Table 1.
Case reports
| Ref. | n | Age (yr) | Gender | Clinical and laboratory findings | Radiology | Localization | Tentative diagnose | Treatment | Histology | Follow up |
| Watanabe et al[32], 2019 | 1 | 70 | Female | Incidental finding | CT unenhanced, low density | Right lobe | HCC | Right partial hepatectomy | Unencapsuled, partly ill defined expansive mass, myofibroblast-, fibroblast cells, inflammatory cells, SMA+, cytokeratins AE1/AE3+; CK7,CK18+, Desmin-, CD68-, IgG4-, ALK- | No recurrence after 7 mo |
| Al-Hussaini et al[24], 2019 | 1 | 8 | Male | FUO, weight loss, hepatomegaly, normal liver enzymes, CRP↑ | MRI: Contrast-enhancing, hyper-intense, well-defined lesion | Right lobe | Infection DD malignancy | Right lobe hepatectomy | Multinucleated giant cells, inflammatory cells, SMA-, ALK-1-, CD-21- CD-23- CD-68+ | No recurrence after 4 mo |
| Lu et al[33], 2018 | 1 | 20 | Male | FUO, jaundice, abdominal pain, CA 19-9↑ | MRI: Multiple lesions, intrahepatic bile duct was significantly dilated | Left lobe | CCC | Biopsy, patient declined operation, PTCD | Spindle cells proliferation and infiltration by mixed inflammatory cells, ALK+, SMA+ | NM |
| Jin et al[5], 2017 | 1 | 42 | Female | Fatigue, fever, pale conjunctivae; Hb↓, Lc↑ | U/S: Hypoechoic mass with unclear border; CT: Low density lesion with mild enhancement | Right lobe | Liver abscess | Right posterior segmentectomy | Chronic inflammatory cells, spindle cells; CD68+, smooth muscle actin, ALK- | No recurrence after 32 mo |
| Mulki et al[22], 2015 | 1 | 50 | Male | Abdominal pain, anorexia, mild fever, hepatomegaly | U/S: 2 hypodense masses, CT: + hepatic vein thrombus | Right lobe | Abscess with septic thrombus | Initial treatment: Biopsy, pigtail, antibiotics, secondary operation | Plasma cells, inflammatory cells, ALK, IgG4+ | No residual disease |
| Obana et al[25], 2015 | 1 | 69 | Male | FUO, CA 19-9 48 ng/mL (n: < 37 ng/mL), Diabetes mellitus II, Dyslipidemia, hypertension | U/S: Irregularly shaped, low-echoic mass; CT: Peripherally enhanced, MRI: T1W, central portion hyperintense | Right lobe Seg VI | CCC/HCC | Partial hepatectomy | Whitish-yellow mass 2 cm in size , inflammatory cell infiltrates, cholesterol cleft granuloma with focal abscess were observed in the central compartment , IgG4 - | NM |
| Guerrero Puente et al[26], 2015 | 1 | 75 | Male | Weight loss, fever, intermittent night sweat, abdominal pain, CRP↑, leukocytosis, cholestasis hypertension, hypercholesterinemia | CT: 8 cm heterogeneous focal lesion, portal branch thrombosis, lymphadenopathy; MRI: T2W isointense, T1W discretely hypointense, cystic–necrotic areas, perilesional edema | Left lobe | Inflammatory disease | CT-guided biopsy followed by antibiotic therapy | Inflammatory pseudotumour, vimentin+, AML+, desmin−, CD68−, ALK−, with no light chain restriction and a low proliferative index (15%) | Partial remission after 1 mo, almost complete remission after 6 mo |
| Onieva-González et al[27], 2015 | 1 | 70 | Male | Low-grade fever, asthenia, weight loss and oligoarthritis, lung tuberculosis, diabetes, gouty arthritis, renal lithiasis and colon diverticulitis | CT: Thickened gallbladder wall, poorly-defined hypodense lesion of 17 mm in the gallbladder bed, U/S: Nodule; MRI: Hypointense in T2 sequences; PET: No metabolism | Seg. V | Liver abscess | Antibiotic therapy, after 4 mo later fine needle biopsy followed by laparoscopic biopsy and cholecystectomy with the lesion in the gallbladder bed | Lymphoid infiltration without malignancy signs, compatible with an inflammatory pseudotumour | NM |
