A Case of Solitary Cavernous Hemangioma of the Small Intestine with Recurrent Clinical Anemic Attacks in Childhood

Abstract

Hemangiomas of the small intestine are extremely rare in children, and the preoperative diagnosis is difficult. The case presented involves an 11-year-old boy with cavernous hemangioma in the proximal ileum, which was detected by technetium Tc 99m red blood corpuscle (99mTc-RBC) scintigraphy and was successfully resected laparoscopically. The results of this case showed the effectiveness of combining 99mTc-RBC scintigraphy and laparoscopy in the diagnosis and treatment of hemangioma of the small intestine. CASE REPORT An 11-year-old boy was admitted with symptoms of anemia and abdominal pain. Hemoglobin level in his serum was 5.9 mg/dl. Gastrointestinal bleeding was suspected, but a stool sample was negative for blood. The pattern of his anemia was microcytic and hypochromic (mean corpuscular volume [MCV], 59.3µm3 [normal, 82 µm3; range, 83-93 µm3]; mean corpuscular hemoglobin [MCH], 15.8 pg [normal, 28 pg; range, 27-32 pg]; and mean corpuscular hemoglobin concentration [MCHC], 26.6 g/dl [normal, 34 g/dl; range, 32-36 g/dl]). Because iron deficiency anemia was suspected (Fe, 15 µg/dl [range, 50-180 µg/dl]; TIBC, 435 µg/dl [range, 280-380 µg/dl]; ferritin, 2.6 ng/ml [range, 26-240 ng/ml]), iron was administered, and the hemoglobin level in the patient's serum was increased to 10.5 g/dl after 1 month. Four months later, the patient was readmitted to our hospital for an attack of anemia (hemoglobin, 7 mg/dl). At this time, the pattern of anemia was normocytic and normochromic (MCV, 85.4 µm3; MCH, 28.7 pg; MCHC, 33.6 g/dl). Endoscopically, no hemorrhagic lesions were found in his stomach or duodenum. Barium enema and contrast study also showed no lesions. On gallium scintigraphy and Meckel's scintigraphy, no positive lesion was observed. Except for iron deficiency, no other hematologic diseases were seen. Iron was administered, and again his anemia improved. Five months later, when he had a third attack of anemia, 99mTc-RBC scintigraphy was immediately performed, and a positive lesion was found in front of the right common iliac artery (Fig. 1). At that time, hemorrhagic parts were not identified in enhanced computed tomography(CT). Finally, a laparoscopic examination was performed, and hemangioma was found in the proximal ileum, 60 cm from the ileocecal valve(Fig. 2). The hemangioma was resected through a 3-cm incision below the umbilicus, and anastomosis was completed. Histologically, the specimen was a cavernous hemangioma in the submucosal layer(Fig. 3). The patient was discharged 7 days after surgery and is doing well.FIG. 1: The 99mTc red blood corpuscle scintigram using the semi-in vivo markers method. Ten minutes after99m Tc-RBC injection, the abnormal accumulation was found in front of the right common iliac artery. The spot did not spread intralumenally.FIG. 2: The cavernous hemangioma was in the proximal ileum, 60 cm from the ileocecal valve. It was approximately 6 cm in length along the ileum.FIG. 3: Histologic finding of hemangioma of ileum. Many irregularly dilated blood vessels with thin walls were seen, mainly in the submucosal layer.DISCUSSION We report an 11-year-old boy with an intestinal cavernous hemangioma at the proximal ileum, causing recurrent anemic attacks. Solitary cavernous hemangioma accounts for approximately 10% of all benign vascular tumors located in the gastrointestinal tract and is rare, particularly in childhood(1). The preoperative diagnosis is difficult(2,3). 99mTc-RBC scintigraphy was initially useful in finding areas of bleeding (4). In this case, the scintigraphy showed a positive lesion in the patient's abdomen, but the hemorrhagic areas were not pointed out on enhanced CT. The scintigraphy may be effective in detecting congestion of the hemangioma in the submucosal layer of the small intestine in early stages. Finally, laparoscopic examination was performed, and the hemangioma of the proximal ileum was seen. The hemangioma was resected and removed through a 3-cm incision below the umbilicus. No bleeding was observed. The laparoscopic examination was useful for finding intestinal hemangioma so that resection of the tumor could be performed using a minimally invasive technique. In this case, we realized that the combination of 99mTc-RBC scintigraphy and laparoscopic examination is an effective method for finding and treating hemangioma of small intestine.