Intermittent small-bowel obstruction caused by gastric adenocarcinoma in a Meckel's diverticulum

Intermittent small-bowel obstruction caused by gastric adenocarcinoma in a Meckel's diverticulum

Brief Reports placement. A clinical trial with larger numbers of patients is needed to verify the results of the present series. REFERENCES 1. Dorma...

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Brief Reports

placement. A clinical trial with larger numbers of patients is needed to verify the results of the present series.

REFERENCES 1. Dormann AJ, Huchzermeyer H. Endoscopic techniques for enteral nutrition: standards and innovations [review]. Dig Dis 2002;20:145-53. 2. Dormann AJ, Glosemeyer R, Leistner U, Deppe H, Roggel R, Wigginghaus B, et al. Modified percutaneous endoscopic gastrostomy (PEG) with gastropexy: early experience with a new introducer technique. Z Gastroenterol 2000;38:933-8. 3. Kulling D, Sonnenberg A, Fried M, Bauerfeind P. Cost analysis of antibiotic prophylaxis for PEG. Gastrointest Endosc 2000;51:152-6. 4. O’Keeffe F, Carrasco CH, Charnsangavej C, Richli WR, Wallace S, Freedman RS. Percutaneous drainage and feeding gastrostomies in 100 patients. Radiology 1989;172:341-3. 5. DiLorenzo J, Dalton B, Miskovitz P. Percutaneous endoscopic gastrostomy. What are the benefits, what are the risks? [review] Postgrad Med 1992;91:277-81.

6. Lee MJ, Saini S, Brink JA, Morrison MC, Hahn PF, Mueller PR. Malignant small bowel obstruction and ascites: not a contraindication to percutaneous gastrostomy. Clin Radiol 1991;44:332-4. 7. Ryan JM, Hahn PF, Mueller PR. Performing radiologic gastrostomy or gastrojejunostomy in patients with malignant ascites. AJR Am J Roentgenol 1998;171:1003-6.

Department of Internal Medicine and Hepatogastroenterology, Klinikum I, Minden, Germany. Reprint requests: PD Dr. med. Arno Josef Dormann, Department of Internal Medicine and Hepatogastroenterology, Klinikum Minden I, Friedrichstr. 17, 32427 Minden, Germany. Copyright ª 2005 by the American Society For Gastrointestinal Endoscopy 0016-5107/2005/$30.00 + 0 PII: S0016-5107(04)02449-6

Intermittent small-bowel obstruction caused by gastric adenocarcinoma in a Meckel’s diverticulum Fabrizio Parente, MD, Andrea Anderloni, MD, Pietro Zerbi, MD, Marco Lazzaroni, MD, Gianluca Sampietro, MD, Piergiorgio Danelli, MD, Gabriele Bianchi Porro, MD

A 52-year-old white man was hospitalized with a 6-month history of recurrent, self-limited episodes of pain in the right lower abdomen. Each episode was associated with abdominal distension and constipation but not vomiting, and would last for several days. There was no hematochezia, but the patient had noted significant weight loss (13 kg in 6

months; body mass index declined from 25.6 to 21 kg/m2). During one of the episodes, the patient had been hospitalized at another institution, where routine biochemical tests, colonoscopy, EGD, and transabdominal US were normal. Barium contrast radiography of the small bowel disclosed an area of narrowing in the distal ileum, with some small nodular defects and slight proximal dilatation, findings that were interpreted as caused by probable Crohn’s disease. The patient was given a corticosteroid parenterally, with some benefit, and was discharged on a tapering dose of orally administered prednisolone, followed by oral administration of mesalazine. He remained well for less than 1 month but then developed further episodes of abdominal pain and was referred to us for evaluation. At admission, the examination was unremarkable. Laboratory test results included the following: Hb, 10.8 g/ dL (normal: 13-17.5 g/dL); red blood cell count, 397  104/ mm3 (450-600/mm3); and white blood cell count, 6700/mm3 (4000-10,000/mm3). A test for fecal occult blood was positive (2 of 3 specimens). Serum electrolytes, tests of liver and renal function, as well as tumor markers (including alpha fetoprotein, carcinoembryonic antigen, and carbohydrate-associated antigen 19-9) were within normal ranges. The erythrocyte sedimentation rate was 32 mm/h (0-10 mm/ h), and C-reactive protein was 3.1 mg/dL (0-1 mg/dL). Transabdominal US revealed 3 large, roundish, hypoechoic areas (5, 2.5, and 2 cm, respectively, in diameter) in

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Meckel’s diverticulum is the most common congenital anomaly of the GI tract (incidence 0.5%-2% in autopsy series). It is caused by incomplete obliteration of the omphalomesenteric duct, which typically disappears during the 5th to 6th week of gestation.1 Although most Meckel’s diverticulae do not cause clinical symptoms and usually are discovered incidentally at laparotomy or autopsy, symptoms from complications (e.g., bleeding, bowel obstruction, perforation) may occur in up to 22% of adults.2 A primary malignant tumor arising within a Meckel’s diverticulum is extremely uncommon, with an incidence that ranges from 1.5% to 3.2%; most are stromal or carcinoid tumors.3 To our knowledge, only a few well-documented cases of adenocarcinoma arising from gastric tissue within a Meckel’s diverticulum are reported in English language publications.3-5 We report here a case of intermittent smallbowel obstruction caused by primary gastric adenocarcinoma arising within a Meckel’s diverticulum.

