OJRA  Vol.3 No.2 , May 2013
Larngeal Manifestations of Relapsing Polychondritis

The incidence of Relapsing Polychondritis is estimated to be 3.5 cases per million with only 600 cases being reported in the world. It can affect any age ranging from 5 to 84 years with predominance in the fourth and fifth decade. Male to female ratio varies from 1:1 to 1:3. It is believed to be an immunologic reaction to collagen Type II which is predominantly presented in the cartilaginous structures of the body and in the eye. The diagnosis is based on the presence of three or more clinical signs, one clinical sign in addition to histological confirmation, or involvement of two or more sites with a favorable response to treatment based on Mc Adam diagnostic criteria. The otolaryngologic manifestations of RP span the ears, nose, larynx and tracheobronchial tree. The symptoms include change in voice quality, respiratory discomfort, stridor, dyspnea, cough, chocking, anterior neck tenderness mainly over the thyroid cartilage and cricoid cartilages, aspiration and difficulty in swallowing. The laryngeal manifestations should be evaluated using high resolution computerized tomography, Magnetic resonance imaging, pulmonary function testing, plain radiography and pulmonary function testing.

Cite this paper
A. Hamdan and D. Sarieddine, "Larngeal Manifestations of Relapsing Polychondritis," Open Journal of Rheumatology and Autoimmune Diseases, Vol. 3 No. 2, 2013, pp. 108-112. doi: 10.4236/ojra.2013.32016.

[1]   R. Jaksch-Wartenhorst, “Polychondropathia,” Wien Archives of Internal Medicine, Vol. 6, 1923, pp. 93-100.

[2]   A. F. Uber, “Eiven Fall Von Systematisierter Chondromalacia,” Virchows Archiv für pathologische Anatomie und Physiologie und für klinische Medizin, Vol. 297, No. 3, 1936, pp. 445-479. doi:10.1007/BF01892722

[3]   T. R. Harwood, “Diffuse Perichondritis, Chondritis, and Iritis,” Archives of Pathology, Vol. 65, No. 1, 1958, pp. 81-87.

[4]   M. C. Pearson, M. H. Kline and D. V. Newcomer, “Relapsing Polychondritis, Chondritis, and Iritis,” New England Journal of Medicine, Vol. 263, No. 2, 1960, pp. 51-58. doi:10.1056/NEJM196007142630201

[5]   W. B. Bean, C. C. Drevets and J. S. Chapman, “Chronic Atrophic Polychondtritis,” Medicine, Vol. 37, No. 4, 1958, pp. 353-363. doi:10.1097/00005792-195812000-00004

[6]   W. B. Reed, “Relapsing Polychondritis (Diffuse Chondrolysis),” Archives of Dermatology, Vol. 106, No. 3, 1972, p. 412. doi:10.1001/archderm.106.3.412b

[7]   H. Spencer, “Pathology of the Lung,” 3rd Edition, WB Saunders, Philadelphia, 1977, pp. 759-761.

[8]   H. S. Luthra, “Relapsing Polychondritis,” In: J. H. Klippel and P. A. Dieppe, Eds., Rheumatology, Vol. 27, Mosby, St. Louis, 1998, pp. 1-4.

[9]   D. E. Trentam and C. H. Le, “Relapsing Polychondritis,” Annals of Internal Medicine, Vol. 129, No. 2, 1998, pp. 114-122. doi:10.7326/0003-4819-129-2-199807150-00011

[10]   C. Frances, R. E. Rasi, J. L. Laporte, M. Rybojad, T. Papo and J. C. Piette, “Dermatologic Manifestation of Relapsing Polychondritis: A Study of 200 Cases at a Single Center,” Medicine, Vol. 8, No. 3, 2001, pp. 173-179. doi:10.1097/00005792-200105000-00003

[11]   C. R. Arkin and A. T. Masi, “Relapsing Polychondritis: Review of Current Status and Case Report,” Seminars in Arthritis and Rheumatism, Vol. 5, No. 1, 1975, pp. 41-62. doi:10.1016/0049-0172(75)90022-0

[12]   F. W. Arudell and J. R. Haserick, “Familial Chronic Atrophic Polychondritis,” Archives of Dermatology, Vol. 82, No. 3, 1960, pp. 439-441.

