Multicentric Castleman disease of hyaline‑vascular variant with paraneoplastic pemphigus results in abnormal lung function: Report of 3 cases

Cen,Jiemei; Zeng,Wen; Pan,Mianluan; Qiu,Ye; Zhang,Jianquan

doi:10.3892/etm.2023.11968

Multicentric Castleman disease of hyaline‑vascular variant with paraneoplastic pemphigus results in abnormal lung function: Report of 3 cases

Authors:
- Jiemei Cen
- Wen Zeng
- Mianluan Pan
- Ye Qiu
- Jianquan Zhang
View Affiliations

Affiliations: Department of Respiratory Medicine, The Eighth Affiliated Hospital, Sun Yat‑Sen University, Shenzhen, Guangdong 518033, P.R. China, Department of Respiratory Medicine, The First Affiliated Hospital of Guangxi Medical University, Nanning, Guangxi 530000, P.R. China
Published online on: April 20, 2023 https://doi.org/10.3892/etm.2023.11968
Article Number: 269
Copyright: © Cen et al. This is an open access article distributed under the terms of Creative Commons Attribution License.

Metrics: Total Views: 0 (Spandidos Publications: | PMC Statistics: )

Metrics: Total PDF Downloads: 0 (Spandidos Publications: | PMC Statistics: )

Cited By (CrossRef): 0 citations Loading Articles...

Abstract

Castleman disease (CD) is clinically divided into unicentric CD (UCD) and multicentric CD (MCD). Hyaline‑vascular variant (HV) is the most common pathological type of UCD, while the plasma cell type (PC) is the most common type of MCD and thus, hyaline‑vascular variant multicentric CD (HV‑MCD) is a rare type of CD. In addition, its etiology has remained elusive. The present study retrospectively analyzed the medical records of 3 patients diagnosed as HV‑MCD admitted to The First Affiliated Hospital of Guangxi Medical University (Guangxi, China) between January 2007 and September 2020. A total of 2 males and 1 female were admitted. The areas involved varied considerably. Respiratory symptoms were seen in 3 cases, along with fever, weight loss and splenomegaly. Damage to the skin and mucous membranes resulted in oral ulcers when accompanied by paraneoplastic pemphigus (PNP). Dry and wet rales were found in all patients. All 3 cases were complicated with PNP and had hypoxemia and obstructive ventilation dysfunction. In accordance with PC‑MCD, it manifested as lymph node enlargement and may involve several lymph nodes. Computed tomography mainly indicated bronchiectasis and mediastinal lymph node enlargement. In 1 case, chemotherapy failed after local mass excision, 1 case remitted after chemotherapy but the lung lesion was irreversible and 1 case was untreated and soon died of respiratory failure. The cases of HV‑MCD with pulmonary involvement were induced by small airway lesions and associated with poor prognosis. Respiratory symptoms along with systemic symptoms were common.

