Successful Wireless Capsule Endoscopy for a 2.5-Year-Old Child: Obscure Gastrointestinal Bleeding From Mixed, Juvenile, Capillary Hemangioma-Angiomatosis of the Jejunum
The lesion responsible for obscure gastrointestinal bleeding in the pediatric population may not be determined with standard primary endoscopic methods. Wireless capsule endoscopy, now a first-line modality for evaluation of the small bowel in the adult population, is a tool that may be useful among children. We report a case of a 2.5-year-old girl who presented with melenic stools. Upper and lower endoscopy, Meckel scans, and mesenteric angiography yielded negative results. Wireless capsule endoscopy identified numerous abnormal, dilated, blood vessels in the proximal jejunum, with associated fresh blood. The patient underwent surgical exploration, with resection of the affected portion of the jejunum. Pathologically, the dilated blood vessels were consistent with mixed, juvenile, capillary hemangioma-angiomatosis of developmental or congenital origin. The patient fared well postoperatively, with no additional bleeding in 9 months of follow-up monitoring. This case report highlights the use of capsule endoscopy in the diagnosis and successful treatment of gastrointestinal bleeding in a young infant. This is the youngest reported patient treated with the use of wireless capsule endoscopy in the pediatric population.
Obscure gastrointestinal bleeding represents ∼5% of all gastrointestinal bleeding.1 A source for the bleeding among these patients with visible gastrointestinal blood loss, recurrent or persistent iron deficiency anemia, or positive fecal occult blood tests is not uncovered with initial primary endoscopic methods (upper and lower gastrointestinal endoscopy) and a variety of other imaging techniques.2 These patients often undergo fruitless, costly, frustrating, and invasive studies.
Earlier research by Gong et al3 and Meron4 culminated in the development of the first human study of capsule endoscopy to image the whole of the small intestine directly.5 Since that time, >170000 patients have undergone capsule endoscopy throughout the world.6 A growing body of literature attests to the value of this diagnostic modality in the adult population, particularly for the investigation of obscure gastrointestinal bleeding.7–13 In this report, we describe the successful outcome of capsule endoscopy for a 2.5-year-old child with obscure gastrointestinal bleeding.
The patient, a previously healthy, 2.5-year-old, white girl (height: 82 cm; weight: 12 kg), presented with a 2-day history of increasing anorexia, weakness, and tarry stools. Her hemoglobin level was 3.9 g/dL, and she was admitted to the PICU.
There was no history of abdominal pain, nausea, vomiting, weight loss, or fever. Recently the patient had received 1 dose of a nonsteroidal antiinflammatory drug. Review of systems, medical history, and family history yielded negative results.
On physical examination, the patient's skin and conjunctivae were very pale. Her heart rate was 150 beats per minute, blood pressure 100/65 mm Hg, respiratory rate 30 breaths per minute and regular, weight 50th percentile, height 10th percentile, head circumference 75th percentile, temperature 36.6°C, and oxygen saturation 100% with room air. Results of a systematic general physical examination were not noteworthy. No skin, mucous membrane, or muscle lesions, such as angiomas, telangiectasia, abnormal pigmentation, or limb swelling, were noted. The abdomen was soft and nontender. There was no hepatosplenomegaly, mass, or bruit. A rectal examination confirmed melenic stool. Admission laboratory data were as follows: hemoglobin level, 3.9 g/dL (normal: 13.0–17.0 g/dL); hematocrit, 11.6% (normal: 36.0–45.5%); mean corpuscular volume, 85.3 fL (normal: 78.0–100.0 fL); mean corpuscular hemoglobin concentration, 33.8 g/dL (normal: 32.0–36.5 g/dL); white blood cell count, 3.6 × 103 cells per μL (normal: 4.2–11.0 × 103 cells per μL); platelet count, 25.0 × 104 platelets per μL (normal: 140–350 × 104 platelets per μL); prothrombin time, 12.4 seconds (normal: 8.5–12.0 seconds); international normalized ratio, 1.20; serum iron concentration, 27 μg/dL (normal: 49–181 μg/dL); ferritin, 5 ng/mL (normal: 30–300 ng/mL).
The patient was given a transfusion of packed red blood cells, to yield a hemoglobin level of 10.6 g/dL, and was treated with famotidine. However, she continued to pass large melenic stools, and her hemoglobin level decreased. Gastric lavage revealed no blood. Endogastroduodenoscopy to the descending duodenum and colonoscopy to the terminal ileum yielded normal findings. Two Meckel scans were negative. A 99Tc red blood cell scan and a computed tomographic scan of the abdomen and pelvis yielded negative results. Celiac and mesenteric arteriography with late-phase venous films also yielded negative findings for arteriovenous malformations or other vascular lesions.
