Double Meckel’s Diverticulum Presenting as Acute Appendicitis: A Case Report and Literature Review

Double Meckel’s Diverticulum Presenting as Acute Appendicitis: A Case Report and Literature Review

The Journal of Emergency Medicine, Vol. 44, No. 4, pp. e321–e324, 2013 Copyright Ó 2013 Elsevier Inc. Printed in the USA. All rights reserved 0736-467...

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The Journal of Emergency Medicine, Vol. 44, No. 4, pp. e321–e324, 2013 Copyright Ó 2013 Elsevier Inc. Printed in the USA. All rights reserved 0736-4679/$ - see front matter

http://dx.doi.org/10.1016/j.jemermed.2012.11.001

Clinical Communications: Adults DOUBLE MECKEL’S DIVERTICULUM PRESENTING AS ACUTE APPENDICITIS: A CASE REPORT AND LITERATURE REVIEW Arif Emre, MD,* Sami Akbulut, MD,† Mehmet Yilmaz, MD,† Murat Kanlioz, MD,* and Burcu Elif Aydin, MD‡ *Department of Surgery, Malatya State Hospital, Malatya, Turkey, †Department of Surgery, Division of Liver Transplantation, Inonu University Faculty of Medicine, Malatya, Turkey, and ‡Department of Pathology, Malatya State Hospital, Malatya, Turkey Reprint Address: Sami Akbulut, MD, Department of Surgery, Division of Liver Transplantation, Inonu University Faculty of Medicine, 44280, Malatya, Turkey

, Abstract—Background: Meckel’s diverticulum is the most common developmental anomaly of the gastrointestinal tract, affecting 1–3% of the general population. This anomaly is caused by incomplete obliteration of the omphalomesenteric duct during the 7th week of gestation and is located in the last meter of the ileum in 90% of patients. Meckel’s diverticulum is often an incidental finding during a laparotomy for other causes, such as acute appendicitis, and occasional complications are bleeding, obstruction, diverticulitis, and perforation. Objective: Primary aim of this study was to present and share an extremely rare case of double Meckel’s diverticulum. Methods: A 20-year-old woman who was admitted due to symptoms of right lower quadrant pain, nausea, and vomiting. A laparotomy with a McBurney’s incision was performed because of a presumed diagnosis of acute appendicitis. The surgical exploration revealed a double Meckel’s diverticulum localized 50 cm proximal to the ileocecal valve, but no acute appendicitis. The operation was finished after a diverticulectomy and appendectomy. No complications occurred during the postoperative follow-up. Conclusions: We conducted a literature review of studies published in the English language on double Meckel’s diverticulum, accessed via PubMed, Medline, and the Google Scholar databases. We found only five case reports of such a Meckel’s diverticulum variant. Ó 2013 Elsevier Inc.

INTRODUCTION Meckel’s diverticulum (MD), a remnant of the vitelline duct that normally disappears at the end of the 7th week of gestation, is the most common congenital abnormality of the small intestine. It arises from the antimesenteric border of the terminal ileum as a true diverticulum that contains all layers of the intestinal wall (1–3). MD is present in 1–3% of the population. It is located in the last meter of the ileum in 90% of patients and is usually 2.9–5 cm long and 1.9 cm wide (4,5). A male preponderance exists, with a male to female ratio of 1.5–5:1 (3,6,7). Although MD remains mostly asymptomatic in affected individuals, a 4–6% probability of symptomatic presentation occurs. Notably, incidence decreases with age. The clinical presentation of MD includes intestinal obstruction, bleeding, diverticulitis, and perforation (2,5). Obstruction of various types is the most common presenting symptom in the adult population, occurring in almost 40% of patients. Diverticulitis is the second most common presentation, and is noted in 12.7–53.3% of all symptomatic cases. These patients often present with signs and symptoms mimicking acute appendicitis, and the diagnosis is usually confirmed during a surgical exploration (2,3,7). Our purpose is to present an extremely rare case of double Meckel’s diverticulum that clinically mimicked acute appendicitis.

, Keywords—double Meckel’s diverticulum; duplication; congenital anomaly; unusual presentation

The authors declare that there is no conflict of interest.

