Vascular malformations of the small intestine manifesting as chronic anemia: Two pediatric cases managed by single-site umbilical laparoscopic surgery

Abstract

Introduction: Vascular malformations affecting abdominal viscera, especially the gastrointestinal tract, are less common than that in other body segments. Nonetheless, it seems to be one of the important causes of gastrointestinal bleeding in not only adults but also children as well. It occurs during the development stage of vascular system, and may increase in severity as the child grows.

Presentation of case: We present here two cases of lesions developed at the small intestine in an 8-year-old girl and 3-year-old girl, which were identified during the management for chronic anemia. Although there were some limitations associated with diagnosis, a histology confirmed the presence of arteriovenous malformations in both cases, they were successfully treated with surgical resection, especially minimal invasive procedure.

Discussion: Vascular malformations of abdominal viscera, especially the small intestine, are rare clinical manifestations in pediatric patients but are among the important causes of acute massive or chronic obscure LGI bleeding. Unless there is significant GI bleeding, patients are usually treated for anemia with obscure LGI bleeding. In the present study, selective angiography was useful in one case and CT enterogram with angiography was useful in the other case.

Conclusion: Considering the rarity and possibility of gastrointestinal bleeding due to vascular malformations, it is necessary to be regarded as one of differential diagnosis when managing a lower gastrointestinal bleeding in pediatric patients. Besides, a minimal invasive procedure could be suggested as a good surgical option when necessary.

Keywords: Minimal invasive; Pediatric; Small intestine; Vascular malformations.

Fig. 1

Selective angiography of Case 1 shows an early venous drainage with vascular tangle in distal jejunal branch (white arrow).

Fig. 2

Gross appearance shows tortuous vascular branch around mesenteric surface of the jejunum.

Fig. 3

Multiple dilated vascular structures of variable sizes and the thickness of the wall affecting the bowel submucosa, muscularis propria, and subserosa (H&E stain, ×20). In inlet, the abnormally dilated vein (left side of image) and artery (right side of image) (Elastic fiber stain, ×100).

Fig. 4

Enhanced CT angiography of Case 2 shows a segmental, circumferential wall thickening (A) and multifocal punctate enhancement on portal venous phase (B).

Fig. 5

Gross appearance shows a markedly dilated, engorged vascular structure encircling the wall of small intestine and a small discrete lesion apart from the main lesion in inlet.