Giant lymph node hyperplasia (GLNH) or Castleman disease is a heteroge
nous group of atypical lympho-proliferative disorders. Two main histol
ogic variants, the hyaline vascular variant and the plasma cell varian
t, have been recognized. Although localized GLNH can often be managed
successfully with surgery, optimal therapy for multifocal disease has
yet to be identified. We report two cases of GLNH treated with 2-chlor
o-deoxyadenosine (2-CDA), a synthetic purine analogue. 2-CDA was utili
zed based on its relative lymphocytic toxicity and the putative pathop
hysiologic process in GLNH being either hamartomatous overgrowth (hyal
ine-vascular variant) or immune dysfunction and lymphoproliferation (p
lasma cell variant). One patient with unresectable localized hyaline-v
ascular GLNH has had a 9-month continuous complete remission following
two courses of 2-CDA therapy followed by radiation therapy. The secon
d patient with disseminated plasma cell type had a partial response to
two cycles of 2-CDA therapy; however, further cycles were not given d
ue to development of possible early neurotoxicity. Although the optima
l management of non-resectable GLNH is yet to be determined, 2-CDA app
ears to be a viable therapeutic option for patients with this disease
process.