Coagulation‐related inflammatory pseudotumor of liver

Abstract

The laboratory findings were: albumin 2.92 (normal: 3.5–5 g/dl), bilirubin 2.00/0.51 (normal: 0.2–1/0–0.2 mg/dl), Glutamic Oxaloacetic Transaminase 29 (normal: 10–42 IU/l), Glutamic Pyruvic Transaminase 18 (normal: 10–40 IU/l), alkaline phosphatase 85 (normal: 32–92 IU/l), gamma glutamyl transpeptidase 38 (normal: 7–64 IU/l), White blood cell 5390 (normal: 4140–10,520/μl), hemoglobin 11.6 (normal: 13.4–17.2 g/dl), platelets 73,000 (normal: 160,000–370,000/μl), C‐reactive protein 4.62 (normal: <5 mg/l), prothrombin time 13.6/11.4. The cytology results showed normal hepatocyte with normal scattered bile duct cells (Figure 1(F)). [...]cytology analysis demonstrated no malignant cells.

Hepatic inflammatory pseudotumors (HIPT) have been regarded as benign liver lesions but possessing some radiographic findings of liver cancers. Assumed etiologies included trauma, infection, vascular causes, and autoimmunity.¹ Here, we report a case of HIPT related to coagulation abnormality that might be a new etiology.

A 55‐year‐old male complained epigastralgia for days before his scheduled regular visit for chronic hepatitis B (CHB). The symptom was not related to meals. Physical examination revealed mild resistance over epigastric area, few spiderangioma over neck, chest and palmer erythema. Abdominal ultrasound revealed heterogeneous, hypoechoic mass like lesions over left lobe of liver (Figure 1(A)). The tumors were central hypodense with persisting peripheral enhancement on computed tomography (CT) (Figure 1(B)). The laboratory findings were: albumin 2.92 (normal: 3.5–5 g/dl), bilirubin 2.00/0.51 (normal: 0.2–1/0–0.2 mg/dl), Glutamic Oxaloacetic Transaminase 29 (normal: 10–42 IU/l), Glutamic Pyruvic Transaminase 18 (normal: 10–40 IU/l), alkaline phosphatase 85 (normal: 32–92 IU/l), gamma glutamyl transpeptidase 38 (normal: 7–64 IU/l), White blood cell 5390 (normal: 4140–10,520/μl), hemoglobin 11.6 (normal: 13.4–17.2 g/dl), platelets 73,000 (normal: 160,000–370,000/μl), C‐reactive protein 4.62 (normal: <5 mg/l), prothrombin time 13.6/11.4. The tumor markers were alpha‐fetoprotein <3.0 (normal: <9 ng/ml), Carcinoembryonic Antigen 2.82(normal: 0–5 ng/ml). Portal vein thrombosis and thrombosis of superior mesenteric artery progressively developed 1–2 weeks later (Figure 1(C),(D)). Echo‐guided fine‐needle aspiration was done due to high suspicion of liver malignancy. The cytology results showed normal hepatocyte with normal scattered bile duct cells (Figure 1(F)). Warfarin (starting dose 5 mg QD for 1 week, then tapering to 2.5 mg QD for 5 weeks) was prescribed because of no evidence of malignancy and benign thrombosis was highly suspected. His symptoms were relieved a few days later and both the thrombi and tumors resolved on the follow‐up imaging studies. (Figure 1(E),(G)).

1 FIGURE. (A) Ultrasound images revealed hypo‐echoic lesions over left lower liver. (B) Computed tomography (CT) with contrast enhancement 1 day after ultrasound showed heterogeneous, hypodense lesions with persisting peripheral enhancement. (C) Portal vein is clear at initial ultrasound scan. (D) Portal vein thrombosis was noted when ultrasound scan before fine‐needle aspiration procedure. (E) Portal vein is clear again after one course of Coumadin. (F) Cytology examination showed no malignancy like cells, only normal hepatocytes and normal bile duct cells. (G) Only hypoenhance residue observed at follow enhanced CT image

Inflammatory pseudotumors (IPTs) represent a group of benign lesions involving many organs. The most common sites are lung and liver.² Definitive diagnosis of IPTs remains a clinical challenge with its non‐specific clinical symptoms, laboratory, and imaging findings. The most common presentations include right upper quadrant pain, fever, and abdominal distension. Laboratory tests usually revealed normal value except slightly elevated liver enzymes.³ The radiologic features of IPTs may be puzzled with other malignancy of liver.⁴ Appropriate management is another challenge. IPTs may spontaneously regress under conservative treatment in most cases.⁴ Surgery remains to be the consideration for patients having severe symptoms.⁵

Here, our purpose is to report an unusual scenario from coagulation‐related inflammatory pseudotumor, which successfully resolved following administering of anticoagulation therapy. The finding of central hypodense with peripheral enhancement usually suggested hepatocellular carcinoma or cholangiocarcinoma. However, two points provide strong implication on its benign etiology, first of which is rapid development of portal vein thrombosis within 12 days that might be infrequent findings in malignancy. Second, cytology analysis demonstrated no malignant cells. When liver malignancy was suspected, benign lesions should always be considered to avoid unnecessary invasive procedure. Coagulation‐related inflammatory pseudotumor of liver can be treated by anticoagulation therapy and resolved quickly.

CONFLICT OF INTEREST

The authors declare no potential conflict of interest.