Lymphangiomatosis of the sigmoid colon - a rare cause of lower gastrointestinal bleeding: A case report and review of the literature

Abstract

Abstract. The present study reports the case of a 79-year-old Chinese man who presented to The First Affiliated Hospital of Xi'an Jiaotong University (Xi'an, China) for the treatment of lower gastrointestinal bleeding. Multiple cystic masses in the sigmoid colon were observed with colonoscopy, and through endoscopic ultrasound (EUS), these cystic masses were confirmed to be echo-free and to exhibit septal walls in the submucosal layer; in consequence, lymphangiomatosis of the sigmoid colon was diagnosed. Considering the repeated bleeding, laparoscopy-assisted partial sigmoid colon resection was performed. The excised specimens were multiple vesicular and soft masses of ~1 cm in diameter, which were located in the submucosal layer and were surrounded by flat endothelial cells. Immunohistochemistry revealed that the specimens were positive for the specific lymphatic endothelial marker D2-40. The pathological diagnosis was consistent with the EUS findings. In the 2-year follow-up after the operation, no bleeding or other complications were noticed.

Lymphangiomatosis of the ileum with perforation: A case report and review of the literature

“…The GI tract may be affected, but the incidence of lymphangioma of the intestinal wall is very low, and it is even rarer in the small bowel (less than 1%) [ 9 , 10 ]. To the best of our knowledge, all cases reported in the literature with GI tract involvement are summarized in Table 1 [ 1 , 2 , 7 , [10] , [11] , [12] , [13] , [14] , [15] , [16] , [17] , [18] ].…”

“…The cells were positive for CD31, CD34 and D2-40. Ki-67 was about 1%.9-month history of intermittent melena, weakness and palpitationabdominal MR, abdominal CT, PET-CT and biopsyresection of the abdominal mass and a segment of 60 cm of the ileum invaded by the abdominal massafter surgery symptoms improved and follow-up laboratory tests showed normal red blood cell count and hemoglobin levelLu G, Li H et al [17]79-year-old mansigmoid colonmultiple cystic masses (colonscopy), with spetal walls in the submucosal layercysts located in the submucosal layer surrounded by flat endothelial cells that were positive for D2-40 at the immunoistochemistryintermittent attacks of bowel bleeding and abdominal discomforts for 3 monthscolonscopy, endoscopic ultrasound and biopsylaparoscopy-assisted partial sigmoid colon resectionin the 2-year follow-up after the operation, no bleeding or other complications were noticedXue L, Guo WG et al [18]58-year-old manlower esophaguslongitudinally protruding mass covered with normal esophageal mucosa and a lesion outside but adjacent to the wall of the esophagusmultiple dilated lymphatic vessels of a different sizes filled with pink beneath squamous epitheliumdysphagia of 7 monthsesophagogastroscopy, esophageal ultrasonography, chest CT and biopsya right lateral thoracotomy was performed fot the resection of the cysts, first the lesion outside and than that protruding in to the esophageal lumenthe postoperative course was uneventful and at the patient was discharged on th 10th postoperative day…”

Lymphangioma of Colon Presenting as an Intramural Tumor

“…15,16,18,19 The role of endoscopic ultrasound (EUS) in the definitive and differential diagnosis of CLCs has been widely reported. 11,15,[18][19][20] EUS with the advantage of direct access to the surface of the colonic mucosa, can provide precise information about the layer of origin and contour of the submucosal tumor, especially with high accuracy in the diagnosis. On EUS, CLCs are confined to the submucosal layer with well-defined margins containing anechoic cystic spaces with septa and intact muscularis propria.…”

“…Endothelial cells stain positive with lymphatic endothelial markers such as D2-40 marker and also for vascular markers including CD34-, CD31-, factor VIII-related antigen and VEGFR3. 11,13…”