Recurrent and Hodgkin’s lymphoma-associated multicentric Castleman’s disease in head and neck region
Keywords:Angio-follicular lymph hyperplasia, Castleman’s disease, Giant lymph node hyperplasia, Lymphoproliferative disorder, Rituximab
Background: Castleman’s disease (CD) is a rare lymphoproliferative disorder. It can involve single (unicentric CD) or multiple (multicentric CD) lymph nodal regions. It occurs predominantly in mediastinum, and treatment options include surgery, radiotherapy, chemotherapy, and monoclonal antibodies.
Methods: Here, we describe two cases of CD which presented with stridor. The first case was a 38-year-old male, a recurrent multicentric CD in retropharyngeal and cervical lymph nodal regions, treated with radiotherapy and rituximab. The second case was a 25-year-old male, a multicentric CD in lower cervical lymph nodal region, treated with steroids and radiotherapy. He subsequently developed Hodgkin’s lymphoma and was treated for the same with chemotherapy and involved-field radiation therapy (IFRT).
Results: Post-treatment, both the patients were asymptomatic and progression-free at 15 months and 42 months follow-up, respectively.
Conclusions: Combined modality of treatment with radiotherapy and chemotherapy or monoclonal antibodies offers good local control in multicentric CD.
Keller AR, Hochholzer L, Castleman B. Hyaline-vascular and plasma-cell types of giant lymph node hyperplasia of the mediastinum and other locations. Cancer. 1972;29:670-83.
Castleman B, Iverson L, Menendez VP. Localized mediastinal lymph node hyperplasia resembling thymoma. Cancer. 1956;9:822-30.
Bowne WB, Lewis JJ, Filippa DA. The management of unicentric and multicentric Castleman’s disease: a report of 16 cases and a review of the literature. Cancer. 1999;85:706-17.
Bucher P, Chassot G, Zufferey G. Surgical management of abdominal and retroperitoneal Castleman’s disease, World J.Surg. Oncol. 2005;3:33.
Hakozaki M, Tajino T, Yamada H. Intramuscular Castleman’s disease of the deltoid: a case report and review of the literature. Skeletal Radiol. 2010;39:715-19.
Lee J, Ban JY, Won KY. Expression of EGFR and follicular dendritic markers in lymphoid follicles from patients with Castleman’s disease. Oncol Rep. 2008;20:851-56.
Menke DM, Camoriano JK, Banks PM. Angiofollicular lymph node hyperplasia: a comparison of unicentric, multicentric, hyaline vascular, and plasma cell types of disease by morphometric and clinical analysis. Mod Pathol. 1992;5:525-30.
Cronin DM, Warnke RA. Castleman disease: an update on classification and the spectrum of associated lesions. Adv Anat Pathol. 2009;16:236-46.
Fajgenbaum DC, Uldrick TS, Bagg A. International, evidence-based consensus diagnostic criteria for HHV-8–negative/idiopathic multicentric Castleman disease. Blood. 2017;129(12):1646-57.
Tazi I, Rachid M, Quessar A. A rare association of Castleman’s disease and nephrotic syndrome. Saudi J Kidney Dis Transpl. 2011;22:116-19.
De MG, De VS, Fabris M. Systemic connective tissue disease complicated by Castleman’s disease: report of a case and review of the literature. Haematologica. 2004;89:03.
Van Rhee F, Stone K, Szmania S. Castleman disease in the 21st century: an update on diagnosis, assessment, and therapy. Clin Adv Hematol Oncol. 2010;8:486-498.
Chronowski GM, Ha CS, Wilder RB. Treatment of unicentric and multicentric Castleman disease and the role of radiotherapy. Cancer. 2001;92:670-6.
Neuhof D, Debus J. Outcome and late complications of radiotherapy in patients with unicentric Castleman disease. Acta Oncol. 2006;45:1126-31.
Noh OK, Lee S, Lee JW. Cases report of unicentric Castleman’s disease: revisit of radiotherapy role. Radiat Oncol J. 2013;31(1):48-54.
Liu AY, Nabel CS, Finkelman BS. Idiopathic multicentric Castleman’s disease: a systematic literature review. Lancet Haematol. 2016;3(4):e163-e75.
Van Rhee F, Wong RS, Munshi N. Siltuximab for multicentric Castleman’s disease: a randomised, double-blind, placebo-controlled trial. Lancet Oncol. 2014;15(9):966-74.
Deisseroth A, Ko CW, Nie L. FDA approval: siltuximab for the treatment of patients with multicentric Castleman disease. Clin Cancer Res. 2015;21(5):950-54.
Nishimoto N, Sasai M, Shima Y. Improvement in Castleman’s disease by humanized anti-interleukin-6 receptor antibody therapy. Blood. 2000;95(1):56-61.
Talat N, Belgaumkar AP, Schulte KM. Surgery in Castleman’s disease: a systematic review of 404 published cases. Ann Surg. 2012;255(4):677-84.
Bower M, Newsom-Davis T, Naresh K. Clinical features and outcome in HIV-associated multicentric Castleman’s disease. J Clin Oncol. 2011;29(18):2481-86.
Pria AD, Pinato D, Roe J. Relapse of HHV8-positive multicentric Castleman disease following rituximab-based therapy in HIV-positive patients. Blood. 2017;129(15):2143-47.