopic examination showed proliferation and infiltration of langerhans cells. Immunohistochemically, Langerhans cells showed strong cytoplasmic staining with S-100 protein and with HLA-DR. He was treated with right pleurodesis, but pneumothorax recurred 14 days later. We reported a case of bilateral spontaneous pneumothoraces in 18 year old with pulmonary histiocytosis-X, which is confirmed by eosinophilic granuloma in Video-associated thoracoscopic lung biopsy.
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KEYWORD
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Pulmonary Histiocytosis X, Pneumothorax
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