An Unusual Cause of Obscure Gastrointestinal Bleeding

Abstract

Question: A 53-year-old woman had been seeing a doctor regularly for liver cirrhosis. Her Child-Pugh score was 8 points and the ascites was well-controlled with diuretics. Her regular blood tests revealed progressive anemia, although she denied any event of melena, hematochezia, or bleeding elsewhere. Her hemoglobin level decreased from 9.0 to 4.5 mg/dL in 2 months and the examinations of gastrointestinal tract were suggested. Esophagogastroduodenoscopy revealed esophageal varices without bleeding stigmata and colonoscopy was normal. Because the bleeding was thought to originate from the small bowel, she underwent capsule endoscopy (CE), which revealed the submucosal mass in the ileum (Figure A). She was referred to our hospital for the further investigations and a double-balloon endoscopy (DBE) with retrograde procedure was done. DBE demonstrated the same lesion as shown on CE (Figure B). This lesion seemed to be a submucosal tumor according to the form of the lesion on endoscopic images. It also had an ulcer on the top part. The surrounding mucosa of the ulcer looked slightly red which was a feature of inflammatory pathologic change (Figure A, B). Therefore, we considered this lesion as the cause of bleeding. Endoscopic ultrasonography (EUS) with the through-the-scope sonoprobe was performed to identify the origin of the mass (Figure C). This EUS examination showed the tumor originating from the first to the third layer, corresponding with the mucosal and submucosal layers. In this case, endoscopic resection would be difficult because the lesion was very close to the muscle layer on EUS. We recommended the patient to receive surgical treatment. The surgical specimen (Figure D) demonstrated tumor involvement of the muscle layer (blue arrow). What is the diagnosis? Look on page 909 for the answer and see the Gastroenterology web site (www.gastrojournal.org) for more information on submitting your favorite image to Clinical Challenges and Images in GI. The pathologic section revealed proliferation of fibroblasts and infiltration of inflammatory cells, such as plasma cells and eosinophils compatible with an inflammatory fibroid polyp (IFP; Figure E), which was the final diagnosis. No progression of anemia was observed after surgery, which confirmed IFP as the cause of anemia in this patient. IFP of the small bowel is an extremely rare condition in adults. It mainly manifest as abdominal pain and obstruction, and rarely manifests as gastrointestinal bleeding.1Wysocki A.P. Taylor G. Windsor J.A. Inflammatory fibroid polyps of the duodenum: a review of the literature.Dig Surg. 2007; 24: 162-168Crossref PubMed Scopus (57) Google Scholar The etiology and pathogenesis of IFP is unknown, but it is postulated to be a consequence of an extreme body reaction to intestinal trauma or a variant of localized eosinophilic gastroenteritis, given that it has marked eosinophilic infiltration. The classical endoscopic appearance of IFP is of a solitary, subpedunculated, submucosal tumor with erosion or ulcer on top. However, such characteristic appearances are demonstrated only in 20% of patient and in some cases, it is difficult to differentiate IFP from the other submucosal tumors. On EUS, IFP typically originates from the second and third layers as a homogeneous and low echoic tumor with ill-defined borders. However, IFP arising from the subserosal layer was also reported.2Anthony P.P. Morris D.S. Vowles K.D. Multiple and recurrent inflammatory fibroid polyps in three generations of a Devon family: a new syndrome.Gut. 1984; 25: 854-862Crossref PubMed Scopus (66) Google Scholar When IFP is limited to the submucosal layer, endoscopic resection is the treatment of choice.3Tada S. Iida M. Yao T. et al.Endoscopic removal of inflammatory fibroid polyps of the stomach.Am J Gastroenterol. 1991; 86: 1247-1250PubMed Google Scholar When the tumor is larger or located in a deeper layer, surgical treatment is recommended. We have reported a case with small bowel submucosal tumor presenting with progressive anemia. The tumor contacts the muscle layer and the surgical treatment was successful. EUS was the key examination to determine the treatment of the choice for this IFP.