Korean J Gastroenterol.  2022 Jan;79(1):41-44. 10.4166/kjg.2021.146.

Inflammatory Myofibroblastic Tumor Misdiagnosed as Intrahepatic Cholangiocarcinoma

• Park JW
• Han SH
• Kim DK

• KMID: 2525522
• DOI: http://doi.org/10.4166/kjg.2021.146

Abstract

An inflammatory myofibroblastic tumor (IMT) is a rare tumor that is currently classified as an intermediate cancer according to the World Health Organization classification system. The pathophysiology of its occurrence is still unknown. Imaging tests, such as CT or MRI, can be helpful in diagnosis, but the final diagnosis is confirmed by a pathological examination through a biopsy and immunohistochemistry stain. The patient, in this case, presented an asymptomatic intrahepatic mass discovered incidentally on an imaging examination. Initially, intrahepatic cholangiocarcinoma was suspected, but she was finally diagnosed with IMT through a histological examination after a liver resection.

Keyword

Inflammatory myofibroblastic tumor; Cholangiocarcinoma; Liver; Hepatectomy

Figure

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  Fig. 1 Abdominal sonography shows 5.6 cm sized heterogeneous hyperechoic mass in S6.

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  Fig. 2 Computed tomography scan, 5.8 cm sized irregular hypodense mass in the (A) pre-phase, (B) arterial phase, and (C) portal phase.

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  Fig. 3 Magnetic resonance imaging. An approximately 5.8 cm irregular-shaped mass in the S6 subcapsular area. (A) T1 weighted image shows low signal intensity, (B) T2 weighted image shows slight high signal intensity, (C) diffusion-weighted image shows restricted diffusion.

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  Fig. 4 Positron emission tomography and computed tomography shows ill-defined, uneven hypermetabolic lesion (SUVmax 5.17) in segment 6 of liver.

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  Fig. 5 Spindle cell proliferative lesion with inflammatory cell infiltration and necrosis (hematoxylin and eosin stain, ×200).

Reference

1. Fletcher CDM, Unni KK, Mertens F. Pathology and genetics of tumours of soft tissue and bone. IARC Press;Lyon:

2. Poh CF, Priddy RW, Dahlman DM. 2005; Intramandibular inflammatory myofibroblastic tumor--a true neoplasm or reactive lesion? Oral Surg Oral Med Oral Pathol Oral Radiol Endod. 100:460–466. DOI: 10.1016/j.tripleo.2004.07.005. PMID: 16182167.

3. Gómez-Román JJ, Sánchez-Velasco P, Ocejo-Vinyals G, Hernández-Nieto E, Leyva-Cobián F, Val-Bernal JF. 2001; Human herpesvirus-8 genes are expressed in pulmonary inflammatory myofibroblastic tumor (inflammatory pseudotumor). Am J Surg Pathol. 25:624–629. DOI: 10.1097/00000478-200105000-00009. PMID: 11342774.

4. Coffin CM, Patel A, Perkins S, Elenitoba-Johnson KS, Perlman E, Griffin CA. 2001; ALK1 and p80 expression and chromosomal rearrangements involving 2p23 in inflammatory myofibroblastic tumor. Mod Pathol. 14:569–576. DOI: 10.1038/modpathol.3880352. PMID: 11406658.

5. Lewis JT, Gaffney RL, Casey MB, Farrell MA, Morice WG, Macon WR. 2003; Inflammatory pseudotumor of the spleen associated with a clonal Epstein-Barr virus genome. Case report and review of the literature. Am J Clin Pathol. 120:56–61. DOI: 10.1309/BUWNMG5RV4D09YYH. PMID: 12866373.

6. Sürer E, Bozova S, Gökhan GA, Gürkan A, Elpek GO. 2009; Inflammatory myofibroblastic tumor of the liver: a case report. Turk J Gastroenterol. 20:129–134. PMID: 19530046.

7. Park JY, Choi MS, Lim YS, et al. 2014; Clinical features, image findings, and prognosis of inflammatory pseudotumor of the liver: a multicenter experience of 45 cases. Gut Liver. 8:58–63. DOI: 10.5009/gnl.2014.8.1.58. PMID: 24516702. PMCID: PMC3916688.

