Clinical disease due to human T cell lymphotropic virus type 1 (HTLVâ1), a retrovirus endemic in certain regions of the world, is rarely reported after solid organ transplantation. In 2009, universal deceased donor organ screening for HTLVâ1 was discontinued in the United States. We report the first case of donorâderived HTLVâ1âassociated myelopathy in a kidney transplant recipient from the United States. The patient, who was HTLVâ1âseronegative prior to transplantation, likely acquired HTLVâ1 infection from a seropositive organ donor. In this era when screening of donors and recipients for HTLV infection is not mandatory, clinicians should be vigilant in recognizing the risk and potential occurrence of this donorâderived infection in recipients with epidemiologic exposures.