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First Case of Refractory Celiac Disease and Ulcerative Jejunitis in a Child Revealed By Capsule Endoscopy
Abstracts
M1403 Capsule Endoscopy in Children: Overall Usefulness and Special Value in Intestinal Crohn’s Disease Vaibhav Mehendiratta, Michael Dimarino, Anthony Infantolino, Sidney Cohen, Mitchell I. Conn, David M. Kastenberg, Leo Katz, Ilene Fardella, Lenore Miranda, Tracy Henwood Objective: To review the experience with Capsule Endoscopy (CE) in pediatric population at a tertiary care center focusing on the utility of CE to diagnose small bowel Crohn’s disease and assess disease activity. Methods: Thirty four patients aged less than 20 years underwent capsule endoscopy at the Jefferson Digestive Diseases Institute, Philadelphia, PA, for a total of 36 studies between May, 2004 to September, 2006. Pillcam SB (Given Imaging, Yoqneam, Israel) capsule was used in all studies. Medical records were reviewed for demographic data, pre-procedure diagnosis, indications, number of complete and incomplete examinations, gastric and small bowel transit times, findings and complications of CE. Results: Two patients failed to complete the study secondary to retention of capsule in the stomach; requiring endoscopic removal in one case and resulting in vomiting of the capsule in other.These patients were excluded from final analysis which represents experience from 32 patients and 34 studies. Mean age of patients was 14 years (Range:7-19 years). Females comprised 62.5% of all patients. Indications included abdominal pain (19 of 34, 56% studies), GI bleeding (47%), diarrhea (41%), anemia (35%) and weight loss/failure to thrive (9%).Capsule was easily swallowed by all patients. Mean gastric transit time was 42 minutes.Capsule failed to visualize the cecum in 18% studies. All 34 capsules were excreted spontaneously and no complications like abdominal pain, bowel obstruction or bleeding were observed.Abnormal findings were seen in 25 of 34 (74%) studies. Crohn’s disease activity, in the form of aphthae, linear and serpiginous ulcers, and fissures involving distal small bowel was visualized in 3 of 5 (60%) patients with already diagnosed Crohn’s disease involving small and/or large intestine. Four patients who were overall negative for Crohn’s disease using conventional radiographic and endoscopic studies had diagnostic findings consistent with Crohn’s disease on CE.These findings were most commonly located in the ileum, but also involved duodenum and jejunum. Other abnormal capsule findings included erythema (in 21% of 34 studies), erosions (18%), fresh blood (15%), ulcers (15%), gastritis (15%) and mucosal fold thickening (12%). Conclusion:A) Capsule endoscopy is a safe and valuable diagnostic tool in pediatric population with a potential to expedite diagnosis and treatment of small bowel disease similar to the adult experience. B) Capsule endoscopy is especially useful in detecting the presence, activity and severity of small bowel Crohn’s disease. C) Capsule endoscopy offers the possibility of improved diagnosis while limiting radiation exposure in children with Crohn’s disease.
