title: Combined antiviral-immunosuppressive treatment in human T-lymphotrophic virus 1-Sjögren-associated myelopathy.
abstract: 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.
authors.affiliation: Department of Neurology, University Hospital and Medical School of Geneva, Micheli-du-Crest 24, CH-1211 Geneva, Switzerland.
authors.forename: Caroline
authors.initials: C
authors.lastname: Pot
authors.forename: Carlo
authors.initials: C
authors.lastname: Chizzolini
authors.forename: Natalia
authors.initials: N
authors.lastname: Vokatch
authors.forename: Jean-Marie
authors.initials: JM
authors.lastname: Tiercy
authors.forename: Camillo
authors.initials: C
authors.lastname: Ribi
authors.forename: Theodor
authors.initials: T
authors.lastname: Landis
authors.forename: Fabienne
authors.initials: F
authors.lastname: Perren
mesh_terms: 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
chemical_list: D000998:Antiviral Agents; D007166:Immunosuppressive Agents; D063065:Organophosphonates; D019259:Lamivudine; D000068698:Tenofovir; D000225:Adenine; D011241:Prednisone; D008775:Methylprednisolone