International Journal of StudentS ReSeaRch

Volume 2 Issue 1 Year 2012 www.ijsronline.com

CASE SERIES

Adult intussusception by tumor in ileum: a diagnostic dilemma

Juan de Dios Diaz-Rosales, Lenin Enriquez-Dominguez, Jose R Castillo-Moreno, Pedro G Gutierrez-Ramirez
ABSTRACT Intussusception in adults is an infrequent cause of intestinal obstruction; preoperative diagnosis is difficult as symptoms can be intermittent and long standing. This relatively rare entity differs from the childhood form in its etiology, presentation and treatment. We present two cases of ileo-colic intussusception in adults, with a clinical presentation that mimicked complicated appendicitis. These cases remind us that both ileal tumors and intussusception must be included in the diagnosis of lower abdominal pain. Key Words: Intussusception, carcinoid tumor, non-Hodgkin lymphoma, ileocecal valve. Introduction The intussusception exists when a proximal segment of bowel (intussusceptum) telescopes into the lumen of the adjacent distal segment (intussuscipiens) [1]. Although it is very common in pediatric population (1-4 cases by 1000 live births), this entity is rare in adults (1-3 cases by each millions of habitants by year), and represents 1% of patient with bowel obstructions and 5% of all intussusceptions [2]. In this report, we present two cases of ileo-colic intussusceptions in adults, both with a pre-operative diagnosis of complicated acute appendicitis and postoperative diagnosis of intussusception by a distal ileal tumor. Case Presentation Case 1 An 18-year-old Mexican male, without pathologic medical history was admitted in the emergency room (ER) of General Hospital of Ciudad Juarez (Mxico) with abdominal pain syndrome. He had a fifteen-day history of abdominal cramping pain (mild to moderate severity) in inferior right quadrant (IRQ); nausea and vomiting, hyperthermia, and malaise were present at the time of presentation. He was treated by his family physician with analgesics and antibiotics without improvement, and the symptoms persisted and the pain became severe and intractable. On examination, the patient was conscious, alert and oriented. He was afebrile with a heart rate (HR) of 92 beats per minute, respiratory rate (RR) 26 cycles per minute and blood pressure (BP) of 100/70 mmHg. Abdominal examination revealed a mass in the in Right Lower Quadrant (RLQ). McBurneys sign, Psoas sign and Obturator sign were positive. Blood work up showed raised white blood cell count at 11,680 per mm3. Other hematological parameters were unremarkable. Plain abdominal films demonstrated signs of intestinal obstruction, air-fluid levels in small bowel and absence of air in the large bowel. Ultrasonography revealed a mass in the RLQ, and evidence of free fluid in
Department of Surgery, Hospital General De Ciudad Juarez, Universidad Autonoma De Ciudad Juarez, Mexico Corresponding Author Juan de Dios Diaz-Rosales, Email: [email protected]

lower abdomen. The pre-operative diagnosis of acute appendicitis and Meckels diverticulum was made. The patient underwent an emergent median laparotomy. During the operation, an ileo-ceco-colic intussusception was found; during reduction, a small mass into the ileal lumen was discovered. It was located in the region of distal ileum (Figure 1A). A right hemicolectomy with resection of affected ileal segments (approximately 20 cm) was performed. The continuity of the digestive tube was reestablished by primary double-layer end-to-end ileumtransvers anastomosis with 3.0 silk and 3.0 polyglactin 910. The anatomical pathology report revealed a submucosal tumor of the ileum, about 3 cm x 3 cm, with features of nonHodgkin lymphoma (intermediate grade) (Figure 2A). The post-operative period was uncomplicated and he was discharged on the 7th day following surgery. At the time of discharge, his presenting symptoms resolved and was referred to the oncology service for further assessment.

