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September 2006

Combined Antiviral-Immunosuppressive Treatment in Human T-Lymphotrophic Virus 1–Sjögren–Associated Myelopathy

Author Affiliations

Author Affiliations: Departments of Neurology (Drs Pot, Vokatch, Landis, and Perren) and Immunology (Drs Chizzolini, Tiercy, and Ribi), University Hospital and Medical School of Geneva, Geneva, Switzerland.

Arch Neurol. 2006;63(9):1318-1320. doi:10.1001/archneur.63.9.1318

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.

Design  Case report.

Setting  University hospital.

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

Main Outcome Measures  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.