結節性硬化症に伴う多巣性微小結節性肺細胞過形成を呈したと考えられる2症例
多巣性微小結節性肺細胞過形成（multifocal micronodular pneumocyte hyperplasi（aMMPH））は，Ⅱ型肺胞上皮様細胞の増殖からなる多巣性結節性病変で，結節性硬化症（tuberous sclerosis complex（TSC））に併発する疾患である．自覚症状はあっても軽微であり，進行は緩徐で治療を要しないとされている．われわれは，TSCに併発した，特徴的な画像所見を持つMMPHと考えられる2症例を経験したので報告する．症例1：幼児期にTSCと診断された20歳女性．18歳時の胸部CTで両肺に散在する結節影やスリガラス影を指摘された．症例2：36歳時にTS...
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| Published in | Journal of UOEH Vol. 39; no. 2; pp. 133 - 141 |
|---|---|
| Main Authors | , , , , , , , , , , , |
| Format | Journal Article |
| Language | English Japanese |
| Published |
学校法人 産業医科大学
2017
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| Subjects | |
| Online Access | Get full text |
| ISSN | 0387-821X 2187-2864 |
| DOI | 10.7888/juoeh.39.133 |
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| Abstract | 多巣性微小結節性肺細胞過形成（multifocal micronodular pneumocyte hyperplasi（aMMPH））は，Ⅱ型肺胞上皮様細胞の増殖からなる多巣性結節性病変で，結節性硬化症（tuberous sclerosis complex（TSC））に併発する疾患である．自覚症状はあっても軽微であり，進行は緩徐で治療を要しないとされている．われわれは，TSCに併発した，特徴的な画像所見を持つMMPHと考えられる2症例を経験したので報告する．症例1：幼児期にTSCと診断された20歳女性．18歳時の胸部CTで両肺に散在する結節影やスリガラス影を指摘された．症例2：36歳時にTSCと診断された44歳女性．43歳時の胸部CTで上葉優位にランダム分布の小さなスリガラス影を認めた．症例1は2年間，症例2は4年間に渡り，呼吸器症状も画像変化も認めない．肺生検の同意が得られず，病理学的な診断ができていないが，血液検査で有意な所見はなく，10 mm以下の結節影やスリガラス影が散在する特徴的画像所見や経年的に不変である経過などから，TSCに伴うMMPHと考えた．2症例とも肺病変に対する治療は行っていない．MMPHは稀な疾患であるが，TSC患者において両肺に多発する結節影やスリガラス影が認められた際にはMMPHである可能性を考慮すべきである． |
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| AbstractList | 多巣性微小結節性肺細胞過形成（multifocal micronodular pneumocyte hyperplasi（aMMPH））は，Ⅱ型肺胞上皮様細胞の増殖からなる多巣性結節性病変で，結節性硬化症（tuberous sclerosis complex（TSC））に併発する疾患である．自覚症状はあっても軽微であり，進行は緩徐で治療を要しないとされている．われわれは，TSCに併発した，特徴的な画像所見を持つMMPHと考えられる2症例を経験したので報告する．症例1：幼児期にTSCと診断された20歳女性．18歳時の胸部CTで両肺に散在する結節影やスリガラス影を指摘された．症例2：36歳時にTSCと診断された44歳女性．43歳時の胸部CTで上葉優位にランダム分布の小さなスリガラス影を認めた．症例1は2年間，症例2は4年間に渡り，呼吸器症状も画像変化も認めない．肺生検の同意が得られず，病理学的な診断ができていないが，血液検査で有意な所見はなく，10 mm以下の結節影やスリガラス影が散在する特徴的画像所見や経年的に不変である経過などから，TSCに伴うMMPHと考えた．2症例とも肺病変に対する治療は行っていない．MMPHは稀な疾患であるが，TSC患者において両肺に多発する結節影やスリガラス影が認められた際にはMMPHである可能性を考慮すべきである． |
| Author | 赤田, 憲太朗 野口, 真吾 坂本, 憲穂 石本, 裕士 西田, 千夏 迎, 寛 山﨑, 啓 星野, 鉄兵 花香, 未奈子 下野, 昌幸 矢寺, 和博 城戸, 貴志 |
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| References | 1. Popper HH, Juettner-Smolle FM & Pongratz MG (1991): Micronodular hyperplasia of type Ⅱ pneumocytes–a new lung lesion associated with tuberous sclerosis. Histopathology 18: 347−354 25. Behnes CL, Schütze G, Engelke C, Bremmer F, Gunawan B, Radzun HJ & Schweyer S (2013): 13-year-old tuberous sclerosis patient with renal cell carcinoma associated with multiple renal angiomyolipomas developing multifocal micronodular pneumocyte hyperplasia. BMC Clin Pathol 13: 4 28. Suzuki K, Seyama K, Hayashi T, Yamashiro Y, Shiraishi A & Kuwatsuru R (2013): Reversed halo sign in tuberous sclerosis complex. Case Rep Radiol 2013: 428501 (doi: 10.1155/2013/428501. Epub 2013 Sep 15) 13. Yamanaka A, Kitaichi M, Fujimoto T, Hirai T, Hori H & Konishi F (2000): Multifocal micronodular pneumocyte hyperplasia in a postmenopausal woman with tuberous sclerosis. Virchows Arch 436: 389−392 29. Lee IS, Shim HS, Lee HJ & Kang YA (2014): Multifocal micronodular pneumocyte hyperplasia mimicking miliary tuberculosis. Am J Respir Crit Care Med 189: 225−226 3. Kobashi Y, Sugiu T, Mouri