Antiretroviral activity of 5-azacytidine during treatment of a HTLV-1 positive myelodysplastic syndrome with autoimmune manifestations.
Myelodysplastic syndromes (MDS) are often accompanied by autoimmune phenomena. The underlying mechanisms for these associations remain uncertain, although T cell activation seems to be important. Human T-lymphotropic virus (HTLV-1) has been detected in patients with myelodysplastic syndromes, mostly in regions of the world which are endemic for the virus, and where association of HTLV-1 with rheumatological manifestation is not rare. We present here the case of a 58 year old man who presented with cytopenias, leukocytoclastic vasculitis of the skin and glomerulopathy, and was diagnosed as MDS (refractory anemia with excess blasts - RAEB 1). The patient also tested positive for HTLV-1 by PCR. After 8 monthly cycles of 5-azacytidine he achieved a complete hematologic remission. Following treatment, a second PCR for HTLV-1 was carried out and found to be negative. This is the first report in the literature of a HTLV-1-positive MDS with severe autoimmune manifestations, which was treated with the hypomethylating factor 5-azacitidine, achieving cytogenetic remission with concomitant resolution of the autoimmune manifestations, as well as HTLV-1-PCR negativity. HTLV-1-PCR negativity may be due to either immune mediated clearance of the virus, or a potential antiretroviral effect of 5-azacytidine. 5-azacytidine is known for its antiretroviral effects, although there is no proof of its activity against HTLV-1 infection in vivo.
1st Department of Internal Medicine, Haematology Unit, National and Kapodistrian University of Athens, Laikon General Hospital, Athens 11527, Greece. pandiamantopoulos@gmail.com
Panagiotis T
PT
Diamantopoulos
Maria
M
Michael
Olga
O
Benopoulou
Efthymia
E
Bazanis
George
G
Tzeletas
John
J
Meletis
George
G
Vayopoulos
Nora-Athina
NA
Viniou
D044966:Anti-Retroviral Agents / Q000008:administration & dosage*; D000964:Antimetabolites, Antineoplastic / Q000008:administration & dosage*; D001327:Autoimmune Diseases / Q000188:drug therapy*; D001374:Azacitidine / Q000008:administration & dosage*; D015490:HTLV-I Infections / Q000188:drug therapy*; D015368:Human T-lymphotropic virus 1 / Q000187:drug effects* / Q000235:genetics / Q000302:isolation & purification; D006801:Humans; D008297:Male; D008875:Middle Aged; D009190:Myelodysplastic Syndromes / Q000188:drug therapy*; D016133:Polymerase Chain Reaction; D016896:Treatment Outcome
D044966:Anti-Retroviral Agents; D000964:Antimetabolites, Antineoplastic; D001374:Azacitidine