We present an adult case of intussusception caused by subserosal inflammatory pseudotumor of the terminal ileum. Enteric intussusception was diagnosed by characteristic Xray finding, so called "beak-like" filling defect in the terminal ileum. An exploratory laparotomy revealed focal subserosal thickening in the terminal ileum, and partial ileocolectomy was performed. Microscopically, we identified a pseudotumor that extended through the muscularis propria into serosa. The pseudotumor was composed of fibrous stroma and chronic inflammatory cells with calcification

Aramaki Takahisa

Fujiwara Takashi

Higami Yoshikazu

Ishigaki Masaru

Isomoto Hajime

Kanazawa Shigeo

Kohno Shigeru

Mizuta Yohei

Mukai Shigenori

Murase Kunihiko

Murata Ikuo

Nagae Takaaki

Nagata Hiroshi

Nishiyama Jin

Shimokawa Isao

Takeshima Fuminao

Nagasaki University's Academic Output SITE: NAOSITE

Acta Med. Nagasaki 44 : 73-76Case ReportSubserosal Inflammatory Pseudotumor Causing IntussusceptionHiroshi NAGATA 1), Kunihiko MURASE 3), Jin NISHIYAMA 1), Takahisa ARAMAKI 1), Shigenori MUKAI 2), Takaaki NAGAE 2), Takashi FUJIWARA 2), Shigeo KANAZAWA 2), Masaru ISHIGAKI 2), Hajime ISOMOTO 3), Fuminao TAKESHIMA 3), Yohei MIZUTA 3), Yoshikazu HIGAMI 4), Isao SHIMOKAWA 4), Ikuo MURATA 5), Shigeru KOHNO 3)1) Department of Internal Medicine, Mitsugi General Hospital 2) Department of Surgery, Mitsugi General Hospital 3) Second Department of Internal Medicine, Nagasaki University School of Medicine 4) First Department of Pathology, Nagasaki University School of Medicine 5) Department of Pharmacotherapeutics, Nagasaki University Postgraduate School of Pharmaceutical Science We present an adult case of intussusception caused by subserosal inflammatory pseudotumor of the terminal ileum. Enteric intussusception was diagnosed by characteristic X-ray finding, so called "beak-like" filling defect in the termi-nal ileum. An exploratory laparotomy revealed focal subserosal thickening in the terminal ileum, and partial ileocolectomy was performed. Microscopically, we identified a pseudotumor that extended through the muscularis propria into serosa. The pseudotumor was composed of fibrous stroma and chronic inflammatory cells with calcification.Key words: intussusception, inflammatory pseudotumor,            subserosaCASE REPORT A 29-year-old male presented in September 1998 with a sudden-onset lower abdominal pain accompa-nied by episodes of nausea and vomiting. Past history revealed ileus one year earlier. These symptoms wors-ened gradually, and he was admitted to our hospital for further evaluation. On physical examination, the abdomen was flat and soft with tenderness in the lower abdomen. Laboratory data demonstrated mild anemia (hemoglobin 11.5g/ dl) but other values were within normal limits.  Plain X-ray of the abdomen showed intestinal ob-struction with dilated small bowel loops and air-fluid levels suggesting small bowel obstruction (Fig. 1). HeIntroduction Intussusception is primarily a disease of infants and children, and is relatively rare in adults, where it is sometimes difficult to diagnose."' The preoperative di-agnosis in the majority of patients is bowel obstruc-tion."') In adults, it is usually caused by benign'-') or malignant neoplasms,10-13) and benign lesions in the small intestine constitute half of the cases.") Inflammatory bowel lesions rarely cause intussusception.15-'S) We re-port here a rare adult case of intussusception caused by a subserosal inflammatory pseudotumor in the ter-minal ileum.Address Correspondence: Kunihiko Murase, M.D. Second Department of Internal Medicine, Nagasaki University School of Medicine, 1-7-1 Sakamoto, Nagasaki 852-8501, Japan TEL: +81-95-849-7273 FAX: +81-95-849-7285Fig. 1. Plain X-ray of the abdomen showing intestinal ob-struction with dilated small bowel loops and air fluid level.was treated by naso-jejunal tube. X-ray examination of the small intestine with gastrografin showed a "beak-like" filling defect in the terminal ileum (Fig. 2). Ultrasonography failed to show a "target-like" appear-ance. Abdominal CT showed cecal wall thickening (Fig. 3). Barium enema showed filling defect in the cecum (Fig. 4). Colonoscopy revealed edematous and reddish mucosa but no tumor was found in the cecum (Fig. 5), and biopsy showed no evidence of neoplasia. These finding suggested that the patient had an intu-ssusception.  