On the 8th day after admission, after informed consent was obtained from the parents, M2A capsule endoscopy (Given Imaging, Norcross, GA) was performed. With general anesthesia and intubation of the airway, the capsule was passed into the esophagus by the anesthesiologist. Then a pediatric upper gastrointestinal endoscope was introduced into the esophagus, and the capsule was pushed easily, with very little pressure from the tip of the endoscope, into the stomach with direct observation. An endoscopic foreign-body net was used to trap the capsule and place it successfully in the second portion of the duodenum, where it was released from the net. The capturing of the capsule with the foreign-body net in the stomach was performed simply, and the release of the capsule into the duodenum required minor manipulation of the net and endoscope (opening and closing the net and varying the positioning of the tip of the endoscope so that the capsule would fall from the net into an open, small-bowel lumen not undergoing peristalsis). The endoscope and net were then removed. The reason for placing the capsule in the duodenum, and not releasing it in the stomach, was to avoid any possibility of prolonged retention of the capsule in the stomach. The entire length of the small intestine was observed, and the capsule was noted to enter the cecum just over 2 hours after placement in the duodenum. The positive finding, identified 43 minutes after commencement of the study, was the presence of multiple abnormal, dilated, blood vessels in a segment of proximal jejunum (Fig 1), where a small amount of fresh blood was present. A diagnosis of jejunal vascular malformation with active bleeding was made. With the use of the surface abdominal wall sensors as guides, it was thought that the lesion was within the reach of pediatric enteroscopy.
The patient was prepared for exploratory laparotomy. With general anesthesia, a pediatric colonoscope was passed into the proximal jejunum and the zone of abnormal vascular ectasia was identified. With the endoscope in position, laparoscopy was performed. The surgeon described multiple small networks of blood vessels on the serosal surface at the site of the mucosal lesions. Similar vascular lesions were present on the serosal surface of several other segments of jejunum, and there was a single small, serosal, ileal lesion. The mesentery was also involved diffusely. The 20-cm length of jejunum shown endoscopically to involve the mucosal surface was resected, with primary end-to-end anastomosis. The pathologic findings are presented in Figs 2 to ⇓4. Microscopic examination revealed a remarkable vascular proliferative lesion with dilated arterioles and capillaries extending from the mucosal surface to the serosal surface and into the mesentery. In addition, there were numerous clusters of poorly developed, immature, vascular structures in the wall of the jejunum. This lesion had the features of a mixed, juvenile, capillary hemangioma-angiomatosis, of developmental or malformative origin.14
The patient fared well postoperatively, and no additional bleeding occurred in 9 months of follow-up monitoring. Brain MRI scans revealed no vascular abnormalities.
In August 2001, the Food and Drug Administration (FDA) approved wireless capsule endoscopy (Given Diagnostic Imaging System; Given Imaging) as an adjunctive tool for the evaluation of small-intestinal diseases. By July 2003, the FDA had approved it as a first-line modality for the evaluation of all suspected small-bowel disorders. In January 2004, the FDA approved its use for pediatric patients between 10 and 18 years of age.
In a multicenter outcomes analysis of obscure gastrointestinal bleeding, Pennazio et al15 showed clearly the diagnostic capabilities of capsule endoscopy in 100 adult cases. The patients had undergone a total of 620 negative diagnostic tests (endogastroduodenoscopy, colonoscopy, small-bowel follow-through radiographs, enteroclysis, enteroscopy, 99Tc red blood cell scans, Meckel scans, computed tomographic scans, and laparoscopy). Overall, capsule endoscopy findings were positive in 47% of cases, suggestive in 15%, and negative in 38%. The authors stressed the fact that the best candidates for capsule endoscopy were those with ongoing obscure overt gastrointestinal bleeding, for whom positive findings were present in 92.3% of cases. These patients were examined while actively bleeding (ie, they had melena during the procedure).
In 2003, Mallet et al16 described preliminary results among children. Seidman et al17 described potential applications of wireless capsule endoscopy among pediatric patients with obscure gastrointestinal bleeding. In that study of 30 patients, 10 to 18 years of age, arteriovenous malformations were found in 75% of the capsule studies. For 1 patient, the capsule was retained for 10 days at the site of small-bowel stenosis but was passed spontaneously after a 4-day course of corticosteroid therapy.