M1404 Clinical Outcomes of Pediatric Crohn’s Disease Diagnosed By Capsule Endoscopy Libia C. Moy, Jeremiah J. Levine Background: Several studies have demonstrated the utility of capsule endoscopy (CE) in the diagnosis of Crohn’s disease (CD). The aim of this study is to correlate the findings of the CE with clinical outcomes in children with small intestinal CD. Methods: We retrospectively reviewed the charts of 20 consecutive patients that underwent CE study (11/02-3/06) and were diagnosed with CD with small bowel involvement. Patients were followed for at least 6 months after the study. The CE findings were divided into 3 categories: erosions, ulcerations and stricture formation. A successful clinical outcome was defined as resolution of symptoms and/or normalization of the blood test(s) that prompted CE. The study was approved by the IRB of North Shore/LIJ Health System. Results: A total of 20 patients were diagnosed with CD disease based on CE; 3 patients were excluded due to lack of follow-up in our institution. There were 9 males and 8 females; mean age 15.3 2.8 years. The indications for CE were: 8 known CD patients with persistent pain and/or weight loss, 6 patients without known CD with abdominal pain and/or weight loss, 1 patient with presumed ulcerative colitis and growth failure, 2 patients had occult GI bleeding and anemia. Six patients were ASCA positive, 6 patients were negative and IBD markers were not sent in 5 patients. The CE findings showed 6 patients with erosions, 6 patients with ulcerations and 5 patients with strictures. Of the 6 patients with erosions, only 1 (16%) had a positive clinical outcome, 3 were still symptomatic on follow up and 2 were found to have vascular disease. Of the 6 patients with ulcerations, 4 (66%) had a positive clinical outcome and 2 patients were still symptomatic. Of the 5 patients with stricture, 4 (80%) had a positive outcome (3 had ileocolic resection) and one was still symptomatic but was not taking his medications. Of the 6 patients who were ASCA positive; 2 were found to have erosions, 2 had ulcerations on the CE and 2 had strictures; only the patients with strictures had a positive clinical outcome. Conclusion: Based on CE findings, 80% of the patients with strictures had a positive outcome compared with 66% of patients with ulcerations and only 16% of the patients with erosions. ASCA positivity did not correlate with findings on CE or clinical outcome. We conclude that CE findings of ulcerations or strictures are highly suggestive of CD and respond to therapy for the CD while erosions alone are not a sufficient to diagnose CD and should not be used to guide medical therapy. Further prospective studies using larger populations are needed to further understand the significance of the CE findings.
AB250 GASTROINTESTINAL ENDOSCOPY Volume 65, No. 5 : 2007
M1405 Carbon Dioxide, a Safe Insufflation Agent in Pediatric Endoscopy Tuvia Marciano, Theresa Guerin, Susan Cohen, Yolanda Rivas Background: The use of carbon dioxide (CO2) insufflation in endoscopy is effective in reducing post procedural abdominal pain and discomfort in adults. CO2 retention does not occur in sedated adults, as it is rapidly absorbed from the bowel and excreted via the respiratory system. To date, there are no published studies examining the safety of CO2 insufflation in pediatric patients. Methods: We performed a chart review of all patients who underwent an endoscopic procedure under conscious sedation between June 2006 and November 2006. Only charts that accurately documented the end tidal carbon dioxide (ETCO2) levels were considered for this study. ETCO2 was measured via nasal cannula using a Philips M1026B gas analyzer. The identification of a normal ETCO2 wave pattern was used to assure validation of measurements. The ETCO2 was measured prior to the administration of anesthesia, at the start of the procedure, and every ten minutes thereafter. A final measurement was obtained ten minutes after the procedure was completed. All patients were sedated by the same anesthesiologist and received a combination of one or more of the following agents, fentanyl, ketamine, midazolam, and propofol. Results: A total of 50 patients meeting the above criteria were identified. Air insufflation was used in 27 patients, and CO2 insufflation in 23 patients. The mean age was 16 7 and 15 7 years, respectively. There was no significant difference in the mean ETCO2 measurement between the air group (35 mmHg) and the CO2 group (38 mm Hg). It was noted that patients in the CO2 group received significantly less propofol than patients in the air group (2.26 mg/kg vs. 4.75 mg/kg; p ! 0.05 two tailed Mann-Whitney test). Recovery times and post procedural pain assessment also did not differ between the two groups. Conclusion: CO2 insufflation during pediatric endoscopy does not result in increased ETCO2 levels. In addition, these patients require less propofol when compared to patients undergoing air insufflation. We speculate that this difference is the result of rapid clearance of the insufflation agent leading to decreased intraprocedural abdominal distention and less pain. These finding suggest that CO2 insufflation is safe and may possibly diminish the intraprocedural discomfort. A prospective study is warranted.
