Complicated course of biliary inflammatory myofibroblastic tumor mimicking hilar cholangiocarcinoma: A case report and literature review

Table 1 Main laboratory findings

	Value	Normal range
White blood cell count	10.04	4.0 x 109/L-9.8 x 109/L
Hemoglobin	125	128-160 g/L
Platelet count	328	130 x 109/L-400 x 109/L
C reactive protein	55.4	≤ 5 mg/L
Aspartate aminotransferase	76	< 40 U/L
Alanine aminotransferase	135	< 40 U/L
γ- glutamyl transferase	344	≤ 36 U/L
Alkaline phosphatase	376	40-150 U/L
Total bilirubin	30	< 21 μmol
Albumin	36.2	36-52 g/L
K+	4.3	3.8-5.3 mmol/L
Na+	132	134-145 mmol/L
SPA	95	70%-130%
INR by Owren	1.02	0.90-1.19
Creatinine	51	62-11 µmol/L
Urea	3.7	2.5-7.5 mmol/L
CA 19-9	111.98	< 37 kU/L
CEA	1.0	< 5 mkg/L
AFP	1.30	0.5-5.5 kU/L
HBsAg	0.11 negative	Negative s/co
Anti-HCV	0.11 negative	Negative
AMA: M2	Negative	Negative
ANA	Negative (1:40)	< 1:40
Immunoglobulin G4	0.653	0.08-1.40 g/L

INR: International normalized ratio; SPA: Stago prothrombin assay; CA 19-9: carbohydrate antigen 19-9; CEA: Carcinoembryonic antigen; AFP: Alpha-fetoprotein; HBsAg: Hepatitis B surface antigen; HCV: Hepatitis C virus; AMA: Antimitochondrial; ANA: Antinuclear.

Table 2 Cases of biliary inflammatory myofibroblastic tumors

Ref.	Age /gender	Location/extension	Treatment	Follow- up	Outcome
Haith et al[46]	6/M	Distal CBD	PD and celecoxib	5 mo	NR
Stamatakis et al[47]	13/F	Proximal CBD, cystic duct, CHD	Extrahepatic BD excision	21 mo	NR
Ikeda et al[48]	43/M	Proximal CBD/ intrahepatic ducts, CHD, gall bladder, lymph nodes	Surgery	7 mo	Lung metastasis
Fukushima et al[49]	58/F	Mid-CBD/pancreas and lymph nodes	PD	-	NR
Walsh et al[50]	50/M	Proximal CBD	PD	19 yr	Metastasis
Sobesky et al[51]	51/F	Distal CBD	PD	2 yr	NR
Venkataraman et al[52]	17–52/3 M, 1 F	1 liver mass; 1 liver mass and periportal infiltration; 2 periportal infiltration	-	-	-
Büyükyavuz et al[53]	8/F	Hepatic hilar	Surgery, antibiotics	-	-
Lopez-Tomassetti Fernandez et al[54]	55/F	Distal CBD	PD	4 yr	R
Martín Malagón et al[55]	51/F	Distal CBD	PD	-	-
Kim et al[56]	63/F	Hilar bile duct	Hepatectomy, caudate lobectomy	5 mo	R
Sekaran et al[57]	17/F	Left hepatic, CHD, proximal CBD	Left hepatectomy	6 wk	NR
Abu-Wasel et al[58]	55/F	Distal CBD	Extrahepatic BD excision	14 mo	NR
Vasiliadis et al[2]	70/F	Mid-CBD	Extrahepatic BD excision	8 mo	NR
D’Cunha et al[59]	12/F	Distal CBD	Debulking, corticosteroids	-	NR
Verma et al[1]	24/F	Mid-CBD	Extrahepatic BD excision	12 mo	NR
Karimi et al[36]	12/F	Hepatic duct bifurcation	CBD resection	-	R
Present case	70/F	Proximal CBD, CHD	Corticosteroids, chemotherapy	24 mo	Disease progression

M: Male; F: Female; CBD: Common bile duct; PD: Pancreatoduodenectomy; NR: No recurrence; CHD: Common hepatic duct; BD: Bile duct; US: Ultrasound; R: Recurrence.

Table 3 Possible treatment options for inflammatory myofibroblastic tumors

Possible treatment options for IMTs
High-dose steroids
Low dose steroids
Non-steroidal anti-inflammatory drugs (e.g., celecoxib)
Vinblastine and methotrexate
Anaplastic lymphoma kinase inhibitors
Ifosfamide-based chemotherapy
Vinorelbine and low-dose cyclophosphamide
Vincristine and actinomycin-D
Cyclosporine, azathioprine
Radical surgical treatment (when anatomically and physiologically feasible)

IMTs: Inflammatory myofibroblastic tumors.