Rare Inflammatory Myofibroblastic Tumor of Pancreas in a Child With Cholestasis Presentati
Background: Inflammatory myofibroblastic tumors (IMTs) are rare with unknown etiology. As pancreas involvement is rare in IMTs, here, we report a case of a girl with IMT, referred to our hospital.
Case presentation: A 4-year-old girl presented with chief complaints of generalized itching and jaundice. Abdominopelvic computed tomography (CT) scans with contrast showed a homogeneous isodense mass lesion in the head of the pancreas with a compressive effect on the distal part of the common bile duct (CBD). Dilatation of intrahepatic bile ducts and CBD (8mm) was observed. Magnetic resonance cholangiopancreatography (MRCP) examination showed dilated gallbladder without stones and intrahepatic/extrahepatic bile ducts. The CBD was dilated and a mass was found in the head of the pancreas. Immune-histochemical studies revealed spindle myoﬁbroblastic tissues with lymphoplasmacyte and eosinophil inﬁltration. All of them were compatible with pancreatic IMTs. The surgery improved the symptoms.
Conclusions: The IMTs of the pancreas can have symptoms like pancreatic cancer. The careful evaluation by imaging and pathology is recommended.
2. Nonaka D, Birbe R, Rosai J. So‐called inflammatory myofibroblastic tumour: a proliferative lesion of fibroblastic reticulum cells? Histopathology. 2005;46(6):604-13.
3. Gómez-Román JJ, Sánchez-Velasco P, Ocejo-Vinyals G, Hernández-Nieto E, Leyva-Cobián F, Val-Bernal JF. Human herpesvirus-8 genes are expressed in pulmonary inflammatory myofibroblastic tumor (inflammatory pseudotumor). The American journal of surgical pathology. 2001;25(5):624-9.
4. Lewis JT, Gaffney RL, Casey MB, Farrell MA, Morice WG, Macon WR. Inflammatory pseudotumor of the spleen associated with a clonal Epstein-Barr virus genome: case report and review of the literature. American journal of clinical pathology. 2003;120(1):56-61.
5. Jones EC, Clement PB, Young RH. Inflammatory pseudotumor of the urinary bladder. A clinicopathological, immunohistochemical, ultrastructural, and flow cytometric study of 13 cases. The American journal of surgical pathology. 1993;17(3):264-74.
6. Gleason BC, Hornick JL. Inflammatory myofibroblastic tumours: where are we now? Journal of clinical pathology. 2008;61(4):428-37.
7. Yamamoto H, Oda Y, Saito T, Sakamoto A, Miyajima K, Tamiya S, et al. p53 Mutation and MDM2 amplification in inflammatory myofibroblastic tumours. Histopathology. 2003;42(5):431-9.
8. Yamamoto H, Yoshida A, Taguchi K, Kohashi K, Hatanaka Y, Yamashita A, et al. ALK, ROS 1 and NTRK 3 gene rearrangements in inflammatory myofibroblastic tumours. Histopathology. 2016;69(1):72-83.
9. Coffin CM, Hornick JL, Fletcher CD. Inflammatory myofibroblastic tumor: comparison of clinicopathologic, histologic, and immunohistochemical features including ALK expression in atypical and aggressive cases. The American journal of surgical pathology. 2007;31(4):509-20.
10. Yamamoto H, Watanabe K, Nagata M, Tasaki K, Honda I, Watanabe S, et al. Inflammatory myofibroblastic tumor (IMT) of the pancreas. Journal of hepato-biliary-pancreatic surgery. 2002;9(1):116-9.
11. Nikitakis N, Brooks J, Frankel B, Papadimitriou J, Sauk J. Inflammatory myofibroblastic tumor of the oral cavity: review of the literature and presentation of an ALK-positive case. Oral Surgery, Oral Medicine, Oral Pathology, Oral Radiology, and Endodontology. 2004;98(2):197-8.
12. Battal M, Kartal K, Tuncel D, Bostanci O. Inflammatory myofibroblastic pancreas tumor: a case report. Clinical case reports. 2016;4(12):1122.
13. Pungpapong S, Geiger XJ, Raimondo M. Inflammatory myofibroblastic tumor presenting as a pancreatic mass: a case report and review of the literature. Jop. 2004;5(5):360-7.
14. Walsh SV, Evangelista F, Khettry U. Inflammatory myofibroblastic tumor of the pancreaticobiliary region: morphologic and immunocytochemical study of three cases. The American journal of surgical pathology. 1998;22(4):412-8.
15. Tomazic A, Gvardijancic D, Maucec J, Homan M. Inflammatory myofibroblastic tumor of the pancreatic head–a case report of a 6 months old child and review of the literature. Radiology and oncology. 2015;49(3):265-70.
16. Panda D, Mukhopadhyay D, Datta C, Chattopadhyay BK, Chatterjee U, Shinde R. Inflammatory myofibroblastic tumor arising in the pancreatic head: a rare case report. Indian Journal of Surgery. 2015;77(6):538-40.
17. Dulundu E, Sugawara Y, Makuuchi M. Inflammatory myofibroblastic tumor of the pancreas–a case report. Biosci Trends. 2007;1:167-9.
18. Qanadli SD, d'Anthouard F, Cugnec J-P, Frija G. Plasma cell granuloma of the pancreas: CT findings. Journal of computer assisted tomography. 1997;21(5):735,6.
19. Coffin CM, Watterson J, Priest JR, Dehner LP. Extrapulmonary inflammatory myofibroblastic tumor (inflammatory pseudotumor). A clinicopathologic and immunohistochemical study of 84 cases. The American journal of surgical pathology. 1995;19(8):859-72.
20. Liu H-K, Lin Y-C, Yeh M-L, Chen Y-S, Su Y-T, Tsai C-C. Inflammatory myofibroblastic tumors of the pancreas in children: A case report and literature review. Medicine. 2017;96(2).
21. Meis JM, Enzinger FM. Inflammatory fibrosarcoma of the mesentery and retroperitoneum. A tumor closely simulating inflammatory pseudotumor. The American journal of surgical pathology. 1991;15(12):1146-56.
22. Kim JY, Song JS, Park H, Byun JH, Song K-B, Kim K-p, et al. Primary mesenchymal tumors of the pancreas: single-center experience over 16 years. Pancreas. 2014;43(6):959-68.
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