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A 32-year-old woman presented with half a year history of intermittent anemia. Physical
examination revealed anemic appearance, and the laboratory tests showed iron deficiency
anemia (hemoglobin was 62 g/L). Esophagogastroduodenoscopy was normal, whereas colonoscopy
showed dark-red coffee-like liquids filling the colon and terminal ileum. Computer
tomography angiography scan showed localized thickening, swelling, and low-density
of the local intestinal wall in the left middle and lower abdomen, without obvious
enhancement. A double-balloon enteroscopy revealed a hemispherical nodular mucosal
swelling lesion covered with white granular villi at the jejunum segment, and the
lesion manifested active bleeding when the endoscopic tube slightly squeezing it (Fig. 1a). Then, exploratory laparotomy was performed, and a 10 cm long lesion was observed
in the bowel. Longitudinal anatomical gross specimen, varied sizes and shapes of white
nodular eminences crowded and distributed on the lumen surface (Fig. 1b). Pathological diagnosis: hemolymphangioma accompanied by hemorrhage; the lesion
was confined to the mucosal and submucosal layer (Fig. 2a,b,c).
Fig. 1(a) Double-balloon enteroscopy demonstrated a hemispherical nodular mucosal swelling
lesion covered with white granular villi at the jejunum segment.(b) Completly resection
of lesion and longitudinal gross specimen dissection, varied sizes and shapes of white
nodular eminences crowded and distributed on the lumen surface.
Fig. 2Microscopic examination. (a) Low magnifification,H&E stain.The lesion was composed
of dilated thin-walled lumen, some of which were dilated and congested, some of which
were filled with lymphatic fluid,involving to the submucosa and mucosa of the small
bowel.(b) High magnifification,D2–40 stain showed positive lymphatic endothelial cell
membrane but negative vascular endothelial cell membrane. (c) High magnifification,CD-31stain
showed positive cell membrane of vascular and lymphatic endothelial cells.
