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CASE REPORT Table of Contents   
Year : 2009  |  Volume : 52  |  Issue : 2  |  Page : 210-212
Inflammatory pseudotumor of the liver: Two case reports and a review of the literature


1 Department of Pathology, Shiraz University of Medical Sciences, Shiraz, Iran
2 Department of Surgery, Shiraz University of Medical Sciences, Shiraz, Iran
3 Department of Radiology, Shiraz University of Medical Sciences, Shiraz, Iran

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   Abstract 

Rare cases of inflammatory pseudotumor (IPT) in two adolescents are reported. Both of them presented with significant weight loss and were operated upon with the impression of liver abscess and malignant liver tumor.
These two cases are reported to emphasize IPT of the liver as a differential diagnosis of hepatic masses in children because recognition of this condition before operation can avoid unnecessary surgery.

Keywords: Adolescence, inflammatory pseudotumor, liver

How to cite this article:
Geramizadeh B, Tahamtan MR, Bahador A, Sefidbakht S, Modjalal M, Nabai S, Hosseini SA. Inflammatory pseudotumor of the liver: Two case reports and a review of the literature. Indian J Pathol Microbiol 2009;52:210-2

How to cite this URL:
Geramizadeh B, Tahamtan MR, Bahador A, Sefidbakht S, Modjalal M, Nabai S, Hosseini SA. Inflammatory pseudotumor of the liver: Two case reports and a review of the literature. Indian J Pathol Microbiol [serial online] 2009 [cited 2020 Jul 12];52:210-2. Available from: http://www.ijpmonline.org/text.asp?2009/52/2/210/48920



   Introduction Top


Inflammatory pseudotumor (IPT) occurs in many organs. Hepatic IPT, also known as inflammatory myofibroblastic tumor, is a rare benign lesion, which is increasingly recognized because of improvements in imaging techniques [1] and is an important differential diagnosis in patients with liver space-occupying lesions. [2] Imaging is usually not reliable for detecting their character and surgical biopsy is needed for final diagnosis. [3]

This tumor is also uncommon in children and has rarely been reported in this age group. [4],[5]

Herein we report the clinical, paraclinical and pathological findings in two children with the final diagnosis of IPT.


   Case Reports Top


Case 1

A 14-year-old boy presented with chills, fever, anorexia and more than 8 Kg weight loss since 2 months before admission.

Laboratory investigation showed a normal liver function test (LFT) and leuckocytosis (22,700/L).

An abdominal computed tomography (CT) scan showed a well-defined heterogeneous mass measuring 5.9 5.4cm in the lower part of the left liver lobe with central areas of necrosis and a slightly hyperdense rim.

With the impression of liver abscess, the patient was operated upon to perform surgical drainage. In our center, surgical drainage is preferred for abscesses larger than 5cm in patients with no response to antibiotic and percutaneous drainage. [6] Gross examination showed a well-defined creamy grey-colored mass with a vague whorling appearance [Figure 1]. Sections of the mass showed numerous plasma cells with varying degrees of fibroblastic proliferation admixed with lymphocytes, eosinophils and macrophages [Figure 2].

The post-operative course was uneventful and now, after 1 year, he is doing well.

Case 2

A 15-year-old hepatitis B surface antigen (HBS)-positive boy presented with weight loss and anorexia. All the laboratory tests, including LFTs, were normal, except for leuckocytosis (16,500/L).

Imaging studies showed a well-defined liver mass. Fine needle aspiration was performed, which indicated low cullarity and was insufficient for diagnosis. He was operated with the clinical diagnosis of hepatocellular carcinoma and a 6cm liver mass was excised.

Gross of the mass showed a well-defined 6cm liver mass, which, on microscopy, showed predominantly fibroblastic proliferation as well as many plasma cells and eosinophils. Now, after about 2 years, he is completely well.


   Discussion Top


Liver IPT was first described by Pack and Baker in 1953. [7] Since then, this tumor has been increasingly recognized, probably because of the advances in imaging techniques. [8] Pre-operative findings of IPTs can mimic malignant tumors and thus it is very important to establish a differential diagnosis for this tumor and malignant liver tumors. [1]

This tumor can also appear as infectious or parasitic liver abscess. [7]

Herein we aimed to analyze our experience about the clinical, imaging and pathologic findings of IPT in two cases of the pediatric age group.

Both of our cases were male and were children and both presented with significant weight loss and anorexia. One of them also had chills and fever because of which his pre-operative diagnosis was liver abscess, although all the cultures were negative.

The other had no sign of an inflammatory process and because of positive HBS and significant weight loss; pre-operative diagnosis was a malignant liver tumor, most probably hepatocellular carcinoma.

Most of the previous reported cases had also presented with weight loss. [7]

Pre-operative diagnosis of the majority of the reported cases had been malignant tumors and abscess and IPT was rarely the clinical impression before surgery. [9]

The CT scan revealed variable contrast enhancements as reported in the literature. [10] Cellular composition of the IPT is highly variable in the reported series. [11]

In one of our cases, the predominant cells were plasma cells and in the other one, fibroblasts were admixed with different proportions of eosinophils and plasma cells, which explains the variations in the imaging findings. [8]

The main difference between IPT and liver abscess is in histology, i.e. in the IPT, the whole liver parenchyma is replaced by collagenous tissue and chronic inflammatory cells, mostly plasma cells, but in liver abscess, foci of acute inflammatory cells and necrosis are present. [10]

The exact nature of IPT is most probably an exuberant inflammatory response to tissue injury, [3] the etiology of which is unclear, but infectious agents emerged as the most likely cause of IPT. [12] Meanwhile, cholangitis was also considered as the cause of hepatic IPT. [13] Studies from Japan reported several cases of IPT in association with autoimmune diseases of the pancreaticobiliary system, such as autoimmune sclerosing pancreatitis and primary sclerosing cholangitis. [14],[15]

There are theories about the neoplastic rather than the inflammatory nature of the IPTs, [16] but unique and consistent chromosomal abnormality has not been identified [17] and these observations await a wider confirmation.

