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Inflammatory myofibroblastic tumor: An unusual hepatic tumor mimicking liver abscess
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Clinics and Research in Hepatology and Gastroenterology (2016) xxx, xxx—xxx
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Inflammatory myofibroblastic tumor: An unusual hepatic tumor mimicking liver abscess Yan-Wen Jin , Fu-Yu Li , Nan-Sheng Cheng ∗ Department of Biliary Surgery, West China Hospital, Sichuan University, No. 37, Guo Xue Xiang, Chengdu, Sichuan 610041, PR China
A 42-year-old woman presented in our department with 1 month of fatigue and repeated high fever. Highest body temperature was 39.2 ◦ C. The body temperature decreased to normal range after she was treated with anti-infection medicine in local hospital but fever relapsed once the treatment ceased. There was no other systemic symptom. She had hepatitis B history and denied previous radiotherapy or industrial chemical exposure. Physical examination showed she was an ill-appearing woman with pale conjunctivae. Her abdomen was soft, not tender or distended, and had no organomegaly or palpable masses. Laboratory examinations revealed hemoglobin of 80 g/l and a hematocrit of 25%. The white blood cell count was 11.6 × 109 /l and neutrophilia accounted for 82.4%. Level of C-reactive protein significantly elevated (CRP, 124.8 mg/L). Quantitation of hepatitis B virus DNA was 3.5 × 105 IU/ml. Liver function and serum level of tumor markers were within normal range (alpha-fetoprotein, 1.47 ng/ml). Abdomen ultrasonography revealed a hypoechoic mass in liver with an unclear border (Fig. 1A). Contrast-enhanced CT confirmed that there was a 6.5 × 4.9 cm low density lesion with mild enhancement in right posterior lobe of liver (Fig. 1B). A liver abscess was the
∗
Corresponding author. E-mail address: [email protected] (N.-S. Cheng).
tentative preoperative diagnosis based on clinical symptoms and imaging studies. The patient eventually underwent right posterior sectionectomy. Microscopically, the lesion was composed of chronic inflammatory cells and spindle cells (Fig. 2A). On immunohistochemical staining, the tumor cells were positive for CD68 (Fig. 2B) and smooth muscle actin (Fig. 2C), and negative for anaplastic lymphoma kinase (ALK). These findings were consistent with the diagnosis of inflammatory myofibroblastic tumor (IMT) of liver. Postoperative recovery of the patient was well and no recurrence was found in 32 months follow-up. IMT is an uncommon neoplasm which is composed of dominant spindle cell proliferation with a variable inflammatory component. It is now generally accepted that IMT is a true neoplasm with intermediate biological potential but the possibility of malignant transformation exists, and the incidence of distant metastasis is low. Morbidity of IMT in liver is extremely rare and the accurate incidence is not known. The etiology and pathogenesis of IMT in liver remain unclear. Infection, vascular diseases, pylephlebitis after appendicitis, Epstein-Barr virus infection and autoimmune disease have been hypothesized in pathogenesis [1]. The clinical presentation includes local mass, upper abdominal pain, nausea, vomit, intermittent fever, anemia, weight loss and so on. However, these symptoms are not spe-
http://dx.doi.org/10.1016/j.clinre.2016.11.008 2210-7401/© 2016 Elsevier Masson SAS. All rights reserved.
Please cite this article in press as: Jin Y-W, et al. Inflammatory myofibroblastic tumor: An unusual hepatic tumor mimicking liver abscess. Clin Res Hepatol Gastroenterol (2016), http://dx.doi.org/10.1016/j.clinre.2016.11.008
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Y.-W. Jin et al.
Figure 1 Abdominal ultrasonography and computed tomographic scan: ultrasonography revealed a hypoechoic mass in liver (A); CT showed a low density lesion with mild enhancement in right posterior lobe of liver (B).
Figure 2 Pathological examination and immunohistochemical staining: lesion was composed of chronic inflammatory cells and spindle cells (hematoxylin and eosin stain, ×200) (A); spindle cells were positive for CD68 (B) and smooth muscle actin (C) (immunostaining, ×200).
cific for IMT of liver [2]. The diagnosis of IMT in liver is difficult. Laboratory findings in IMT cases are generally normal. Due to the low sensitivity and reliability of needle aspiration cytology or core biopsy in diagnosis of IMT, surgical resection for biopsy remains the optimal choice [3]. There are many treatment options for IMT of liver, including surgery, steroids, non-steroidal anti-inflammatory drugs, antibiotics, radiotherapy and chemotherapy, but surgical management with clear margin is still the best choice for IMT of liver [4]. Recently, ALK inhibitors may provide a new therapeutic option in patients with ALK-expressing IMT [5]. It is difficult to delineate the prognostic parameters of IMT in liver. A recent study revealed a low recurrence rate [2], but significant recurrence rate (25%) existed in another study [6]. Now, IMT of liver is still a challenge in clinical practice.
