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  Volume 96
Numero 3
maggio-giugno 2010 		I documenti sono in formato PDF, consultabili utilizzando Acrobat Reader
 
Inflammatory myofibroblastic tumor of the stomach in an adult woman: a rare intermittent cause of gastric outlet obstruction

Fatih Albayrak, Hakan Dursun, Yavuz Albayrak, Sare Altas, Abdullah Uyanik, Rahsan Yildirim
1Department of Internal Medicine, Faculty of Medicine, Ataturk University, Erzurum; 2Department of General Surgery and Burn Unit, Erzurum Training and Research Hospital, Erzurum; 3Department of Pathology, Faculty of Medicine, Ataturk University, Erzurum, Turkey

Key words: inflammatory myofibroblastic tumor, endoscopy, spindle cell.

abstract

Background. Inflammatory myofibroblastic tumor is a neoplasm of intermediate biological potential that frequently recurs and rarely metastasizes.
Case report. We report a rare case of intermittent gastric outlet obstruction by an inflammatory myofibroblastic tumor of the cardia.
Results. A 56-year-old woman presented at the gastroenterology department with a two-day history of hematemesis and melena. She had intermittent nausea and vomiting complaints, which had manifested periodically for about five months. Upper gastrointestinal endoscopy demonstrated a mass of 6 cm in diameter, which was resected. Histological examination revealed ulcerated mucosal granulation-like tissue with myofibroblastic spindle cell proliferation in a storiform pattern.
Conclusions. In order to avoid unnecessary aggressive therapy, gastric IMT should be taken into account when a gastric mass accompanied by the various clinical manifestations of IMT is found in an adult. Free full text available at www.tumorionline.it

Introduction
 Inflammatory myofibroblastic tumor (IMT) has been defined as “a neoplasm of intermediate biological potential that frequently recurs and rarely metastasizes”1. IMT is also called inflammatory pseudotumor, plasma cell granuloma, inflammatory myofibroblastoma, and inflammatory myofibrohistiocytic proliferation. It is an inflammatory solid tumor that contains spindle cells, myofibroblasts, plasma cells, lymphocytes, and histiocytes2,3. Inflammatory pseudotumors rarely occur in the stomach2,3. A thorough literature search identified only 6 reported cases in the English-language medical literature so far2-6. We report, for the first time, a rare cause of intermittent gastric outlet obstruction in the form of an IMT of the cardia.

Case report
A 56-year-old woman presented at the gastroenterology department with a 2-day history of hematemesis and melena. She complained of intermittent nausea and vomiting, which had manifested periodically for about 5 months. There were no other symptoms. On examination, the patient appeared pale. Her body temperature was 36.7 °C, the respiratory rate 16/min, heart rate 110 beats/min, and blood pressure 90/50 mmHg. Findings on cardiopulmonary examination were normal. The abdomen was soft and not tender. Her medical background and family history were unremarkable and physical examination was normal. Blood tests included a hemoglobin level of 5.9 g/dL, a white blood cell count of 11,800/mm 3, and a platelet count of 372,000/μL. Coagulogram, liver function tests, creatinine and BUN were normal. Fluid resuscitation was started immediately and the patient was transfused with 2 units of red blood cells. After stabilization, an esophagogastroduodenoscopy was planned. Upper gastrointestinal endoscopy demonstrated a mass of 6 cm in diameter (Figure 1). The tumor was a large polyp originating from the cardia and extending towards the pylorus. There was no active bleeding. Stacked (bite-on-bite) forceps biopsy specimens were obtained, which showed only normal mucosa. A computed tomography (CT) scan of the abdomen demonstrated a well-defined lesion in the gastric body (Figure 2). On ultrasound and CT no evidence of metastatic disease or infiltration of adjacent organs was found. However, because of the size of the tumor, which was suggestive of malignancy, surgical excision was performed. At laparotomy, a round, rubbery mass with an extragastric component was excised; a definite diagnosis could not be made based on frozen-section biopsies, so a partial gastrectomy (Billroth I) was performed. The surgical procedure was completed without complications, and the patient tolerated the procedure well. Macroscopic examination of the resected portion of the stomach revealed a tumor mass that involved the entire thickness of the gastric wall. Most of the mass was a polyp; one portion consisted of a 11 × 7 × 5 cm solid white nodule. Histological sections of the tumor mass showed ulcerated mucosal granulation-like tissue, with myofibroblastic spindle cell proliferation in a storiform pattern. The mitotic activity was low, and cellular atypia was minimal. An increased number of plasma cells occurred singly and in clusters and lymphoid aggregates were present (Figure 3). Immunoperoxidase stains showed reactivity of the spindle cells to vimentin (Figure 4) and smooth-muscle actin, while being negative for c-kit, CD34, desmin, S-100, CD31 and anaplastic lymphoma kinase-1. These findings were consistent with IMT. The size of this gastric inflammatory pseudotumor was very uncommon. The patient has been symptom-free over 8 months of follow-up.













