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Saadah, O. I.1; Albokhari, S. M.1
1Department of Pediatrics, King Abdulaziz University Hospital, Jeddah, Saudi Arabia
Submitted by: firstname.lastname@example.org
Introduction: Anti-TNF-á has been reported to be effective in adult patients with fistulizing Crohn’s disease, but no data is available to support its use in infants. Herein, we report the use of Infliximab in an Arab infant with infantile Crohn’s disease with perianal fistulas.
Methods: Case report
Results: This is an 11 months old female infant. She was born at term with birth weight of 2.3 kg. She was initially breast fed supplemented with infant formula. At 4 weeks of age she started to have dyschezia and bloody streaking of stools. At the age of 3 months she had sigmodoscopy done which showed ulcerated rectal mucosa which was attributed to allergy for which she was put on elemental formula. At 6 months of age she had little response despite exclusive elemental formula intake and she was not gaining weight. She had a fleshy skin tag around the anus with skin ulceration over the sacrum. A month later the sacral ulcer has healed with the use of antibiotics but a fistula was opened into the right labia majora. She was the first child to consanguineous parents with no family history of IBD. Laboratory data included hemoglobin of 8.6 g/dl, C-reactive protein (CRP) of 89 and serum albumin of 13 g/L. Stool specimens for culture and microscopy were negative, and a Mantoux test was negative. Immune work up was normal, as well as testing for food allergens. Colonoscopy showed evidence of pancolitis more pronounced distally. Histopathology of the colonic biopsy shows chronic active colitis but no granuloma. She was managed initially with total parenteral nutrition for 8 weeks in addition to corticosteroids, salazopyrine and metronidazole. Repeated colonoscopy after 8-weeks of TPN showed moderate improvement in the mucosal inflammation, but no improvement in the fistula. Azathioprine was added to help on healing the fistula. 4 months later, the perianal disease was getting worse inspite of optimal immunosuppressive therapy, with another two fistulas opening into the perianal area with more ulceration at the surrounding areas. Infliximab was started after as an intravenous infusion at a dose of 5 mg/kg, followed by a second dose after 2 weeks, and then every 8 weeks. Patient had an allergic skin reaction and irritability during the second dose which was aborted using antihistamine and iv corticosteroid and the infusion was resumed after a short suspension without problems. At 4 weeks after the start of infliximab the stool became formed with no blood or mucus and the output from the fistula became less and the ulcerated skin around the fistula openings started to heal.
Conclusion: Infliximab is effective and probably safe in young infants with Crohn’s disease and severe perianal fistulas.
© 2004 Lippincott Williams & Wilkins, Inc.
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