| Chang et al[50], 2014 | 1 | 38 | Male | Fatigue, abdominal distension and weight loss, jaundice, hepatomegaly, bilateral ankle edema | U/S: Complex mass; CT: Large cystic or necrotic mass; MRI: T2W: Cystic portion hyperintense to liver parenchyma, surrounded by a hypointense rim. T2W: Hyperintense compared to liver parenchyma | Bilateral | N/A | Ultrasound-guided and open biopsy, followed by resection | Cellular spindle-cell proliferation with heavy inflammatory infiltrate consisting primarily of plasma cells and lymphocytes | Recurrence |
| You et al[35], 2014 | 1 | 43 | Male | Chronic cough, right-upper-quadrant pain, anorexia for 3 mo, leukozytosis, elevated platelet count | U/S: 18 cm mass with slightly echogenic center; MRI: Large mass with central dark area and some peripheral spokes; CT: Mass, 20 cm × 17 cm × 18 cm, with extensions into the medial segment of the left hepatic lobe, hypervascular nodular area with enhanced density at the periphery and hypoattenuating density centrally | Right lobe | Fibrolamellar hepatocellular carcinoma or CCC | Percutaneous needle core biopsy > NM | Bland spindle cell proliferation amidst small mature lymphocytes, numerous plasma cells, histiocytes, and few neutrophils. Spindle cells showed a storiform pattern with large areas of necrosis; cytokeratin (CAM 5.2) -, cytokeratin 5/6 -, actin-, CD34-, CD117-, DOG-1-, desmin-, CD68-, S100-, Pan-melanoma-. Spindle cells were negative for CD21, CD23, CD35, ALK-1. Epstein-Barr virus-encoded small RNA in situ hybridization (EBER) showed large numbers of Epstein-Barr virus positive cells, including some spindle cells | NM |
| Durmus et al[36], 2014 | 1 | 67 | Female | Moderate diffuse abdominal tenderness, focus over epigastrium | U/S: Heterogeneous hypoechogenic tumor; CT: Contrast enhancing mass with irregular confluent non-enhancing areas in the center with a hypodense late enhancing rim and no wash-out in the late phase, MRI: In T1W hypointense borders, well defined without fatty components. T2W showed a heterogeneous slightly hyperintense lesion with an ill-defined hyperintense rim | Segment IV | Malignancy | Left hemihepatectomy with partial excision of the adherent abdominal wall and diaphragm | Tumor with fibrosis and partially necrotic tissue infiltrated by inflammatory cells, predominantly plasma cells, and also pigmented macrophages and granulocytes | NM |
| Wong et al[37], 2013 | 1 | 56 | Female | Right-upper-quadrant abdominal pain, renal transplant | U/S: 2 cm × 2.4 cm mass in the left hepatic lobe with associated biliary duct dilatation, MRI: atrophic left liver lobe with multiple strictures and distal duct dilatation. 2-cm lesion at the origin of the left hepatic duct | Left lobe | Primary hepatic tumor | Surgical resection | Dense hyalinised stroma and scattered, histiocytic and lymphocytic inflammation | NM |
| Kruth et al[38], 2012 | 1 | NM | NM | FUO CRP↑ | Gastroscopy, CT lung and abdomen, MRI: 3.3 cm lesion | Seg. VI | Adenoma, focal nodular hyperplasia or HCC | Surgical resection | NM | No recurrence after 1 yr |
| Chablé-Montero et al[39], 2012 | 1 | 23 | Female | Fever, diaphoresis, right-upper-quadrant abdominal pain | U/S and CT: Heterogenous rounded hepatic lesion of 7 cm in greatest dimension | Right lobe | Pyogenic hepatic abscess | Antibiotics, later right hepatic lobectomy | Grossly a non-encapsulated but well demarcated hepatic tumor with central necrosis of 11 cm in greatest dimension; microscopically: Spindle myofibroblastic cells arranged in fascicles. Leukocytes, lymphocytes, plasma cells, SMA+ | NM |
| Kayashima et al[30], 2011 | 1 | 57 | Female | Asymptomaticlaparoscopic calculous cholecystectomy 3 yr ago | U/S: 3 liver masses, CT: 1 intra- and 2 extrahepatic lesions; MRI: three high‐intensity lesions; PET: Abnormal accumulation in all lesions | Right lobe | CCC | Surgical resection (tiny black‐colored nodules within the abdominal cavity and spilled gallstones) | Inflammatory granuloma located at liver parenchyma | No recurrence after 6 mo |
