Early Detection of Pulmonary Lymphangioleiomyomatosis by Spontaneous Pneumothorax A Case Report
A 31-year-old female was admitted because of dyspnea. A chest X-ray showed pneumothorax. A computed tomography (CT) scan of the chest revealed scattered thin-walled cystic lesions in both lung fields. Pulmonary lymphangioleiomyomatosis (LAM) was confirmed by a thoracoscopic lung biopsy, which reveal...
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| Published in | Kita Kantō igaku (The Kitakanto Medical Journal) Vol. 53; no. 3; pp. 303 - 307 |
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| Main Authors | , , , , |
| Format | Journal Article |
| Language | English Japanese |
| Published |
The Kitakanto Medical Society
2003
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| Subjects | |
| Online Access | Get full text |
| ISSN | 1343-2826 1881-1191 |
| DOI | 10.2974/kmj.53.303 |
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| Abstract | A 31-year-old female was admitted because of dyspnea. A chest X-ray showed pneumothorax. A computed tomography (CT) scan of the chest revealed scattered thin-walled cystic lesions in both lung fields. Pulmonary lymphangioleiomyomatosis (LAM) was confirmed by a thoracoscopic lung biopsy, which revealed the nodular proliferation of immature smooth muscle cells that stained positive for HMB-45, desmin, α-smooth muscle actin, progesterone receptor and estrogen receptor when examined using immunostaining techniques. Medroxyprogesterone was prescribed. This report describes the early detection of LAM using chest CT scans and a thoracoscopic lung biopsy in a patient that experienced a spontaneous pneumothorax. |
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| AbstractList | A 31-year-old female was admitted because of dyspnea. A chest X-ray showed pneumothorax. A computed tomography (CT) scan of the chest revealed scattered thin-walled cystic lesions in both lung fields. Pulmonary lymphangioleiomyomatosis (LAM) was confirmed by a thoracoscopic lung biopsy, which revealed the nodular proliferation of immature smooth muscle cells that stained positive for HMB-45, desmin, α-smooth muscle actin, progesterone receptor and estrogen receptor when examined using immunostaining techniques. Medroxyprogesterone was prescribed. This report describes the early detection of LAM using chest CT scans and a thoracoscopic lung biopsy in a patient that experienced a spontaneous pneumothorax. |
| Author | Dobashi, Kunio Mori, Masatomo Iwasaki, Yasuki Tomizawa, Hiroko Mita, Yoshinori |
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| References | 4. Kitaichi M, Nishimura K, Itoh H, et al. Pulmonary lymphangioleiomyomatosis : a report of 46 patients including a clinicopathologic study of prognostic factors. Am J Respir Crit Care Med 1995 ; 151 : 527-533. 9. Matsui K, Takeda K, Yu ZX, et al. Downregulation of estrogen and progesterone receptors in the abnormal smooth muscle cells in pulmonary lymphangioleiomyomatosis following therapy. An immunohistochemical study. Am J Respir Crit Care Med 2000 ; 161 : 1002-1009. 1. Johnson S. Rare diseases. 1. Lymphangioleiomyomatosis : clinical features, management and basic mechanisms. Thorax 1999 ; 54 : 254-264. 7. Oh YM, Mo EK, Jang SH, et al. Pulmonary lymphangioleiomyomatosis in Korea. Thorax 1999 ; 54 : 618-621. 