Angiodysplasia of the Ileum: Case Report of a Rare Cause of Intestinal Bleeding in Children
Stanko Ćavar1*, Mate Škegro2, Dražen Jelašić3 and Jurica Vuković4
1Department of Pediatric Surgery, University Hospital Centre Zagreb, Croatia
2 Department of Abdominal Surgery, University Hospital Centre Zagreb, Croatia
3 Department of Pathology and Cytology, University Hospital Centre Zagreb, Croatia
4Department of Pediatric Gastroenterology, University Hospital Centre Zagreb, Croatia
Submission: February 05, 2017; Published: April 11, 2017
*Corresponding author: Stanko Ćavar, Department of Pediatric Surgery, University Hospital Centre Zagreb, Balkans, Oporovečka 5 10040 Zagreb, Croatia.
How to cite this article: Stanko Ć, Mate Š, Dražen J, Jurica V. Angiodysplasia of the Ileum: Case Report of a Rare Cause of Intestinal Bleeding in Children. Acad J Ped Neonatol. 2017; 4(3): 555694. DOI: 10.19080/AJPN.2017.04.555694
Abstract
Introduction: Angiodysplasia can occur throught the gastrointestinal tract and ileum is one of the rarest locations for angiodiysplasias in the overall population. There may be multiple angiodysplasias in one gastrointestinal part or they can coexist in several different gastrointestinal location. Gastrointestinal angiodysplasia is most often founded in patients older than 60 years.
Case presentation: This is a case about a 14 year old Croatian white female who initially presented with symptoms of syncope and iron deficiency anemia. Scintigraphy with Technetium (TC)- 99m pertechnetate reveiled abnormal focal activity in a segment of the small intestine in the right abdominal part and we believed that this was due to a Meckel’s diverticulum. During the operation we found an angiodysplasia of the ileum.
Conclusion: This case highlights the importance of considering a gastrointestinal angiodysplasia in an acute pediatric gastrointestinal bleeding patient and as a differential diagnosis of anemia. During an operation, children with gastrointesinal bleeding require careful examination of the gastrointestinal tract and should be carried out in all cases where an initial laparoscopy failed to identify a cause of bleeding.
Keywords: Angiodysplasia; Vascular malformation; Gastrointestinal hemorrhage; Ileum; Children; Case report
Introduction
The prevalence of gastrointestinal angiodysplasia in the overall population is not well known. In the western population the jejunum was the most common location of angiodysplasias (80%), duodenum (51%), stomach (22.8%), right colon (11.4%) and ileum (5.7%) [1]. There are a few cases in the literature about gastrointestinal angiodysplasias in the children [2-4]. Histological confirmation shows dilated vessels in the mucosa and submucosa, sometimes covered with single layer of surface epithelium [5].
Case presentation
A 14 year old Croatian white female presented with a history of syncope and iron deficiency anemia. She had juvenile arthritis and she was treated with methotrexate, methylprednisolonum, meloxicamum and acidum folicum. She was pale and hemodynamically stable. Her hemoglobin was 63g/L, red blood cells was 2.2 x 1012/L, Fe 2 umol/L, and had a positive fecal occult blood. Scintigraphy with Technetium (TC)- 99m pertechnetate and SPECT (single photon emission computed tomography) abdominis showed activity in the stomach and abnormal focal activity in a segment of the small intestine in the right abdominal part and we believed that this was due to a Meckel’s diverticulum. During laparoscopic examination we could not find any source of bleeding so we proceeded with a laparotomy. We saw blood in the lumen of ileum and three lesions were identified on palpation of the small intestine at approximately 90cm. orally from the valvulae Bauhini. A 25cm portion of ileum was resected and a primary anastomosis was performed (Figure 1). Hystological findings were consistent with angiodysplastic lesions, there were a multiple tortuous thin-walled vessels in submucosa with anastomosis beetwen vessels (Figure 2). She recovered well and was discharged on seventh post operative day. During a one year follow up she was without simptoms such as rebleeding or anemia.
Discussion
Angiodysplasia account for 60%-75% of bleeding sources in the small bowel and are generally found in the proximal small bowel [1,6]. Exact data is not known for children yet. We found sources of bleeding in ileum which is the rarest location for angiodysplasias. The bleeding in angiodysplasia tend to be recurrent and chronic [7]. Nevertheless, profuse acute bleeding can be the reason for orthostasis and hypotension. Almost 50% of patients presenting with anaemia are found to have angiodysplasia [8]. In our case the first clinical manifestation was syncope and iron deficiency anemia but we seldom think about angiodisplasia in the children. Sometimes, the diagnostic examinations of causes of bleeding from the gastrointestinal are very difficult and inacurate. Angiodysplasia is usually diagnosed by endoscopy, capsule endoscopy, MSCT or MR angiography and standard angiography [1,6,9,10]. In one serie selective viscerale angiography was the- most valuable investigation, identifying the probable cause of bleeding in 53% of all patients [8]. Angiography is an invasive diagnostic tool, has many complications, is dependent on the quality of angiography and expirience of the physician carrying out the radiological interpretation. Sometimes surgery may be required for detection of angiodysplasias. In one serie, exploratory laparotomy detected 21% of identified causes of bleeding and was particularly valuable in younger patients. Other examination methods such as small bowel radiography and radionuclide bleeding scan are not satisfactory for detecting small bowel lesions [6]. In our case, a radionuclide scan was misleading. We believed that the source of bleeding was from a Meckel’s divetriculum. During laparoscopy we failed to find a Meckels’ diverticulum nor the source of bleeding. We proceeded with a laparotomy and performed a careful examination from the stomach to the rectum and found multiple angiodysplasias in the ileum. Treatment angiodysplasia ranged from observation to endoscopic traetment, medications, laser or surgery. The rebleeding rate after endoscopic therapy for angiodysplasia of the small bowel is about 45% [11]. Surgical resection is the method of choice for lesions that have been clearly identified as the source of severe bleeding and for multiple lesions with reccurent bleeding [1,3,6,8,9]. Failing to find a source of bleeding can be the cause of death. In patients who experience symptomatic blood loss due to angiodysplasia, aproximately 40-50% will experience rebleeding [12]. So we need to identify these lesions and perforrm a definitive treatment. We resected part of the ileum that had angiodysplasia and a primary anastomosis was performed which proved to be a safe choice.
Conclusion
This case highlights the importance of considering a gastrointestinal angiodysplasia in an acute pediatric gastrointestinal bleeding patients and during diagnostic of anemia. Scintigraphy with Technetium (TC)- 99m pertechnetate and SPECT abdominis can’t distinguish beetwen Meckel’s diverticulum and angiodysplasia. During an operation in children with gastrointesinal bleeding, a careful examination of the gastrointestinal tract should be carried out in all cases where an initial laparoscopy failed to identify a cause of bleeding. Resection of the bowel with a primary anastomosis is a definiteve therapy for angiodysplasia.
Compliance with Ethical Standards
Ethical approval
All procedures performed in studies involving human participants were in accordance with the ethical standards of the institutional and/or national research committee and with the 1964 Helsinki declaration and its later amendments or comparable ethical standards.
Informed consent
Informed consent was obtained from all indivdual participants included in the study.
Conflict of Interest
Author Stanko Ćavar, Author Mate Škegro, Author Dražen Jelašić, and Author Jurica Vuković declare that they have no conflict of interest.
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