Ethical approval The patient gave written informed consent.
Funding None.
Author contributions All authors cared for the patient and wrote the report. Manuscript is approved by all authors for publication.
Disclosure of interest The authors declare that they have no competing interest.
References [1] Cheuk W, Chan JK, Shek TW, Chang JH, Tsou MH, Yuen NW, et al. Inflammatory pseudotumor-like follicular dendritic cell tumor: a distinctive low-grade malignant intra-abdominal neoplasm
Please cite this article in press as: Jin Y-W, et al. Inflammatory myofibroblastic tumor: An unusual hepatic tumor mimicking liver abscess. Clin Res Hepatol Gastroenterol (2016), http://dx.doi.org/10.1016/j.clinre.2016.11.008
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Inflammatory myofibroblastic tumor: An unusual hepatic tumor mimicking liver abscess with consistent Epstein-Barr virus association. Am J Surg Pathol 2001;25:721—31. [2] Yang X, Miao R, Yang H, Chi T, Jiang C, Wan X, et al. Retrospective and comparative study of inflammatory myofibroblastic tumor of the liver. J Gastroenterol Hepatol 2015;30: 885—90. [3] Koea JB, Broadhurst GW, Rodgers MS, McCall JL. Inflammatory pseudotumor of the liver: demographics, diagnosis, and the case for nonoperative management. J Am Coll Surg 2003;196: 226—35.
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[4] Tang L, Lai EC, Cong WM, Li AJ, Fu SY, Pan ZY, et al. Inflammatory myofibroblastic tumor of the liver: a cohort study. World J Surg 2010;34:309—13. [5] Tothova Z, Wagner AJ. Anaplastic lymphoma kinase-directed therapy in inflammatory myofibroblastic tumors. Curr Opin Oncol 2012;24:409—13. [6] Coffin CM, Watterson J, Priest JR, Dehner LP. Extrapulmonary inflammatory myofibroblastic tumor (inflammatory pseudotumor). A clinicopathologic and immunohistochemical study of 84 cases. Am J Surg Pathol 1995;19:859—72.
Please cite this article in press as: Jin Y-W, et al. Inflammatory myofibroblastic tumor: An unusual hepatic tumor mimicking liver abscess. Clin Res Hepatol Gastroenterol (2016), http://dx.doi.org/10.1016/j.clinre.2016.11.008
ARTICLE IN PRESS
CLINRE-953; No. of Pages 3
Clinics and Research in Hepatology and Gastroenterology (2016) xxx, xxx—xxx
Available online at
ScienceDirect www.sciencedirect.com
IMAGE OF THE MONTH
Inflammatory myofibroblastic tumor: An unusual hepatic tumor mimicking liver abscess Yan-Wen Jin , Fu-Yu Li , Nan-Sheng Cheng ∗ Department of Biliary Surgery, West China Hospital, Sichuan University, No. 37, Guo Xue Xiang, Chengdu, Sichuan 610041, PR China
A 42-year-old woman presented in our department with 1 month of fatigue and repeated high fever. Highest body temperature was 39.2 ◦ C. The body temperature decreased to normal range after she was treated with anti-infection medicine in local hospital but fever relapsed once the treatment ceased. There was no other systemic symptom. She had hepatitis B history and denied previous radiotherapy or industrial chemical exposure. Physical examination showed she was an ill-appearing woman with pale conjunctivae. Her abdomen was soft, not tender or distended, and had no organomegaly or palpable masses. Laboratory examinations revealed hemoglobin of 80 g/l and a hematocrit of 25%. The white blood cell count was 11.6 × 109 /l and neutrophilia accounted for 82.4%. Level of C-reactive protein significantly elevated (CRP, 124.8 mg/L). Quantitation of hepatitis B virus DNA was 3.5 × 105 IU/ml. Liver function and serum level of tumor markers were within normal range (alpha-fetoprotein, 1.47 ng/ml). Abdomen ultrasonography revealed a hypoechoic mass in liver with an unclear border (Fig. 1A). Contrast-enhanced CT confirmed that there was a 6.5 × 4.9 cm low density lesion with mild enhancement in right posterior lobe of liver (Fig. 1B). A liver abscess was the
∗
Corresponding author. E-mail address: [email protected] (N.-S. Cheng).
