An interesting case of inflammatory myofibroblastic tumor presenting as cholangiocarcinoma

Karimi, Mehrdad; Tizmaghz, Adnan; Shabestanipour, Ghazaal

doi:10.1016/j.ijscr.2018.03.040

Cited by 7 publications

(8 citation statements)

References 11 publications

Supporting

Mentioning

Contrasting

Order By: Relevance

“…In general, hepatic lesions are well-circumscribed with variable enhancement patterns, usually heterogenous or peripheral in the arterial phase. [4][5][6][7] Brush cytology obtained by endoscopic retrograde cholangiopancreatography (ERCP) is frequently inconclusive, and therefore diagnosis is usually histological. Direct cholangioscopy with biopsy is an option.…”

Section: Discussionmentioning

confidence: 99%

Inflammatory Myofibroblastic Tumour of the Common Bile Duct

et al. 2022

View full text Add to dashboard Cite

Inflammatory myofibroblastic tumour is a rare entity of indeterminate biological potential with a reduced tendency for recurrence and metastasis. Although it can arise from multiple organs, the bile duct is a very rare site of origin. We report the case of a 75-year-old asymptomatic male with elevated gamma-glutamyl transferase [1575 U/L (12 - 64 U/L)] and alkaline phosphatase [271 U/L (40 - 150 U/L)]. Computed tomography showed a 17 mm hypervascular lesion in the confluence of the right and left hepatic ducts, with bile duct ectasia and right liver lobe atrophy. The patient was initially managed as having a Klatskin tumour and underwent right hepatectomy. Histology showed a spindle cell proliferation with an inflammatory infiltrate of lymphocytes, plasma cells and collagen-rich stroma, consistent with an inflammatory myofibroblastic tumour. He was discharged 30 days after admission, and nine months later remains asymptomatic. His liver function tests have normalized and follow-up tests are unremarkable.

show abstract

Section: Discussionmentioning

confidence: 99%

Inflammatory Myofibroblastic Tumour of the Common Bile Duct

et al. 2022

View full text Add to dashboard Cite

show abstract

“…Biliary IMT is rarely reported and usually occurs in adults. Heretofore, there are only 16 documented cases of biliary IMT, of which one is published in Russian (Table 1 )[ 21 - 26 ]. Among these studies, the age of onset varies from 6 to 70 years, and 5 of the 16 cases were male.…”

Section: Discussionmentioning

confidence: 99%

“…Further imaging examination such as computed tomography (CT) and MRI may also be required. Since intrahepatic IMT usually has similar features upon CT and MRI with cholangiocarcinoma, a differential diagnosis by image examination is unreliable[ 26 - 29 ]. Clinicians are trying more discerning methodologies.…”

Section: Discussionmentioning

confidence: 99%

Infant biliary cirrhosis secondary to a biliary inflammatory myofibroblastic tumor: A case report and review of literature

Yuan¹,

Shu²,

Zhang³

et al. 2022

WJCC

View full text Add to dashboard Cite

BACKGROUND A biliary inflammatory myofibroblastic tumor (IMT) is a rare type of mesenchymoma that, although it has a broad age spectrum, usually occurs in adults. Diagnosis is difficult because biliary IMTs often exhibit nonspecific clinical symptoms and imaging features, resulting in delayed or inappropriate treatment. Although most IMTs are benign, some show malignant properties such as infiltration, recurrence, and metastasis. CASE SUMMARY Here, we retrospectively describe a 10-month-old infant who was admitted to our hospital due to stubborn jaundice. The patient responded poorly to routine medical treatment and his clinical manifestations and laboratory tests lacked specificity, so we turned to repeated ultrasound scans and other imaging examinations. As both hepatosplenic ultrasonography and diffusion-weighted magnetic resonance imaging demonstrated a space-occupying lesion, an exploratory laparotomy was performed. The final diagnosis made over two mo after the disease onset was infant biliary cirrhosis caused by a biliary IMT, which partially infiltrated into the liver. This infant is the youngest case of biliary IMTs that has been reported till now. The patient underwent an incomplete resection of the mass and Kasai Portoenterostomy. However, because of cirrhosis, he also received a paternal liver transplant. Since some IMTs show malignant properties, we proceeded with a three-year of follow-up; however, no recurrence or metastasis has been noted. CONCLUSION Neoplastic disease such as IMTs should be considered when routine medical treatment of obstructive jaundice is not successful. Observation of dynamic imaging changes is helpful for diagnosis. Periodic follow-up is necessary for IMTs.

show abstract

“…Despite reported successful treatment, complete reduction and tumor regression using a conservative approach (corticosteroids, NSAIDs, antibiotics, chemotherapy), tumors identified as IMTs by histopathology are locally progressive and often need surgical resection, especially if medical therapy is not effective[ 36 , 37 ]. Although there are no approved recommendations for operative hepatobiliary IMTs treatment, the literature suggests that patients with resectable IMTs should be managed with radical surgical resection when it is anatomically and physiologically feasible[ 5 , 35 - 37 ].…”

Section: Discussionmentioning

confidence: 99%

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

Strainiene¹,

Sedleckaite²,

Jarašūnas³

et al. 2021

WJCC

View full text Add to dashboard Cite

BACKGROUND The inflammatory myofibroblastic tumor (IMT) is a rare, idiopathic, usually benign, mass-forming disease with myofibroblastic proliferation and a varying amount of inflammatory cells. Although it can affect various organs, the biliary tract is a rare localization of primary IMT, clinically, endoscopically and radiologically imitating cholangiocarcinoma. The treatment options are based only on clinical practice experience. CASE SUMMARY A 70-year-old woman was referred to our center due to progressive fatigue, weight loss, abdominal pain, night sweats, and elevated liver enzymes. Magnetic resonance cholangiopancreatography and endoscopic retrograde cholangiopancreatography (ERCP) revealed proximal common hepatic duct and hilar biliary strictures extending bilaterally to lobular bile ducts. Although initial clinical, endoscopic and radiological signs were typical for hilar cholangiocarcinoma, histological examination showed no signs of malignancy. In total, 8 biopsies using different approaches were performed (several biopsies from dominant stricture during ERCP and direct cholangioscopy; ultrasound-guided liver biopsy; diagnostic laparoscopy with liver and lymph node biopsies). Histological examination revealed signs of IMT, and the final diagnosis of biliary IMT was stated. Although IMT is usually a benign disease, in our case, it was complicated. All pharmacological treatment measures were ineffective. The patient still needs permanent stenting, suffers from recurrent infections and mechanical jaundice. Despite that, the patient already survived 24 mo. CONCLUSION IMT presenting with hilar biliary strictures is a unique diagnostic and clinical challenge as it is indistinguishable from cholangiocarcinoma, and there are no evidence-based treatment options. Our goal is to increase the understanding of this rare disease and its possible course.

show abstract

An interesting case of inflammatory myofibroblastic tumor presenting as cholangiocarcinoma

Cited by 7 publications

References 11 publications

Inflammatory Myofibroblastic Tumour of the Common Bile Duct

Inflammatory Myofibroblastic Tumour of the Common Bile Duct

Infant biliary cirrhosis secondary to a biliary inflammatory myofibroblastic tumor: A case report and review of literature

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

Contact Info

Product

Resources

About