Short-term efficacy of the IL6 receptor antibody tocilizumab in patients with HIV-associated multicentric Castleman disease: report of two cases

Abstract

Multicentric Castleman disease (MCD) is a lymphoproliferative disorder caused by human herpesvirus 8 (HHV8) infection HIV associated MCD (HIV-MCD) presents with various clinical symptoms. Many HIV-negative MCD patients are often treated with anti-human interleukin-6 (IL6) receptor monoclonal antibodies (tocilizumab), and successful results have been reported. IL-6 plays an important role in the development of both HIV-positive and HIV-negative MCD; however, the efficacy of tocilizumab in HIV-MCD patients is unknown. We herein report the clinical and biologic courses of two HIV-MCD patients treated with tocilizumab. In both cases, a significant and rapid clinical improvement was observed after the first infusion. However, the treatment efficacy was not maintained for a long period, and relapse occurred at 15 and 22 weeks, respectively. Both patients received rituximab and subsequently achieved complete clinical remission. Our report, in addition to data presented in the literature, suggests that tocilizumab could be an initial treatment option in patients with HIV-MCD.

Clinical and laboratory characterization of 114 cases of Castleman disease patients from a single centre: paraneoplastic pemphigus is an unfavourable prognostic factor

“…Rituximab has been used in HIV‐ and/or HHV8‐positive MCD patients (Gérard et al , , ). Anti‐interleukin 6 (IL6) receptor antibodies have long been used for MCD treatment (Galeotti et al , ; Nagao et al , ). The humanized anti‐IL6 monoclonal antibody, siltuximab, was recently proven to be effective against MCD in a randomized multi‐centre clinical trial (van Rhee et al , ) and has now been approved for use in MCD patients in US and Europe (Markham & Patel, ).…”

Is tocilizumab a potential therapeutic option for refractory unicentric Castleman disease?

“…Biochemical improvement in inflammatory markers was also noted . Three small case series, using the same regimen of tocilizumab and each including 2 to 3 patients, reported an overall response rate of 100% with a follow‐up ranging up to 4 years . Tocilizumab has also been reported to be beneficial in PNP associated with MCD …”

Intrapulmonary unicentric Castleman disease mimicking peripheral pulmonary malignancy

“…This form of disease is typically accompanied by increased concentrations of circulating cytokines, primarily IL‐6. Patients with multicentric disease have recently been treated using antibodies against the IL‐6 receptor, such as tocilizumab and siltuximab, with relatively satisfactory results …”