M1406 First Case of Refractory Celiac Disease and Ulcerative Jejunitis in a Child Revealed By Capsule Endoscopy Terry E. Sigman, Florin Costea, Chantal Bernard, Ernest G. Seidman Celiac disease is defined as a gluten sensitive enteropathy, which in most cases improves on a gluten free diet. Refractory celiac disease is a rare condition in which the small bowel enteropathy persists despite being on a strict gluten free diet and may be associated with ulcerative jejunitis and can predispose to enteropathy associated T-cell lymphoma (EATL). We present a 9-year-old girl who first presented with a history of persistent abdominal pain and vomiting, a normal CBC and albumin. The patient was found to have selective IgA deficiency, yet a slightly positive IgA anti-tissue transglutaminase and positive anti-endomyseal antibody tests, as well as an elevated serum total IgE level. An initial endoscopy revealed a few aphtous ulcerations in the bulb. Histology of the duodenum revealed patchy increased eosinophilic infiltrates (30-40/HPF) with glandular infiltration. There was also patchy mild villous blunting, and mildly increased intraepithelial lymphocytes (IEL) focally. A colonoscopy was macroscopically normal, but histology also showed focal areas with mild active colitis, characterized by eosinophilic infiltrates in the lamina propria. The findings were not considered diagnostic of Crohn’s or celiac disease. She was confirmed to be HLADQ2 positive. A wireless videocapsule endoscopy (CE) was then performed, which revealed features typical of celiac disease: delayed appearance of villi in mid jejunum; scalloping of folds with a mosaic pattern to the proximal small bowel mucosa. Surprisingly, several fibrincovered and actively bleeding mucosal ulcerations were seen in the jejunum. The ulcerative jejunitis was confirmed by push enteroscopy. Histological findings now confirmed typical findings of celiac disease in the distal duodenum and jejunum to 115 cm (subtotal villous atrophy, increased crypts and IEL) focally, with spared areas. T cell markers were normal and no clonality suggestive of EATL were found. After 6 months on a gluten free diet, a repeat CE revealed notable improvement but persistence of fewer jejunal ulcerations and areas with villous atrophy. A repeat push enteroscopy was performed and repeat samples for T-cell markers and clonality are pending. Ulcerative jejunitis due to refractory celiac disease has never previously been reported in the pediatric age group (!21 years). The use of CE was of utmost importance in this child, as we would not have otherwise diagnosed the ulcerative jejunitis.
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M1403 Capsule Endoscopy in Children: Overall Usefulness and Special Value in Intestinal Crohn’s Disease Vaibhav Mehendiratta, Michael Dimarino, Anthony Infantolino, Sidney Cohen, Mitchell I. Conn, David M. Kastenberg, Leo Katz, Ilene Fardella, Lenore Miranda, Tracy Henwood Objective: To review the experience with Capsule Endoscopy (CE) in pediatric population at a tertiary care center focusing on the utility of CE to diagnose small bowel Crohn’s disease and assess disease activity. Methods: Thirty four patients aged less than 20 years underwent capsule endoscopy at the Jefferson Digestive Diseases Institute, Philadelphia, PA, for a total of 36 studies between May, 2004 to September, 2006. Pillcam SB (Given Imaging, Yoqneam, Israel) capsule was used in all studies. Medical records were reviewed for demographic data, pre-procedure diagnosis, indications, number of complete and incomplete examinations, gastric and small bowel transit times, findings and complications of CE. Results: Two patients failed to complete the study secondary to retention of capsule in the stomach; requiring endoscopic removal in one case and resulting in vomiting of the capsule in other.These patients were excluded from final analysis which represents experience from 32 patients and 34 studies. Mean age of patients was 14 years (Range:7-19 years). Females comprised 62.5% of all patients. Indications included abdominal pain (19 of 34, 56% studies), GI bleeding (47%), diarrhea (41%), anemia (35%) and weight loss/failure to thrive (9%).Capsule was easily swallowed by all patients. Mean gastric transit time was 42 minutes.Capsule failed to visualize the cecum in 18% studies. All 34 capsules were excreted spontaneously and no complications like abdominal pain, bowel obstruction or bleeding were observed.Abnormal findings were seen in 25 of 34 (74%) studies. Crohn’s disease activity, in the form of aphthae, linear and serpiginous ulcers, and fissures involving distal small bowel was visualized in 3 of 5 (60%) patients with already diagnosed Crohn’s disease involving small and/or large intestine. Four patients who were overall negative for Crohn’s disease using conventional radiographic and endoscopic studies had diagnostic findings consistent with Crohn’s disease on CE.These findings were most commonly located in the ileum, but also involved duodenum and jejunum. Other abnormal capsule findings included erythema (in 21% of 34 studies), erosions (18%), fresh blood (15%), ulcers (15%), gastritis (15%) and mucosal fold thickening (12%). Conclusion:A) Capsule endoscopy is a safe and valuable diagnostic tool in pediatric population with a potential to expedite diagnosis and treatment of small bowel disease similar to the adult experience. B) Capsule endoscopy is especially useful in detecting the presence, activity and severity of small bowel Crohn’s disease. C) Capsule endoscopy offers the possibility of improved diagnosis while limiting radiation exposure in children with Crohn’s disease.
