Abstract

BackgroundVarious terms have been used to describe vascular lesions in the intestine, including angiodysplasia, arteriovenous malformation, and telangiectasia. Such lesions are common in adults and are typified by angiodysplasia, a type of arteriovenous malformation. In contrast, these lesions are rarely seen in the pediatric population. Angiodysplasia may cause gastrointestinal bleeding, which is sometimes an indication for treatment. Considering the high rate of recurrence after surgical treatment, conservative treatments are mainly chosen. We herein report an extremely rare case of a prolapsed colon due to an arteriovenous malformation successfully treated by resection in a 1-year-old girl. We also highlight the differences between pediatric and adult cases.Case presentationA girl developed bloody stools at 7 months of age. She visited another hospital at 1 year of age because of continuing moderate hematochezia and recent onset of rectal prolapse. Colonoscopy showed a protruding lesion located 15 cm from the anal verge, suggesting a submucosal vascular abnormality. Contrast-enhanced computed tomography and magnetic resonance imaging at our hospital revealed the localized lesion with dilated blood vessels in part of the sigmoid colon; no other lesions were present in the gastrointestinal tract. Laparoscopic-assisted sigmoidectomy was performed. A subserosal vascular lesion was visualized and resected using end-to-end anastomosis. Pathologic examination of the 2.2 × 2.7-cm segment revealed several abnormally enlarged and ectatic blood vessels in the submucosa extending into the subserosa. The lesion was diagnosed as an arteriovenous malformation. The patient had a good clinical course without recurrence at the 2-year follow-up.ConclusionsAn arteriovenous malformation in the sigmoid colon may rarely cause intussusception and prolapse of the colon. Complete resection is a radical and potentially effective treatment. Computed tomography and colonoscopy were useful for evaluation of the lesion in the present case.

Highlights

  • Various terms have been used to describe vascular lesions in the intestine, including angiodysplasia, arteriovenous malformation, and telangiectasia

  • Computed tomography and colonoscopy were useful for evaluation of the lesion in the present case

  • In children, vascular anomalies such as infantile hemangiomas are more commonly seen in the skin and soft tissue, while they are rare in the GI tract [1, 2]

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Summary

Conclusions

Vascular anomalies such as infantile hemangiomas are more commonly seen in the skin and soft tissue, while they are rare in the GI tract [1, 2]. The lesion in our case was pathologically diagnosed as an AVM, the imaging findings were similar to those of a venous malformation, with the absence of an obvious feeding artery on CT and the presence of bowel wall thickening on MRI. This may be because most of the vascular abnormality in this case was composed of veins; too few elastic arteries were present to affect the imaging. We chose the term “AVM” to describe the lesion in this report This case suggests that pediatric AVMs in the intestine are different from adult angiodysplasia from the viewpoints of genesis and pathology.

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