Hepatic sinusoidal obstruction syndrome (HSOS) is a rare hepatic vascular disorder characterized by intrahepatic congestion, liver injury, and post-sinusoidal portal hypertension, and it is frequently associated with hematopoietic stem cell transplantation. In this study, we observed a case of HSOS associated with the ingestion of , a pyrrolizidine alkaloid (PA)-containing Chinese herb, in a patient with alcoholic cirrhosis. The patient was a 43-year-old man with chief complaints of physical asthenia and a loss of appetite for more than a month. The diagnosis of HSOS combined with alcoholic cirrhosis was confirmed via the histopathological examination of liver tissues. With proper supportive and symptomatic care and anticoagulation therapy using low-molecular-weight heparin, the patient's condition was stabilized. Because of its nonspecific symptoms in the early stage and a lack of information about PA consumption, PA-induced HSOS (PA-HSOS) has been long neglected, especially in patients with underlying liver diseases. Early identification and intervention are critical for optimizing outcomes. Further efforts are needed to supervise the use of PA-containing herbal medicines and identify accurate biomarkers for PA-HSOS.