K, Irei T, Nakata M & Oka M (2008): Clinicopathological analysis of multifocal micronodular pneumocyte hyperplasia associated with tuberous sclerosis in Japan. Respirology 13: 1076−1081 11. Guinee D, Singh R, Azumi N, Singh G, Przygodzki RM, Travis W & Koss M (1995): Multifocal micronodular pneumocyte hyperplasia: a distinctive pulmonary manifestation of tuberous sclerosis. Mod Pathol 8: 902−906 22. Incedayi M, Sonmez G & Basekim C (2011): Massive bilateral renal anigiomyolipomatosis and multifocal micronodular pneumocytes hyperplasia associated with tuberous sclerosis: a case report. Wien Klin Wochenschr 123: 674−676 6. Roach ES, Gomez MR & Northrup H (1998): Tuberous sclerosis complex consensus conference: revised clinical diagnostic criteria. J Child Neurol 13:624−628 30. Sun Q, Cai HR, Mark EJ, Miao LY, Wu HY, Zhou Q, Chen J, Zhang W & Meng FQ (2015): Multifocal micronodular pneumocyte hyperplasia in a Chinese man masquerading as miliary tuberculosis. Int J Clin Exp Pathol 8: 2165−2170 24. Miravet Sorribes L, Mancheño Franch N & Batalla Bautista L (2013): Multifocal micronodular pneumocyte hyperplasia in a patient with tuberous sclerosis. Arch Bronconeumol 49: 36−37 8. Maruyama H, Ohbayashi C, Hino O, Tsutsumi M & Konishi Y (2001): Pathogenesis of multifocal micronodular pneumocyte hyperplasia and lymphangioleiomyomatosis in tuberous sclerosis and association with tuberous sclerosis genes TSC1 and TSC2. Pathol Int 51: 585−594 16. Kobayashi T, Satoh K & Ohkawa M (2005): Multifocal micronodular pneumocyte hyperplasia associated with tuberous sclerosis. Acta Radiol 46: 37−40 12. Wu K & Tazelaar HD (1999): Pulmonary angiomyolipoma and multifocal micronodular pneumocyte hyperplasia associated with tuberous sclerosis. Hum Pathol 30: 1266−1268 5. Oka T (2005): Tuberous sclerosis related pulmonary lesions other than lymphangioleiomyomatosis. Kokyu 24: 505−514 (in Japanese) 15. Fujitaka K, Isobe T, Oguri T, Yamasaki M, Miyazaki M, Kohno N, Takeshima Y & Inai K (2002): A case of micronodular pneumocyte hyperplasia diagnosed through lung biospy using thoracoscopy. Respiration 69: 277−279 18. Kobashi Y, Yoshida K, Miyashita N, Niki Y, Matsushima T & Irei T (2005): Multifocal micronodular pneumocyte hyperplasia in a man with tuberous sclerosis. Intern Med 44: 462−466 27. Ogawa R, Miyagawa M, Ide K, Akamune A, Ohtsuki Y & Mochizuki T (2013): Exacerbation and remission of pulmonary micronodules with lymphangioleiomyomatosis around the time of childbirth. Jpn J Radiol 31: 633−636 10. Corrin B, Liebow AA & Friedman PJ (1975): Pulmonary lymphangiomyomatosis. A review. Am J Pathol 79: 348−382 14. Cancellieri A, Poletti V & Corrin B (2002): Respiratory failure due to micronodular type II pneumocyte hyperplasia. Histopathology 41: 263−265 21. Chang MH & Yao-Peng H (2010): Lymphangioleiomyomatosis, multifocal micronodular pneumocyte hyperplasia, and clear-cell micronodules in a 39-year-old woman with tuberous sclerosis: a case report. Int J Surg Pathol 18: 522−525 17. Ristagno RL, Biddinger PW, Pina EM & Meyer CA (2005): Multifocal micronodular pneumocyte hyperplasia in tuberous sclerosis. AJR Am J Roentgenol 184: S37−S39 2. Muir TE, Leslie KO, Popper H, Kitaichi M, Gagné E, Emelin JK, Vinters HV & Colby TV (1998): Micronodular pneumocyte hyperplasia. Am