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Unusual gastric Crohn’s disease treated with infliximab - a case report
S190
Abstracts
Purpose: Azathioprine and 6 mercaptopurine are commonly used agents for the treatment of inflammatory bowel disease. Leukopenia is a well recognized side effect and serial monitoring of the CBC is necessary. We report a case of 6 – mercaptopurine (6 –MP) induced neutropenia treated with G–CSF in a 36 –year old man with Crohn’s disease (CD). The patient is status post subtotal colectomy with ileostomy in December 1997 with an ileo–rectal anastomosis in May 1998. He subsequently developed recurrent disease characterized by perianal fistulas, frequent loose stools and rectal bleeding. He was treated elsewhere with mesalamine, serial infusions of infliximab, 6 –MP, budesonide and antibiotics. In February 2002, his dose of 6 –MP was increased from 125 mg to 150 mg daily. At that time, his WBC count was 3,600 /mm3. He was admitted to Boston Medical Center in March 2002 with fever and severe perianal pain. An exam under anesthesia prior to admission revealed distal proctitis with anal ulcerations and a perianal fistula. A seton was placed. CT showed rectal wall thickening and perirectal inflammation. The WBC was 1,800/mm3 on admission with an absolute neutrophil count (ANC) of 558. 6 –MP was discontinued. Broad spectrum antibiotics were administered but high fevers persisted. His WBC decreased to 600/mm3 on hospital day 6 (ANC of 132). G–CSF (neupogen®) 300 mcg/day sq was initiated on the third hospital day and continued for a total of 7 days. Within five days of therapy with G–CSF, his WBC count was 2,800/mm3 with an ANC greater than 900/mm3. Peak WBC was 12,300/mm3. The patient defervesced and antibiotics were discontinued. Leukopenia did not recur following discontinuation of G–CSF. The patient underwent a diverting ileostomy and has done well. Previous reports have described the safety and efficacy of G–CSF in patients with cyclic neutropenia associated with CD as well as mesalamine–induced leukopenia. In a small open labeled study in 11 patients, GM–CSF given sq daily for 8 weeks was beneficial in the treatment of fistulizing and mucosal CD. No patient worsened and side effects were not significant. Placebo controlled studies are needed to determine the efficiacy of G–CSF and GM–CSF as a primary treatment for CD. In summary, our case demonstrates that G–CSF was effective and safe in the management of 6 –MP induced neutropenia.
580 INFANT WITH PANCREATIC ASCITES: AN UNUSUAL PRESENTATION OF AN UNUSUAL DISORDER Miguel Saps, M.D., Seema Khan, M.D., Alka Goyal, M.D., Barbara Gaines, M.D., Nader Youssef, M.D., Manisha Harpavat, M.D. and Carlo Di Lorenzo, M.D.*. Pediatric Gastroenterology and Pediatric Surgery, Children’s Hospital of Pittsburgh, University of Pittsburgh, Pittsburgh, PA. Purpose: Pancreatic ascites is a rare pediatric diagnosis and only 12 cases affecting infants have been reported. Thus, there is limited knowledge regarding the etiology, management and prognosis of this condition. Methods: We report our experience with the diagnosis and management of a 4 –mo– old male with pancreatic ascites. Case: A 4 –mo– old previously healthy male with an unremarkable birth history was evaluated for increasing abdominal distention for 9 days. Initially, he had associated fever and irritability attributed to acute otitis media, and improved once treated with amoxicillin. He also had non– bilious vomiting and diarrhea lasting one day during the course of antibiotic treatment. On examination he appeared well, afebrile, anicteric and had no peripheral edema. The abdomen was diffusely distended, soft, non–tender, without masses but with a fluid thrill. Pertinent investigations included: serum amylase 23 IU/L, lipase 2279 IU/L, albumin 2.3 g/dL, sodium 125mEq/L, potassium 6.1mEq/L, platelets 1044 x109/L, elevated ascitic fluid lipase, and serum–ascites albumin gradient ⬍1.1. Abdominal CT showed a large amount of ascites, edema and a cyst within the head of the pancreas. Liver enzymes, viral titres, blood cultures, calcium phosphorus, magnesium, lipid profile, sweat test, and hereditary pancreatitis tests were normal. This asymptomatic child with ascites was managed with bowel rest, total parenteral nutrition, albumin infusions, furosemide, subcutaneous