8. Ueda M, Yukihide Y, Ogawa K, et al. 2003; A case of inflammatory pseudotumor of the liver hilum successfully treated with aggressive hepatectomy. J Pediatr Surg. 38:E9–E11. DOI: 10.1016/j.jpedsurg.2003.08.004. PMID: 14614735.

9. Nagarajan S, Jayabose S, McBride W, et al. 2013; Inflammatory myofibroblastic tumor of the liver in children. J Pediatr Gastroenterol Nutr. 57:277–280. DOI: 10.1097/MPG.0b013e31829e0b3b. PMID: 23974058.

10. Ogawa T, Yokoi H, Kawarada Y. 1998; A case of inflammatory pseudotumor of the liver causing elevated serum CA19-9 levels. Am J Gastroenterol. 93:2551–2555. DOI: 10.1111/j.1572-0241.1998.00717.x. PMID: 9860425.

11. Miettinen M, Sobin LH, Sarlomo-Rikala M. 2000; Immunohistochemical spectrum of GISTs at different sites and their differential diagnosis with a reference to CD117 (KIT). Mod Pathol. 13:1134–1142. DOI: 10.1038/modpathol.3880210. PMID: 11048809.

12. Goldsmith PJ, Loganathan A, Jacob M, et al. 2009; Inflammatory pseudotumours of the liver: a spectrum of presentation and management options. Eur J Surg Oncol. 35:1295–1298. DOI: 10.1016/j.ejso.2009.04.003. PMID: 19515527.

13. Cook JR, Dehner LP, Collins MH, et al. 2001; Anaplastic lymphoma kinase (ALK) expression in the inflammatory myofibroblastic tumor: a comparative immunohistochemical study. Am J Surg Pathol. 25:1364–1371. DOI: 10.1097/00000478-200111000-00003. PMID: 11684952.

14. Pack GT, Baker HW. 1953; Total right hepatic lobectomy; report of a case. Ann Surg. 138:253–258. DOI: 10.1097/00000658-195308000-00012. PMID: 13066016. PMCID: PMC1609266.

15. Tang L, Lai EC, Cong WM, et al. 2010; Inflammatory myofibroblastic tumor of the liver: a cohort study. World J Surg. 34:309–313. DOI: 10.1007/s00268-009-0330-x. PMID: 20033408.

16. Yamaguchi J, Sakamoto Y, Sano T, Shimada K, Kosuge T. 2007; Spontaneous regression of inflammatory pseudotumor of the liver: report of three cases. Surg Today. 37:525–529. DOI: 10.1007/s00595-006-3433-0. PMID: 17522777.

17. Butrynski JE, D'Adamo DR, Hornick JL, et al. 2010; Crizotinib in ALK-rearranged inflammatory myofibroblastic tumor. N Engl J Med. 363:1727–1733. DOI: 10.1056/NEJMoa1007056. PMID: 20979472. PMCID: PMC3014292.

18. Xiang J, Liu X, Wu S, Lv Y, Wang H. 2012; Multiple inflammatory myofibroblastic tumor of the duodenum: case report and literature review. J Gastrointest Surg. 16:1442–1445. DOI: 10.1007/s11605-012-1883-9. PMID: 22528570.

19. Koea JB, Broadhurst GW, Rodgers MS, McCall JL. 2003; Inflammatory pseudotumor of the liver: demographics, diagnosis, and the case for nonoperative management. J Am Coll Surg. 196:226–235. DOI: 10.1016/S1072-7515(02)01495-3. PMID: 12595051.

20. Bertocchini A, Lo Zupone C, Callea F, et al. 2011; Unresectable multifocal omental and peritoneal inflammatory myofibroblastic tumor in a child: revisiting the role of adjuvant therapy. J Pediatr Surg. 46:e17–e21. DOI: 10.1016/j.jpedsurg.2011.01.007. PMID: 21496520.

21. Johnson K, Notrica DM, Carpentieri D, Jaroszewski D, Henry MM. 2013; Successful treatment of recurrent pediatric inflammatory myofibroblastic tumor in a single patient with a novel chemotherapeutic regimen containing celecoxib. J Pediatr Hematol Oncol. 35:414–416. DOI: 10.1097/MPH.0b013e3182915cef. PMID: 23669730.

22. Kovach SJ, Fischer AC, Katzman PJ, et al. 2006; Inflammatory myofibroblastic tumors. J Surg Oncol. 94:385–391. DOI: 10.1002/jso.20516. PMID: 16967468.