We encountered a late pulmonary complication after umbilical cord blood transplantation (UCBT) that has not been previously reported. High-resolution CT (HRCT) findings of this disease were compared with the pathology. HRCT obtained on inspiration showed dilated thick-walled bronchioli, and innumerable centrilobular linear and branching structures in the bilateral middle and lower lobes. Neither mosaic perfusion nor air-trapping was seen in HRCT on inspiration and expiration. These HRCT findings were atypical compared with those of former bronchiolitis obliterans (BO) after bone marrow transplant (BMT). Pathologic specimens obtained by open lung biopsy showed thickening of the wall from the distal bronchioli to the alveolar ducts due to submucosal and intraepithelial infiltration of lymphocytes, histiocytes and foamy macrophages, which was not accompanied by organizing changes. These changes resemble lymphocytic bronchiolitis in lung transplant recipients, which was well correlated with HRCT findings. We think that our case was a new late pulmonary complication after UCBT.