J Gastrointest Canc DOI 10.1007/s12029-014-9579-x

CASE REPORT

Appendiceal Mucinous Cystadenoma Intussuscepted into the Cecum on a Patient with Rectal Carcinoma: A Case Report Zhenqiang Sun & Haijiang Wang & Xianbo Yu & Zeliang Zhao & Qisan Wang

# Springer Science+Business Media New York 2014

Introduction Appendiceal mucinous cystadenoma intussusception is rare but is readily ruptured and may form pseudomyxoma peritonei [1–3]. This condition is difficult to diagnose preoperatively due to its similarity to appendicitis or to atypical symptoms. A computed tomography (CT) scan, radioscopy, or colonoscopy can differentiate appendiceal mucinous cystadenoma intussusception from a cecal polyp or mass [4, 5]. Misdiagnosis may lead to treatment with false-polypectomy colonoscopy, which can result in appendiceal perforation and mucinous cystadenoma rupture. Etiologies include anatomical variations of the appendix and pathological conditions [6]. Especially in patients with rectal cancer, pseudomyxoma peritonei is difficult to detect.

Case Description A 57-year-old woman presented to our hospital with a 6month history of hematochezia and a 1-month period of changes in defecation habits. She reported that mild swelling and pain had occurred in the right lower quadrant of her abdomen three times during the past 3 years, lasting

Z. Sun : H. Wang (*) : X. Yu : Z. Zhao : Q. Wang Department of Gastrointestinal Surgery, Affiliated Tumor Hospital, Xinjiang Medical University, 789 Suzhou East Street, Xinshi District, Urumqi 830011, China e-mail: [email protected] Z. Sun Research Laboratory of Disease Genomics, Cancer Research Institute, Central South University, Changsha 410008, China

approximately 90 min each time. There was no history of anemia, abdominal distension, constipation, nausea, or vomiting. She had lost approximately 5 kg in weight within the past 6 months. Upon presentation, the patient had tenderness in the right lower quadrant of her abdomen, but no rebound tenderness or muscular tension upon abdominal examination. A mobile and soft mass of approximately 5 cm×7 cm in diameter could be palpitated. A CT scan revealed a concentric ring pattern in the right lower quadrant of her abdomen (Fig. 1). A colonoscopy revealed a round neoplasm in the ileocecus (Fig. 2) and placenta percreta, which were removed; a biopsy showed active inflammatory cells. There was also a round cyst of 3 cm in diameter in the left attachment area. The plasma levels of carcinoembryonic antigen and carbohydrate antigen 19–9 were 74 μg/L and 509.4 U/L, respectively. Based on colonoscopy, pathology, and CT, possible diagnoses were moderately differentiated carcinoma of the rectum (cT4N1M0), ileocecal intussusception, appendicular cystis, or left oviduct cystis. During an exploratory operation, we observed that approximately two thirds of the appendix (15 cm×5 cm) was intussuscepted into the cecum (Fig. 3); the appendix could slide in and out of the cecum. An intact resection of both the appendix and ileocecum was performed. Histopathology during surgery showed appendicular mucinous cystadenoma without rupture. There was no pseudomyxoma in other organs of the abdominal or pelvic cavities. Because of the extent of the initial excision, no further enlarging resection or chemotherapy infusion therapy was necessary. The left oviduct cyst was excised, and a biopsy of the left mesonephric source mesosalpinx and radical resection of the rectal carcinoma were carried out. Postoperative pathology confirmed appendicular mucinous cystadenoma (Fig. 4) and rectal medium-low differentiated adenocarcinoma of the ulcer type with Duke’s C (pT4N2M0). Subsequently, the

J Gastrointest Canc

Fig. 1 The CT scan revealed a concentric ring pattern of approximately 5 cm×7 cm in diameter in the right lower quadrant of the abdomen (indicated by a white arrow)

patient was treated with regular chemotherapy for rectal carcinoma.

Discussion Appendiceal mucinous cystadenoma is uncommon. Although its etiology is unclear, it has been reported in patients with appendiceal anatomical variations and pathological conditions [7, 8]. The inflammation causes the interface between the appendix and the cecum prone to stenosis or occlusion. In appendiceal mucinous cystadenomas, secreted mucus gathers in the appendiceal cavity, causing it to expand and thinning the mucosal wall, finally forming an oval or round cyst [9, 10]. In the case reported here, the symptoms were not typical of acute appendicitis. She presented with typical symptoms of rectal cancer, including hematochezia and alteration of defecation habits and anal feeling.

