Chronic anemia caused by ileal hemangioma in an infant

An 11-month-old female infant presented with a 5-month history of intermittently bloody stools. She was admitted with poor appetite and activity for the past one month. Physical examination showed pallor and normal vital signs, and laboratory tests showed severe anemia with hemoglobin level of 2.7 g/dL. There was no evidence of a heterotopic gastric mucosa in the technetium-99m (Tc-99m) pertechnetate scan. The source of bleeding could not be identified after upper gastrointestinal endoscopy and colonoscopy. Video capsule endoscopy was not performed because of the limited technology; however, Tc-99m-labeled red blood cell (RBC) scan confirmed that there was active gastrointestinal (GI) bleeding (Fig. 1a). Due to the abnormal clinical findings, a decision to perform a surgical intervention was made, and a laparoscopic approach was selected. Intraoperatively, a 4-cm transmural ileal hemangioma was observed 55 cm proximal to the ileocecal valve (Fig. 1b), and resection and ileo-ileostomy were performed. The anastomosis was hand-sewn intracorporeally, and a Harmonic scalpel (Ethicon Endosurgery, Cincinnati, OH) was used for division of the mesentery. The resected specimen was placed in an extraction bag and removed via the umbilical incision. Histopathological examination revealed dilated and anastomosing vascular channels lined by bland-looking endothelial cells within the submucosa and intramuscular spaces. These endothelial cells showed positive staining for clusters of differentiation 31 (CD31) and CD34 and negative staining for podoplanin (D2-40) stain, consistent with the clinical diagnosis. In infants, severe anemia may cause an irreversible delay in mental, motor, and behavioral development. It may cause serious health issues, restrict recovery, or even result in death if the diagnosis is made too late to be effectively treated. Although this patient received blood transfusion due to severe anemia, there were no specific associated symptoms or physical examination findings. Compared to Meckel's diverticulum, patients with GI hemangioma may have an extremely low hemoglobin level and require multiple transfusions. Meckel's diverticulum was initially suspected because of the patient's age; however, the finding of Meckel's scan was negative, but the Tc-99m-labeled RBC scan showed positive results. Small bowel bleeding should be considered even though it is rare, as it is responsible for several obscure GI bleeding cases. Pooled cases, with an averaged mean age of 49 years (range 0–75 years), showed that the introduction of capsule endoscopy, balloon-assisted enteroscopy, and radiographic imaging were important for the preoperative diagnosis of small bowel hemangiomas.1Fu J.X. Zou Y.N. Han Z.H. Yu H. Wang X.J. Small bowel racemose hemangioma complicated with obstruction and chronic anemia: a case report and review of literature.World J Gastroenterol. 2020; 26: 1674-1682Crossref Scopus (8) Google Scholar However, less cases of small bowel hemangioma have been reported in children, for whom preoperative examination could be challenging due to limited techniques and patient cooperation. Therefore, Meckel's scan and Tc-99m-labeled RBC scan are noninvasive options for infants. The therapeutic option in our case was surgery, which was consistent with these pooled cases, and the patient's hemoglobin increased to 13.2 g/dL after the surgical resection procedure. Infantile hemangiomas are the most common benign tumors of infancy, with a prevalence of 4·5%2Léauté-Labrèze C. Harper J.I. Hoeger P.H. Infantile haemangioma.Lancet. 2017; 390: 85-94Abstract Full Text Full Text PDF PubMed Scopus (248) Google Scholar and most of them are located in the cephalic region. Routine treatment is not indicated unless there are life-threatening risks or anatomic distortions. Cutaneous hemangiomas have been reported in relation to GI hemangioma in a few cases.3Soukoulis I.W. Liang M.G. Fox V.L. Mulliken J.B. Alomari A.I. Fishman S.J. Gastrointestinal infantile hemangioma: presentation and management.J Pediatr Gastroenterol Nutr. 2015; 61: 415-420Crossref PubMed Scopus (25) Google Scholar However, the appearance of this patient was generally normal and could be easily ignored, as there was an absence of superficial hemangioma. In conclusion, although infantile hemangioma in the GI tract is rare and hard to diagnose preoperatively, it should be considered when GI bleeding is confirmed without specific clinical presentations or exact origin. The authors have no conflicts of interest relevant to this article.