12. Avila NA, Chen CC, Chu SC, et al. Pulmonary lymphangioleiomyomatosis : correlation of ventilation-perfusion scintigraphy, chest radiography, and CT with pulmonary function tests. Radiology 2000; 214 : 441-446. 10.松島秀和, 高柳 昇, 川田一郎ら.高齢者びまん性過誤腫性肺脈管筋腫症の1例.日呼吸会誌2001;39 : 519-523. 13. 松村輔二, 岡田克典, 島田和佳ら.東日本における肺移植希望患者の臨床的検討.日呼吸会誌2000;38 : 839-843. 5. Logginidou H, Ao X, Russo I, et al. Frequent estrogen and progesterone receptor immunoreactivity in renal angiomyolipomas from women with pulmonary lymphangioleiomyomatosis. Chest 2000 ; 117 : 25-30. 11.中込一之, 大部 幸, 山本夏男ら.気管支喘息として治療されていた肺リンパ脈管筋腫症の1例.日呼吸会誌2001;39 : 567-571. 3. 野村 智, 南 眞司, 藤村政樹ら.肺リンパ脈管筋腫症を伴った結節性硬化症の1例.日呼吸会誌2001;39 : 944-948. 2. Seyama K, Kira S, Takahashi H, et al. Longitudinal follow-up study of 11 patients with pulmonary lymphangioleiomyomatosis : diverse clinical courses of LAM allow some patients to be treated without anti-hormone therapy. Respirology 2001 ; 6 : 331-340. 8. Johnson SR, Tattersfield AE. Clinical experience of pulmonary lymphangioleiomyomatosis in the UK. Thorax 2000 ; 55 : 1052-1057. 6. 佐藤輝彦, 瀬山邦明, 児玉裕三ら.肺リンパ脈管筋腫症 (Pulmonary lymphangioleiomyomatosis ; LAM) に合併する自然気胸の臨床的意義.日呼吸会誌2001;39 : 656-663. |
| References_xml | – reference: 3. 野村 智, 南 眞司, 藤村政樹ら.肺リンパ脈管筋腫症を伴った結節性硬化症の1例.日呼吸会誌2001;39 : 944-948. – reference: 12. Avila NA, Chen CC, Chu SC, et al. Pulmonary lymphangioleiomyomatosis : correlation of ventilation-perfusion scintigraphy, chest radiography, and CT with pulmonary function tests. Radiology 2000; 214 : 441-446. – reference: 7. Oh YM, Mo EK, Jang SH, et al. Pulmonary lymphangioleiomyomatosis in Korea. Thorax 1999 ; 54 : 618-621. – reference: 9. Matsui K, Takeda K, Yu ZX, et al. Downregulation of estrogen and progesterone receptors in the abnormal smooth muscle cells in pulmonary lymphangioleiomyomatosis following therapy. An immunohistochemical study. Am J Respir Crit Care Med 2000 ; 161 : 1002-1009. – reference: 13. 松村輔二, 岡田克典, 島田和佳ら.東日本における肺移植希望患者の臨床的検討.日呼吸会誌2000;38 : 839-843. – reference: 4. Kitaichi M, Nishimura K, Itoh H, et al. Pulmonary lymphangioleiomyomatosis : a report of 46 patients including a clinicopathologic study of prognostic factors. Am J Respir Crit Care Med 1995 ; 151 : 527-533. – reference: 10.松島秀和, 高柳 昇, 川田一郎ら.高齢者びまん性過誤腫性肺脈管筋腫症の1例.日呼吸会誌2001;39 : 519-523. – reference: 8. Johnson SR, Tattersfield AE. Clinical experience of pulmonary lymphangioleiomyomatosis in the UK. Thorax 2000 ; 55 : 1052-1057. – reference: 11.中込一之, 大部 幸, 山本夏男ら.気管支喘息として治療されていた肺リンパ脈管筋腫症の1例.日呼吸会誌2001;39 : 567-571. – reference: 5. Logginidou H, Ao X, Russo I, et al. Frequent estrogen and progesterone receptor immunoreactivity in renal angiomyolipomas from women with pulmonary lymphangioleiomyomatosis. Chest 2000 ; 117 : 25-30. – reference: 6. 佐藤輝彦, 瀬山邦明, 児玉裕三ら.肺リンパ脈管筋腫症 (Pulmonary lymphangioleiomyomatosis ; LAM) に合併する自然気胸の臨床的意義.日呼吸会誌2001;39 : 656-663. – reference: 1. Johnson S. Rare diseases. 1. Lymphangioleiomyomatosis : clinical features, management and basic mechanisms. Thorax 1999 ; 54 : 254-264. – reference: 2. Seyama K, Kira S, Takahashi H, et al. Longitudinal follow-up study of 11 patients with pulmonary lymphangioleiomyomatosis : diverse clinical courses of LAM allow some patients to be treated without anti-hormone therapy. Respirology 2001 ; 6 : 331-340. |
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| Title | Early Detection of Pulmonary Lymphangioleiomyomatosis by Spontaneous Pneumothorax A Case Report |
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