AJG – Vol. 97, No. 9, Suppl., 2002
octreotide, and paracentesis without improvement. At this stage an endoscopic retrograde cholangio–pancreatography demonstrated a disrupted pancreatic duct and a cystic fluid collection. A pancreatic stent did not prevent re–accumulation of ascites. An exploratory laporatomy was then performed and revealed a cyst. Cystogastrostomy showed rapid resolution of pancreatitis and ascites. The patient has had no recurrence of pancreatic ascites during a 2–mo follow– up on a regular diet. Conclusions: Pancreatic ascites is an uncommon complication of pancreatitis that may produce few to no symptoms in infants. We speculate that our patient had pancreatitis, possibly viral in origin, complicated by a disrupted duct and/or ruptured cyst leading to ascites formation. Medical management was ineffective. Surgery appears to have been curative. 581 UNUSUAL GASTRIC CROHN’S DISEASE TREATED WITH INFLIXIMAB – A CASE REPORT Michael G. Firth, M.D. and Charlene M. Prather, M.D.*. Division of Gastroenterology and Hepatology, Saint Louis University School of Medicine, St Louis, MO. Purpose: This is an unusual case report of treatment of severe gastric Crohns disease treated with infliximab. A 35 year– old African American female presented to the hospital with a 2 week history of epigastric abdominal pain, associated with recurrent nausea and vomiting. The patient reported prior good health, although she had an unexplained weight loss of 15 lbs over 2 months prior to this acute illness. Her review of systems was otherwise normal. She denied any anorexia, bowel irregularity, or hematochesia. She noted no fever, chills or night sweats. Medications were limited to oral contraceptives, with no NSAID use. She admitted to smoking 1⁄2 ppd of cigarettes, but denied alcohol or illicit drug use. Family history was unremarkable. On physical examination, the patient appeared weak and fatigued, and had recurrent wretching. VS were normal. She had no skin lesions nor lymphadenopathy. Her abdominal exam revealed moderately severe epigastric tenderness, with no perirectal abnormalities. Her WBC was 10,200 with 35% bands. The electrolytes, amylase/lipase, HCG, and hepatic functions were normal, except for an albumin of 2.5. An abdominal CT scan showed severe concentric thickening of the stomach with possible thickening of the cecum and terminal ileum. EGD demonstrated marked diffuse thickening and ulceration throughout the antrum and duodenal bulb. Colonoscopy showed a few isolated aphthous and linear ulcerations in the ascending colon, cecum and terminal ileum. Gastric histology revealed marked inflammatory changes and ulceration extending through the muscularis mucosa, but without clear granulomas. Studies of the colon and ileum showed architectural distortion, chronic inflammation and cryptitis, consistent with Crohns disease. AFB and immune histochemical stains were negative. The patient was started on therapy with methylprednisolone 60 mg IV and pantoprazole 40 mg IV daily. Her symptoms gradually improved over 5 days, but worsened twice when switched to oral prednisone. Repeat EGD at 2 weeks showed little clinical change. Azathioprine 50 mg/day was added and the patient received an initial dose of infliximab 5mg/kg IV. Her symptoms markedly improved over the following week. She was given additional doses of infliximab at 2, 8 and 16 weeks with continued clinical remission.This case represents an unusual presentation of gastric Crohns disease with very limited distal small bowel involvement, and shows the clinical effectiveness of infliximab in this setting. 582 A MODEL PROGRAM FOR THE MANAGEMENT OF CHRONIC HEPATITIS C IN LARGER PRACTICES Brian R. Willis, M.D. and Elaine R. Baker, M.D.*. Gastroenterology, Kaiser Permanente Northwest, Portland, OR and Gastroenterology, Kaiser Permanente Northwest, Portland, OR. Purpose: An individualized, time–intensive, and “first come,first serve” approach to the care of chronic Hepatitis C patients is commonplace in the
