Article Text

Download PDFPDF

Multiple intestinal haemangiomas presenting as intussusception and bleeding
  1. Khalil ElGendy,
  2. Amro Salem
  1. Department of Colorectal Surgery, King Fahad Specialist Hospital, Dammam, Eastern Province, Saudi Arabia
  1. Correspondence to Dr Khalil ElGendy, k.m.elgendy{at}gmail.com

Statistics from Altmetric.com

Description

A 17-year-old girl was referred to the colorectal surgery unit with a history of multiple hospital admissions due to recurrent abdominal pain, lower gastrointestinal (GI) bleeding and severe anaemia with haemoglobin reaching a low of 3 g/dL in one episode. Clinical examination was unremarkable. Colonoscopy revealed two lesions suggestive of haemangiomas at the rectosigmoid region and at the hepatic flexure (figure 1). Nuclear medicine GI bleeding scintigraphic scanning revealed active bleeding from the hepatic flexure. The patient was admitted due to an acute episode of intestinal obstruction and bleeding per rectum. CT angiogram (figure 2) revealed intussusception at the small bowel with characteristic findings in the form of a sausage-shaped mass with visible layers in the coronal (A) and axial (B) views and target sign in another axial view (C) associated with haemangioma as the leading point (size 20.3 mm×16.9 mm). In addition, another haemangioma was identified (figure 2D) with no findings suggestive of the active bleeding site.

Figure 1

Colonoscopy revealing haemangiomas at (A) rectosigmoid and (B) hepatic flexure.

Figure 2

CT angiogram showing diagnostic findings of intussusception in the form of elongated sausage-shaped mass in the coronal (A) and axial views (B) with multiple layers of the intussuscepted bowel associated with haemangioma as leading lesion (C) which also shows the target sign. Another haemangioma within the small bowel was identified (D).

Surgical exploration was offered and revealed the intussusception, with identification of the haemangioma as the leading point after the reduction (figure 3). Enterotomy was performed and revealed multiple haemangiomas close to the intussusception site, present over a segment of 20 cm, requiring small bowel resection of that segment and primary anastomosis. Colotomy, ligation and excision were done over the pre-identified haemangiomas in the hepatic flexure and rectosigmoid region. The pathological examination was reported to be multiple cavernous haemangioma. The patient had a smooth postoperative recovery with follow-up to 1 year without any attacks of abdominal pain or bleeding.

Figure 3

Intraoperative findings of intussusception (A), and haemangioma after resection of the intussusception (B) and after enterotomy (C).

Multiple intestinal haemangiomas are a very rare disorder. Adult intussusceptions are mostly caused by leading lesions, with very few cases caused by haemangiomas.1 Colonic involvement is uncommon, with solitary lesions usually involving the rectosigmoid.2 However, there is no reported case of multiple intestinal haemangiomas involving both the small and large bowel with a presentation of intussusception. Such cases are challenging and need comprehensive and extensive investigation to reach the diagnosis, find the extent of the disease and bleeding site, and prepare a subsequent management plan.3 Investigations may include: CT angiogram, scintigraphic studies and capsule endoscopy. MRI proved to be more accurate in demonstrating the extension of the lesions in selective cases.4

Learning points

  • Multiple intestinal haemangiomas are very rare disorders and can present with bleeding and intussusception.

  • A variety of investigations are required to diagnose such cases, which might include CT, MRI, scintigraphic scanning and endoscopy including capsule one.

  • Surgery can be offered in selective cases with appropriate preoperative work up and diagnosis.

References

View Abstract

Footnotes

  • Contributors KE was involved in case scenario, literature review and manuscript preparation; AS was involved in management of the case, informed consent and final revision.

  • Competing interests None.

  • Patient consent Obtained.

  • Provenance and peer review Not commissioned; externally peer reviewed.

Request Permissions

If you wish to reuse any or all of this article please use the link below which will take you to the Copyright Clearance Center’s RightsLink service. You will be able to get a quick price and instant permission to reuse the content in many different ways.