[13]   T. Papo, B. Wechsler, O. Bletry, A. M. Piette, P. Godeau and J. C. Piette, “Pregnancy in Relapsing Polychondritis,” Arithritis and Rheumatism, Vol. 40, No. 7, 1997, pp. 1245-1249. doi:10.1002/art.8

[14]   J. M. Foidart, S. Abe, G. R. Martin, T. M. Zizic, E. V. Barnett, T. J. Lawley, et al., “Antibodies to Type II Collagen in Relapsing Polychondritis,” New England Journal of Medicine, Vol. 299, No. 22, 1978, pp. 1203-1207. doi:10.1056/NEJM197811302992202

[15]   L. Giroux, F. Paquin, M. J. Guerard-Desjardins and A. Lefaivre, “Relapsing Polychondritis: An Autoimmune Disease,” Seminars in Arthritis and Rheumatism, Vol. 13, No. 2, 1983, pp. 182-187. doi:10.1016/0049-0172(83)90005-7

[16]   J. S. Courtenay, M. J. Dallman, A. D. Dayan, A. Martin and B. Mosedale, “Immunization against Heterologous Type II Collagen Induces Arthritis in Mice,” Nature, Vol. 283, No. 5748, 1980, pp. 666-668. doi:10.1038/283666a0

[17]   L. P. McAdam, M. A. O’Hanlan, R. Bluestone and C. M. Pearson, “Relapsing Polychondritis: Prospective Study of 23 Patients and a Review of the Literature,” Medicine, Vol. 55, No. 3, 1976, pp. 193-215. doi:10.1097/00005792-197605000-00001

[18]   J. M. Damiani and H. L. Levine, “Relapsing Polychondritis Report of Ten Cases,” Laryngoscope, Vol. 89, 1979, pp. 929-944.

[19]   C. J. Michet, C. H. McKenna, H. S. Luthra and W. M. O’Fallon, “Relapsing Polychondritis: Survival and Predictive Role of Early Disease Manifestations,” Annals of Internal Medicine, Vol. 104, No. 1, 1986, pp. 74-78. doi:10.7326/0003-4819-104-1-74

[20]   J. M. Damiani and H. L. Levine, “Relapsing Polychondritis Report of Ten Cases,” Laryngoscope, Vol. 89, No. 6, 1979, pp. 929-943.

[21]   R. O. Williams, D. G. Williams and R. N. Maini, “Anti-Type II Collagen ELISA Increased Disease Specificity Following Removal of Anionic Contaminants from Salt-Fractionated Type II Collagen,” Journal of Immunological Methods, Vol. 147, No. 1, 1992, pp. 93-100. doi:10.1016/S0022-1759(12)80033-0

[22]   L. P. McAdam, M. A. O’Hanlan, R. Bluestone and C. M. Pearson, “Relapsing Polychondritis: Prospective Study of 23 Patients and a Review of the Literature,” Medicine, Vol. 55, No. 3, 1976, pp. 193-215. doi:10.1097/00005792-197605000-00001

[23]   J. L. Port, A. Khan and R. R. Barbu, “Computed Tomography of Relapsing Polychondritis,” Computerized Medical Imaging and Graphics, Vol. 17, No. 2, 1993, pp. 119-123. doi:10.1016/0895-6111(93)90053-P

[24]   M. Oddone, P. Toma, A. Taccone, G. Hanau, A. Delogu and G. Gemme, “Relapsing Polychondritis in Childhood: a Rare Observation Studied by CT and MRI,” Pediatric Radiology, Vol. 22, No. 7, 1992, pp. 537-538. doi:10.1007/BF02013006

[25]   H. R. Neeson, K. B. Remley, A. E. Stillman and F. L. Rimell, “Dynamic MR Imaging of the Larynx and Trachea,” The American Society of Neuroradiology 34th Annual Meeting, Seattle, 23-27 June 1996.