J Surg Pathol 22: 465−472 23. Ishii M, Asano K, Kamiishi N et al (2012): Tuberous sclerosis diagnosed by incidental computed tomography findings of multifocal micronodular pneumocyte hyperplasia: a case report. J Med Case Rep 6: 352 9. Tanaka M, Hirata H, Wataya-Kaneda M, Yoshida M & Katayama I (2016): Lymphangioleiomyomatosis and multifocal micronodular pneumocyte hyperplasia in Japanese patients with tuberous sclerosis complex. Respir Investig 54: 8−13 4. Taniguchi N, Konno S, Nasuhara Y, Matsuno Y, Oka T & Nishimura M (2011): A case of multifocal micronodular pneumocyte hyperplasia without clinical findings of tuberous sclerosis. Nihon Kokyuki Gakkai Zasshi 49: 355−359 (in Japanese) 31. Franz DN, Brody A, Meyer C, Leonard J, Chuck G, Dabora S, Sethuraman G, Colby TV, Kwiatkowski DJ & McCormack FX (2001): Mutational and radiographic analysis of pulmonary disease consistent with lymphangioleiomyomatosis and micronodular pneumocyte hyperplasia in women with tuberous sclerosis. Am J Respir Crit Care Med 164: 661−668 33. Hayashi T, Kumasaka T, Mitani K, Yao T, Suda K & Seyama K (2010): Loss of heterozygosity on tuberous sclerosis complex genes in multifocal micronodular pneumocyte hyperplasia. Mod Pathol 23: 1251−1260 20. Saito M, Yuasa T, Satoh S, Nanjo H, Tsuchiya N & Habuchi T (2008): Developing multiple lung nodules in a renal transplant recipient with tuberous sclerosis who had undergone bilateral nephrectomy due to renal cell carcinomas. Int J Urol 15: 257−258 26. Shimoyama T, Kawasaki T, Tanoue M & Kimura B (2014): Pulmonary adenocarcinoma in a patient with tuberous sclerosis complex with multifocal micronodular pneumocyte hyperplasia. Jpn J Lung Canc 54: 135−140 (in Japanese) 7. Okamura A & Yamauchi H (1978): Pulmonary manifestation of tuberous sclerosis-Its relationship to pulmonary hamartoangiomyomatosis. Jpn J Chest Dis 37: 216−222 (in Japanese) ISSN: 03853667 19. Kamiya H, Shinoda K, Kobayashi N, Kubo K, Nomura T, Morita T & Fujii T (2006): Tuberous sclerosis complex complicated by pulmonary multinodular shadows. Intern Med 45: 275−278 32. Maruyama H, Seyama K, Sobajima J, Kitamura K, Sobajima T, Fukuda T, Hamada K, Tsutsumi M, Hino O & Konishi Y (2001): Multifocal micronodular pneumocyte hyperplasia and lymphangioleiomyomatosis in tuberous sclerosis with a TSC2 gene. Mod Pathol 14: 609−614 |
| References_xml | – reference: 19. Kamiya H, Shinoda K, Kobayashi N, Kubo K, Nomura T, Morita T & Fujii T (2006): Tuberous sclerosis complex complicated by pulmonary multinodular shadows. Intern Med 45: 275−278 – reference: 26. Shimoyama T, Kawasaki T, Tanoue M & Kimura B (2014): Pulmonary adenocarcinoma in a patient with tuberous sclerosis complex with multifocal micronodular pneumocyte hyperplasia. Jpn J Lung Canc 54: 135−140 (in Japanese) – reference: 7. Okamura A & Yamauchi H (1978): Pulmonary manifestation of tuberous sclerosis-Its relationship to pulmonary hamartoangiomyomatosis. Jpn J Chest Dis 37: 216−222 (in Japanese) ISSN: 03853667 – reference: 21. Chang MH & Yao-Peng H (2010): Lymphangioleiomyomatosis, multifocal micronodular pneumocyte hyperplasia, and clear-cell micronodules in a 39-year-old woman with tuberous sclerosis: a case report. Int J Surg Pathol 18: 522−525 – reference: 9. Tanaka M, Hirata H, Wataya-Kaneda