M1404 Clinical Outcomes of Pediatric Crohn’s Disease Diagnosed By Capsule Endoscopy Libia C. Moy, Jeremiah J. Levine Background: Several studies have demonstrated the utility of capsule endoscopy (CE) in the diagnosis of Crohn’s disease (CD). The aim of this study is to correlate the findings of the CE with clinical outcomes in children with small intestinal CD. Methods: We retrospectively reviewed the charts of 20 consecutive patients that underwent CE study (11/02-3/06) and were diagnosed with CD with small bowel involvement. Patients were followed for at least 6 months after the study. The CE findings were divided into 3 categories: erosions, ulcerations and stricture formation. A successful clinical outcome was defined as resolution of symptoms and/or normalization of the blood test(s) that prompted CE. The study was approved by the IRB of North Shore/LIJ Health System. Results: A total of 20 patients were diagnosed with CD disease based on CE; 3 patients were excluded due to lack of follow-up in our institution. There were 9 males and 8 females; mean age 15.3 2.8 years. The indications for CE were: 8 known CD patients with persistent pain and/or weight loss, 6 patients without known CD with abdominal pain and/or weight loss, 1 patient with presumed ulcerative colitis and growth failure, 2 patients had occult GI bleeding and anemia. Six patients were ASCA positive, 6 patients were negative and IBD markers were not sent in 5 patients. The CE findings showed 6 patients with erosions, 6 patients with ulcerations and 5 patients with strictures. Of the 6 patients with erosions, only 1 (16%) had a positive clinical outcome, 3 were still symptomatic on follow up and 2 were found to have vascular disease. Of the 6 patients with ulcerations, 4 (66%) had a positive clinical outcome and 2 patients were still symptomatic. Of the 5 patients with stricture, 4 (80%) had a positive outcome (3 had ileocolic resection) and one was still symptomatic but was not taking his medications. Of the 6 patients who were ASCA positive; 2 were found to have erosions, 2 had ulcerations on the CE and 2 had strictures; only the patients with strictures had a positive clinical outcome. Conclusion: Based on CE findings, 80% of the patients with strictures had a positive outcome compared with 66% of patients with ulcerations and only 16% of the patients with erosions. ASCA positivity did not correlate with findings on CE or clinical outcome. We conclude that CE findings of ulcerations or strictures are highly suggestive of CD and respond to therapy for the CD while erosions alone are not a sufficient to diagnose CD and should not be used to guide medical therapy. Further prospective studies using larger populations are needed to further understand the significance of the CE findings.
