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7Pm INTESTINE/COLON/IBD 42 PYLORIC METAPLASIA AS a HISTOLOGICAL CLUE for DIAGNOSIS of CROHN's DISEASE in the ABSENCE of GRANULOMA

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7Pm INTESTINE/COLON/IBD 42 PYLORIC METAPLASIA AS a HISTOLOGICAL CLUE for DIAGNOSIS of CROHN's DISEASE in the ABSENCE of GRANULOMA Clinical Vignette Poster Session I Thursday, October 20, 2011 5pm - 7pm INTESTINE/COLON/IBD 42 PYLORIC METAPLASIA AS A HISTOLOGICAL CLUE FOR DIAGNOSIS OF CROHN'S DISEASE IN THE ABSENCE OF GRANULOMA. Paul, Adam 1; Malkani, Anjali 1; Twaddell, William 1, 2, 1. Pediatrics, University of Maryland Children's Hospital, Baltimore, MD, United States. 2. Pathology, University of Maryland Medical Center, Baltimore, MD, United States. Pyloric metaplasia of colonic mucosa is a rare pathologic finding that occurs due to replacement of inflamed and ulcerated mucosa with regenerative tissue. It is a well documented feature of Crohn’s disease in the pathology literature, but is underrepresented in pediatric gastroenterology literature. We present a 16 year old female presenting with a one month history of generalized abdominal pain, nausea, vomiting, and significant weight loss worsening over the last 3 days. Abdominal CT showed significant terminal ileum thickening, and luminal narrowing, as well as features of early intramural abscess formation. Infectious work up was negative, but acute phase reactants and stool calprotectin were elevated. She was started on piperacillin/tazobactam and metronidazole for abscess treatment. Once the abscess had improved radiographically, she underwent endoscopy and colonoscopy which showed localized granular mucosa in the cecum, and moderate inflammation at the IC valve with marked swelling, with a visually normal recto‐sigmoid and transverse colon. Intubation of the IC valve was not possible due to the degree of swelling. However, biopsies from cecum showed focal acute cryptitis as well as crypt distortion and pyloric metaplasia. The remainder of the colonic, duodenal, gastric and esophageal biopsies were normal. She was started on high dose steroids and azathioprine with resolution of symptoms. Despite being a rare and nonspecific finding, pyloric metaplasia should be looked for to support biopsy diagnosis of Crohn’s disease in the absence of granuloma formation, as it is not a typical feature of Ulcerative Colitis. 43 A SEVERE EXACERBATION OF ULCERATIVE COLITIS AFTER RITUXIMAB THERAPY IN A PATIENT WITH IDIOPATHIC THROMBOCYTOPENIC PURPURA . Raizner, Aileen 1; Phatak, Uma Padhye1; Pashankar, Dinesh S1, 1. Pediatrics, Yale University, New Haven, CT, United States. Background: Rituximab (an anti‐CD20 antibody) is widely used for autoimmune disorders including Idiopathic Thrombocytopenic Purpura (ITP). Severe ulcerative colitis (UC) has been reported to occur following rituximab therapy in a child with nephrotic syndrome (Pediatrics 2010) and an adult with Grave’s disease (Gut 2008). Effects of rituximab in patients with both UC and an autoimmune disorder have not been reported. Case Report : A 17‐year‐old male presented with a history of bloody stools and abdominal pain. His father had a colectomy for severe UC. Blood work showed Hb 10 g/dL and platelets 14,000/uL. UC was suspected but endoscopy was deferred due to thrombocytopenia. ITP was subsequently diagnosed. He failed to respond to iv immunoglobulin and Rho (D) Immune Globulin. He received 3 weekly doses of rituximab 750 mg. Two weeks after the third infusion, he presented with severe abdominal pain and progressive bloody diarrhea occurring 25 times per day (stool output 2 liters/day). Blood work showed hemoglobin 8.4g/dL, platelets 150,000/uL,C‐reactive protein 171mg/dL, a positive p‐ANCA , and negative ASCA antibody. Stool tests were negative for infection. An upper endoscopy with histology was unremarkable. A sigmoidoscopy revealed severe ulcerations. Histology revealed severe, chronic active colitis without granulomata. Flow cytometry from 6 weeks through 16 weeks after rituximab therapy revealed virtually no circulating B cells, consistent with anti CD20 therapy, and normal circulating T cells. He was hospitalized and treated with bowel rest, parenteral nutrition and high dose intravenous steroids for a week. He slowly improved and presently is doing well on tapering prednisone and 6‐MP therapy. Conclusions‐ Although rituximab therapy led to improvement in ITP, it also led to a severe exacerbation of UC in our patient. This clinical pattern of severe colitis with B cell depletion is similar to the previous reports. We recommend caution and vigilant monitoring when using rituximab in patients with both UC and autoimmune disorders. 44 MESALAMINE‐INDUCED EOSINOPHILIC PNEUMONIA IN AD 15 YEAR OL BOY WITH CROHN’S DISEASE. Lucia, Chantal 1; Garcia, Jennifer 2; Reeves‐Garcia, Jesse 1; Muinos, William 1; Gomara, Roberto 1; Ambati, Shashikanth 1; Hernandez, Erick 1, 1. Pediatric Gastroenterology, Miami Children's hospital, Miami, FL, United States. 