CASE REPORT

Brief Reports

Figure 2. A, Capsule endoscope image showing polypoid lesion with central depression. B, Capsule endoscope image showing large irregular ulceration in distal ileum.

the right hepatic lobe (confirmed by CT as strongly suggestive of hepatic metastases) and a thickened bowel loop in the right flank (maximum thickness 9 mm). The latter finding had an irregular outer profile and a hypoechoic echo pattern, with significant proximal dilatation, suggesting the presence of an organic stricture. Moreover,

mesenteric hypertrophy with roundish hypoechoic lymph nodes also was noted (Fig. 1). EGD, colonoscopy, and push enteroscopy (to 175 cm) were normal. Capsule endoscopy disclosed a polypoid lesion with central ulceration and a large irregular ulcer in the ileum immediately proximal to the area of luminal narrowing, where the capsule was retained (Fig. 2). Considered together, the findings were interpreted as strongly suggestive of a small bowel tumor, and the patient underwent exploratory laparotomy. At surgery, there was a bulky tumor arising from a broadbase diverticulum in the antimesenteric border of the ileum, approximately 70 cm proximal to the ileocecal valve. The outer margin of the mass was inflamed and was adherent to the ascending colon. Mesenteric lymph nodes and liver metastases (>5 lesions involving both hepatic lobes; judged to be surgically unresectable) were present. The adhesions were taken down to separate the diverticulum from the colon, which was not infiltrated by the tumor. The capsule endoscope, retained a few centimeters

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Figure 1. A, US image (transverse section) showing marked bowel wall thickening of an ileal loop with a hypoechoic echo pattern and irregular outer profile. B, US image of enlarged lymph node (>2 cm) in surrounding mesentery.

Brief Reports

Figure 3. Intraoperative view of capsule endoscope lodged in stricture a few inches from the Meckel’s diverticulum. Figure 5. Photomicrograph of ileal specimen, showing moderately differentiated intestinal-type gastric adenocarcinoma (H&E; orig. mag. 4). Inset, moderately differentiated intestinal type gastric adenocarcinoma (H&E, orig. mag. 40).

At a 4-month follow-up, he was alive, with no evidence of abdominal-systemic disease progression.

DISCUSSION

proximal to an ileal stricture, was extracted through an enterotomy (Fig. 3). Then, a long segment of ileum (40 cm) bearing the mass and the diverticulum was resected with end-to-end anastomosis by stapling. Histopathologic evaluation of the resection specimen revealed a Meckel’s diverticulum (5  7 cm) on the antimesenteric ileal border, which had an indurated wall and irregular, hyperemic mucosa (Fig. 4). A tight stricture was present a few centimeters proximal to the diverticulum and immediately below the enterotomy defect, where the capsule had been retrieved. Microscopic examination demonstrated a moderately differentiated adenocarcinoma arising from ectopic gastric mucosa within the Meckel’s diverticulum that had infiltrated the wall to the serosa (Fig. 5). Multiple lymph-node metastases were present (pT3 N1 M1). Helicobacter pylori was not detected by light or electron microscopy. The postoperative course was uneventful, and the patient was discharged home with adjuvant chemotherapy.

The lifetime risk of symptoms in patients with a Meckel’s diverticulum is not greater than 4%6; 40% of the patients with symptoms are children younger than 10 years of age who usually present with acute or chronic GI bleeding. Acute or intermittent small-bowel obstruction is a relatively common clinical presentation of a Meckel’s diverticulum in adult patients. It usually results from complications such as intussusception, volvulus, or internal hernia; less frequently, it may also be caused by neoplasm. The usual tumors that arise within Meckel’s diverticulum are stromal and carcinoid tumor and fibroma. Adenocarcinoma is extremely rare, and is thought to arise mainly from heterotopic tissue present within the diverticulum, including pancreatic tissue, as well as duodenal, jejunal, colonic, and gastric mucosa. Because the frequency of ectopic gastric tissue in Meckel’s diverticulum is only 10% to 20%,7 other cell types (e.g., intestinal cells) are more likely to be involved in the carcinogenetic process. The present case is one of a few described in English language publications3-5 in which the tumor can be assumed to have arisen from ectopic gastric tissue. Factors potentially implicated in the carcinogenetic development of tumors in Meckel’s diverticulum remain uncertain. Some investigators speculate that ectopic gastric mucosa within the diverticulum may have a greater malignant potential than the normal small-bowel mucosa, and, therefore, such ectopic tissue increases the risk of cancer.3 Conversely, H pylori (which was present in the

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Figure 4. Gross resection specimen of ileum, showing Meckel’s diverticulum on antimesenteric border (speculum) with tumor arising from margins and invading ileum and mesentery.

