Castleman’s Disease of the Pleura: Case Presentation and Review of Literature


Purpose. Castleman’s Disease is a very rare medical disease and the presence of this condition in the pleura is even rarer and there are only few reported cases. The presence of symptoms is also unusual and has been mostly described in multicentric cases of castleman’s disease instead of solitary lesions (unicentric castleman). We report a case of unicentric pleural castlema’s disease with presence of constitutional symptoms. Methods. A 36-year-old male without any significant past medical history who presented to the emergency due to an incidental large pleural effusion seen in chest radiograph, but referring chronic constitutional symptoms consistent of significant weight loss and chronic cough. Results. Chest computed tomography revealed a mass and several attemps with minimally invase approach failed to identify the lesion. The mass was excised through VATS but due to its significant vascularity, it had to be converted to open thoracotomy. Conclusion. The treatment of unicentric lesions is mainly complete surgical excision and the prognosis is excellent with some chances of recurrence. There are less than one dozen of reported pleural castleman reported in the literature. This case evidence than unicentric lesion may exhibit constitutional symptoms and confirmed than lesions above 5 cm can be removed by VATS but the chances for conversion to an open thoracotomy are high.

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P. Loarte, N. Araguez, R. Flores, L. Wu and R. Cohen, "Castleman’s Disease of the Pleura: Case Presentation and Review of Literature," International Journal of Clinical Medicine, Vol. 3 No. 4, 2012, pp. 311-317. doi: 10.4236/ijcm.2012.34062.

Conflicts of Interest

The authors declare no conflicts of interest.