At laparotomy, the intussusception, was spontaneouslyresolved but there was focal thickening of the serosa at the distal 20 cm of the terminal ileum. Therefore, partial ileocolectomy was performed to prevent a caus-ing of recurrent ileus. Grossly, the mucosa appeared intact. Microscopically, the lesion extended through the muscularis propria into the serosa (Fig. 6A). The lesion was composed of fibrous stroma and chronic in-flammatory cells including lymphocytes, plasma cells (Fig. 6B) with calcification (Fig. 6C) providing a diag-nosis of inflammatory pseudotumor. Postoperative course was satisfactory, and the patient remains in good health two years after the operation.Fig. 3. Abdominal CT showing cecal wall thickening.Fig. 2. X-ray examination of the small intestine with gastrografin showing the so called "beak-like" filling defect in the terminal ileum.Fig. 5. Colonoscopy showed an edematous and reddish mucosa with no tumor in the cecum.Fig. 4. Barium enema showing a filling defect in the cecum.Fig. 6AFig. 6B Fig. 6CMicroscopically, the lesion extended through the muscularis propria into the serosa (A) (X 10). The pseudotumor was composed of fibrous stroma and a mixture of chronic inflammatory cells including lymphocytes, plasma cells (B) (x200), and contained areas of calcification (C) (X200).Discussion Intussusception in adults is almost invariably caused by some preexisting lesions, including benign tumors, such as leiomyoma,5.6' hamartoma,''8' and neurofibroma,9' or malignant neoplasms.'° 13' Furthermore, trauma,"' post-operative adhesion '20 2J Meckel's diverticulum,2 22' and lymphoid hyperplasia23' are other potential predispos-ing factors in the production of intussusception.  Inflammatory pseudotumor is not a true neoplasia. It occurs rarely in the small intestine, and a solitary orpolypoid lesion may present clinically as intussusception and bowel obstruction.""' In the small intestine in-flammatory pseudotumors are commonly located in the mucosal layer of the ileum."") In our case, the pseudotumor was located in the muscularis propria and serosa, that are rare sites for this tumor. The true nature of pseudotumor in the present case is obscure. There are several pathoetiologic factors as for inflam-matory pseudotumor reported in the litereture.15 21' Although the microscopic findings were similar to the post-operative change, 1120) he has no history of abdominal operation. Ramsden et al.") reported a 76-year-old manwith recurrent intussusception associated with vascu-lar proliferation."' The present case did not have such a vascular change. One of the histologic feature in our case is calcification within the tumor. Entero-colic cal-cification is probably the most common radiographic manifestation of schistosomal infestation of the gastroin-testinal tract.25' Kimura et al.26> reported a patient with a complete invagination of the cecum that contained two large laminated calcified fecaliths. Most of the eggs of the schistosomioma are deposited in the submucosa, whereas serosal calcification is rare. 25,26) In the present case, there were no granulomas, fecalis nor pathogens. Subsequently, etiology of the pseudotumor was not determined.  Mesenteric panniculitis is an extremely rare disease in which the adipose tissue is replaced by fibrosis, ne-crosis, and calcification.27> However, our case did not show any of the features of mesenteric panniculitis.  