View Figures

View References

1	Oksenhendler E, Boutboul D, Fajgenbaum D, Mirouse A, Fieschi C, Malphettes M, Vercellino L, Meignin V, Gérard L and Galicier L: The full spectrum of Castleman disease: 273 patients studied over 20 years. Br J Haematol. 180:206–216. 2018.PubMed/NCBI View Article : Google Scholar
2	Biçakçioğlu P, Gülhan SS, Acar LN, Ağaçkiran Y, Özkara Ş, Kaya S and Karaoğlanoğlu N: Intrathoracic Castleman disease. Turk J Med Sci. 44:197–202. 2014.PubMed/NCBI View Article : Google Scholar
3	Dispenzieri A and Fajgenbaum DC: Overview of Castleman disease. Blood. 135:1353–1364. 2020.PubMed/NCBI View Article : Google Scholar
4	Haap M, Wiefels J, Horger M, Hoyer A and Müssig K: Clinical, laboratory and imaging findings in Castleman's disease-The subtype decides. Blood Rev. 32:225–234. 2018.PubMed/NCBI View Article : Google Scholar
5	van Rhee F, Oksenhendler E, Srkalovic G, Voorhees P, Lim M, Dispenzieri A, Ide M, Parente S, Schey S, Streetly M, et al: International evidence-based consensus diagnostic and treatment guidelines for unicentric Castleman disease. Blood Adv. 4:6039–6050. 2020.PubMed/NCBI View Article : Google Scholar
6	Kligerman SJ, Auerbach A, Franks TJ, Franks TJ and Galvin JR: Castleman disease of the thorax: Clinical, radiologic, and pathologic correlation: From the radiologic pathology archives. Radiographics. 36:1309–1332. 2016.PubMed/NCBI View Article : Google Scholar
7	Zhang X, Rao H, Xu X, Li Z, Liao B, Wu H, Li M, Tong X, Li J and Cai Q: Clinical characteristics and outcomes of Castleman disease: A multicenter study of 185 Chinese patients. Cancer Sci. 109:199–206. 2018.PubMed/NCBI View Article : Google Scholar
8	Hill AJ, Tirumani SH, Rosenthal MH, Shinagare AB, Carrasco RD, Munshi NC, Ramaiya NH and Howard SA: Multimodality imaging and clinical features in Castleman disease: Single institute experience in 30 patients. Br J Radiol. 88(20140670)2015.PubMed/NCBI View Article : Google Scholar
9	Ota H, Kawai H and Matsuo T: Unicentric castleman's disease arising from an intrapulmonary lymph node. Case Rep Surg. 2013(289089)2013.PubMed/NCBI View Article : Google Scholar
10	Sarana B, Jaal J, Tamm H and Laisaar T: Resection of unicentric interlobar Castleman disease with following adjuvant radiotherapy. SAGE Open Med Case Rep. 5(2050313x17744481)2017.PubMed/NCBI View Article : Google Scholar
11	Mimouni D, Anhalt GJ, Lazarova Z, Aho S, Kazerounian S, Kouba DJ, Mascaro JM Jr and Nousari HC: Paraneoplastic pemphigus in children and adolescents. Br J Dermatol. 147:725–732. 2002.PubMed/NCBI View Article : Google Scholar
12	van Rhee F and Munshi NC: Castleman disease. Hematol Oncol Clin North Am. 32:xiii–xiv. 2018.PubMed/NCBI View Article : Google Scholar
13	Boutboul D, Fadlallah J, Chawki S, Fieschi C, Malphettes M, Dossier A, Gérard L, Mordant P, Meignin V, Oksenhendler E and Galicier L: Treatment and outcome of unicentric castleman disease: A retrospective analysis of 71 cases. Br J Haematol. 186:269–273. 2019.PubMed/NCBI View Article : Google Scholar
14	Larroche C, Cacoub P, Soulier J, Oksenhendler E, Clauvel JP, Piette JC and Raphael M: Castleman's disease and lymphoma: Report of eight cases in HIV-negative patients and literature review. Am J Hematol. 69:119–126. 2002.PubMed/NCBI View Article : Google Scholar
15	Wang ST, Wang QP, Li J, Zhang T, Zhang L and Mao YY: Amyloidosis secondary to intrapulmonary Castleman disease mimicking pulmonary hyalinizing granuloma-like clinical features: A rare case report. Medicine (Baltimore). 98(e15039)2019.PubMed/NCBI View Article : Google Scholar
16	Zhang H, Zhang JQ, Zhong XN, Liu GN, Li MH, Bai J, He ZY and Deng JM: Two case reports of Castleman disease with pulmonary involvement. Int J Tuberc Lung Dis. 19:362–365. 2015.PubMed/NCBI View Article : Google Scholar
17	Hwangbo Y, Cha SI, Lee YH, Lee SY, Seo H, Oh S, Kim M, Choi SH, Park TI and Shin KM: A case of multicentric castleman's disease presenting with follicular bronchiolitis. Tuberc Respir Dis (Seoul). 74:23–27. 2013.PubMed/NCBI View Article : Google Scholar
