Intraluminal gastrointestinal bleeding can be a life-threatening condition. In case of OGIB this is rarely the case when esophagogastroduodenoscopy and colonoscopy with adequate preparation have been performed[5,6], which often results in a diagnostic dilemma. In 75% of OGIB patients, the lesion is ultimately found in the small intestine, and occasionally causes recurrent or persistent bleeding.
Small bowel bleeding is relatively rare, comprising only about 5% of gastrointestinal bleeding[1,7]. Concerning the common causes of small bowel bleeding, the causative disease varies by age. In patients under 40 years of age, inflammatory bowel disease, the most common cause, is followed by Dieulafoy’s lesions, neoplasms, Meckel’s disease and Polysosis syndrome. In those over 40, angioectasia including arteriovenous malformation and hemangioma is followed by Dieulafoy’s lesions, neoplasms and NSAIDs ulcers. In addition, Yamamoto et al reported that the frequency of angioectasia-related bleeding among small bowel bleeding cases was 20%.
Of all primary small bowel tumor cases, 47% are benign. Among them, small bowel hemangioma is a relatively rare primary small bowel tumor, histologically defined as a benign tumor. Wilson et al reported that hemangioma constitutes approximately 10% of benign small bowel tumors and that most reported cases have occurred in the jejunum and ileum. Concerning the initial common symptoms of small bowel hemangioma, the occurrence frequency of iron-deficiency anemia, pain and intussusception are 41%, 31% and 13%, respectively. Macroscopically, Chen et al summarized previous endoscopy reports of intestinal hemangiomas describing the lesions as typically submucosal, purple to red, soft and pedunculated. Histologically, hemangiomas can be broadly classified in capillary, cavernous and mixed type, with the cavernous type being the most common. They generally consist of numerous dilated, irregular blood-filled spaces or sinuses lined by layers of endothelial cells.
Recent advances in endoscopic technique including VCE and DBE have allowed preoperative diagnosis of small bowel hemangioma. VCE can be recommended as part of the routine work-up in patients with obscure bleeding, and it is not contraindicated except in patients with stenosis of the intestine. Compared with VCE, DBE has the advantage of biopsy and therapeutic potential, such as preoperative localization, coagulation and hemostasis by clipping, whereas Xin indicated that successful total enteroscopy is achieved in only 1.6% of patients through the antegrade procedure.
In recent years, the therapeutic options including minimally invasive laparoscopic surgery represented by our cases and even non-surgical endoscopic approaches for small bowel hemangioma have been proposed. The laparoscopic approach to gastrointestinal diseases is now widely accepted, but it is generally difficult to locate the lesion by palpation, especially in the jejunum and ileum. A previous study reported on colonic tattooing in animal model with various agents, such as methylene blue, indigo carmine, toludine blue, lymphazurine, hematoxylin, eosin, indocyanine green (ICG) and India ink. Only ICG and India ink tattoos persisted for more than 48 h, while ICG was associated with allergic reactions and systemic toxicity. Therefore, endoscopic marking with India ink is used widely as a visualization technique for colorectal cancer, to define the operative location. India ink tattooing has a low incidence of complications (0.22%), and remains for a prolonged duration[17,18]. In the present case, we selected this visualization technique for video-assisted single-port laparoscopic enterectomy, expecting a minimally invasive effect. Concerning non-surgical endoscopic approaches, several clinical studies have reported that endoscopic mucosal resection can be a useful tool for small bowel hemangioma[19-21]. However, in addition to the common endoscopic complications including intestinal perforation and lesion persistence, the endoscopic approach for small bowel hemangioma with rich vascularity has the potential for flooding, given the potential for misconception of the lesion depth. Chen et al also recommended careful consideration of the indications for endoscopic surgery. As one of the methods to overcome these potential risks, laparoscopic and endoscopic cooperative surgery (LECS) has been suggested as a new concept for tumor dissection. In recent years, Kanaji et al demonstrated safe and total laparoscopic resection of hemangiomas in the third portion of the duodenum using the LECS technique. Therefore, LECS may become a useful therapeutic option for small bowel hemangioma.
In conclusion, we have documented two cases of small bowel hemangioma found in examinations for OGIB. The preceding implementation of VCE made the selective decision of DBE insertion easy, and the endoscopic process facilitated early treatment, resulting in avoidance of progression to life-threatening status. In the present case report (Case 2), CECT showed small nodule enhancement in the lesion. Therefore, the preceding CECT with a characteristic of rapid and minimally invasive technique may omit VCE, making it a useful algorithm for further early treatment. These findings imply that various preoperative endoscopic contrivances may result in safer, more minimally invasive treatment.
Two cases of small bowel hemangioma found in examinations for obscure gastrointestinal bleeding (OGIB) were treated with combination of laparoscopic and endoscopic modalities.
Preoperative enteroscopy and imaging tests in each case suggested that the lesion was a small intestinal hemangioma or arteriovenous malformation.
Iron-deficiency anemia was diagnosed, with one case showing severe anemia.
Video capsule endoscopy (VCE) showed the precise location of the lesion, and double-balloon enteroscopy (DBE) revealed raised lesions with or without spout bleeding in the small intestine.
They diagnosed Case 1 and Case 2 as cavernous hemangioma and capillary hemangioma, respectively.
The patients were treated with combination of laparoscopic and endoscopic modalities.
There are few reports on treatments combining VCE, DBE on fluoroscopy, preoperative endoscopic marking, and single-port laparoscopic surgery.
There are no uncommon terms used in this manuscript.
Experiences and lessons
The authors share this case as important knowledge for the appropriate treatment process for small bowel hemangioma.
These two case reports demonstrate that small bowel hemangioma found as an examination for OGIB were successfully treated with combination of laparoscopic and endoscopic modalities.