In conclusion, we have described a rare case of intussusception caused by subserosal inflammatory pseudotumor of the terminal ileum, although the etiol-ogy of the lesion remains obscure.References1) Laws HL, Aldrete JS. Small-bowel obstruction: a review of 465    cases. South Med J 69:733-4, 1976 2) Gordon RS, O'Dell KB, Namon AJ, Becker LB. Intussusception in    the adult; a rare disease. J Emerg Med 9:337-42, 1991 3) Reijne HA, Joosten HJ, de Boer HH. Diagnosis and treatment of    adult intussusception. Am J Surg 158:25-8, 1989 4) Dharia KM, Marino AW Jr, Mancini HW. Enterocolic intussusception    in adults. Dis Colon Rectum 15:194-200, 1972 5) Mansberg VJ, Mansberg G, Doust BD. Jejunojejunal intussusception    secondary to leiomyoma. Austral Radiol 40:72-4, 1996 6) Gourtsoyiannis NC, Bays D, Malamas M. Radiological appearances    of small intestinal leiomyomas. Clin Radiol 45:94-103, 1992 7) Yamagami T, Tokiwa K, Iwai N. Myoepithelial hamartoma of the    ileum causing intussusception in an infant. Pediatr Surg Intern    12:206-7, 1997 8) Gonzalvez J, Marco A, Andujar M et al. Myoepithelial hamartoma   of the ileum: a rare cause of intestinal intussusception in children.   Eur J Pediatr Surg 5:303-4, 19959) Watanuki F, Ohwada S, Hosomura Y et al. Small ilea] neurofibroma     causing intussusception in a non-neurofibromatosis patient. J    Gastroenterol 30:113-6, 1995 10) Hurwitz LM, Gerlten SL. Colonoscopic diagnosis of ileocolic    intussusception. Gastroint. Endoscopy 32:217-8, 1986 11) Felix EL, Cohen MH, Bernstein AD, Schwartz JH. Adult intussusception:    case report of recurrent intussusception and review of the litera-   ture. Am J Surg 131:758-61, 1976 12) Boyle MJ, Arkell LJ, Williams JT. Ultrasonic diagnosis of adult    intussusception. Am J Gastroenterol 88:617-8, 1993 13) Begos DG, Sandor A, Modlin IM. The diagnosis and management    of adult intussusception. Am J Surg 173:88-94, 1997 14) Azar T, Berger DL. Adult intussusception. Ann Surg 226;134-8,    1997 15) LiVolsi VA, Perzin KH. Inflammatory pseudotumors (inflammatory    fibrous polyps) of the small intestine: A clinicopathologic study.    Am J Dig Dis 20:325-36, 1975 16) Nkanza NK, King M, Hutt MS. Intussusception due to inflamma-   tory fibroid polyps of the ileum: a report of 12 cases from Africa.    Br J Surg 67:271-4, 1980 17) Winkler H, Zelikovski A, Gutman H et al. Inflammatory fibroid    polyp of the jejunum causing intussusception. Am J Gastroenterol    81:598-601, 1986 18) Myint MA, Medeiros LJ, Sulaiman RA et al. Inflammatory    pseudotumor of the ileum. A report of a multifocal, transmural le-   sion with regional lymph node involvement. Arch Pathol Lab Med    118:1138-42, 1994 19) Komadina R, Smrkolj V. Intussusception after blunt abdominal    trauma. J Trauma. 45:615-6, 1998 20) Rozsos 1. Ileal intussusception, caused by fibroma, after cholecystectomy    performed under microlaparotomy. Orvosi Hetilap 135:1645-7, 1994 21) Mordkin R, Gibbons MD, Cain MP. Ileal-ileal intussusception: an    unusual complication following enterocystoplasty. Urology 47:138-   9, 1996 22) Daneman A, Myers M, Shuckett B, et al. Sonographic appearances    of inverted Meckel diverticulum with intussusception. Pediatr    Radiol 27:295-8, 1997 23) Pang LC. Intussusception revisited: clinicopathologic analysis of    261 cases, with emphasis on pathogenesis. South Med J 82:215-28,   1989 24) Ramsden KL, Newman J, Moran A. Florid vascular proliferation in    repeated intussusception mimicking primary angiomatous lesion. J    Clin Pathol 46:91-2, 1993 25) Fataar S, Bassiony H, Hamed MS et al. Radiographic spectrum of    rectocolonic calcification from schistosomiasis. Am J Roentgenol   142:933-6, 1984 26) Kimura H, Satou T, Takahashi N et al. Intussusception of the    cecum with calcified fecaliths. Am J Gastroenterol 90:318-9, 199527) Wexner SD, Attiyeh FF. Mesenteric panniculitis of the sigmoid    colon. Report of two cases. Dis Colon Rectum 30:812-5, 1987

Subserosal Inflammatory Pseudotumor Causing Intussusception

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Subserosal Inflammatory Pseudotumor Causing Intussusception

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