AB250 GASTROINTESTINAL ENDOSCOPY Volume 65, No. 5 : 2007
M1405 Carbon Dioxide, a Safe Insufflation Agent in Pediatric Endoscopy Tuvia Marciano, Theresa Guerin, Susan Cohen, Yolanda Rivas Background: The use of carbon dioxide (CO2) insufflation in endoscopy is effective in reducing post procedural abdominal pain and discomfort in adults. CO2 retention does not occur in sedated adults, as it is rapidly absorbed from the bowel and excreted via the respiratory system. To date, there are no published studies examining the safety of CO2 insufflation in pediatric patients. Methods: We performed a chart review of all patients who underwent an endoscopic procedure under conscious sedation between June 2006 and November 2006. Only charts that accurately documented the end tidal carbon dioxide (ETCO2) levels were considered for this study. ETCO2 was measured via nasal cannula using a Philips M1026B gas analyzer. The identification of a normal ETCO2 wave pattern was used to assure validation of measurements. The ETCO2 was measured prior to the administration of anesthesia, at the start of the procedure, and every ten minutes thereafter. A final measurement was obtained ten minutes after the procedure was completed. All patients were sedated by the same anesthesiologist and received a combination of one or more of the following agents, fentanyl, ketamine, midazolam, and propofol. Results: A total of 50 patients meeting the above criteria were identified. Air insufflation was used in 27 patients, and CO2 insufflation in 23 patients. The mean age was 16 7 and 15 7 years, respectively. There was no significant difference in the mean ETCO2 measurement between the air group (35 mmHg) and the CO2 group (38 mm Hg). It was noted that patients in the CO2 group received significantly less propofol than patients in the air group (2.26 mg/kg vs. 4.75 mg/kg; p ! 0.05 two tailed Mann-Whitney test). Recovery times and post procedural pain assessment also did not differ between the two groups. Conclusion: CO2 insufflation during pediatric endoscopy does not result in increased ETCO2 levels. In addition, these patients require less propofol when compared to patients undergoing air insufflation. We speculate that this difference is the result of rapid clearance of the insufflation agent leading to decreased intraprocedural abdominal distention and less pain. These finding suggest that CO2 insufflation is safe and may possibly diminish the intraprocedural discomfort. A prospective study is warranted.
M1406 First Case of Refractory Celiac Disease and Ulcerative Jejunitis in a Child Revealed By Capsule Endoscopy Terry E. Sigman, Florin Costea, Chantal Bernard, Ernest G. Seidman Celiac disease is defined as a gluten sensitive enteropathy, which in most cases improves on a gluten free diet. Refractory celiac disease is a rare condition in which the small bowel enteropathy persists despite being on a strict gluten free diet and may be associated with ulcerative jejunitis and can predispose to enteropathy associated T-cell lymphoma (EATL). We present a 9-year-old girl who first presented with a history of persistent abdominal pain and vomiting, a normal CBC and albumin. The patient was found to have selective IgA deficiency, yet a slightly positive IgA anti-tissue transglutaminase and positive anti-endomyseal antibody tests, as well as an elevated serum total IgE level. An initial endoscopy revealed a few aphtous ulcerations in the bulb. Histology of the duodenum revealed patchy increased eosinophilic infiltrates (30-40/HPF) with glandular infiltration. There was also patchy mild villous blunting, and mildly increased intraepithelial lymphocytes (IEL) focally. A colonoscopy was macroscopically normal, but histology also showed focal areas with mild active colitis, characterized by eosinophilic infiltrates in the lamina propria. The findings were not considered diagnostic of Crohn’s or celiac disease. She was confirmed to be HLADQ2 positive. A wireless videocapsule endoscopy (CE) was then performed, which revealed features typical of celiac disease: delayed appearance of villi in mid jejunum; scalloping of folds with a mosaic pattern to the proximal small bowel mucosa. Surprisingly, several fibrincovered and actively bleeding mucosal ulcerations were seen in the jejunum. The ulcerative jejunitis was confirmed by push enteroscopy. Histological findings now confirmed typical findings of celiac disease in the distal duodenum and jejunum to 115 cm (subtotal villous atrophy, increased crypts and IEL) focally, with spared areas. T cell markers were normal and no clonality suggestive of EATL were found. After 6 months on a gluten free diet, a repeat CE revealed notable improvement but persistence of fewer jejunal ulcerations and areas with villous atrophy. A repeat push enteroscopy was performed and repeat samples for T-cell markers and clonality are pending. Ulcerative jejunitis due to refractory celiac disease has never previously been reported in the pediatric age group (!21 years). The use of CE was of utmost importance in this child, as we would not have otherwise diagnosed the ulcerative jejunitis.
www.giejournal.org