Abstracts
Purpose: Azathioprine and 6 mercaptopurine are commonly used agents for the treatment of inflammatory bowel disease. Leukopenia is a well recognized side effect and serial monitoring of the CBC is necessary. We report a case of 6 – mercaptopurine (6 –MP) induced neutropenia treated with G–CSF in a 36 –year old man with Crohn’s disease (CD). The patient is status post subtotal colectomy with ileostomy in December 1997 with an ileo–rectal anastomosis in May 1998. He subsequently developed recurrent disease characterized by perianal fistulas, frequent loose stools and rectal bleeding. He was treated elsewhere with mesalamine, serial infusions of infliximab, 6 –MP, budesonide and antibiotics. In February 2002, his dose of 6 –MP was increased from 125 mg to 150 mg daily. At that time, his WBC count was 3,600 /mm3. He was admitted to Boston Medical Center in March 2002 with fever and severe perianal pain. An exam under anesthesia prior to admission revealed distal proctitis with anal ulcerations and a perianal fistula. A seton was placed. CT showed rectal wall thickening and perirectal inflammation. The WBC was 1,800/mm3 on admission with an absolute neutrophil count (ANC) of 558. 6 –MP was discontinued. Broad spectrum antibiotics were administered but high fevers persisted. His WBC decreased to 600/mm3 on hospital day 6 (ANC of 132). G–CSF (neupogen®) 300 mcg/day sq was initiated on the third hospital day and continued for a total of 7 days. Within five days of therapy with G–CSF, his WBC count was 2,800/mm3 with an ANC greater than 900/mm3. Peak WBC was 12,300/mm3. The patient defervesced and antibiotics were discontinued. Leukopenia did not recur following discontinuation of G–CSF. The patient underwent a diverting ileostomy and has done well. Previous reports have described the safety and efficacy of G–CSF in patients with cyclic neutropenia associated with CD as well as mesalamine–induced leukopenia. In a small open labeled study in 11 patients, GM–CSF given sq daily for 8 weeks was beneficial in the treatment of fistulizing and mucosal CD. No patient worsened and side effects were not significant. Placebo controlled studies are needed to determine the efficiacy of G–CSF and GM–CSF as a primary treatment for CD. In summary, our case demonstrates that G–CSF was effective and safe in the management of 6 –MP induced neutropenia.
580 INFANT WITH PANCREATIC ASCITES: AN UNUSUAL PRESENTATION OF AN UNUSUAL DISORDER Miguel Saps, M.D., Seema Khan, M.D., Alka Goyal, M.D., Barbara Gaines, M.D., Nader Youssef, M.D., Manisha Harpavat, M.D. and Carlo Di Lorenzo, M.D.*. Pediatric Gastroenterology and Pediatric Surgery, Children’s Hospital of Pittsburgh, University of Pittsburgh, Pittsburgh, PA. Purpose: Pancreatic ascites is a rare pediatric diagnosis and only 12 cases affecting infants have been reported. Thus, there is limited knowledge regarding the etiology, management and prognosis of this condition. Methods: We report our experience with the diagnosis and management of a 4 –mo– old male with pancreatic ascites. Case: A 4 –mo– old previously healthy male with an unremarkable birth history was evaluated for increasing abdominal distention for 9 days. Initially, he had associated fever and irritability attributed to acute otitis media, and improved once treated with amoxicillin. He also had non– bilious vomiting and diarrhea lasting one day during the course of antibiotic treatment. On examination he appeared well, afebrile, anicteric and had no peripheral edema. The abdomen was diffusely distended, soft, non–tender, without masses but with a fluid thrill. Pertinent investigations included: serum amylase 23 IU/L, lipase 2279 IU/L, albumin 2.3 g/dL, sodium 125mEq/L, potassium 6.1mEq/L, platelets 1044 x109/L, elevated ascitic fluid lipase, and serum–ascites albumin gradient ⬍1.1. Abdominal CT showed a large amount of ascites, edema and a cyst within the head of the pancreas. Liver enzymes, viral titres, blood cultures, calcium phosphorus, magnesium, lipid profile, sweat test, and hereditary pancreatitis tests were normal. This asymptomatic child with ascites was managed with bowel rest, total parenteral nutrition, albumin infusions, furosemide, subcutaneous