Brief Reports

stomach of our patient), has been shown to be capable of colonizing ectopic gastric mucosa8; it also is a well-known carcinogen that is implicated in the pathogenesis of mucosa-associated lymphoid tissue lymphoma and gastric adenocarcinoma. However, a role for H pylori in the pathogenesis of the tumor in our patient remains doubtful, because the organism could not be detected by either light or electron microscopy in the resected Meckel’s diverticulum. The preoperative diagnosis of neoplasm in a Meckel’s diverticulum is extremely difficult, because clinical features and radiologic findings are nonspecific and may mimic those of Crohn’s disease or other small-bowel tumors. When neoplasia is diagnosed or suspected before surgery (as in the present case), it is likely to be advanced.3 Radiographic contrast studies, including small-bowel enteroclysis, may define complications associated with the tumor (e.g., bowel obstruction) but usually do not demonstrate the type of lesion with certainty. US and CT may reveal an intestinal stricture or bowel-wall thickening because of the tumor but are of little help in detecting the Meckel’s diverticulum. When there is ectopic gastric mucosa in the diverticulum, technetium Tc 99m scintigraphy may be of value because the radioisotope is taken up by gastric mucosa; sensitivity is about 80%.9 In the case presented, the diagnosis of adenocarcinoma of Meckel’s diverticulum as the cause of ileal obstruction was not established before surgery, but an organic ileal stricture, presumably neoplastic, was demonstrated by US and capsule endoscopy. The latter test has significantly improved small-bowel imaging, so that it now constitutes the imaging procedure of choice for suspected small-intestinal bleeding. Other potential indications for capsule endoscopy include inflammatory bowel disease, surveillance of polyposis syndromes, and suspected enteric tumors (as in the present case), although reported experience with the latter indication is limited.10 These conditions are poorly characterized by standard imaging studies, and only intraoperative enteroscopy might constitute a valid alternative to capsule endoscopy. Retention of the capsule at the stricture, previously considered a potential cause of obstruction,

may now be regarded as a marker at surgery for the obstructive lesion.

REFERENCES 1. Jay GD, Margulis RR, McGraw AB, Northrip RR. Meckel’s diverticulum: a survey of 103 cases. Arch Surg 1950;61:158-69. 2. Mackey WC, Dineen P. A fifty-year experience with Meckel’s diverticulum. Surg Gynecol Obstet 1983;156:56-64. 3. Kusumoto H, Yoshitake H, Mochida K, Kumashiro R, Sano C, Inutsuka S. Adenocarcinoma in Meckel’s diverticulum: report of a case and review of 30 cases in the English and Japanese literature. Am J Gastroenterol 1992;87:910-3. 4. Lin P, Koffron A, Heilizer T, Theodoropoulus P, Pasikov D, Lujan H. Gastric adenocarcinoma of Meckel’s diverticulum as a cause of colonic obstruction. Am Surg 2000;66:627-30. 5. Rieber JM, Weinshel E, Nguyen T, Sidhu G, Bini E. Synchronous gastric adenocarcinomas in a patient with Meckel’s diverticulum. J Clin Gastroenterol 2001;33:78-80. 6. Turgeon DK, Barnett JL. Meckel’s diverticulum. Am J Gastroenterol 1990;85:777-81. 7. Rutheford RB, Akers DR. Meckel’s diverticulum: a review of 148 pediatric patients, with special reference to the pattern of bleeding and to mesodiverticular vascular bands. Surgery 1966;59:618-26. 8. Morris A, Nicholson G, Zwi J, Vanderwee M. Campylobacter pylori infection in Meckel’s diverticula containing gastric mucosa. Gut 1989;30:1233-5. 9. Wine CR, Nahrwold DL, Waldhausen JA. Role of technetium scan in the diagnosis of Meckel’s diverticulum. J Pediatr Surg 1974;8:885-8. 10. de Mascarenhas-Saraiva MN, da Silva Araujo Lopes LM. Small bowel tumors diagnosed by wireless-capsule endoscopy: report of five cases. Endoscopy 2003;35:861-4.

Department of Gastroenterology (FP, AA) ; Pathology Unity (PZ); Department of Gastroenterology (ML); Department of General Surgery (GS, PD); Department of Gastroenterology L. Sacco University Hospital, Milan, Italy (GBP). Reprint requests: Dr. Fabrizio Parente, MD, Department and Chair of Gastroenterology, L. Sacco University Hospital, Via G.B.Grassi, 74, 20157 Milan, Italy. Copyright ª 2005 by the American Society for Gastrointestinal Endoscopy 0016-5107/2005/$30.00 + 0 PII: S0016-5107(04)02450-2

A simple technique for maximal tip deflection of an upper endoscopy Kantaro Hisatomi, MD, Yasuyuki Kondo, MD, Masaru Tauchi, MD, Tatsuro Yanagawa, MD, Yukihiro Sakurai, MD

It sometimes is difficult, during upper endoscopy, to perform certain maneuvers (e.g., procurement of biopsy specimens, injection, clip application, variceal ligation)

when the target lesion is in the fornix or near the cardia of the stomach, because the ability to deflect the tip of the endoscope is limited. When a relative stiff accessory device,

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