Intussusception in adulthood is unusual. We describe herein a rare case of adult ileoileal intussusception caused by an ileal lipoma, which was diagnosed preoperatively and was confirmed at the operation to have reduced spontaneously. A 68-year-old woman experienced sudden-onset colicky pain in the upper abdomen accompanied by vomiting and was brought to our hospital by ambulance. Physical examination revealed a distended abdomen and tenderness in the upper abdomen. Laboratory findings showed slightly elevated inflammatory parameters. Abdominal computed tomography (CT) showed a target sign in the ileum, which is a typical sign of intussusception. Additional caudal-side scans showed a homogenous and fatty mass measuring 2.5 cm that was considered to be the leading point for the invagination. These findings led to a preoperative diagnosis of intussusception induced by a lipoma. The patient underwent emergency surgery. Laparotomy revealed a yellowish, soft ileal tumor measuring 2.5 cm in diameter and that the intussusception had already been reduced at laparotomy. Approximately 15 cm of the ileum's length, including the tumor, was resected, and an end-to-end anastomosis was performed. Adult intussusception caused by an ileal lipoma is a rare condition. However, CT is the most useful tool for making a definite preoperative diagnosis based on its typical findings

Ishimoto, Tatsuro

Itamoto, Toshiyuki

Oishi, Koichi

Sueoka, Satoshi

Hiroshima University Institutional Repository

Hiroshima J. Med. Sci.Vol. 65, No. 3·4, 65~68, December, 2016HIJM 65–1065Intussusception involves the invagination of one segment of bowel together with its mesenteric fold into the lumen of an adjacent segment of bowel. Intussusception in adulthood is unusual, with an incidence of approximately 2-3 cases per 1,000,000 people per year7).  The occurrences of intussuscep-tions in adults account for less than 5% of all cases of intussusceptions and almost 1% of bowel ob-structions1,2,9).  There are major differences in the clinical presentation, diagnostic approach, and management of intussusception between the pedi-atric and adult populations8).Lipomas of the small intestine are rare benign tumors, and are most commonly encountered inci-dentally because they are usually asymptomatic. Symptomatic lipomas manifest with hemorrhage or intestinal obstruction18), and intussusception also constitutes a rare clinical entity.We describe herein a rare case of adult ileoileal intussusception caused by an ileal lipoma, which was diagnosed preoperatively and was confirmed to have reduced spontaneously at operation.CASE PRESENTATIONA 68-year-old woman experienced sudden-onset colicky pain in the upper abdomen with vomiting and was brought to our hospital by ambulance.  Her medical history included hypertension and arrhyth-mia.  She had undergone appendectomy for appendi-citis at the age of 22 years and cholecystectomy for acute cholecystitis at the age of 45 years.  She had begun chronic hemodialysis due to end-stage renal disease 8 years previously.  Physical examination re-vealed a distended abdomen and tenderness in the upper abdomen.  Laboratory findings showed slightly elevated inflammatory parameters.  Her white blood cell count was 9,200/μL, and her C-reactive protein level was 0.3 mg/dL.  Abdominal computed tomogra-phy (CT) showed a target sign in the ileum, which is a typical sign of an intussusception (Fig. 1a).  The intussusception corresponded to the entero-enteric type.  Further caudal-side scans showed a fatty (-100 Hounsfield Units) homogenous mass measuring 2.5 A Case of Spontaneously Reduced Ileoileal Intussusception Caused by a LipomaSatoshi SUEOKA1), Toshiyuki ITAMOTO1,2,*), Koichi OISHI1) and Tatsuro ISHIMOTO3)1) Department of Gastroenterological Surgery, Hiroshima Prefectural Hospital, Japan2) Department of Gastroenterological and Transplant Surgery, Applied Life Sciences, Institute of Biomedical and Health Sciences, Hiroshima University3) Department of Transplant Surgery, Hiroshima Prefectural HospitalABSTRACTIntussusception in adulthood is unusual.  