[1] D. Bornekamp, K. M. Horton, R. H. Hruban and E. K. Fishman, “Castleman Disease: The Great Mimic,” RadioGraphics, Vol. 31, No. 6, 2011, pp. 1793-1807. doi:10.1148/rg.316115502
[2] J. Herrada, F. Cabanillas, L. Rice, et al., “The Clinical Behavior of Localized and Multicentric Castleman Disease,” Annals of Internal Medicine, Vol. 128, No. 8, 1998, pp. 657-662.
[3] T. W. Muskardin, B. A. Peterson and J. A. Molitor, “Castleman Disease and Associated Autoimmune Disease,” Current Opinion in Rheumatology, Vol. 24, No. 1, 2012, pp. 76-83. doi:10.1097/BOR.0b013e32834db525
[4] S. P. Reynold, A. R. Gibbs, R. Weeks, et al., “Massive Pleural Effusion: An Unusual Presentation of Castleman’s Disease,” European Respiratory Journal, Vol. 5, No. 9, 1992, pp. 1150-1153.
[5] H. E. El-Osta and R. Kurzrock, “Castleman’s Disease: From Basic Mechanism to Molecular Therapeutics,” The Oncologist, Vol. 16, 2011, pp. 497-511. doi:10.1634/theoncologist.2010-0212
[6] B. Roca, “Castleman’s Disease. A Review,” AIDS Reviews, Vol. 11, 2009, pp. 3-7.
[7] K. Yoshizaki, T. Matsuda, N. Nishimoto, et al., “Pathogenic Significance of Interleukin-6 (IL-6/BSF-2) in Castleman’s Disease,” Blood, Vol. 74, No. 4, 1989, pp. 1360-1367.
[8] I. S. Abdul-Rahman, A. M. Al-Amri and K. Q. Ghallab, “Castleman’s Disease: A Study of a Rare Lymphoproliferative Disorder in a University Hospital,” Clinical Medicine: Blood Disorders, Vol. 2, 2009, pp. 5-19.
[9] B. Bandera, C. Ainsworth, J. Shikle, E. Rupard and M. Roach, “Treatment of Unicentric Castleman Disease with Neoadjuvant Rituximab,” Chest, Vol. 138, No. 5, 2009, pp. 1239-1241. doi:10.1378/chest.09-2084
[10] A. Dham and B. A. Peterson, “Castleman Disease,” Current Opinion in Hematology, Vol. 14, No. 4, 2007, pp. 354-359. doi:10.1097/MOH.0b013e328186ffab
[11] C. S. Restre-po, M. M. Chen, S. Martinez-Jimenez, J. Carrillo and C. Restrepo, “Chest Neoplasm with Infectious Etiologies,” World Journal of Radiology, Vol. 3, No. 12, 2011, pp. 279-288. doi:10.4329/wjr.v3.i12.279
[12] C. H. Chen, H. C. Liu, T. T. Hung and T. P. Liu, “Possible Roles of Epstein-Barr Virus in Castleman Disease,” Journal of Cardiothoracic Surgery, Vol. 4, No. 31, 2009, pp. 1-5.
[13] D. M. Cronin and R. A. Warnke, “Castleman Disease. An Update on Classification and the Spectrum of Associated Lesions,” Advances in Anatomic Pathology, Vol. 16, No. 4, 2009, pp. 236-246. doi:10.1097/PAP.0b013e3181a9d4d3
[14] H. J. Lach-mann, J. A. Gilbertson, J. D. Gillmore, P. N. Hawkins and M. B. Pepys, “Unicentric Castleman’s Disease Complicates by Systemic AA Amyloidosis: A Curable Disease,” International Journal of Medicine, Vol. 95, No. 4, 2002, pp. 211-218.
[15] J. B. Alpert, D. Nonaka, A. Chachoua, et al., “Increasing Dyspnea Due to an Anterior Mediastinal Mass,” Chest, Vol. 139, No. 1, 2011, pp. 217-223. doi:10.1378/chest.10-0383
[16] B. F. Menezes, R. Mor-gan, M. Azad, “Multicentric Catleman’s Disease: A Case Report,” Journal of Medical Case Reports, Vol. 1, No. 78, pp. 2007.
[17] S. F. Ko, M. J. Hseih, S. H. Ng, et al., “Imaging Spectrum of Castleman’s Disease,” American Journal of Roentgenology, Vol. 182, No. 3, 2004, pp. 769-775.
[18] K. Enomoto, I. Nakamichi, K. Hamada, et al., ”Unicentric and Multicentric Castleman’s Disease,” British Journal of Radiology, Vol. 80, No. 949, 2007, pp. e24-e26. doi:10.1259/bjr/93847196
[19] S. D. Chang and R. F. Thoeni, “Castleman’s Disease Presenting as an Adnexal Mass: Ultrasound, CT and MRI Features,” British Journal of Radiology, Vol. 77, No. 914, 2004, pp. 161-163. doi:10.1259/bjr/30098414
[20] K. Ohara, K. Narita, H. Ishikawa, et al., “Local Recurrence of Castleman Disease,” Kyobu Geka, Vol. 62, No. 2, 2009, pp. 136-139.
[21] T. Ushio, K. Yoshimura and A. Kojima, “A Case of Castleman’s Disease That Recurred Nine Years after Initial Surgical Removal,” Nihon Kyobu Shikkan Gakkai Zasshi, Vol. 32, No. 12, 1994, pp. 1175-1180.
[22] N. Yaris, M. Cakir, M. Kalyoncu, et al., “Castleman Disease: A Case with Atypical Presentation,” Pediatric Hematology Oncology, Vol. 21, 2004, pp. 203-208. doi:10.1080/08880010490276908
[23] H. Kurotaki, M. Kaimori and K. Nagai, “Recurred Castleman’s Disease Containing a Fibrohistiocytic Nodular Lesion with Vascular Occlusion,” Pathology International, Vol. 43, No. 10, 1993, pp. 603-607. doi:10.1111/j.1440-1827.1993.tb03237.x
[24] S. L. Han, X. X. Chen, X. F. Zheng, et al., “The Clinicopathological Behavior and Surgical Treatment of Abdominal Castleman’s Disease,” Singapore Medical Journal, Vol. 51, No. 10, 2010, pp. 813-816.
[25] S. Shetty, R. A. Brenes, L. Panait and J. Sanchez, “Video Assisted Thoracoscopic Resection of a Posterior Mediastinal Castleman’s Tumor,” Journal of Cardiothoracic Surgery, Vol. 6, No. 113, 2011.
[26] P. A. Seirafi, E. Ferguson and F. H. Edwards, “Thoracoscopic Resection of Castleman Disease, Case Report and Review,” Chest, Vol. 123, 2003, pp. 280-282. doi:10.1378/chest.123.1.280
[27] I. A. C. De Vries, M. M. S. van Acht, et al., “Neoadjuvant Radiotherapy of Primary Irresectable Unicentric Castleman’s Disease: A Case Report and Review of the Literature,” Radiation Oncology, Vol. 5, No. 7, 2010.
[28] N. Talat and K. M. Schulte, “Castleman’s Disease: Systematic Analysis of 416 Patients from the Literature,” The Oncologist, Vol. 16, 2011, pp. 1316-1324. doi:10.1634/theoncologist.2011-0075
[29] P. Farruggia, A. Trizzino, N. Scibetta, et al., “Castleman’s Disease in Childhood: Report of Three Cases and Review of the Literature,” Italian Journal of Pediatrics, Vol. 35, No. 50, 2011.
[30] N. S. Paramothayan and J. Barron, “New Criteria for the Differentiation between Transudates and Exudates,” Journal of Clinical Pathology, Vol. 55, No. 1, 2002, pp. 69-71. doi:10.1136/jcp.55.1.69
[31] A. S. Leong, K. Cooper and F. J. Leong, “Manual of Diagnostic Cytology,” 2nd Edition, Greenwich Medical Media, Ltd., Cambridge, 2003, p. 29.
[32] S. F. Ko, S. H. Ng, M. J. Hsieh, et al., “Castleman Disease of the Pleura: Expe-rience with Eight Surgically Proven Cases,” Annals of Thoracic Surgery, Vol. 76, No. 1, 2003, pp. 219-224. doi:10.1016/S0003-4975(03)00133-4
[33] J. O’Driscoll, “Giant Lymph Node Hyperplasia Presenting as a Pleural Mass,” Irish Medical Journal, Vol. 78, No. 5, 1985, pp. 132-133.
[34] M. G. Alexandrakis, F. H. Passam, D. Ky-riakou and D. Bouros, “Pleural Effusions in Hematologic Malignancies,” Chest, Vol. 25, No. 4, 2004, pp. 1546-1555. doi:10.1378/chest.125.4.1546
[35] R. Burrah, V. Deshmane, S. Althaf, et al., “Castleman’s Disease of the Pleura,” General Thoracic and Cardiovascular Surgery, Vol. 59, No. 8, 2011, pp. 572-574. doi:10.1007/s11748-010-0676-x
[36] O. Ichiguchi and M. Yoshioka, “Castleman’s Disease in the Cardiophrenic Angle Resected Thoracoscopically; Report of a Case,” Kyobu Geka, Vol. 62, No. 2, 2009, pp. 161-163.

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