Ileo-ileal intussusception caused by lymphangioma of the small bowel treated by single-incision laparoscopic-assisted ileal resection

Abstract

Intraabdominal lymphangiomas are uncommon; additionally, those affecting the gastrointestinal tract are rare and account for less than 1% of cases. Intussusception caused by a cystic lymphangioma of the small bowel is extremely rare. The patient was a 20-year-old woman who visited our emergency room with a complaint of abdominal pain. A computed tomography image revealed ileo-ileal intussusception with a leading hypovascular mass measuring 1 cm in a diameter. Single-incision laparoscopic-assisted ileal resection was performed. The surgical specimen consisted of a soft polycystic mass. Macroscopically, a pedunculated polyp with a convolutional pattern was found. Microscopically, the inner surfaces of the cysts were covered with a single layer of endothelial cells. On immunohistochemical examination, the endothelial cells were partially positive for D2-40 and CD34. Smooth muscle cells were also found around the cysts. The lesion was diagnosed as a cystic lymphangioma. Dozens of cases of small bowel lymphangiomas have previously been reported. Of these, cases with intussusception were very rare. This is the first case of small bowel intussusception due to lymphangioma treated by single-incision laparoscopic-assisted surgery.

Small bowel lymphangioma causing ileo-ileal intussusception in adults

“…To our knowledge and with literature review there has been one more case reported of ileo-ileal intussusception has occurred due to lymphangioma in an adult [7] . The definitive treatment for gastrointestinal cystic lymphangioma is resection, with resection of the involved segment of bowel the most preferred procedure.…”

A case report of recurrent intussusception caused by small bowel lymphangioma in an adult

“…There have been several case reports on the patients with small intestinal lymphangioma causing intussusception in adults [ 1 , 3 , 4 , 6 ]. In all these reports, the patients presented with abdominal pain with or without nausea.…”

“…Lymphangiomas are benign tumors resulting from congenital lymphatic system malformation [ 3 ], generally located in the head and neck regions or the axilla. The gastrointestinal lymphangioma accounts for only 1% of all [ 4 , 5 ]. Small bowel lymphangioma causing intussusception is rare, there have only been a few reports published [ 1 , 3 , 4 , 6 ].…”

Jejunal lymphangioma causing intussusception in an adult: An unusual case with review of the literature

“…Although there has been evidence that ADPKD predisposes to renal cancer, there is new evidence that it could predispose to other types of cancer, specifically along the GI tract or in the liver [ [9] , [10] , [11] , [12] ]. Gene mutations could predispose individuals to tumor formation, through signaling pathway disruptions, specifically suppression of the apoptotic pathway [ 3 , 11 , 13 , 14 ]. The signaling derangements, specifically signaling inhibitors, found in ADPKD and solid tumors are strikingly similar [ 15 ].…”

“…Lymphangiomas are benign tumors comprised of congenitally dilated lymphatic channels and occur most often in the axilla, head and neck [ [1] , [2] , [3] ]. Lymphangioma of the GI tract comprises only 1% of lymphangioma diagnoses, but with increased utilization of endoscopic procedures the incidental discovery of these tumors in GI tract is increasing, leading us to believe the are more common than previously reported [ 2 , 4 ].…”