AJG – Vol. 97, No. 9, Suppl., 2002
octreotide, and paracentesis without improvement. At this stage an endoscopic retrograde cholangio–pancreatography demonstrated a disrupted pancreatic duct and a cystic fluid collection. A pancreatic stent did not prevent re–accumulation of ascites. An exploratory laporatomy was then performed and revealed a cyst. Cystogastrostomy showed rapid resolution of pancreatitis and ascites. The patient has had no recurrence of pancreatic ascites during a 2–mo follow– up on a regular diet. Conclusions: Pancreatic ascites is an uncommon complication of pancreatitis that may produce few to no symptoms in infants. We speculate that our patient had pancreatitis, possibly viral in origin, complicated by a disrupted duct and/or ruptured cyst leading to ascites formation. Medical management was ineffective. Surgery appears to have been curative. 581 UNUSUAL GASTRIC CROHN’S DISEASE TREATED WITH INFLIXIMAB – A CASE REPORT Michael G. Firth, M.D. and Charlene M. Prather, M.D.*. Division of Gastroenterology and Hepatology, Saint Louis University School of Medicine, St Louis, MO. Purpose: This is an unusual case report of treatment of severe gastric Crohns disease treated with infliximab. A 35 year– old African American female presented to the hospital with a 2 week history of epigastric abdominal pain, associated with recurrent nausea and vomiting. The patient reported prior good health, although she had an unexplained weight loss of 15 lbs over 2 months prior to this acute illness. Her review of systems was otherwise normal. She denied any anorexia, bowel irregularity, or hematochesia. She noted no fever, chills or night sweats. Medications were limited to oral contraceptives, with no NSAID use. She admitted to smoking 1⁄2 ppd of cigarettes, but denied alcohol or illicit drug use. Family history was unremarkable. On physical examination, the patient appeared weak and fatigued, and had recurrent wretching. VS were normal. She had no skin lesions nor lymphadenopathy. Her abdominal exam revealed moderately severe epigastric tenderness, with no perirectal abnormalities. Her WBC was 10,200 with 35% bands. The electrolytes, amylase/lipase, HCG, and hepatic functions were normal, except for an albumin of 2.5. An abdominal CT scan showed severe concentric thickening of the stomach with possible thickening of the cecum and terminal ileum. EGD demonstrated marked diffuse thickening and ulceration throughout the antrum and duodenal bulb. Colonoscopy showed a few isolated aphthous and linear ulcerations in the ascending colon, cecum and terminal ileum. Gastric histology revealed marked inflammatory changes and ulceration extending through the muscularis mucosa, but without clear granulomas. Studies of the colon and ileum showed architectural distortion, chronic inflammation and cryptitis, consistent with Crohns disease. AFB and immune histochemical stains were negative. The patient was started on therapy with methylprednisolone 60 mg IV and pantoprazole 40 mg IV daily. Her symptoms gradually improved over 5 days, but worsened twice when switched to oral prednisone. Repeat EGD at 2 weeks showed little clinical change. Azathioprine 50 mg/day was added and the patient received an initial dose of infliximab 5mg/kg IV. Her symptoms markedly improved over the following week. She was given additional doses of infliximab at 2, 8 and 16 weeks with continued clinical remission.This case represents an unusual presentation of gastric Crohns disease with very limited distal small bowel involvement, and shows the clinical effectiveness of infliximab in this setting. 582 A MODEL PROGRAM FOR THE MANAGEMENT OF CHRONIC HEPATITIS C IN LARGER PRACTICES Brian R. Willis, M.D. and Elaine R. Baker, M.D.*. Gastroenterology, Kaiser Permanente Northwest, Portland, OR and Gastroenterology, Kaiser Permanente Northwest, Portland, OR. Purpose: An individualized, time–intensive, and “first come,first serve” approach to the care of chronic Hepatitis C patients is commonplace in the