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Dermatomyositis, complicated with pneumomediastinum, successfully treated with cyclosporine A: a case report and review of literature

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Abstract

We report a rare case of patient with dermatomyositis (DM) who developed spontaneous pneumomediastinum (PnM) and subcutaneous emphysema. She was successfully treated with oral prednisolone and cyclosporine A (CsA). We reviewed the cases of PnM in patients with DM treated with CsA. A review of four previously reported cases revealed that treatment with systemic glucocorticoid and CsA was effective for the DM and PnM. We indicate that initial and early treatment of the patients with DM and PnM with CsA enabled rapid tapering of the dose of glucocorticoid and improved the disease.

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References

  1. Dickey BF, Myers AR (1984) Pulmonary disease in polymyositis and dermatomyositis. Semin Arthritis Rheum 41:137–140

    Google Scholar 

  2. King TE, Cherniack RM, Schwarz MI (1994) The lungs and connective tissue disease. In: Murray JF, Nadel JA (eds) Textbook of respiratory medicine. Saunders, Philadelphia, pp 1850–1872

    Google Scholar 

  3. Yarborough JW, Sealy WC, Millar JA (1975) Thoracic surgical problems associated with rheumatoid arthritis. J Thorac Cardiovasc Surg 69:347–354

    Google Scholar 

  4. Sawker LA, Easom HF (1971) Recurrent spontaneous pneumothoraces in systemic lupus erythematosus. Chest 60:604–605. doi:10.1378/chest.60.6.604

    Article  Google Scholar 

  5. Edwards WG, Dines DE (1966) Recurrent spontaneous pneumothorax in diffuse scleroderma: report of a case. Dis Chest 49:96–98. doi:10.1378/chest.49.1.96

    Article  PubMed  Google Scholar 

  6. Korkmaz C, Ozkan R, Akay M, Hakan T (2001) Pneumomediastinum and subcutaneous emphysema associated with dermatomyositis. Rheumatology 40:476–478. doi:10.1093/rheumatology/40.4.476

    Article  PubMed  CAS  Google Scholar 

  7. Neves FS, Shinjo SK, Carvalho JF, Levy-Neto M, Borges CT (2007) Spontaneous pneumomediastinum and dermatomyositis may be a not so rare association: report of a case and review of the literature. Clin Rheumatol 26:105–107. doi:10.1007/s10067-005-0109-x

    Article  Google Scholar 

  8. Kono H, Inokuma S, Nakayama H, Suzuki M (2000) Pneumomediastinum in dermatomyositis: association with cutaneous vasculopathy. Ann Rheum Dis 59:361–372. doi:10.1136/ard.59.5.372

    Article  Google Scholar 

  9. Yamanashi Y, Maeda H, Konishi F, Hiyama K, Yamana S, Ishioka S et al (1999) Dermatomyositis associated with rapidly progressive fatal interstitial pneumonitis and pneumomediastinum. Scand J Rheumatol 28:58–61. doi:10.1080/03009749950155805

    Article  Google Scholar 

  10. Jansen TL, Barrera P, van Engelen BG, Cox N, Laan RF, van de Putte LB (1998) Dermatonyositis with subclinical myositis and spontaneous pneumomediastinum with pneumothorax: case report and review of the literature. Clin Exp Rheumatol 16:733–735

    PubMed  CAS  Google Scholar 

  11. Englund P, Lindroos E, Nennesmo I, Klareskog L, Lundberg IE (2001) Skeletal muscle fibers express major histocompatibility complex class II antigens independently of inflammatory infiltrates in inflammatory myopathies. Am J Pathol 159:1263–1273

    PubMed  CAS  Google Scholar 

  12. Inukai A, Kuru S, Liang Y, Takano A, Kobayashi Y, Sakai M et al (2000) Expression of HLA-DR and its enhancing molecules in muscle fibers in polymyositis. Muscle Nerve 23:385–392. doi:10.1002/(SICI)1097-4598(200003)23:3<385::AID-MUS10>3.0.CO;2-T

    Article  PubMed  CAS  Google Scholar 

  13. Nagaraju K, Raben N, Merritt G, Loeffler L, Kirk K, Plotz P (1998) A variety of cytokines and immunologically relevant surface molecules are expressed by normal human skeletal muscle cells under proinflammatory stimuli. Clin Exp Immunol 113:407–414. doi:10.1046/j.1365-2249.1998.00664.x