M, Yoshida M & Katayama I (2016): Lymphangioleiomyomatosis and multifocal micronodular pneumocyte hyperplasia in Japanese patients with tuberous sclerosis complex. Respir Investig 54: 8−13 – reference: 16. Kobayashi T, Satoh K & Ohkawa M (2005): Multifocal micronodular pneumocyte hyperplasia associated with tuberous sclerosis. Acta Radiol 46: 37−40 – reference: 8. Maruyama H, Ohbayashi C, Hino O, Tsutsumi M & Konishi Y (2001): Pathogenesis of multifocal micronodular pneumocyte hyperplasia and lymphangioleiomyomatosis in tuberous sclerosis and association with tuberous sclerosis genes TSC1 and TSC2. Pathol Int 51: 585−594 – reference: 22. Incedayi M, Sonmez G & Basekim C (2011): Massive bilateral renal anigiomyolipomatosis and multifocal micronodular pneumocytes hyperplasia associated with tuberous sclerosis: a case report. Wien Klin Wochenschr 123: 674−676 – reference: 3. Kobashi Y, Sugiu T, Mouri K, Irei T, Nakata M & Oka M (2008): Clinicopathological analysis of multifocal micronodular pneumocyte hyperplasia associated with tuberous sclerosis in Japan. Respirology 13: 1076−1081 – reference: 18. Kobashi Y, Yoshida K, Miyashita N, Niki Y, Matsushima T & Irei T (2005): Multifocal micronodular pneumocyte hyperplasia in a man with tuberous sclerosis. Intern Med 44: 462−466 – reference: 1. Popper HH, Juettner-Smolle FM & Pongratz MG (1991): Micronodular hyperplasia of type Ⅱ pneumocytes–a new lung lesion associated with tuberous sclerosis. Histopathology 18: 347−354 – reference: 10. Corrin B, Liebow AA & Friedman PJ (1975): Pulmonary lymphangiomyomatosis. A review. Am J Pathol 79: 348−382 – reference: 14. Cancellieri A, Poletti V & Corrin B (2002): Respiratory failure due to micronodular type II pneumocyte hyperplasia. Histopathology 41: 263−265 – reference: 11. Guinee D, Singh R, Azumi N, Singh G, Przygodzki RM, Travis W & Koss M (1995): Multifocal micronodular pneumocyte hyperplasia: a distinctive pulmonary manifestation of tuberous sclerosis. Mod Pathol 8: 902−906 – reference: 32. Maruyama H, Seyama K, Sobajima J, Kitamura K, Sobajima T, Fukuda T, Hamada K, Tsutsumi M, Hino O & Konishi Y (2001): Multifocal micronodular pneumocyte hyperplasia and lymphangioleiomyomatosis in tuberous sclerosis with a TSC2 gene. Mod Pathol 14: 609−614 – reference: 17. Ristagno RL, Biddinger PW, Pina EM & Meyer CA (2005): Multifocal micronodular pneumocyte hyperplasia in tuberous sclerosis. AJR Am J Roentgenol 184: S37−S39 – reference: 2. Muir TE, Leslie KO, Popper H, Kitaichi M, Gagné E, Emelin JK, Vinters HV & Colby TV (1998): Micronodular pneumocyte hyperplasia. Am J Surg Pathol 22: 465−472 – reference: 13. Yamanaka A, Kitaichi M, Fujimoto T, Hirai T, Hori H & Konishi F (2000): Multifocal micronodular pneumocyte hyperplasia in a postmenopausal woman with tuberous sclerosis. Virchows Arch 436: 389−392 – reference: 23. Ishii M, Asano K, Kamiishi N et al (2012): Tuberous sclerosis diagnosed by incidental computed tomography findings of multifocal micronodular pneumocyte hyperplasia: a case report. J Med Case Rep 6: 352 – reference: 25. Behnes CL, Schütze G, Engelke C, Bremmer F, Gunawan B, Radzun HJ & Schweyer S (2013): 13-year-old tuberous sclerosis patient with renal cell carcinoma associated with multiple renal angiomyolipomas developing multifocal