Fig. 3 In intraoperative exploration, the appendix was intussuscepted into the cecum (indicated by a white arrow)

Appendiceal mucinous cystadenoma must be differentiated from mesenteric cysts or ovarian cysts. When intussuscepted into the cecum, appendiceal mucinous cystadenomas become more difficult to diagnose [11–13]. Multiple tests are helpful, including a barium enema, CT scan, ultrasonic inspection, and colonoscopy. In a CT scan, the intussuscepted appendix has the appearance of concentric rings like that of other types of cecal intussusception (Fig. 1). However, it is very difficult to preoperatively explicitly diagnose appendicular mucinous cystadenoma. By colonoscopy, intussusception of the appendix may appear as a pedunculated polyp. Endoscopists should be aware of this unusual condition, as excision may result in peritonitis [7]. In the case reported here, colonoscopy showed a neoplasm in the cecum and a small piece of tissue was taken for biopsy; fortunately, this procedure did not lead to perforation. Therefore, upon colonoscopy, intussusception biopsy is not advocated. Surgical treatment depends on the associated pathology. Antibiotic treatment is usually ineffective. If the appendix is not split, it can be completely cured by intact resection; if split, the fragments usually disseminate and implant in the enterocoelia and pelvic cavity, forming pseudomyxoma peritonei that are nearly impossible to cure.

Conclusion

Fig. 2 Colonoscopy revealed a round neoplasm (indicated by a white arrow) of 3 cm in diameter in the ileocecus that displayed signs of inflammation

It is rare that appendiceal mucinous cystadenoma results in appendiceal intussusception into the cecum, especially in patients with rectal carcinoma. Although most cases are diagnosed during surgical exploration, it is helpful to use various methods preoperatively. Extraction of tissues for biopsy during colonoscopy is not advocated as this procedure can lead to perforation and pseudomyxoma peritonei formation. An intact excision is essential in the treatment of appendicular intussusception with appendiceal mucinous neoplasms.

J Gastrointest Canc Fig. 4 Postoperative pathology demonstrated appendicular mucinous cystadenoma, indicated by white arrows (H&E staining, ×200 magnification)

Conflict of Interest The authors declare that they have no conflict of interest.

References 1. Pulighe F, Paliogiannis P, Cossu A, et al. Molecular analysis of appendiceal mucinous cystadenoma and rectal adenocarcinoma in a patient with urothelial carcinoma: a case report. J Med Case Rep. 2013;7(1):170. 2. Sturniolo G, Barbuscia M, Taranto F, et al. Mucocele of the appendix. Two case reports. G Chir. 2011;32(11–12):487–90. 3. Laalim SA, Toughai I, el Benjelloun B, et al. Appendiceal intussusception to the cecum caused by mucocele of the appendix: laparoscopic approach. Int J Surg Case Rep. 2012;3(9):445–7. 4. Duncan JE, DeNobile JW, Sweeney WB. Colonoscopic diagnosis of appendiceal intussusception: case report and review of the literature. JSLS. 2005;9:488–90. 5. Ram AD, Peckham C, Akobeng AK, Thomas AG, David TJ, Patel L. Inverted appendix mistaken for a polyp during colonoscopy and leading to intussusception. J Cyst Fibros. 2005;4:203–4.

6. Yildirim U, Ozaydin I, Erdem H, et al. Intussusception of the appendix induced by sessile serrated adenoma: a case report. Turk Patoloji Derg. 2012;28(3):290–2. 7. T˘aban S, Dema A, Laz˘ar D, Sporea I, et al. An unusual “tumor” of the cecum: the inverted appendiceal stump. Romanian J Morphol Embryol. 2006;47:193–6. 8. Thomas RE, Maude K, Rotimi O. A case of an intussuscepted neuroendocrine carcinoma of the appendix. World J Gastroenterol. 2006;12(6):971–3. 9. Kaya C, Yazici P, Omeroglu S, et al. Laparoscopic appendectomy for appendiceal mucocele in an 83-year-old woman. World J Gastrointest Surg. 2013;5(6):207–9. 10. Yoshida Y, Sato K, Tada T, et al. Two cases of mucinous cystadenoma of the appendix successfully treated by laparoscopy. Case Rep Gastroenterol. 2013;7(1):44–8. 11. Kalogiannidis I, Mavrona A, Grammenou S, et al. Endometrial adenocarcinoma and mucocele of the appendix: an unusual coexistence. Case Rep Obstet Gynecol. 2013;2013:892378. 12. Zaharie F, Tomuş C, Mocan L, et al. Intestinal obstruction secondary to appendiceal mucocele. Chirurgia (Bucur). 2012;107(6):802–4. 13. Alese OB, Irabor DO. Mucinous cystadenoma of the appendix: a case report. Afr Health Sci. 2010;10(1):99–100.

Appendiceal mucinous cystadenoma intussuscepted into the cecum on a patient with rectal carcinoma: a case report.

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