Ethics Statement
Ethics Statement is not applicable for case report according to the Medical Ethics Committee of the Third Hospital of Zhenjiang Affiliated Jiangsu University, but Informed consent was obtained from the patients for publication. The study was conducted in accordance with the Declaration of Helsinki.
Case 1
A 49-year-old man consumed Tusanqi three times a day after a fall injury (3 g each time). After taking it for more than 1 month, he complained of fatigue, abdominal distension, and abdominal pain for one week. Routine blood biochemical indexes were as follows: total bilirubin (TBIL), 40.6 µmol/L; albumin (ALB), 33.2 g/L; alanine aminotransferase (ALT), 490 U/L; aspartate aminotransferase (AST), 892.3 U/L; alkaline phosphatase (ALP), 136.0 U/L; gamma-glutamyl transferase (GGT), 83 U/L. Routine bloodwork findings were as follows: hemoglobin (HB), 134 g/L; platelet count (PLT), 152 × 109/L; white blood cell count (WBC), 6.8 × 109/L; C-reactive protein (CRP), 72.49 mg/mL; prothrombin time (PT), 16.0 s; international standardized ratio (INR), 1.44. Computed tomography (CT) showed diffuse enlargement of the liver, and a plain scan showed an uneven decrease in the density of the liver parenchyma. The hepatic parenchyma in the arterial and venous phases showed characteristic "map" and uneven enhancement, the hepatic vein was not clearly observed, and ascites was complicated (Fig. 1A).
HSOS was diagnosed after admission and the patient was treated with low-molecular-weight heparin and warfarin. After 40 days of anticoagulation treatment, his clinical symptoms disappeared, and the biochemical indices and imaging results were normal (Fig. 1B). After discharge, the patient insisted on oral warfarin treatment, and the goal of achieving INR 2–2.5 was monitored weekly. At the third and eighth months after cure, a liver biopsy was performed. Viral hepatitis (A–E), anti-nuclear antibody, anti-smooth muscle antibody, and anti-mitochondrial antibody tests were negative, and the immunoglobulin level was within the normal range. The patient was tested for INR and other indices weekly after discharge, and the warfarin anticoagulation treatment was discontinued after 3 months. No abnormal manifestations were observed in the biochemical indices or imaging characteristics.
Case 2
A 52-year-old woman intermittently used Tusanqi 1–2 times daily (3 g each time). Symptoms of fatigue, abdominal distension, and pain occurred after 15 days. Routine blood biochemical indexes were as follows: WBC, 4.13 × 109/L; HB, 149 g/L; PLT, 123 × 109/L; PT, 15.0 s; INR, 1.17; TBIL, 48.9 µmol/L; ALB, 26.0 g/L; ALT, 1215 U/L; AST, 1347 U/L; ALP, 118 U/L; GGT, 251 U/L. Diffuse liver swelling was evident on ultrasound; the echo of the liver parenchyma was thickened and dense, and its distribution was uneven. A "patchy" echo reduction area along the hepatic vein was visible. A small amount of peritoneal effusion was noted. A plain CT scan showed that the density of the liver parenchyma was unevenly decreased. The hepatic parenchyma in the arterial and venous phases showed a characteristic "map" and uneven enhancement, and the hepatic vein was thin and unclear. Enlarged lymph nodes were observed in the hilar region (Fig. 1C).
After the diagnosis of HSOS was made, the patient was treated with low-molecular-weight heparin. After 50 days of treatment, her symptoms disappeared, the biochemical indexes were normal, the findings on CT imaging were close to normal (Fig. 1D), and she was discharged from the hospital and treated with rivaroxaban tablets. The anticoagulant therapy was stopped after 3 months. Similarly, viral hepatitis (A–E), Epstein–Barr virus, and cytomegalovirus infections and other liver diseases, such as autoimmune liver diseases, were not detected.
Pathological Findings
For the first time for case one, liver histology showed no obvious inflammation or necrosis in most of the liver tissues. Congestion was observed in the hepatic sinuses in a few areas, and patchy and banded inflammatory and necrotic zones were still present. Fibrous embolism and perisinusoidal fibrosis were observed in the central vein (Fig. 2). The second liver pathology showed lesser inflammation and necrosis than the first. Thin fibrous bands were observed around the portal area; a small amount of inflammatory cell infiltration, obvious expansion of the hepatic sinuses, perisinusoidal fibrosis, and fibrotic embolism in the central vein were noted; and CD34 immunohistochemical staining showed that the sinus endothelium was normal (Fig. 3).
A liver biopsy was performed in the fourth month of outpatient follow-up in second case. Pathological features included no obvious inflammation and necrosis, obvious expansion of the hepatic sinuses, increased red blood cells in the sinuses, mild fibrosis around the sinuses, fibrosis deposition in the sinuses, and no embolism in the central vein. Immunohistochemical staining results for CD31 in the sinus endothelium were normal (Fig. 4).