Treatment of Idiopathic Multicentric Castleman’s Disease With Sequential Thalidomide-Cyclophosphamide-Prednisone After Siltuximab:Report of One Case

Yue DANG; Jian LI; Yaping LUO; Lu ZHANG

doi:10.3881/j.issn.1000-503X.16284

PDF(486 KB)

Acta Academiae Medicinae Sinicae ›› 2025, Vol. 47 ›› Issue (3) : 483-486. DOI: 10.3881/j.issn.1000-503X.16284

Treatment of Idiopathic Multicentric Castleman’s Disease With Sequential Thalidomide-Cyclophosphamide-Prednisone After Siltuximab:Report of One Case

Yue DANG ¹ ,
Jian LI ¹ ,
Yaping LUO ² ,
Lu ZHANG ¹

Author information +

History +

Abstract

Castleman’s disease is a rare polyclonal lymphoproliferative disorder.This article reports the diagnosis and treatment of a 45-year-old female patient with idiopathic multicentric Castleman’s disease.The patient presented recurrent fever,enlarged lymph nodes,and elevated levels of inflammation markers.After multiple serological examinations and tissue biopsies,she was diagnosed with hyaline vascular-type Castleman’s disease.Initially,the patient received siltuximab targeting interleukin-6,which significantly improved her condition.Considering the cost and convenience of long-term treatment,she subsequently switched the therapy to an oral treatment regimen of thalidomide,cyclophosphamide,and prednisone (TCP),which maintained disease control.This report aims to highlight the diagnostic complexity and diversity of treatment options for idiopathic multicentric Castleman’s disease,demonstrating the potential of the TCP regimen as a cost-effective treatment choice.

Key words

Castleman’s disease / siltuximab / thalidomide,cyclophosphamide,and prednisone regimen

Cite this article

EndNote

Ris (Procite)

Bibtex

Download Citations

Yue DANG , Jian LI , Yaping LUO , et al. Treatment of Idiopathic Multicentric Castleman’s Disease With Sequential Thalidomide-Cyclophosphamide-Prednisone After Siltuximab:Report of One Case[J]. Acta Academiae Medicinae Sinicae. 2025, 47(3): 483-486 https://doi.org/10.3881/j.issn.1000-503X.16284

References

List( Publishing order | Descend order by publishing year | Descend order by cited within ) Chart analysis

[1]	Castleman B, Towne VW. Case records of the Massachusetts General Hospital:case No.40231[J]. N Engl J Med, 1954, 250(23):1001-1005.DOI:10.1056/nejm195406102502308. Cited in this article [1]

[2]	Carbone A, Borok M, Damania B, et al. Castleman disease[J]. Nat Rev Dis Primers, 2021, 7(1):84.DOI:10.1038/s41572-021-00317-7. Cited in this article [1]

[3]	Vora K, Dash A, Bach A, et al. A case of Castleman’s disease that presented as a retroperitoneal mass[J]. Nat Rev Urol, 2007, 4(5):285-288.DOI:10.1038/ncpuro0800. Cited in this article [1]

[4]	Fajgenbaum DC, Uldrick TS, Bagg A, et al.International, evidence-based consensus diagnostic criteria for HHV-8-negative/idiopathic multicentric Castleman disease[J]. Blood, 2017, 129(12):1646-1657.DOI:10.1182/blood-2016-10-746933. Cited in this article [1]

[5]	van Rhee F, Oksenhendler E, Srkalovic G, et al. International evidence-based consensus diagnostic and treatment guidelines for unicentric Castleman disease[J]. Blood Adv, 2020, 4(23):6039-6050.DOI:10.1182/bloodadvances.2020003334. Cited in this article [2]

[6]	Zhang X, Rao H, Xu X, et al. Clinical characteristics and outcomes of Castleman disease:a multicenter study of 185 Chinese patients[J]. Cancer Sci, 2018, 109(1):199-206.DOI:https://doi.org/10.1111/cas.13439. Cited in this article [1]

[7]	中华医学会血液学分会淋巴细胞疾病学组, 中国抗癌协会血液肿瘤专业委员会, 中国Castleman病协作组. 中国Castleman病诊断与治疗专家共识(2021年版)[J]. 中华血液学杂志, 2021, 42(7):529-534.DOI:10.3760/cma.j.issn.0253-2727.2021.07.001. Cited in this article [2]

[8]	van Rhee F, Wong RS, Munshi N, et al. Siltuximab for multicentric Castleman’s disease:a randomised,double-blind,placebo-controlled trial[J]. Lancet Oncol, 2014, 15(9):966-974.DOI:10.1016/S1470-2045(14)70319-5. Cited in this article [2]

[9]	张路, 李剑. 白细胞介素-6靶向治疗前时代的中国Castleman病诊疗现状[J]. 协和医学杂志, 2023, 14(5):911-914.DOI:10.12290/xhyxzz.2023-0227. Cited in this article [1]

[10]

Morra

, Pierson

, Shilling

, et al. Predictors of response to anti-IL 6 monoclonal antibody therapy (siltuximab) in idiopathic multicentric Castleman disease:secondary analyses of phase Ⅱ clinical trial data[J]. Br J Heamatol, 2019, 184(2):232-241.DOI:10.1111/bjh.15588.

Cited in this article [1]

[11]	Garbers C, Heink S, Korn T, et al. Interleukin-6:designing specific therapeutics for a complex cytokine[J]. Nat Rev Drug Discov, 2018, 17(6):395-412.DOI:10.1038/nrd.2018.45. Cited in this article [1]

[12]	Zhang L, Zhao AL, Duan MH, et al. Phase 2 study using oral thalidomide-cyclophosphamide-prednisone for idiopathic multicentric Castleman disease[J]. Blood, 2019, 133(16):1720-1728.DOI:10.1182/blood-2018-11-884577. Cited in this article [1]

[13]	Fajgenbaum DC, Langan RA, Japp AS, et al. Identifying and targeting pathogenic PI3K/AKT/mTOR signaling in IL-6-blockade-refractory idiopathic multicentric Castleman disease[J]. J Clin Invest, 2019, 129(10):4451-4463.DOI:10.1172/jci126091. Cited in this article [1]

[14]	Puzik A, Thiel A, Faust K, et al. Thalidomide has anti-inflammatory properties in neonatal immune cells[J]. Innate Immun, 2013, 19(1): 42-52.DOI:10.1177/1753425912449881. Cited in this article [1]

[15]	Shortt J, Hsu AK, Johnstone RW. Thalidomide-analogue biology:immunological,molecular and epigenetic targets in cancer therapy[J]. Oncogene, 2013, 32(36):4191-4202.DOI:10.1038/onc.2012.599. Cited in this article [1]

[16]	Dong Y, Zhang L, Nong L, et al. Effectiveness of rituximab-containing treatment regimens in idiopathic multicentric Castleman disease[J]. Ann Hematol, 2018, 97(9):1641-1647.DOI:10.1007/s00277-018-3347-0. Cited in this article [1]

[17]	Harada T, Kikushige Y, Miyamoto T, et al. Peripheral helper-T-cell-derived CXCL13 is a crucial pathogenic factor in idiopathic multicentric Castleman disease[J]. Nat Commun, 2023, 14(1):6959.DOI:10.1038/s41467-023-42718-0. Cited in this article [1]

[18]	Pierson SK, Katz L, Williams R, et al. CXCL13 is a predictive biomarker in idiopathic multicentric Castleman disease[J]. Nat Commun, 2022, 13(1):7236.DOI:10.1038/s41467-022-34873-7. Cited in this article [1]