    Article  PubMed  CAS  Google Scholar 

  14. Engel AG, Arahata K, Emslie-Smith A (1990) Immune effector mechanisms in inflammatory myopathies. Res Publ Assoc Res Nerv Ment Dis 68:141–157

    PubMed  CAS  Google Scholar 

  15. Arahata K, Engel AG (1984) Monoclonal antibody analysis of mononuclear cells in myopathies I: quantitation of subsets according to diagnosis and sites of accumulation and demonstration and counts of muscle fibers invaded by T cells. Ann Neurol 16:193–208. doi:10.1002/ana.410160206

    Article  PubMed  CAS  Google Scholar 

  16. Yamadori I, Fujita J, Kajitani H, Bandoh S, Tokuda M, Ohtsuki Y et al (2001) Lymphocyte subsets in lung tissues of interstitial pneumonia associated with untreated polymyositis/dermatomyositis. Rheumatol Int 21:89–93. doi:10.1007/s00296-001-0146-y

    Article  PubMed  CAS  Google Scholar 

  17. Chino Y, Murata H, Goto D, Matsumoto I, Tsutsumi A, Sakamoto T et al (2006) T cell receptor BV gene repertoire of lymphocytes in bronchoalveolar lavage fluid of polymyositis/dermatomyositis patients with interstitial pneumonitis. Int J Mol Med 17:101–109

    PubMed  CAS  Google Scholar 

  18. Giese T, Zeier M, Schemmer P, Uhl W, Schoels M, Dengler T et al (2004) Monitoring of NFAT-regulated gene expression in the peripheral blood of allograft recipients: a novel perspective toward individually optimized drug doses of cyclosporine A. Transplantation 77:339–344. doi:10.1097/01.TP.0000109260.00094.01

    Article  PubMed  CAS  Google Scholar 

  19. Saggi SJ, Andoh TF, Safirstein R, Bennett WM (2004) Cyclosporin induces renal proto-oncogene RNA message and increased transforming growth factor beta prior to renal fibrosis: modification by calcium channel blockade in the salt-repleted rat. Nephrology 9:58–64. doi:10.1111/j.1440-1797.2003.00230.x

    Article  PubMed  CAS  Google Scholar 

  20. Fuleihan R, Ramesh N, Horner A, Ahern D, Belshaw PJ, Alberg DG et al (1994) Cyclosporin A inhibits CD40L expression in T lymphocytes. J Clin Invest 93:1315–1320. doi:10.1172/JCI117089

    Article  PubMed  CAS  Google Scholar 

  21. Qushmaq KA, Chalmers A, Esdaile JM (2000) Cyclosporine A in the treatment of refractory adult polymyositis/dermatomyositis population-based experience in 6 patients and literature review. J Rheumatol 27:2855–2859

    PubMed  CAS  Google Scholar 

  22. Terao M, Ozawa K, Inui S, Murota H, Yokomi A, Itami S (2007) A case of dermatomyuositis complicated with pneumomediastinum. Mod Rheumatol 17:156–159. doi:10.1007/s10165-006-0556-5

    Article  PubMed  Google Scholar 

  23. Kuroda T, Morikawa H, Satou T, Tanabe Y, Murakami S, Ito S et al (2003) A case of dermatomyositis complicated with pneumomediastinum successfully treated with cyclosporine A. Clin Rheumatol 22:45–48. doi:10.1007/s10067-002-0675-0

    Article  PubMed  CAS  Google Scholar 

  24. Nonomura Y, Koike R, Nishio J, Tsubata R, Kohsaka H, Kubota T, Miyasaka N (2001) A case of dermatomyositis complicated with pneumomediastinum that was successfully treated with cyclosporin A. Ryumachi 41:653–658

    PubMed  CAS  Google Scholar 

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Correspondence to Wan-Hee Yoo.

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Kim, HJ., Hong, YK. & Yoo, WH. Dermatomyositis, complicated with pneumomediastinum, successfully treated with cyclosporine A: a case report and review of literature. Rheumatol Int 29, 1101–1104 (2009). https://doi.org/10.1007/s00296-008-0822-2

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  • DOI: https://doi.org/10.1007/s00296-008-0822-2

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