The optimal therapy method for IPT of the liver is controversial. On the one hand, they are benign with no malignant potential and on the other hand, complete spontaneous regression with antibiotics having been reported without surgery. [1] However, the pre-operative impression of the reported tumors had not been IPT, as in our cases. Thus, correct pre-operative histologic diagnosis by biopsy can avoid unnecessary surgery. [3]

In conclusion, in hepatic masses of children, IPT should be kept in mind as a possible cause, especially when the patient is symptomatic. Hence, percutaneous biopsy can be performed to obtain a histologic confirmation to obviate unnecessary surgery.

 
   References Top

1.Koea JB, Broadhurst GW, Rodgers MS, McCall SL. Inflammatory pseudotumor of the liver: Demographic, diagnosis and the case for nonoperative management. J Am Coll Surg 2003;196:226-35.  Back to cited text no. 1    
2.Karnak I, Senocak ME, Ciftici AO, Caglar M, Bingol-kologlu M, Tanyel FC, et al . Inflammatory myofibroblastic tumors in children: Diagnosis and treatment. J Pediatr Surg 2001;36:908-12.  Back to cited text no. 2    
3.Czauderna P, Schaarschmidt K, Komasara L, Harmis D, Lempe M, Vorpahl K, et al . Abdominal inflammatory masses mimicking neoplasia in children: Experience of two centers. Pediatr Surg Int 2005;21:346-50.  Back to cited text no. 3    
4.Bankole-Sinni R, Coulibaly B, Denoulet D, Nandiolo R, Mobiot L, Oulai. Inflammatory pseudotumor of the liver in a child: Case report. Med Trop (Mars) 1997;57:269-72.  Back to cited text no. 4    
5.Gosavi A, Agashe S, Phansopkar M, Jadhav SS, Agrawal BR. Inflammatroy pseudotumor of liver: A case report. Indian J pathol Microbiol 1997;40:81-3.  Back to cited text no. 5    
6.Tan YM, Chung AY, Chow PK, Cheow PC, Wong WK, Ooi LL, et al . An appraisal of surgical and percutaneous drainage for pyogenic liver abscesses larger than 5 cm. Ann Surg 2005;241:485-90.  Back to cited text no. 6  [PUBMED]  [FULLTEXT]
7.Pack GT, Baker HW. Total right hepatic lobectomy: Report of a case. Ann Surg 1953;138:253-8.  Back to cited text no. 7  [PUBMED]  [FULLTEXT]
8.Tsou YK, Lin CJ, Liu NJ, Lin CH, Lin SM. Inflammatory pseudotumor of the liver: Report of eight cases, including three unusual cases and a literature review. J Gastroenterol Hepatol 2007;22:2143-7.  Back to cited text no. 8  [PUBMED]  [FULLTEXT]
9.Park KS, Jang BK, Chung WJ, Cho KB, Hwang JS, Kang YN, et al . Inflammatory pseudotumor of the liver: A clinical review of 15 cases. Korean J Hepatol 2006;12:429-38.  Back to cited text no. 9  [PUBMED]  [FULLTEXT]
10.Fukuya T, Honda H, Matsumata T, Kawanami T, Shimoda Y, Muranaka T, et al . Diagnosis of IPT of the liver: Value of CT. AJR Am J Roentgenol 1994;193:1087-91.  Back to cited text no. 10    
11.Shek TW, Ng IO, Chan KW. IPT of the liver: Report of four cases and review of the literature. Am J Surg Pathol 1993;17:231-8.  Back to cited text no. 11  [PUBMED]  
12.Horiuchi R, Uchida T, Kojima T, Shikata T. Inflammatory pseudotumor of the liver: Clinicopathologic study and review of the literature. Cancer 1990;65:1583-90.  Back to cited text no. 12  [PUBMED]  
13.Yoon KH, Ha HK, Lee JH, Suh JH, Kim MH, Kim PN, et al . Inflammatory pseudotumor of the liver in patients with recurrent pyogenic cholangitis: CT - histopathologic correlation. Radiology 1999;211:373-9.  Back to cited text no. 13    
14.Toda K, Yasuda I, Nishigaki Y, Enya M, Yamada T, Nagura K, et al . Inflammatory pseudotumor of the liver with primary sclerosing cholangitis. J Gastroenterol 200;35:304-9.  Back to cited text no. 14    
15.Sasahira N, Kawabe T, Nakamura A, Shimura K, Shimura H, Itobayashi E, et al . Inflammatory pseudotumor of the liver and peripheral eosinophilia in autoimmune pancreatitis. World J Gastroenterol 2005;11:922-5.  Back to cited text no. 15  [PUBMED]  [FULLTEXT]
16.Sirvent N, Coindre JM, Pedeutour F. Inflammatory myofibroblastic tumors. Ann Pathol 2002;22:453-60.  Back to cited text no. 16  [PUBMED]  [FULLTEXT]
17.SantaCruz KS, McKinley TM, Powell RD Jr, Hermreck AS, Sonnino RE. Inflammatory myofibroblastic tumor of the gastroesophageal junction in childhood. Pediatr Pathol Mol Med 2002;21:49-56.  Back to cited text no. 17    

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Correspondence Address:
Bita Geramizadeh
Transplant Research Center, Pathology Department, Shiraz University of Medical Sciences, Shiraz PO BOX: 71345 - 1864
Iran
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0377-4929.48920

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  [Figure 1], [Figure 2]

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