We describe herein a rare case of adult ileoileal in-tussusception caused by an ileal lipoma, which was diagnosed preoperatively and was confirmed at the operation to have reduced spontaneously.  A 68-year-old woman experienced sudden-onset colicky pain in the upper abdomen accompanied by vomiting and was brought to our hospital by ambulance.  Physical examination revealed a distended abdomen and tenderness in the upper abdomen.  Laboratory findings showed slightly elevated inflammatory parameters.  Abdominal computed tomography (CT) showed a target sign in the ileum, which is a typical sign of intus-susception.  Additional caudal-side scans showed a homogenous and fatty mass measuring 2.5 cm that was considered to be the leading point for the invagination.  These findings led to a pre-operative diagnosis of intussusception induced by a lipoma.  The patient underwent emergency surgery.  Laparotomy revealed a yellowish, soft ileal tumor measuring 2.5 cm in diameter and that the intussusception had already been reduced at laparotomy.  Approximately 15 cm of the ileum's length, including the tumor, was resected, and an end-to-end anastomosis was per-formed.  Adult intussusception caused by an ileal lipoma is a rare condition.  However, CT is the most useful tool for making a definite preoperative diagnosis based on its typical findings.Key words: Intussusception, Computed tomography, Lipoma* Corresponding author: Toshiyuki Itamoto Department of Gastroenterological Surgery, Hiroshima Prefectural Hospital 1-5-54 Ujina-kanda, Minami-ku, Hiroshima 734-8530, Japan Tel: +81-82-254-1818, Fax: +81-82-252-6932, E-mail: t-itamoto@hph.pref.hiroshima.jp66 S. Sueoka et alan end-to-end anastomosis was performed.  The post-operative course was uneventful, and the patient was discharged on the thirteenth postoperative day.Macroscopic assessment of the resected specimen showed the presence of a submucosal tumor that was 2.5 × 2.0 × 1.5 cm in size with the features of lipoma (Fig. 3a, b).  Histological examination revealed ma-ture fat cells, connective tissue, and scattered blood vessels within the removed submucosal tumor. There was no evidence of dysplasia or malignancy (Fig. 3c).  The patient is alive without symptoms 5 years postoperatively.CONCLUSIONSThe exact mechanism that precipitates intussus-ception in adults is still unknown.  However, it is generally suggested that any lesion in the bowel wall or irritant within the bowel lumen could alter the normal peristaltic pattern and thereby initiate an invagination leading to intussusception16).  This pathologic process often results in obstruction.  In-tussusceptions have been classified according to the site of the lesion.  The most common classification system divides intussusception into four categories: enteric, ileocolic, ileocecal, and colonic5,16).  Two-thirds of intussusceptions in adults are of the en-cm that was considered to be the leading point for the invagination (Fig. 1b).  These findings led to a preoperative diagnosis of intussusceptions induced by a lipoma.The patient underwent emergency surgery.  Lapa-rotomy revealed a yellowish, soft ileal tumor measur-ing 2.5 cm in diameter and that the intussusception had already been reduced (Fig. 2).  Approximately 15 cm of ileum, including the tumor, was resected, and Fig. 1. Abdominal computed tomography: An axial image showing a typical target sign (a) and a fatty mass as the leading point of the intussusception (b).Fig. 2. Operative findings: Laparotomy revealed a yellowish, soft ileal tumor measuring 2.5 cm in diameter and that the intussusception had already been reduced.Fig. 3. Macroscopic and histologic findings of the excised specimen.a,b: A submucosal tumor, measuring 2.5 × 2.0 × 1.5 cm.c: Mature fat cells in the submucosa to the muscularis propria of the ileum (hematoxylin and eosin staining, × 100).67A Case of Ileoileal Intussusception Caused by a Lipomathan necessary to remove the tumor without reduc-tion.  