Multicentric Castleman's disease treated with antivirals and immunosuppressants

Abstract

A patient negative for human immunodeficiency virus (HIV) developed multicentric Castleman's disease (MCD) and Kaposi's sarcoma (KS) associated with active human herpesvirus 8 (HHV-8) infection. He was treated with sequential antiviral therapy, chemotherapy, and corticosteroids. HHV-8 levels were monitored throughout the course of the patient's illness, and were found to rise on relapse. No consistent change in HHV-8 levels was found with antiviral therapy. We demonstrate that in this patient antiviral therapy was clinically ineffective, and did not alter HHV-8 levels, but that corticosteroid and combination chemotherapy led to clinical improvement. Despite the implication of HHV-8 as a cause of MCD, few studies have correlated HHV-8 levels with clinical response.

Fifty years of multicentric Castleman's disease

“…All the patients had a reduction in KSHV viral load with the ganciclovir therapy, accompanying the resolution of their symptoms. However, the use of foscarnet and cidofovir antiviral therapy was ineffective in an HIV-negative MCD patient with proven KSHV viraemia and treatment with corticosteriods in combination with chlorambucil chemotherapy was required to achieve a clinical response (60). Furthermore the KSHV viral load rose in this patient with the commencement of anti-herpesvirus therapy; this may denote that the antiviral therapy was ineffective in this case, or that once the MCD is established KSHV has a less prominent role and antiviral therapy is less effective than immunotherapy or chemotherapy.…”

HIV-associated multicentric Castleman disease

“…In studies with cidofovir, zero of seven patients responded. (24, 80–81) Two of four patients achieved remission with foscarnet. (19, 76, 81, 82) Ganciclovir and valgangiclovir induced remission in one patient and reduced frequency of relapse in two additional patients.…”

Therapeutic options for human herpesvirus-8/Kaposi’s sarcoma-associated herpesvirus-related disorders

“…Also, there are case reports describing successful control of KS, PEL and MCD by antiherpes drugs with or without cytotoxic chemotherapy [73,[103][104][105]. Despite these encoraging observations, other groups have reported poor therapeutic effects of antiherpes agents in KSHVrelated disorders [54,106,107]. Therefore, it is unclear at present if inhibition of lytic KSHV replication is beneficial in treating these diseases.…”