We report a case of transfusion-related acute lung injury (TRALI) occurring during the third session of artificial liver therapy in a patient with hyperbilirubinemia caused by drug-induced liver injury. The patient’s symptoms improved after treatment with non-invasive ventilator-assisted ventilation, methylprednisolone sodium succinate, diuretics, and other interventions. This case reminds clinicians that transfusion-related acute lung injury can also occur rarely during artificial liver therapy. Early diagnosis and timely management are essential to reduce patient mortality.