A Case of Relapsing Polychondritis Successfully Treated with Combination of a Glucocorticoid and Cyclosporine

Koichiro  Takahashi; Hiroshi  Inoue; Hironori  Sadamatsu; Hitomi  Umeguchi; Naoko  Sueoka-Aragane; Shinya  Kimura

doi:10.4236/ijcm.2015.67057

Koichiro Takahashi, Hiroshi Inoue, Hironori Sadamatsu, Hitomi Umeguchi, Naoko Sueoka-Aragane, Shinya Kimura^*

Abstract:

Relapsing polychondritis is a rare cartilaginous inflammatory disease affecting the external ear, nose, peripheral joints and tracheobronchial tree. It is characterized by recurrent inflammation and degeneration of cartilage and connective tissue. A 72-year-old man complained of dyspnea, cough and wheezing for 2 months. Diffuse wall thickening and narrowing from the trachea to segmental bronchus were seen on chest CT. Tracheostomy was performed in order to avoid as-phyxia, and he was diagnosed as relapsing polychondritis on the basis of pathology evaluation of a tracheal biopsy specimen. He was treated with high doses of a glucocorticoid, with which his symptoms improved. However, the cough and wheezing recurred after tapering of the glucocorticoid. His symptoms thereafter were improved by combination of the glucocorticoid with cyclosporine. The immunosuppressive agent provided effective treatment for glucocorticoid-resistant relapsing polychondritis.

Keywords: Relapsing Polychondritis, Trachea, Cyclosporine

Cite this paper: Takahashi, K. , Inoue, H. , Sadamatsu, H. , Umeguchi, H. , Sueoka-Aragane, N. and Kimura, S. (2015) A Case of Relapsing Polychondritis Successfully Treated with Combination of a Glucocorticoid and Cyclosporine. International Journal of Clinical Medicine, 6, 439-443. doi: 10.4236/ijcm.2015.67057.

References

[1] McAdam, L.P., O’Hanlan, M.A., Bluestone, R. and Pearson, C.M. (1976) Relapsing Polychondritis: Prospective Study of 23 Patients and a Review of the Literature. Medicine (Baltimore), 55, 193-215.
http://dx.doi.org/10.1097/00005792-197605000-00001

[2] Kent, P.D., Michet Jr, C.J. and Lutha, H.S. (2004) Relapsing Polychondritis. Current Opinion in Rheumatology, 16, 56-61.
http://dx.doi.org/10.1097/00002281-200401000-00011

[3] Letko, E., Zafirakis, P., Baltatzis, S., Voudouri, A., Livir-Rallatos, C., et al. (2002) Relapsing Polychondritis: A Clinical Review. Seminars in Arthritis and Rheumatism, 31, 384-395.
http://dx.doi.org/10.1053/sarh.2002.32586

[4] Ernst, A., Rafeq, S., Boiselle, P., Sung, A., Reddy, C., et al. (2009) Relapsing Polychondritis and Airway Involvement. Chest, 135, 1024-1030. http://dx.doi.org/10.1378/chest.08-1180

[5] Sarodia, B.D., Dasgupta, A. and Mehta, A.C. (1999) Management of Airway Manifestations of Relapsing Polychondritis: Case Reports and Review of Literature. Chest, 116, 1669-1675.
http://dx.doi.org/10.1378/chest.116.6.1669

[6] Lahmer, T., Treiber, M., von Werder, A., Foerger, F., Knopf, A., et al. (2010) Relapsing Polychondritis: An Autoimmune Disease with Many Faces. Autoimmunity Reviews, 9, 540-546.
http://dx.doi.org/10.1016/j.autrev.2010.02.016

[7] Frances, C., El Rassi, R., Laporte, J.L., Rybojad, M., Papo, T., et al. (2001) Dermatologic Manifestations of Relapsing Polychondritis. A Study of 200 Cases at a Single Center. Medicine (i), 80, 173-179.
http://dx.doi.org/10.1097/00005792-200105000-00003

[8] Terato, K., Shimomizu, Y., Katayama, K., Takemitsu, Y., Yamashita, I., et al. (1990) Specificity of Antibodies to Type II Collagen in Rheumatoid Arthritis. Arthritis Rheumatology, 33, 1493-1500.
http://dx.doi.org/10.1002/art.1780331006

[9] Eng, J. and Sabanathan, S. (1991) Airway Complications in Relapsing Polychondritis. Annals of Thoracic Surgery, 51, 686-692.
http://dx.doi.org/10.1016/0003-4975(91)90348-T

[10] Yamashita, H., Takahashi, H., Kubota, K., Ueda, Y., Ozaki, T., et al. (2014) Utility of Fluorodeoxyglucose Positron Emission Tomography/Computed Tomography for Early Diagnosis and Evaluation of Disease Activity of Relapsing Polychondritis: A Case Series and Literature Review. Rheumatology (Oxford), 53, 1482-1490.
http://dx.doi.org/10.1093/rheumatology/keu147

[11] . Trentham, D.E. and Le, C.H. (1998) Relapsing Polychondritis. Annals of Internal Medicine, 129, 114-122.
http://dx.doi.org/10.7326/0003-4819-129-2-199807150-00011

[12] Park, J., Gowin, K.M. and Schumacher Jr., H.R. (1996) Steroid Sparing Effect of Methotrexate in Relapsing Polychondritis. The Journal of Rheumatology, 23, 937-938.

[13] Priori, R., Paroli, M.P., Luan, F.L., Abdulaziz, M., Pivetti Pezzi, P. and Valesini, G. (1993) Cyclosporin A in the Treatment of Relapsing Polychondritis with Severe Recurrent Eye Involvement. British Journal of Rheumatology, 32, 352.
http://dx.doi.org/10.1093/rheumatology/32.4.352

[14] Stewart, K.A. and Mazanec, D.J. (1992) Pulse Intravenous Cyclophosphamide for Kidney Disease in Relapsing Polychondritis. The Journal of Rheumatology, 19, 498-500.

[15] Marie, I., Lahaxe, L., Josse, S. and Levesque, H. (2009) Sustained Response to Infliximab in a Patient with Relapsing Polychondritis with Aortic Involvement. Rheumatology, 48, 1328-1329.
http://dx.doi.org/10.1093/rheumatology/kep224

[16] Wallace, Z.S. and Stone, J.H. (2013) Refractory Relapsing Polychondritis Treated with Serial Success with Interleukin 6 Receptor Blockade. The Journal of Rheumatology, 40, 100-101.
http://dx.doi.org/10.3899/jrheum.120381

[17] Takeda, K., Nagasaka, K. and Miyasaka, N. (2005) Polymyositis/Dermatomyositis and Interstitial Lung Disease: A New Therapeutic Approach with T-Cell-Specific Immunosuppressants. Autoimmunity, 38, 383-392.
http://dx.doi.org/10.1080/08916930500124023