| Huang et al[40], 2012 | 1 | 30 | Male | Right upper abdominal pain; CEA↑; 2 yr after renal transplant | CT: Low-density mass, about 30 mm in diameter, well defined, and with peripheral enhancement | Caudate lobe | HCC or liver abscess | Hepatic caudate lobectomy with complete resection of the mass | Mixture of spindle-shaped myofibroblastic cells and chronic inflammatory cells; SMA+ | NM |
| Beauchamp et al[41], 2011 | 1 | 74 | Female | FUO | CT: Numerous hypodense lesions scattered throughout the liver | NM | NM | Liver biopsy | IMT | NM |
| Al-Jabri et al[29], 2010 | 1 | 69 | Male | Right upper quadrant pain, nausea, vomiting, recent weight loss, rheumatoid arthritis and bronchiectasis, CRP↑, cholestasis (normal Bili) | U/S: Ill-defined area, CT: multiple low attenuation lesions | Right lobe | Cholecystitis, malignancy | Fine needle biopsy | Presence of benign hepatocytes, acellular debris and a mixture of acute and chronic inflammatory cells | No recurrence after 3 mo |
| Salakos et al[43], 2010 | 1 | 10 | Male | Fever, weight loss, fatigue, tachycardia, hepatomegaly, leukocytosis, platelet count ↑ | U/S: Space occupying lesion in the liver; CT: Large lesion with sold and cystic parts and heterogenous enhancement | Right and left lobe | NM | Biopsy followed by conservative treatment (ceftriaxone, clindamycin, NSAR) | Hyperplastic cholangioles, myofibroblasts and fibroblasts, infiltrate of lymphocytes, eosinophils and neutrophils; ALK+ | Partial response after 2 mo, complete response |
| Ueda et al[45], 2009 | 1 | 79 | Male | Leukocytosis | U/S: Hypoechoic lesion, 3 cm in diameter, with several stones. CT: Low density area in segment V; MRI: Lesion of slightly low signal intensity; MRCP: Lesion of moderate-to-high signal intensity on T2W | Right lobe | Inflammation due to cholangitis with intrahepatic bile duct stones | 1. ERCP: Sphincterotomy, antibiotics because of common bile duct stone; 2. Relapse of symptoms 4 wk later > resection | Grossly gray, fibrotic, solid tumor, intrahepatic bile duct stones. Proliferation of diffuse myofibroblastic and mesenchymal cells in a mixed myxoedematous, dense fibrotic stroma, with many small vessels and marked infiltration by various acute and chronic inflammatory cells | No recurrence after 18 mo |
| Sürer et al[7], 2009 | 1 | 48 | Female | Weakness, fever, weight loss, right upper abdominal pain, Lc-, neutrophil 75.3%, liver function normal | U/S: Single hypoechoic lesion in right lobe | Right lobe | NM | Resection | No capsule, light brown, no necrosis, spindle cells, granulation-tissue type vessels, chronic inflammatory cells on loose, edemateous, myxoid stroma, CD 38+, SMA+, ALK+, desmin, EMA- | 2 yr no recurrence after 2 yr |
| Manolaki et al[47], 2009 | 1 | 9 | Female | Fever, mild anorexia, intermittent epigastric pain | U/S: Hypoechoic lesion, lymph node at porta hepatis, CT: hypodense space-occupying lesion | Left lobe | NM | Biopsy, secondary left lateral segmentectomy with lymph node excision | Pale and firm lesion (3.5 cm × 2.5 cm × 3.0 cm) with whitish solid infiltrations extending to the capsule of the liver. Proliferation of spindle-shaped cells arranged in short fascicles with an ill-defined mark. Inflammatory cells, predominantly lymphocytes, plasma cells and eosinophils; vimentin+, SMA+, CD68+,TBC+ | No recurrence after 3 yr |
CT: Computed tomography; MRI: Magnetic resonance imaging; FUO: Fever unknown origin; CRP: C-reactive protein; CCC: Cholangiocarcinoma; HCC: Hepatocellular carcinoma; PTCD: Percutaneous transhepatic cholangio drainage; NM: Not mentioned; U/S: Ultrasonography; Hb: Haemoglobin; LC: Leukocytes; TC: Thrombocytes; T1W: T1-Weighted; T2W: T2-Weighted; Chron Hep B: Chronic Hepatitis B; Seg: Segment; ↑: Increase; ↓: Decrease; WBC : Wight blood cells; SMA: Smooth muscle actin; ERCP: Endoscopic retrograde cholangiopancreatography.