RECEIVED: 18 February 2012; FINAL SUBMISSION RECEIVED: 8 May 2012; ACCEPTED: 2 November 2012 e321

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CASE REPORT A 20-year-old woman was admitted to our Emergency Clinic with complaints of pain in the abdomen, nausea, and vomiting. The patient indicated that the pain had commenced 12 h earlier and exhibited a progressive shift to the right lower quadrant. On checking her vital signs, body temperature; pulse rate; and arterial blood pressure were 37.6 C; 96 beats/min; and 120/70 mm Hg, respectively. Widespread abdominal tenderness was evident on physical examination, although it was more prominent in the right lower quadrant. Guarding and rebound tenderness were detected, particularly in the right lower quadrant. A plain abdominal X-ray study revealed no remarkable pathology other than a gas artifact. Liver and kidney functions tests were within the normal range, and no pathological findings other than occasional red blood cells (2–3 per high power field) were observed in a urinalysis. The complete blood count report was as follows: white blood cells 13,340/mm3 (91% neutrophils); hemoglobin 15.3 g/dL; and platelets 187,000/mL. Considering the patient’s medical history, physical examination findings, and results from the blood tests, a definitive diagnosis of acute appendicitis was established. Exploration through McBurney’s incision revealed that the appendix was normally localized and exhibited no pronounced inflammatory changes; therefore, the small bowel was inspected proximally over its entire length starting from the ileocecal valve. A mildly inflamed double MD was observed 50 cm cephalad to the ileocecal valve, surrounded by minimal reactive fluid (Figure 1). A diverticulectomy and primary repair were undertaken due to the diameter of the ileal segment where the diverticular structures were localized, which was larger than expected. A drain was placed in the right paracolic gutter. The operation was completed with a final appendectomy. No complica-

Figure 1. Intraoperative view of double Meckel’s diverticulum.

Figure 2. The fundic glands of the heterotrophic gastric mucosa intermingled with intestinal mucosa (hematoxylin and eosin,  40,  200).

tions occurred during the postoperative follow-up period. A histopathological evaluation of the specimen revealed a focal ulcerated area in the small intestinal mucosa, whereas intense inflammation, heterotrophic gastric tissue (antral and corporeal-fundic glands), pancreatic acini, and an area of peptic ulcer featured by reactive epithelial changes were documented in the diverticular sections (Figures 2–4). Consequently, the results of the pathological examination indicated double MD. DISCUSSION Congenital gastrointestinal malformations comprise approximately 6% of all congenital anomalies (5). Failure of the vitelline duct to dissolve is one of the most common congenital gastrointestinal anomalies and results in several anomalies, including omphalomesenteric fistulas, enterocysts, fibrous bands connecting the intestine to

Figure 3. The heterotrophic glandular structures related to the gastric antrum (hematoxylin and eosin,  20).

Double Meckel’s Diverticulum

Figure 4. Heterotrophic pancreatic acini and glandular structures related to the gastric antrum (hematoxylin and eosin,  200).

the umbilicus, or MD (3). MD, with or without connection to the umbilicus or to the mesentery, accounts for 90% of all vitelline duct anomalies (3). MD consists of a small outpouching of the gastrointestinal tract due to the incomplete obliteration of the omphalomesenteric duct during the 7th and 8th weeks of gestation. MD is a true diverticulum containing all of the layers of a normal small bowel. The classic presentation of MD, which is usually located 50–100 cm proximal to the ileocecal valve, is before the age of 2 years; it is seen twice as often in males as in females and is found in about 1–3% of the population (5). MD was first described by Fabricus Heldanus in 1598, subsequently reported by Levator in 1671, and then by Ruysch in 1707. However, it was described in detail by Hohann Friedrick Meckel in 1809 (1,3–5). Although MD stems from the antimesenteric surface of the ileum in 90% of cases, rare cases of MD that arise from the mesenteric surface of the ileum, proximal jejunum, or rectum have also been reported. Cases of ‘‘wandering’’ MD have also been published (5). We encountered a sizable number of articles in the English literature on searching for the term Meckel’s diverticulum in the PubMed, Google Scholar, and Medline databases. However, a literature search performed by entering the keywords double MD displayed just five case reports (4,6,8–10). Therefore, we have presented the sixth such case in this study. However, there are insufficient data in the literature to elucidate the ‘‘double’’ developmental anomaly. The clinical presentation in a patient with MD includes intestinal obstruction, bleeding, diverticulitis, and perforation. Bleeding is a major clinical finding in children with symptomatic MD. The recorded incidence of bleeding has been as high as 50%. The primary reason for bleeding is associated with heterotopic tissue, as

