Unusual Case

Small bowel hemangioma diagnosed with : Report of two pediatric cases

A E Jones, B H Ainsworth, A Desai, T T Tsang Department of Paediatric , Norfolk and Norwich University Hospital NHS Trust, Colney Lane, Norwich, NR4 7UY, UK

Address for correspondence: A. E. Jones, 59 Wernddu Road, Ammanford, Dyfed, UK. E-mail: [email protected]

Abstract Hemangiomas of the small bowel usually present with intestinal bleeding. This may be acute with massive Hemangiomas of the small bowel are rare tumors blood loss which can be life threatening or chronic that often present with gastrointestinal bleeding. Diagnosis can be difficult and exploratory with anemia as in our two cases. Other forms of laparotomy has often proved to be the only method presentation include intussusception,[2] small bowel with which to determine the presence and location obstruction and perforation.[1,3] Diagnosis of small of these tumors.We report two cases of small bowel bowel hemangiomas can be difficult and exploratory hemangioma in children aged 10 and 7 years, in laparotomy has often proved to be the only method which the diagnosis was made by laparoscopy. Laparoscopy identifies the affected segment of with which to determine the presence and location bowel and allows delivery to a minimally extended these tumors.[1,3] We report two cases of small bowel umbilical port site. The avoidance of an open hemangioma in children aged 10 and 7 years in which laparotomy helps to reduce post-operative analgesic the diagnosis was made by laparoscopy. requirement and achieves an early return of bowel function. .com). CASE REPORT

Key words: Hemangioma, laparoscopy, pediatric, small bowel The first patient was a 10-year-old girl who was under investigation for recurrent and iron .medknowdeficiency anemia requiring blood transfusions. INTRODUCTION Physical examination was unremarkable. Blood count revealed hemoglobin of 6.8 g/dL, mean cell volume Hemangiomas of the (www are rare (MCV) of 72. Double contrast barium follows through and account for only 0.05% of all intestinal and Technetium 99 meter pertechnetate (Meckel’s) neoplasms.[1] They are commonly found within the scan were normal. In addition, anti-endomysial and small bowel andThis comprise PDFa site 7-10%is hostedavailable of all benignby forMedknow freeautoimmune download Publications antibody from screens were normal. tumors.[1] The commonest location within the small However, tests were positive on a bowel is the mid-.[1] They have a tendency number of occasions. Upper and lower toward multiplicity with solitary tumors being gastrointestinal were arranged, however, extremely rare.[1,2] Hemangiomas have an association before these investigations could be performed, the with systemic angiomatoses such as blue rubber bleb patient presented acutely with collapse and melaena. naevus syndrome, Maffucci’s syndrome, and Klippel- Following fluid resuscitation, she underwent urgent Trenaunay-Weber syndrome. Grossly, an intestinal oesophago-gastro-duodenoscopy (OGD) and colon­ hemangioma is usually soft and polypoid; it is red, oscopy, both of which revealed no abnormality. Lapar­ blue or wine-colored, and varies in size from a few oscopy was undertaken at the same time and revealed millimeters to a large polypoid mass projecting into a 2 cm vascular malformation of the jejunum [Fig­ the lumen or infiltrating the bowel wall.[1] ures 1 and 2], which was resected with primary jeje-

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CMYK29 Jones, et al.: Small bowel hemangioma diagnosed with laparoscopy

to allow resection of the affected bowel segment and primary end-to-end anastomosis. She made an une­ ventful recovery and was found to have normal he­ moglobin and hematinics at follow up. Histology confirmed a cavernous hemangioma extending through the full thickness of the bowel wall.

DISCUSSION

Hemangiomas of the small bowel are rare tumors that often present with gastrointestinal bleeding. They are commonly found in the small bowel and hence often make diagnosis difficult. Initial Figure 1: Laparoscopic view of the jejunal hemangioma investigations in the patient with gastrointestinal bleeding often include upper and lower gastrointestinal , barium contrast study, and a Meckel’s scan. These investigations are often normal and while useful in excluding other diagnoses, do not provide clear indication of a hemangioma. Other investigations such as red blood cell scan and angiography have a rather low yield when the vascular malformation is not actively bleeding and one may have to resort to to determine the presence and location [1,3] these tumors..com). Laparoscopy as employed in our two cases allows a diagnosis to be made without performing a Figure 2: Opened specimen showing ulcerated haemangioma on the laparotomy. The laparoscopy identifies the affected mucosal side of the jejunum segment of bowel and if open excision is subsequently nal end-to-end anastomosis via mini laparotomy .medknowper- required, allows delivery to a minimally extended formed by extending the umbilical trocar incision. umbilical port site. Resection and anastomosis is then She made an uneventful recovery and follow up at 6 performed in the usual manner. A port site is months revealed a normal blood count (wwwand hematin- positioned at the umbilicus for the laparoscope, with ics. Histology of the resected bowel confirmed a je- two lower abdominal ports for instruments. The small junal vascularThis malformation. PDFa site is hostedavailable by forMedknow freebowel download Publicationsis examined from from the ileocecal junction prox­ imally. Laparoscopy can be performed in conjunction The second patient was a 7-year-old girl who initially with upper and lower gastrointestinal endoscopy presented with a three weeks history of lethargy, under the same anesthetic, as in our first case. Prior anorexia, and abdominal pain. Physical examination insufflation of the gastrointestinal tract during en­ was normal. On admission, her hemoglobin was 4 g/ doscopy did not create any difficulties for the lapar­ dL with an MCV of 62 and required blood transfusion. oscopy or identification of the pathology in this case. Fecal occult blood tests were negative. A Meckel’s The avoidance of an open laparotomy helps to re­ scan, abdominal ultrasound scan, OGD and colonos­ duce post-operative analgesic requirement and copy were normal. She underwent laparoscopy, which achieves an early return of bowel function. revealed a 2 cm hemangioma in the distal jejunum. Furthermore, there is the added cosmetic benefit of A mini laparotomy was performed via the umbilicus the small umbilical scar in girls.

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30 CMYK Jones, et al.: Small bowel hemangioma diagnosed with laparoscopy

Laparoscopy in these two cases also allowed a thor­ REFERENCES ough examination of the whole length of small bow­ el using the ‘walking’ maneuver. The good light 1. Boyle L, Lack EE. Solitary cavernous hemangioma of . Case report and literature review. Arch Pathol Med Lab source and magnification enabled easy viewing of 1993;117:939-41. any mesenteric or seromuscular abnormalities. We 2. Morgan DR, Mylankal K, el Barghouti N, Dixon MF. Small bowel did not proceed to intra-operative small bowel hemangioma with local involvement presenting as intussusception. J Clin Pathol 2000;53:552-3. endoscopy as we felt that additional large hemangi­ 3. Ramanujam PS, Venkatesh KS, Bettinger L, Hayashi JT, Rothman omas were unlikely. We were also confident that the MC, Fietz MJ. Hemangioma of the small intestine: Case report resected specimen had had a recent bleed because of and literature review. Am J Gastroenterol 1995;90:2063-4. the eroded appearance of the mucosal surface of the hemangioma when examined intra-operatively. We Cite this article as: Jones AE, Ainsworth BH, Desai A, Tsang TT. Small believe laparoscopy compliments other modalities in bowel hemangioma diagnosed with laparoscopy: Report of two pediatric the management of occult gastrointestinal bleeding cases. J Min Access Surg 2007;3:29-31. and the use of various modalities or in combination Date of submission: 17/08/06, Date of acceptance: 06/11/06 can be considered depending on the circumstances. Source of Support: Nil, Conflict of Interest: None declared.

.com).

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