18	Nabeya D, Yoshimatsu Y and Fujiwara H: A case of acute progressive diffuse interstitial lung disease preceding idiopathic multicentric Castleman disease. Respir Med Case Rep. 31(101216)2020.PubMed/NCBI View Article : Google Scholar
19	Johkoh T, Müller NL, Ichikado K, Nishimoto N, Yoshizaki K, Honda O, Tomiyama N, Naitoh H, Nakamura H and Yamamoto S: Intrathoracic multicentric Castleman disease: CT findings in 12 patients. Radiology. 209:477–481. 1998.PubMed/NCBI View Article : Google Scholar
20	Herrada J, Cabanillas F, Rice L, Manning J and Pugh W: The clinical behavior of localized and multicentric Castleman disease. Ann Intern Med. 128:657–662. 1998.PubMed/NCBI View Article : Google Scholar
21	Chronowski GM, Ha CS, Wilder RB, Cabanillas F, Manning J and Cox JD: Treatment of unicentric and multicentric Castleman disease and the role of radiotherapy. Cancer. 92:670–676. 2001.PubMed/NCBI View Article : Google Scholar
22	Marcelin AG, Aaron L, Mateus C, Gyan E, Gorin I, Viard JP, Calvez V and Dupin N: Rituximab therapy for HIV-associated Castleman disease. Blood. 102:2786–2788. 2003.PubMed/NCBI View Article : Google Scholar
23	Ide M, Kawachi Y, Izumi Y, Kasagi K and Ogino T: Long-term remission in HIV-negative patients with multicentric Castleman's disease using rituximab. Eur J Haematol. 76:119–123. 2006.PubMed/NCBI View Article : Google Scholar
24	Ocio EM, Sanchez-Guijo FM, Diez-Campelo M, Castilla C, Blanco OJ, Caballero D and San Miguel JF: Efficacy of rituximab in an aggressive form of multicentric Castleman disease associated with immune phenomena. Am J Hematol. 78:302–325. 2005.PubMed/NCBI View Article : Google Scholar
25	Corbellino M, Bestetti G, Scalamogna C, Calattini S, Galazzi M, Meroni L, Manganaro D, Fasan M, Moroni M, Galli M and Parravicini C: Long-term remission of Kaposi sarcoma-associated herpesvirus-related multicentric Castleman disease with anti-CD20 monoclonal antibody therapy. Blood. 98:3473–3475. 2001.PubMed/NCBI View Article : Google Scholar
26	Kofteridis DP, Tzagarakis N, Mixaki I, Maganas E, Xilouri E, Stathopoulos EN, Eliopoulos GD and Gikas A: Multicentric Castleman's disease: Prolonged remission with anti CD-20 monoclonal antibody in an HIV-infected patient. AIDS. 18:585–586. 2004.PubMed/NCBI View Article : Google Scholar
27	Bower M, Newsom-Davis T, Naresh K, Merchant S, Lee B, Gazzard B, Stebbing J and Nelson M: Clinical features and outcome in HIV-Associated multicentric castleman's disease. J Clin Oncol. 29:2481–2486. 2011.PubMed/NCBI View Article : Google Scholar
28	Anhalt GJ: Paraneoplastic pemphigus. J Investig Dermatol Symp Proc. 9:29–33. 2004.PubMed/NCBI View Article : Google Scholar
29	Lim JM, Lee SE, Seo J, Kim DY, Hashimoto T and Kim SC: Paraneoplastic pemphigus associated with a malignant thymoma: A case of persistent and refractory oral ulcerations following thymectomy. Ann Dermatol. 29:219–222. 2017.PubMed/NCBI View Article : Google Scholar
30	Wang L, Bu D, Yang Y, Chen X and Zhu X: Castleman's tumours and production of autoantibody in paraneoplastic pemphigus. Lancet. 363:525–531. 2004.PubMed/NCBI View Article : Google Scholar
31	Reichard KK, Robinett S and Foucar MK: Clonal cytogenetic abnormalities in the plasma cell variant of Castleman disease. Cancer Genet. 204:323–327. 2011.PubMed/NCBI View Article : Google Scholar
32	Mihara M, Hashizume M, Yoshida H, Suzuki M and Shiina M: IL-6/IL-6 receptor system and its role in physiological and pathological conditions. Clin Sci (Lond). 122:143–159. 2012.PubMed/NCBI View Article : Google Scholar
33	Luo JM, Li S, Huang H, Cao J, Xu K, Bi YL, Feng RE, Huang C, Qin YZ, Xu ZJ and Xiao Y: Clinical spectrum of intrathoracic Castleman disease: A retrospective analysis of 48 cases in a single Chinese hospital. BMC Pulm Med. 15(34)2015.PubMed/NCBI View Article : Google Scholar