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55–60% of all MDs contain histopathologically heterotrophic tissues (5). Although heterotrophic gastric mucosa (23–60%) and pancreatic tissue (5–16%) have been the most frequently encountered tissues, duodenal mucosa, jejunal mucosa, colonic mucosa, endometriosis, or hepatobiliary tissue are among the rare heterotrophic tissues (3,5). Secretion of acidic and alkaline fluids from the gastric and pancreatic tissues yields additional complications, such as bleeding and ulceration (3,7). Bleeding originates from ectopic mucosa in approximately 90% of cases with manifest bleeding (3). An intestinal obstruction due to MD is the most common symptom presenting in adults (40%) and is the second major clinical finding in children. Ileoileal or ileocolic intussusceptions are likely to be complicated as a result of invagination of the MD into the ileal lumen, particularly in children (5). Additionally, herniation of MD into a hernial sac, a condition known as Littre’s herniation, can also result in an obstruction. On rare occasions, conditions such as stone formation within the MD lumen and inversion or volvulus of MD can also be included among events likely to lead to an obstruction. Another clinical finding is MD, which accounts for 12.7–53.3% of symptomatic cases and is more commonly seen in older patients in whom it is most often misdiagnosed as appendicitis. Therefore, when a normal appendix is seen during an operation for appendicitis, the distal lumen should be examined for a diverticulum, as in our case. Diverticulitis is inflammation due to obstruction by an enterolith, foreign body, or parasite (5). A strong correlation appears to exist between complications and patient sex, age, length, basal diameter of the diverticulum, and the amount of heterotrophic tissue contained. In general, male sex, younger age, diverticular length >3 cm, basal diameter #2 cm, and palpable heterotrophic tissue are individually regarded as risk factors (11). Tumoral transformation from an MD is uncommon and has been reported in 0.5–5.0% of symptomatic MD cases. Tumors rarely originate from the diverticulum; lipoma, hamartoma, melanoma, lymphoma, sarcoma, adenocarcinoma, villous adenoma, leiomyosarcoma, and carcinoid tumors arising from MD have been reported (3,5,11). Foreign bodies, including fish bones, gallstones, enteroliths, marbles, bullets, and phytobezoars, have been reported in <2% of patients with symptomatic MD (3). Despite advances in radiological techniques, the diagnosis of MD is still difficult and is usually made during surgery (11). Plain radiography, barium studies, angiography, computed tomography, ultrasonography, and scintigraphy all play complementary roles in the diagnosis of MD. Plain radiography allows only a diagnosis of an intestinal obstruction or perforation. Ultrasonography, which can reveal a pelvic abscess, a tubular

