Combined antiviral-immunosuppressive treatment in human T-lymphotrophic virus 1-Sjögren-associated myelopathy.
BACKGROUND
In several studies, antiretroviral drugs (principally zidovudine) have been used with success in the treatment of myelopathy associated with human T-lymphotrophic virus 1 (HTLV-1) (tropical spastic paraparesis-HTLV-1-associated myelopathy). The retrovirus HTLV-1 has been implicated as a causative agent of Sjögren syndrome (SS) in clinical reports and murine experiments. Moreover, a recognized complication of primary SS is a myelopathy, which has been shown in case reports to respond to immunosuppressive treatment.


OBJECTIVE
To describe a patient with a rapidly progressive, extensive myelopathy with evidence of HTLV-1 infection and SS (probably secondary to HTLV-1) in whom we achieved spectacular therapeutic success using combined immunosuppressive and antiviral therapy.


METHODS
Case report.


METHODS
University hospital. Patient A young Haitian woman diagnosed with HTLV-1 and SS developed extensive myelopathy leading to severe disability.


METHODS
Clinical and radiological improvement.


RESULTS
Spectacular radiological and clinical recovery as well as stabilization were achieved with combined antiviral and immunosuppressant treatment. Follow-up at 2 years showed no signs of relapse.


CONCLUSIONS
Both tropical spastic paraparesis-HTLV-1-associated myelopathy and Sjögren myelopathy are potentially very disabling. Rapidly progressive myelopathy secondary to SS necessitates the introduction of immunosuppressant therapies. The presence of HTLV-1 may confer the necessity to add antiviral therapy.
Pot
Caroline
C
Department of Neurology, University Hospital and Medical School of Geneva, Micheli-du-Crest 24, CH-1211 Geneva, Switzerland.
Chizzolini
Carlo
C
Vokatch
Natalia
N
Tiercy
Jean-Marie
JM
Ribi
Camillo
C
Landis
Theodor
T
Perren
Fabienne
F
D000225:Adenine / Q000031:analogs & derivatives / Q000627:therapeutic use; D000998:Antiviral Agents / Q000627:therapeutic use*; D004359:Drug Therapy, Combination; D005260:Female; D015490:HTLV-I Infections / Q000150:complications / Q000188:drug therapy* / Q000532:radiotherapy; D006801:Humans; D007166:Immunosuppressive Agents / Q000627:therapeutic use*; D019259:Lamivudine / Q000627:therapeutic use; D008279:Magnetic Resonance Imaging / Q000379:methods; D008775:Methylprednisolone / Q000627:therapeutic use; D008875:Middle Aged; D063065:Organophosphonates / Q000627:therapeutic use; D011241:Prednisone / Q000627:therapeutic use; D011859:Radiography; D012859:Sjogren's Syndrome / Q000150:complications / Q000000981:diagnostic imaging / Q000188:drug therapy*; D000068698:Tenofovir
D000998:Antiviral Agents; D007166:Immunosuppressive Agents; D063065:Organophosphonates; D019259:Lamivudine; D000068698:Tenofovir; D000225:Adenine; D011241:Prednisone; D008775:Methylprednisolone