Figure 1 Gross image. A: Case 1- Mass resected from the ileal lumen. B: Case 2- Transoperative enterotomy of ileum showing a polypoid mass in the inner of the lumen

Case 2 A 25-year-old Mexican female, without pathologic medical history was admitted in the ER of our hospital with abdominal pain syndrome. The patient presented with a three-day history of abdominal discomfort with intermittent abdominal cramping pain of mild to moderate in severity, and localized around the mesogastric area, and progressed gradually into a severe pain with radiation to the right iliac quadrant. She also had nausea accompanied by 3 episodes of vomiting. She was afebrile with stable vital signs. Examination revealed a distended abdomen with rigidity and guarding with raised local temperature. An abdominal mass was palpated in the RLQ; Rovsing and Giordano signs were

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International Journal of StudentS ReSeaRch

Volume 2 Issue 1 Year 2012 www.ijsronline.com
CASE SERIES

elicited. Bowel sounds were absent. Laboratory results were unremarkable except for mild elevated white blood counts at 12,600 per. Plain abdominal films showed air-fluid levels, with the sentinel loop sign in RLQ. A diagnosis of small bowel obstruction by complicated appendicitis was made. The patient underwent an emergency median laparotomy. During surgery, an ileo-cecal intussusception was found and reduced. A small mass of size 2 cm x 3 cm in the lumen of ileum was discovered (Figure 1B). The appendix appeared without evidence of pathology. A resection en-block of the affected segment was performed. The histopathological examination revealed a submucosal endocrine-like tumor of the ileum with homogenous aspect formed by monotonous sheet of cells containing small compact nuclei that were uniform and contained dense granules, forming nests with invasion to serous layer, suggestive of a well differentiated invasive carcinoid tumor (Figure 2B). The patient underwent a second operation on the 3th post-operative day, and a right hemicolectomy was performed, there was no evidence of nodes or metastases (IIIA). Post-operative period was uneventful and she was discharged on the 5th post-operative day. Following discharge she was referred to the oncological service for further evaluation.

secondary to a definable lesion (intra-luminal lesions in 90%) in adults patients [2-4]. The mechanism of development is believed that any lesion in the intestinal wall alters normal peristalsis, and is able to initiate an invagination [5]. The intussusception is broadly classified as: jejuno-jejunal, ileo-ileal, ileo-colic, ileo-cecal-colic, colo-colonic, sigmoidorectal, and appendicico-cecal [5,6]. The adult intussusception presents with a variety of non-specific symptoms that can have an acute, intermittent, and or chronic abdominal pain [7] and bowel obstruction which can either be complete or intermittent [8]. Only about 9 to 10% of adult patients present with the typical triad of abdominal pain, palpable abdominal mass and bloody stool, and hence, the preoperative diagnosis is difficult [9]. In our cases, the core symptom was abdominal pain; insidious in the teenager and acute in adult patient. With such a presenting symptomatology and a palpable mass in the RLQ, a diagnosis of appendicitis was made. Although the large evolution of the boy was atypical for appendicitis, for this, the differential diagnosis was Meckels diverticulum. However intra-operatively, this appendicitismimicking presentation turned out to be an ileo-ceco-colic intussusceptions. Abdominal ultrasound and Computed Tomography (CT) scan are the most effective diagnostic methods [2], although a number of different methods are available: barium imaging, endoscopic examination, and angiographic and radionucleotide studies [9]. Abdominal ultrasound and CT scan have high sensibility and specificity; and both shows a classic image of target or sausage shape mass. Endoscopic examination is therapeutic in children, but in adults it does not have any considerable hydrostatic reduction effect, because of high incidence of underlying anatomical abnormalities [10], like these cases (inner ileal tumors). Our clinical scenario comprised of patients with abdominal pain syndrome in an ER of a secondary health care hospital in Mexico. All the patients presenting with such a symptomatology are clinically evaluated and subjected to an abdominal x-ray and subsequently abdominal ultrasonography. Similar situation exists in many developing countries where facilities of high resolution imaging might not be available specially in secondary health care catering centers and the diagnosis is mostly relied upon clinical impression and basic imaging modalities like the roentgenogram and sonography. In such a setting, like in our cases, a high suspicion of a possible intussusception should be considered as this may pose a diagnostic dilemma to the clinician where facilities of Magnetic Resonance Imaging (MRI) and CT scanning are not available. In adults, surgery is necessary and imperative, because of the high incidence of underlying anatomical abnormalities [5,6]. Most studies agree that resection is necessary; this could be en-block [6], because this entity is secondary to a neoplasm into the lumen of bowel, and majority are malignant. It remains debatable whether reduction should be attempted or whether en-block resection should be carried out without attempting reduction [9,10]. In the first patient, we performed a hemicolectomy, because we suspected that the tumor was malignant. In the second patient, we did know

Figure 2 Histopathological examination (100X magnification). A: Lymphoid tissue with loss of architecture, proliferations of atypical lymphoid cells. B: Cells with eosinophilic cytoplasm and hyperchromic nucleus that depicts a carcinoid tumor.