Discussion
IMTs have been identified in virtually every organ system but are most commonly reported in the lungs. Intraabdominal sites of the disease are most often the liver, followed by stomach, bowel, and spleen. IMT is primarily a disease of children and young adults. Gastric IMT in adults is very rare and to the best of our knowledge the present case is only the seventh reported in an adult2-7.
There has been debate as to whether IMT is a pseudotumor or a neoplasm and whether it is benign or malignant. It is currently regarded as a specific neoplasm within the larger descriptive category of inflammatory sclerosing and fibrosing processes. It is locally recurrent but rarely metastasizes1. The tumors are histopathologically composed of myofibroblastic spindle cells, with an inflammatory cell infiltrate of plasma cells, lymphocytes, and eosinophils8.
IMTs are often large, ranging in size from 3 cm to 10 cm. The gastric cases described to date were located at different sites in the stomach: cardia, antrum, and prepyloric region9. Our patient presented with a large IMT that had prolapsed through the pylorus, causing symptoms due to gastric outlet obstruction. These tumors have been described by Lazure as whitish or translucent, single or multiple, sometimes ulcerated, endoluminal polypoid nodules10. They are infiltrative and often extend through the gastric wall, sometimes reaching adjacent organs including the esophagus, duodenum, peritoneal cavity, spleen, pancreas, and liver5,9-11, as well as adjacent structures such as the falciform ligament or greater omentum1,4. These features mimic malignancy on endoscopy and radiological imaging5,11.
Even with a thorough diagnostic workup, which may include ultrasonography, CT, endoscopy, and percutaneous fine-needle aspiration biopsy, it is difficult to make an accurate preoperative diagnosis of IMT. Confirmation with microscopic examination of gastric specimens is frequently required12. The differential diagnosis of IMT is extensive and depends on tumor site and morphological characteristics, and on the nature of the specimen available for examination1,13. 
In conclusion, gastric IMT is a rare lesion in adults. It is, however, surgically curable and should therefore be accurately differentiated from sarcomas at the time of exploration. In order to avoid unnecessary aggressive therapy, gastric IMT should be taken into account when a gastric mass accompanied by the various clinical manifestations of IMT is found in an adult. Resection of the tumor was the treatment of choice in all cases reviewed12. After surgical excision, endoscopic follow-up is recommended because of the possibility of recurrence at the excision site or development of malignancy in the remote gastric mucosa, as well as the difficulty in predicting the prognosis14.

References
 1. Coffin CM, Hornick JL, Fletcher CD: Inflammatory myofibroblastic tumor: comparison of clinicopathologic, histologic, and immunohistochemical features including ALK expression in atypical and aggressive cases. Am J Surg Pathol, 31: 509-520, 2007.
 2. Al-Taie OH, Mork H, Jenett M, Klein D, Muller JG, Scheurlen M: Fast-growing gastric inflammatory pseudotumor: a rare manifestation of peptic ulcer disease. Endoscopy, 34: 239, 2002
 3. Leon CJ, Castillo J, Mebold J, Cortez L, Felmer R: Inflammatory myofibroblastic tumor of the stomach: an unusual complication after gastrectomy. Gastrointest Endosc, 63: 347-349, 2006.
 4. Park SH, Kim JH, Min BW, Song TJ, Son GS, Kim SJ, Lee SW, Chung HH, Lee JH, Um JW: Exophytic inflammatory myofibroblastic tumor of the stomach in an adult woman: a rare cause of hemoperitoneum. World J Gastroenterol, 14: 136-139, 2008.
 5. Kim KA, Park CM, Lee JH, Cha SH, Park SW, Hong SJ, Seol HY, Cha IH, Mok YJ, Kim YS: Inflammatory myofibroblastic tumor of the stomach with peritoneal dissemination in a young adult: imaging findings. Abdom Imaging, 29: 9-11, 2004.
 6. Kojimahara K, Mukai M, Yamazaki K, Yamada T, Katayama T, Nakada K, Uematsu S, Umezono A, Hosoda Y: Inflammatory pseudotumor of the stomach: report of a highly infiltrative case with electron microscopic and immunohistochemical studies. Acta Pathol Jpn, 43: 65-70, 1993.
 7. Shah SM, Sussman D, Jorda M, Ribeiro A: EUS with EMR of an inflammatory myofibroblastic tumor of the stomach. Gastrointest Endosc, 67: 561-563, 2008.
 8. Coffin CM, Fletcher JA: Inflammatory myofibroblastic tumour. In: Pathology and genetics of tumours of soft tissue and bone. World Health Organization classification of tumours, Fletcher CDM, Unni KK, Mertens F (Eds), pp 91-93, IARC Press, Lyon 2002.
 9. Estêvão-Costa J, Correia-Pinto J, Rodrigues FC, Carvalho JL, Campos M, Dias JA, Carneiro F, Santos NT: Gastric inflammatory myofibroblastic proliferation in children. Pediatr Surg Int, 13:95-99, 1998.
10. Lazure T, Ferlicot S, Gauthier F, Doz F, Couturier J, Fabre M, Bedossa P: Gastric inflammatory myofibroblastic tumors in children: an unpredictable course. J Pediatr Gastroenterol Nutr, 34: 319-322, 2002.
11. Schroeder BA, Wells RG, Sty JR: Inflammatory fibroid polyp of the stomach in a child. Pediatr Radiol, 17: 71-72, 1987. 
12. Deprez PH, Aouattah T, Piessevaux H: Endoscopic removal or ablation of oesophageal and gastric superficial tumours. Acta Gastroenterol Belg, 69: 304-311, 2006.
13. 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, 19: 859-872, 1995.
14. Hamidah A, Khu SY, Thambidorai CR, Muhaizan WM, Zarina AL, Jamal R: Recurrent gastric inflammatory myofibroblastic tumour in a 13-year-old male. Pediatr Surg Int, 23: 601-603, 2007.
 
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