For young children who are unable to swallow the capsule, a variety of endoscopic techniques have been described for placement of the capsule in the stomach and duodenum. These include the use of a foreign-body “Roth net,” as was used for our patient. Foreign-body grasping forceps or snares may also be useful with “frontloading” of the capsule on a gastroscope.17 An innovative capsule-delivery device has been patented by US Endoscopy (Mentor, OH), and FDA approval is pending. It is designed specifically to deliver and to release the capsule into the duodenum for patients with dysphagia, gastroparesis, or anatomic problems; it could be very useful in the pediatric age group. A 3-year-old patient with obscure iron deficiency anemia who underwent successful capsule endoscopy was described from Norway.18 The capsule was placed endoscopically in the duodenal bulb, to avoid the risk of gastric retention. The study revealed a large, hamartomatous, polypoidal tumor, which was successfully resected surgically.
Nonnatural excretion of the capsule has been reported. However, nonnatural excretion of the capsule among children has not been defined. A provisional anecdotal definition for adults is capsule retention for >14 days; for the majority of those adult patients, a small-bowel-obstructing lesion is present. A preliminary report19 described an M2A “patency capsule,” an ingestible biodegradable capsule designed to disintegrate slowly, allowing passage of the remnants through strictures or stenotic areas in the small bowel. The patency capsule is not approved by the FDA for use for any patient, regardless of age, and was not available to us on an emergency basis for the treatment of the child described. The patency capsule is the same size as the M2A capsule endoscope (length: 26 mm; diameter: 11 mm), with a tiny radiofrequency identification tag, and a hand-held scanner is used to determine a signal from the radiofrequency identification tag. In addition, the capsule contains barium. Therefore, if the capsule is not excreted after ingestion, the physician can detect the presence of the patency capsule within the body with the hand-held scanner and then determine the exact location of the obstruction with fluoroscopy. If obstructed, the patency capsule is designed to dissolve after 2 to 3 days and pass naturally. When the M2A patency capsule indicates no obstruction, the regular M2A capsule should pass naturally. The patency capsule may prove to be a useful indicator of the safe passage of the regular M2A capsule through the small bowel among very young children.
The case presented raises an ethical dilemma regarding the use of a medical device such as the M2A capsule for a patient well below the FDA-approved age of 10 years. The American Academy of Pediatrics, in a statement to the Institute of Medicine on August 23, 2004, stated, “Lack of study and FDA approval of medical devices for children is a tremendous barrier to providing pediatric patients access to appropriate therapies. By default, the standard of care for pediatric patients is off-label use of nearly every device.” The child presented in this case had a hemoglobin level of 5 mg/dL, with persistent melena, and multiple previous evaluations (some quite invasive) yielded negative results regarding a source of bleeding. A diagnosis needed to be made, and it was our clinical judgment to proceed with the M2A capsule study for this child. The parents of the child were informed fully, before the procedure, regarding the risks of upper endoscopy, anesthesia, and endoscopic passage of the capsule into the stomach and duodenum and the possibility of nonnatural excretion. We did not seek approval from the institutional review board at our institution because we viewed this clinical situation as innovative therapy rather than research. Future research may be directed at developing a smaller capsule to avoid the possibility of nonnatural excretion. Additional experience with the current M2A system among children may be required before such technology can be developed. Our experience with this 2.5-year-old child, the youngest reported in the literature to have undergone a capsule endoscopic study successfully, underscores the usefulness of video capsule endoscopy in the treatment of acute obscure gastrointestinal bleeding in childhood and infancy.
We would like to thank Henry Mangurten, MD, Chairman of Pediatrics, Advocate Lutheran General Hospital, for his expertise in preparation and review of the manuscript.
- Accepted June 16, 2005.
- Address correspondence to Hymie Kavin, MD, Department of Medicine, Division of Gastroenterology and Nutrition, Advocate Lutheran General Hospital, 1775 Dempster St, Park Ridge, IL 60068. E-mail:
The authors have indicated they have no financial relationships relevant to this article to disclose.
- ↵Given M2A Imaging. Home page. Available at: www.givenimaging.com. Accessed May 25, 2005
- Mylonaki M, Fritscher-Ravens A, Swain P. Wireless capsule endoscopy: a comparison with push enteroscopy in patients with gastroscopy and colonoscopy negative gastrointestinal bleeding. Gut.2003;52 :1122– 1126
- ↵Kemson RL, Fletcher CDM, Evans HL, Hendrickson MR, Sibley RK. Tumors of soft tissues. In: Rosai J, Sobin LH, eds. Atlas of Tumor Pathology. Washington, DC: Armed Forces Institute of Pathology;1998:307– 370
- ↵Boivin ML, Voderholzer W, Lochs H. The M2A patency capsule: the Berlin experience. Gastroenterology.2004;126 (suppl 2):A459
- Copyright © 2006 by the American Academy of Pediatrics