[26]   J. A. Fornadley, D. J. Seibert, B. E. Ostrov and W. S. Warren, “The Role of MRI when Relapsing Polychondritis is Suspected but not Proven,” International Journal of Pediatric Otorhinolaryngology, Vol. 31, No. 1, 1995, pp. 101-107. doi:10.1016/0165-5876(94)01068-9

[27]   J. Eng and S. Sabanathan, “Airway Complications in Relapsing Polychondritis,” The Annals of Thoracic Surgery, Vol. 51, No. 4, 1991, pp. 686-692. doi:10.1016/0003-4975(91)90348-T

[28]   K. S. Lee, A. Ernst, D. E. Trentham, et al., “Relapsing Polychondritis: Prevalence of Expiratory CT Airway Abnormalities,” Radiology, Vol. 240, No. 2, 2006, pp. 565-573. doi:10.1148/radiol.2401050562

[29]   H. E. Eckel, C. Wittekindt, U. Schroeder, J. P. Klussmann and C. Sittel, “Management of Bilateral Arytenoids Cartilage Fixation versus Recurrent Laryngeal Nerve Paralysis,” The Annals of Otology, Rhinology, and Laryngology, Vol. 112, No. 2, 2003, pp. 103-108.

[30]   W. S. Copeman, “Rheumatoid Arthritis of the Cricoarytenoid Joints,” British Medical Journal, Vol. 1, No. 5032, 1957, pp. 1398-1399. doi:10.1136/bmj.1.5032.1398

[31]   H. Bienenstock, G. E. Ehrlich and R. H. Freyberg, “Rheumatoid Arthritis of the Cricoarytenoid Joints: A Clinicopathologic Study,” Arthritis & Rheumatism, Vol. 6, No. 1, 1963, pp. 48-63. doi:10.1002/art.1780060106

[32]   D. L. Dolan, G. B. Lemmon and S. L. Teitelbaum, “Relapsing Polychondritis Analytical Review and Studies on Pathogenesis,” The American Journal of Medicine, Vol. 41, No. 2, 1966, pp. 285-299. doi:10.1016/0002-9343(66)90023-4

[33]   G. J. Gibson and P. Davis, “Respiratory Complications of Relapsing Polychondritis,” Thorax, Vol. 29, No. 6, 1974, pp. 726-731. doi:10.1136/thx.29.6.726

[34]   B. D. Sarodia, A. Dasgupta and A. C. Mehta, “Management of Airway Manifestations of Relapsing Polychondritis: Case Reports and Review of Literature,” Chest Journal, Vol. 116, No. 6, 1999, pp. 1669-1675. doi:10.1378/chest.116.6.1669

[35]   P. D. Kent, C. J. Michet and H. S. Luthra, “Relapsing Polychondritis,” Current Opinion in Rheumatology, Vol. 16, No. 1, 2003, pp. 56-61. doi:10.1097/00002281-200401000-00011

[36]   E. Hilgert, B. Toleti, K. Kruger and I. Nejedlo, “Hoarseness Due to Bamboo Nodes in Patients with Autoimmune Diseases: A Review of Literature,” Journal of Voice, Vol. 22, No. 3, 2008, pp. 343-350. doi:10.1016/j.jvoice.2006.10.009

[37]   R. Minami, T. Miyamura, M. Nakamura, et al., “A Clinical Study of Five Cases Demonstrating Relapsing Polychondritis,” Nihon Rinsho Meneki Gakkai Kaishi, Vol. 32, No. 4, 2009, pp. 269-273. doi:10.2177/jsci.32.269

[38]   T. Wang, Q. Lu, Y. Qin and N. He, “The Diagnosis and Treatment of Relapsing Polychondritis (A Case Report),” Lin Chung Er Bi Yan Hou Tou Jing Wai Ke Za Zhi, Vol. 22, No. 13, 2008, pp. 590-592.

[39]   J. Grosflam and M. E. Weinblatt, “Methotrexate: Mechanism of Action, Pharmacokinetics, Clinical Indications, and Toxicity,” Current Opinion in Rheumatology, Vol. 3, No. 3, 1991, pp. 363-368. doi:10.1097/00002281-199106000-00006

[40]   J. Park, K. M. Gowin, H. R. Schumacher Jr., “Steroid Sparing Effect of Methotrexate in Relapsing Polychondritis,” The Journal of Rheumatology, Vol. 23, No. 5, 1996, pp. 937-938.