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fluid-distended diverticulum at a site far from the cecum, diverticular wall swelling, segmental thickening of the intestinal walls, and invagination, is not sufficiently specific. Abdominal computed tomography scans can aid in the diagnosis if MD is suspected; they may show multiple fluid-filled, dilated ileal loops with distal collapse (11). The most sensitive technique is scintigraphy, and various modifications of imaging techniques have been devised to improve sensitivity. The mucoid cells of the gastric mucosa secrete chloride into the intestinal lumen. Excretion does not depend on the presence of parietal cells. Technetium-99m pertechnetate behaves in a manner analogous to halide anions. The mucoid surface cells of the gastric mucosa, whether located normally or ectopically, actively accumulate and secrete pertechnetate into the intestine. This is the basis for detecting ectopic gastric mucosa in patients with symptomatic MD. Scintigraphy is the most effective modality for children, with a sensitivity of 80–90%, specificity of 95%, and an accuracy of 90%. However, when applied to adults, these values fall to 62%, 9%, and 46%, respectively. This difference can be attributed to the area where the ectopic gastric mucosa is involved. Scintigraphy easily detects an area with larger ectopic gastric mucosa, which is most likely to hemorrhage earlier (5,11). Whether a patient is symptomatic has been an important factor in the approach to MD. How asymptomatic patients, in whom MD was detected incidentally during surgery or for other reasons, should be managed is still controversial, and no controversy exists about the approach to symptomatic cases. In cases of symptomatic MD, the surgical approach can be performed openly or laparoscopically, depending on the type of complication. A diverticulectomy or segmental ileal resection and primary anastomosis are preferred, but the choice is dictated by the type of complication. Although some studies suggest that an asymptomatic diverticulum should not be resected, others suggest the contrary. Artigas et al. concluded that an asymptomatic MD should not be resected due to decreasing prevalence of symptoms with increasing age (12). Cullen et al. concluded that all incidentally discovered diverticula should be resected at any age because the risks of complications from MD do not decrease with age (13).

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CONCLUSIONS The possibility that asymptomatic MD can become symptomatic has always been worrisome. Many studies have suggested that incidental MD should always be removed (3,5). Although the incidence of complications arising from surgical resection of MD is approximately 1%, the life-long risk for complications in patients with MD is 4–6.4% (3,5,11,13). In addition, most large-series studies have reported no reduced risk for MD-related complications with increasing age; so, we recommend diverticulectomy on a routine basis in cases possessing the aforementioned risk factors if MD is detected incidentally during laparotomy or laparoscopy (13). REFERENCES 1. Alevli F, Akbulut S, Dolek Y, Cakabay B, Sezgın A. Patent vitelline duct as a cause of acute abdomen: case report of an adult patient. Turk J Gastroenterol 2011;22:101–3. 2. Yagci G, Cetiner S, Tufan T. Perforation of Meckel’s diverticulum by a chicken bone, a rare complication: report of a case. Surg Today 2004;34:606–8. 3. Yahchouchy EK, Marano AF, Etienne JC, Fingerhut AL. Meckel’s diverticulum. J Am Coll Surg 2001;192:658–62. 4. Yang JG, Ma DQ, Hao RR, Li CL, Zou LF. Detection of double Meckel diverticulum by Meckel scan. Clin Nucl Med 2008;33: 729–30. 5. Uppal K, Tubbs RS, Matusz P, Shaffer K, Loukas M. Meckel’s diverticulum: a review. Clin Anat 2011;24:416–22. 6. Tauro LF, Martis JJ, Shenoy HD. Double Meckel’s diverticulum. J Indian Med Assoc 2009;107:174–5. 7. Korkmaz O, Yilmaz HG, Keles C. Complications of Meckel’s diverticulum in adults. Turkiye Klinikleri J Med Sci 2007;27:518–21. 8. Mazza L, Garino M, Morgando A, Fronda G, Rizzetto M. A double Meckel’s diverticulum as obscure gastrointestinal bleeding cause. Report of a case. Minerva Gastroenterol Dietol 2006; 52:225–31. 9. Albu E, Solomon L, Shah AK, Murthy RM, Gerst PH. Double Meckel’s diverticulum. South Med J 1992;85:1142–4. 10. Tauro LF, George C, Rao BS, Martis JJ, Menezes LT, Shenoy HD. Asymptomatic Meckel’s diverticulum in adults: is diverticulectomy indicated? Saudi J Gastroenterol 2010;16:198–202. 11. Bani-Hani KE, Shatnawi NJ. Meckel’s diverticulum: comparison of incidental and symptomatic cases. World J Surg 2004;28:917–20. 12. Artigas V, Calabuig R, Badia F, Rius X, Allende L, Jover J. Meckel’s diverticulum: value of ectopic tissue. Am J Surg 1986; 151:631–4. 13. Cullen JJ, Kelly KA, Moir CR, Hodge DO, Zinsmeister AR, Melton LJ 3rd. Surgical management of Meckel’s diverticulum. An epidemiologic, population-based study. Ann Surg 1994;220: 564–8.