tentative preoperative diagnosis based on clinical symptoms and imaging studies. The patient eventually underwent right posterior sectionectomy. Microscopically, the lesion was composed of chronic inflammatory cells and spindle cells (Fig. 2A). On immunohistochemical staining, the tumor cells were positive for CD68 (Fig. 2B) and smooth muscle actin (Fig. 2C), and negative for anaplastic lymphoma kinase (ALK). These findings were consistent with the diagnosis of inflammatory myofibroblastic tumor (IMT) of liver. Postoperative recovery of the patient was well and no recurrence was found in 32 months follow-up. IMT is an uncommon neoplasm which is composed of dominant spindle cell proliferation with a variable inflammatory component. It is now generally accepted that IMT is a true neoplasm with intermediate biological potential but the possibility of malignant transformation exists, and the incidence of distant metastasis is low. Morbidity of IMT in liver is extremely rare and the accurate incidence is not known. The etiology and pathogenesis of IMT in liver remain unclear. Infection, vascular diseases, pylephlebitis after appendicitis, Epstein-Barr virus infection and autoimmune disease have been hypothesized in pathogenesis [1]. The clinical presentation includes local mass, upper abdominal pain, nausea, vomit, intermittent fever, anemia, weight loss and so on. However, these symptoms are not spe-
http://dx.doi.org/10.1016/j.clinre.2016.11.008 2210-7401/© 2016 Elsevier Masson SAS. All rights reserved.
Please cite this article in press as: Jin Y-W, et al. Inflammatory myofibroblastic tumor: An unusual hepatic tumor mimicking liver abscess. Clin Res Hepatol Gastroenterol (2016), http://dx.doi.org/10.1016/j.clinre.2016.11.008
+Model CLINRE-953; No. of Pages 3
ARTICLE IN PRESS
2
Y.-W. Jin et al.
Figure 1 Abdominal ultrasonography and computed tomographic scan: ultrasonography revealed a hypoechoic mass in liver (A); CT showed a low density lesion with mild enhancement in right posterior lobe of liver (B).
Figure 2 Pathological examination and immunohistochemical staining: lesion was composed of chronic inflammatory cells and spindle cells (hematoxylin and eosin stain, ×200) (A); spindle cells were positive for CD68 (B) and smooth muscle actin (C) (immunostaining, ×200).
cific for IMT of liver [2]. The diagnosis of IMT in liver is difficult. Laboratory findings in IMT cases are generally normal. Due to the low sensitivity and reliability of needle aspiration cytology or core biopsy in diagnosis of IMT, surgical resection for biopsy remains the optimal choice [3]. There are many treatment options for IMT of liver, including surgery, steroids, non-steroidal anti-inflammatory drugs, antibiotics, radiotherapy and chemotherapy, but surgical management with clear margin is still the best choice for IMT of liver [4]. Recently, ALK inhibitors may provide a new therapeutic option in patients with ALK-expressing IMT [5]. It is difficult to delineate the prognostic parameters of IMT in liver. A recent study revealed a low recurrence rate [2], but significant recurrence rate (25%) existed in another study [6]. Now, IMT of liver is still a challenge in clinical practice.
Ethical approval The patient gave written informed consent.
Funding None.
Author contributions All authors cared for the patient and wrote the report. Manuscript is approved by all authors for publication.
Disclosure of interest The authors declare that they have no competing interest.
References [1] Cheuk W, Chan JK, Shek TW, Chang JH, Tsou MH, Yuen NW, et al. Inflammatory pseudotumor-like follicular dendritic cell tumor: a distinctive low-grade malignant intra-abdominal neoplasm
Please cite this article in press as: Jin Y-W, et al. Inflammatory myofibroblastic tumor: An unusual hepatic tumor mimicking liver abscess. Clin Res Hepatol Gastroenterol (2016), http://dx.doi.org/10.1016/j.clinre.2016.11.008
+Model CLINRE-953; No. of Pages 3
ARTICLE IN PRESS
Inflammatory myofibroblastic tumor: An unusual hepatic tumor mimicking liver abscess with consistent Epstein-Barr virus association. Am J Surg Pathol 2001;25:721—31. [2] Yang X, Miao R, Yang H, Chi T, Jiang C, Wan X, et al. Retrospective and comparative study of inflammatory myofibroblastic tumor of the liver. J Gastroenterol Hepatol 2015;30: 885—90. [3] Koea JB, Broadhurst GW, Rodgers MS, McCall JL. Inflammatory pseudotumor of the liver: demographics, diagnosis, and the case for nonoperative management. J Am Coll Surg 2003;196: 226—35.
3
[4] Tang L, Lai EC, Cong WM, Li AJ, Fu SY, Pan ZY, et al. Inflammatory myofibroblastic tumor of the liver: a cohort study. World J Surg 2010;34:309—13. [5] Tothova Z, Wagner AJ. Anaplastic lymphoma kinase-directed therapy in inflammatory myofibroblastic tumors. Curr Opin Oncol 2012;24:409—13. [6] Coffin CM, Watterson J, Priest JR, Dehner LP. Extrapulmonary inflammatory myofibroblastic tumor (inflammatory pseudotumor). A clinicopathologic and immunohistochemical study of 84 cases. Am J Surg Pathol 1995;19:859—72.
Please cite this article in press as: Jin Y-W, et al. Inflammatory myofibroblastic tumor: An unusual hepatic tumor mimicking liver abscess. Clin Res Hepatol Gastroenterol (2016), http://dx.doi.org/10.1016/j.clinre.2016.11.008