Discussion While intussusceptions in children have an idiopathic etiology in 95% cases, the condition is almost always Diaz-Rosales et. al. Int J Stud Res 2012;2(1):18-20

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International Journal of StudentS ReSeaRch

Volume 2 Issue 1 Year 2012 www.ijsronline.com
CASE SERIES

exact the origin of the tumor, and thus the resection of only the distal ileum and cecum was performed, as we did not have a definitive diagnosis of carcinoid tumor until the pathological evaluation. Following the histopathological analysis, the patient underwent a hemicolectomy without complications. Conclusion A differential diagnosis of small bowel intussusception should be kept in consideration in cases presenting with lower abdominal pain. The clinical presentations of such cases may at times mislead to a definitive diagnosis and often cause a diagnostic dilemma, especially in centers where high resolution imaging modalities are not available. A high suspicion index by the clinician is imperative to not rule out an ileal pathology causing intussusceptions in such cases. The management of such cases consists of surgical resection en-block, however the resection of only the affected segment can be validated when the diagnosis of pathology is reported, and resection en-block could be performed later. References
1. 2. 3. 4. Soni S, Moss P, Jaiganesh T. Idiopathic adult intussusception. Int J Emerg Med 2011;4:8. Azar T, Berger D. Adult intussusception. Ann Surg 1997;226(2):1348. Kim JH, Lee KM, Yun SH, et.al. Ileocecal intussusception in an adult: a case report. Turk J Gastroenterol 2007;18:50-2. Valera JM, Maiza E, Contreras L, Smok G. Tumores de intestino delgado: Experiencia de 11 aos en un centro de referencia. Gastr Latinoam 2004;15:180-5. Krasniqi AS, Hamza AR, Salihu LM et.al. Compound double ileoileal and ileocecocolic intussusception caused by lipoma of the ileum in an adult patient: a case report. J Med Case Reports 2011;5:452. Basar O, Odemis B, Ertugrul E. Ileo-ileal invagination caused by lymphoma. Chin Med J 2007;120:1119-20. Montiel-Jarqun AJ, Garca-Ramrez UN, Reyes-Pramo P, BlancaDaz J, Ruz-Len B. Invaginacin intestinal en el adulto joven. Rev Med Inst Mex Seguro Soc 2008;46:681-4. McCawley N, Collins CG, Barry M, McGuinness J, Leahy AL. Adult intussusception -need for en-bloc resection. Ir J Med Sci 2006;175:746. Wang N, Cui XY, Liu Y, et.al. Adult intussusception: a retrospective review of 41 cases. World J Gastroenterol 2009;15(26):3303-8. Begos DG, Sanor A, Modlin IM. The diagnosis and management of adult intussusception.Am J Surg 1997;173:88-94.

Please cite this paper as: Diaz-Rosales J, EnriquezDominguez L, Castillo-Moreno JR, Gutierrez-Ramirez PG. Adult intussusception by tumor in ileum: a diagnostic dilemma. Int J Stud Res 2012;2(1):18-20. doi: http://dx.doi.org/10.5549/IJSR.2.1.18-20 Received: 23 Nov 2011, Accepted: 15 Mar 2012 This is an Open Access article distributed under the terms of the Creative Commons Attribution License (http://creativecommons.org/licenses/by/3.0), which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.

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Consent Written informed consent was obtained from the patient for publication of this case report and any accompanying images. A copy of the written consent is available for review by the editor-in-chief of this journal. Authors Contributions PGG, LE and JDR participated in clinical diagnosis and surgery of both the patients. JRC, LE and JDR contributed with literature search and drafting of the manuscript. JRC and LE contributed with review of the manuscript. Competing Interests The authors declare that they have no competing interests. Funding Sources of funding- None

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