Therefore, reduction should be attempted if the involved bowel segment is viable but only in pa-tients for whom a definitive benign diagnosis has been made preoperatively9,13,14).The rare case presented here could have a preop-erative definitive diagnosis of ileoileal intussuscep-tion caused by an intestinal lipoma.  Typical CT findings of intussusception and the existence of a li-poma as the leading point contributed to a definitive preoperative diagnosis.  In addition to the rarity of the disease, the present case developed a spontane-ous reduction that was confirmed at operation. There have been few reports of spontaneous reduc-tion of adult intussusception of the appendix12).  An-other report involved a case of intussusception caused by blunt trauma to the abdomen10).  These cases were confirmed to be spontaneously reduced on follow-up CT.  Spontaneous reduction in adult in-tussusception can be a phenomenon occurring in the setting of idiopathic or incidental intussusception. However, to the best of our knowledge, there have been no reports of spontaneous reduction of adult intussusception caused by neoplasms.In conclusion, adult intussusception caused by an intestinal lipoma is a rare condition.  CT is the most useful tool for making a definite preoperative diagnosis based on its typical findings.  Further-more, the present case highlights the possibility of spontaneous reduction of intussusception caused by neoplasms.ACKNOWLEDGEMENTSWe thank all the staff members employed at Hiroshima Prefectural Hospital, for treating and caring for this patient.(Received August 31, 2016)(Accepted September 23, 2016)REFERENCES1. Azar, T. and Berger, D.L. 1997. Adult intussuscep-tion. Ann. Surg. 226: 134-138.2. Balamoun, H. and Doughan, S. 2011. Ileal lipoma. A rare cause of ileocolic intussusception in adults: Case report and literature review. World J. Gastrointest. Surg. 3: 13-15.3. Barbiera, F., Cusmà, S., Di Giacomo, D., Finazzo, M., Lo Casto, A. and Pardo, S. 2001. Adult intesti-nal intussusception: comparison between CT features and surgical findings. Radiol. Med. 102: 37-42.4. Barussaud, M., Regenet, N., Briennon, X., de, Kerviler, B., Pessaux, P., Kohneh- Sharhi, N., et al. 2006. Clinical spectrum and surgical approach of adult intussusceptions: a multicentric study. Int. J. Colorectal. Dis. 21: 834-839.5. Briggs, D.F., Carpathios, J. and Zollinger, R.W. 1961. Intussusception in adults. Am. J. Surg. 101: 109-113.teric type15) that was seen in this case.  The clinical presentation is complicated and non-specific in adults.  Although abdominal pain is the most com-mon presentation, it is accompanied by varying symptoms and usually has a subacute or chronic onset.  Fewer than 20% of cases present acutely with complete bowel obstruction.  The diagnostic radiologic method that is most useful in making the diagnosis of adult intussusception is CT1).  It helps in both making an accurate diagnosis and also for identifying any associated underlying causes.  Pre-vious reports regarding adult intussusception showed that most patients had organic lesions as the leading point, and the majority of the lesions were neoplasms13,16).  A recent report showed that benign (24%) and malignant (21%) neoplasms were the most common etiology for adult intussusception; 30% of cases with any organic leading point were idiopathic15).The sensitivity of CT to diagnose intus-susceptions correctly has been reported to range from 58% to 90%, as verified by subsequent sur-gery3,4,11).Gastrointestinal lipomas are most commonly lo-cated in the colon (65% to 75%), small bowel (20% to 25%), and occasionally in the foregut (< 5%)6). Common symptoms of intestinal lipoma include abdominal pain (68%), intussusception (44%), hemorrhage (29%), vomiting (24%), and obstruc-tive symptoms (18%)17).  Thus, intussusception is a common complication of gastrointestinal lipoma, although the disease is classified as rare.  Lipo-mas account for 5.1% of the causes of adult intus-susception15).  