micronodular pneumocyte hyperplasia. BMC Clin Pathol 13: 4 – reference: 5. Oka T (2005): Tuberous sclerosis related pulmonary lesions other than lymphangioleiomyomatosis. Kokyu 24: 505−514 (in Japanese) – reference: 31. Franz DN, Brody A, Meyer C, Leonard J, Chuck G, Dabora S, Sethuraman G, Colby TV, Kwiatkowski DJ & McCormack FX (2001): Mutational and radiographic analysis of pulmonary disease consistent with lymphangioleiomyomatosis and micronodular pneumocyte hyperplasia in women with tuberous sclerosis. Am J Respir Crit Care Med 164: 661−668 – reference: 15. Fujitaka K, Isobe T, Oguri T, Yamasaki M, Miyazaki M, Kohno N, Takeshima Y & Inai K (2002): A case of micronodular pneumocyte hyperplasia diagnosed through lung biospy using thoracoscopy. Respiration 69: 277−279 – reference: 33. Hayashi T, Kumasaka T, Mitani K, Yao T, Suda K & Seyama K (2010): Loss of heterozygosity on tuberous sclerosis complex genes in multifocal micronodular pneumocyte hyperplasia. Mod Pathol 23: 1251−1260 – reference: 30. Sun Q, Cai HR, Mark EJ, Miao LY, Wu HY, Zhou Q, Chen J, Zhang W & Meng FQ (2015): Multifocal micronodular pneumocyte hyperplasia in a Chinese man masquerading as miliary tuberculosis. Int J Clin Exp Pathol 8: 2165−2170 – reference: 12. Wu K & Tazelaar HD (1999): Pulmonary angiomyolipoma and multifocal micronodular pneumocyte hyperplasia associated with tuberous sclerosis. Hum Pathol 30: 1266−1268 – reference: 28. Suzuki K, Seyama K, Hayashi T, Yamashiro Y, Shiraishi A & Kuwatsuru R (2013): Reversed halo sign in tuberous sclerosis complex. Case Rep Radiol 2013: 428501 (doi: 10.1155/2013/428501. Epub 2013 Sep 15) – reference: 4. Taniguchi N, Konno S, Nasuhara Y, Matsuno Y, Oka T & Nishimura M (2011): A case of multifocal micronodular pneumocyte hyperplasia without clinical findings of tuberous sclerosis. Nihon Kokyuki Gakkai Zasshi 49: 355−359 (in Japanese) – reference: 27. Ogawa R, Miyagawa M, Ide K, Akamune A, Ohtsuki Y & Mochizuki T (2013): Exacerbation and remission of pulmonary micronodules with lymphangioleiomyomatosis around the time of childbirth. Jpn J Radiol 31: 633−636 – reference: 20. Saito M, Yuasa T, Satoh S, Nanjo H, Tsuchiya N & Habuchi T (2008): Developing multiple lung nodules in a renal transplant recipient with tuberous sclerosis who had undergone bilateral nephrectomy due to renal cell carcinomas. Int J Urol 15: 257−258 – reference: 24. Miravet Sorribes L, Mancheño Franch N & Batalla Bautista L (2013): Multifocal micronodular pneumocyte hyperplasia in a patient with tuberous sclerosis. Arch Bronconeumol 49: 36−37 – reference: 29. Lee IS, Shim HS, Lee HJ & Kang YA (2014): Multifocal micronodular pneumocyte hyperplasia mimicking miliary tuberculosis. Am J Respir Crit Care Med 189: 225−226 – reference: 6. Roach ES, Gomez MR & Northrup H (1998): Tuberous sclerosis complex consensus conference: revised clinical diagnostic criteria. J Child Neurol 13:624−628 |
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| Snippet | 多巣性微小結節性肺細胞過形成（multifocal micronodular pneumocyte hyperplasi（aMMPH））は，Ⅱ型肺胞上皮様細胞の増殖からなる多巣性結節性病変で，結節性硬化... |
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| SubjectTerms | リンパ脈管症 多巣性微小結節性肺細胞過形成 結節性硬化症 肺癌 |
| Title | 結節性硬化症に伴う多巣性微小結節性肺細胞過形成を呈したと考えられる2症例 |
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