[41]   A. Chang-Miller, M. Okamura, V. E. Torres, et al., “Renal Involvement in Relapsing Polychondritis,” Medicine, Vol. 66, No. 3, 1987, pp. 202-217. doi:10.1097/00005792-198705000-00004

[42]   K. L. G. Svenson, R. Holmdahl, L. Klareskog, et al., “Cyclosporin a Treatment in a Case of Relapsing Polychondritis,” Scandinavian Journal of Rheumatology, Vol. 13, No. 4, 1984, pp. 329-333. doi:10.3109/03009748409111304

[43]   K. Svenson, S. O. Bohman and R. Hallgren, “Renal Interstitial Fibrosis and Vascular Changes. Occurrence in Patients with Auroimmune Diseases Treated with Cyclosporine,” Formerly Archives of Internal Medicine, Vol. 146, No. 10, 1986, pp. 2007-2010. doi:10.1001/archinte.1986.00360220169028

[44]   A. Anstey, S. Mayou, K. Morgan, R. B. Clague and D. D. Munro, “Relapsing Polychondritis: Autoimmunity to Type II Collagen and Treatment with Cyclosporin A,” British Journal of Dematology, Vol. 125, No. 6, 1993, pp. 588-591. doi:10.1111/j.1365-2133.1991.tb14801.x

[45]   E. H. S. Choy, I. C. Chikanza, G. H. Kingsley and G. S. Panayi, “Chimaeric Anti-CD4 Monoclonal Antibody for Relapsing Polychondritis,” The Lancet, Vol. 338, No. 8764, 1991, pp. 338-450.

[46]   A. D. Ormerod and L. J. Clark, “Relapsing Polychondritis—Treatment with Cyclosporin A,” British Journal of Dematology, Vol. 127, No. 3, 1992, p. 300. doi:10.1111/j.1365-2133.1992.tb00135.x

[47]   P. A. Van der Lubbe, A. M. Miltenburg and F. C. Breedveld, “Anti-CD4 Monoclonal Antibody for Relapsing Polychondritis,” The Lancet, Vol. 337, No. 8753, 1991, p. 1349. doi:10.1016/0140-6736(91)93023-3

[48]   T. T. Tanaka, H. F. Furutani and T. H. Harioka, “Anaesthetic Management of a Patient with Relapsing Polychondritis Undergoing Laparoscopic Surgery,” Anaesth Intensive Care Journal, Vol. 34, No. 3, 2006, pp. 372- 374.

[49]   E. Karaman, C. Duman, H. Cansz, M. Ylmaz and M. Ibrahimov, “Laryngotracheal Reconstruction at Relapsing Polychondritis,” Journal of Craniofacial Surgery, Vol. 21, No. 1, 2010, pp. 211-212. doi:10.1097/SCS.0b013e3181c50f7d

[50]   J. Q. Zhang, Q. Li, C. Bai, Y. Huang, Y. C. Dong, X. P. Yao and Q. Wang, “Clinical Features and Treatment of Relapsing Polychondritis with Involvement of the Respiratory Tract-Report of Thirteen Cases,” Zhonghua Jie He He Hu Xi Za Zhi, Vol. 30, No. 3, 2007, pp. 173-177.

[51]   Y. D. Heman-Ackah and R. T. Sataloff, “Use of Silicone Hearing Aid Mold Material in Laryngotracheal Reconstruction,” The Annals of Otology, Rhinology and Laryngology, Vol. 114, No. 10, 2005, pp. 739-742.

[52]   L. F. Childs, S. Rickert, O. C. Wengerman, R. Lebovics and A. Blitzer, “Laryngeal Manifestations of Relapsing Polychondritis and a Novel Treatment Option,” Journal of Voice, Vol. 26, No. 5, 2012, pp. 587-589. doi:10.1016/j.jvoice.2011.07.012