2. Pediatric Gastroenterology, Miller School of Medicine. University of Miami, Miami, FL, United States. A 15 year‐old boy with Crohn’s Disease presented with a one day history of sharp chest pain worsened by movement and inspiration localized to left upper side of the chest. He denied cough, fever, night sweats, weight loss, diarrhea and rash. 6‐Mercaptopurine (6MP) immunosuppression and Asacol (Mesalamine) had been started five months prior. Additionally, the patient had been treated for Mycobacterium fortuitum in the stool two weeks prior and had returned back from the Dominican Republic four days prior.Physical exam was remarkable for tenderness on the left second rib. A left upper lobe nodule was discovered on chest X‐ Ray that had not been present one month previously. Computed tomography (CT) of the chest showed nonspecific bilateral non‐calcified sub‐centimeter pulmonary nodules with pleural nodules centered in the upper lung zones. Right thoracoscopy and wedge resection was performed. Biopsy results showed eosinophilic pneumonia with no discrete granulomas. Special stains were negative for acid fast and fungal stains. Electron microscopy was negative for Birbeck granules or viral inclusions. The patient remained on 6MP and Asacol was discontinued. There was complete resolution of respiratory symptoms, and repeat CT chest three months later showed no pulmonary nodule. Conclusion: Although the differential for a pulmonary nodule in an immunosuppressed patient with inflammatory bowel disease is broad. The diagnostic findings of eosinophilia, upper lobe involvement, and pulmonary eosinophilia on biopsy are highly suggestive of Mesalamine toxicity. This case illustrates how eosinophilic pneumonia should be considered in the pediatric population who develop pulmonary nodules while on Mesalamine therapy. 45 METASTATIC CROHN’S DISEASE IN A TEENAGE BOY ON INFLIXIMAB. Kutsch, Erika 1; Figueroa, Ernesto 2; Adeyemi, Adebowale 1, 1. Gastroenterology, AI duPont Hospital for Children , Wilmington, DE, United States. 2. Urology, AI duPont Hospital for Children, Wilmington, DE, United States. Case: A 16 year old male presented with weight loss, diarrhea, erythema nodosum and scrotal swelling. Colonoscopy findings included diffuse edema, pseudopolyps, exudate and aphthae. Pathology was consistent with granulomatous colitis. He underwent Infliximab induction, followed by 5mg/kg every 8 weeks. Four months later, scrotal and penile edema reoccured in the absence of bowel symptoms. Infliximab trough was therapeutic (7.6mg/ml). MRI showed marked edema of scrotal sac without evidence of fistula. He was treated with prednisone, metronidazole and topical tacrolimus with improvement over a few weeks. Three months later, he again developed scrotal and penile edema. Repeat colonoscopy revealed colonic mucosal healing. Scrotal biopsy did not show granulomas but lymphohistiocytic infiltration, consistent with cutaneous manifestation of Crohn's disease (CD). A short course of prednisone and infliximab dose increase to 10mg/kg resulted in sustained resolution of his scrotal and penile edema. Discussion: Metastatic CD of the genitourinary system is a rare extraintestinal manifestation that is histologically similar to inflammation seen in the intestinal tract. In patients with metastatic CD, cutaneous manifestation and intestinal involvement do not always follow parallel course as adequate treatment of bowel disease does not predict improvement of cutaneous lesions. Treatment of metastatic CD with different agents, including steroids, cyclosporin A, 6‐ mercaptopurine, sulfasalazine and antibiotics have shown variable success. Treatment with infliximab has shown promising results. Several case reports have suggested its efficacy in treatment when other therapies have been unsuccessful. We hope that maximizing the infliximab dose in our patient will lead to sustained remission of cutaneous findings. 46 MEGALOGASTRIA IN CROHN’S DISEASE. Ahmad, Fareed 1; Steiner, Steven 1, 1. Riley Hospital for Children, Indianapolis, IN, United States. A 16‐year‐old Caucasian female was referred with a one month history of abdominal pain, vomiting, and weight loss. Physical examination was remarkable for mild abdominal tenderness, distension and a perianal skin tag with underlying fissure. Laboratory examination revealed anemia (Hemoglobin 9 gm/dl), elevated inflammatory markers (ESR 38 mm/hr, CRP 2.8 mg/dl), and hypoalbuminemia (2.5 g/dl). Abdominal CT scan (Panel A) demonstrated megalogastria, extending into the right pelvis. Upper gastrointestinal endoscopy revealed a large, fluid‐filled stomach with a thickened, erythematous pylorus and a large duodenal bulb ulcer, with duodenal narrowing (Panel B). There was no evidence of Helicobacter pylori on gastric biopsy. Active chronic duodenitis was observed. Anti‐Saccharomyces cerevisiae antibodies were
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