Namikawa et al14) reviewed 50 cases of adult intussusception caused by a lipoma and reported in the English literature during the past decade.  The cohort comprised 28 men and 22 women with a mean age of 52.1 years.  The lipoma size ranged from to 1.2 to 12.0 cm, with a mean size of 4.9 cm.  Among them, 50% of lipomas were located in the small intestine, and the most fre-quent location was the ileum (21 of 25 cases).  A preoperative diagnosis of intussusception was made in 21 (42%) of 50 patients.  Their case was reported as the sixth case of adult ileoileal intus-susception caused by a lipoma diagnosed preopera-tively14).Surgical resection remains the recommended treatment for adult intussusception due to the large proportion of organic causes and the relatively high incidence of malignancy1,9,13).  The advantages of in-traoperative reduction of the intussusceptions prior to resection, especially when the small bowel is af-fected, are that it may preserve a considerable length of bowel and thereby prevent development of short-bowel syndrome, but most surgeons agree that reduction should not be attempted if there are signs of irreversible bowel ischemia, inflammation, or when malignancy is suspected.  However, the re-section of a long segment of intussusception often necessitates the excision of more of the intestine 68 S. Sueoka et al6. Chou, J.W., Feng, C.L., Lai, H.C., Tsai, C.C., Chen, S.H., Hsu, C.H., et al. 2008. Obscure gastrointestinal bleeding caused by small bowel lipoma. Intern. Med. 47: 1601-1603.7. Coleman, M.J., Hugh, T.B., May, R.E. and Jensen, M.J. 1981. Intussusception in the adult. Aust. N. Z. J. Surg. 51: 179-181.8. Demirkan, A., Yağmurlu, A., Kepenekci, I., Sulaimanov, M., Gecim, E. and Dindar, H. 2009. Intussusception in adult and pediatric patients: two different entities. Surg. Today 39: 861-865.9. Eisen, L.K., Cunningham, J.D. and Aufses, A.H. 1999. Intussusception in adults: institutional review. J. Am. Coll. Surg. 188: 390-395.10. Emet, M., Kocak, A.O., Akbas, I., Karaman, A. and Arslan, S. 2015. Spontaneous reduction of il-eoileal adult intussusception after blunt abdominal injury. Am. J. Emerg. Med. 33: 1542.11. Gayer, G., Apter, S., Hofmann, C., Nass, S., Amitai, M., Zissin, R., et al. 1998. Intussusception in adults: CT diagnosis. Clin. Radiol. 53: 53-57.12. Komine, N., Yasunaga, C., Nakamoto, M., Shima, I., Iso, Y., Takeda, Y., et al. 2004. Intussusception of the appendix that reduced spontaneously during follow-up in a patient on hemodialysis therapy. In-tern. Med. 43: 479-483.13. Nagorney, D.M., Sarr, M.G. and McIlrath, D.C. 1981.Surgical management of intussusception in the adult. Ann. Surg. 193: 230-236.14. Namikawa, T., Hokimoto, N., Okabayashi, T., Kumon, M., Kobayashi, M. and Hanazaki, K. 2012. Adult ileoileal intussusception induced by an ileal lipoma diagnosed preoperatively: report of a case and review of the literature. Surg. Today 42: 686-692.15. Onkendi, E.O., Grotz, T.E., Murray, J.A. and Donohue, J.H. 2011. Adult intussusception in the last 25 years of modern imaging: is surgery still in-dicated? J. Gastrointest. Surg. 15: 1699-1705.16. Weilbaecher, D., Bolin, J.A., Hearn, D. and Ogden, W. 1971. Intussusception in adults. Review of 160 cases. Am. J. Surg. 121: 531-535.17. Zografos, G., Tsekouras, D.K., Lagoudianakis, E.E. and Karantzikos, G. 2005. Small intestinal li-poma as a cause of massive gastrointestinal bleeding identified by intraoperative enteroscopy. A case report and review of the literature. Dig. Dis. Sci. 50: 2251-2254.18. Yao, T., Yao, K., Matake, H., Furukawa, K., Nagae, T., Motomura, A., et al. 2001. Primary small intestinal tumors [in Japanese with English abstract]. Stomach. Intestine. 36: 871-881.

A Case of Spontaneously Reduced Ileoileal Intussusception Caused by a Lipoma

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A Case of Spontaneously Reduced Ileoileal Intussusception Caused by a Lipoma

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