BACKGROUND: Available data on the burden of Human T-cell lymphotropic virus type I/II infection for eastern Africa, limited to Ethiopia, Mozambique, and Rwanda, show prevalence lower than elsewhere in Africa (0% - 1.8%). Even if Tropical Spastic Paraparesis occurs in an endemic form in Ethiopia, its seroprevalence is low. Over a lifetime, it is estimated that 1–2% of Human T-cell lymphotropic virus type I/ II -infected individuals will develop progressive and disabling inflammatory clinical manifestations. We are reporting this case since it signifies the existence of seropositive Tropical Spastic Paraparesis in our setting and the need to properly diagnose this condition.CASE PRESENTATION:  We are reporting a 45 years old female patient from Addis Ababa, Ethiopia, who presented with progressive weakness of the lower limbs and urinary urge incontinence of five years duration. Serology for Human T-cell lymphotropic virus type I/ II antibody was positive. She was diagnosed to have probable tropical spastic paraparesis after fulfilling World Health Organization diagnostic criteria for tropical spastic paraparesis with the level of ascertainment. Symptoms showed transient improvements after providing five days of Methylprednisolone followed by low doses of corticosteroids and Azathioprine. The patient is now significantly disabled and wheelchair-bound. CONCLUSIONS: The patient described here signifies a probable Human T-cell lymphotropic virus type I/ II - associated myelopathy/ tropical spastic paraparesis in Ethiopian women. This case highlights the existence of Human T-cell lymphotropic virus type I/ II - associated myelopathy/ tropical spastic paraparesis within our setting and the need to properly diagnose this condition.KEYWORDS:  Probable HTLV associated myelopathy/ Tropical Spastic Paraparesis, case report, Ethiopia