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* = Presenting author

P329 Intermittent vancomycin and gentamicin as exclusive therapy for severe very early onset Inflammatory Bowel Disease

R. Lev-Tzion, O. Ledder, E. Shteyer, D. Turner*

Shaare Zedek Medical Center, Juliet Keidan Institute of Pediatric Gastroenterology and Nutrition, Jerusalem, Israel

Background

Very Early Onset Inflammatory Bowel Disease (VEO-IBD) is a unique subtype of IBD and many patients are resistant to standard therapy. VEO-IBD is more often associated with monogenic etiologies, particularly those with infantile onset. While children with both PSC and IBD treated with vancomycin have been reported to incidentally show improvement in IBD as well, vancomycin has never been studied as exclusive therapy for VEO-IBD.

Methods

We report here our experience using oral vancomycin and gentamicin (V&G) to successfully treat two patients with VEO-IBD refractory to standard treatments.

Results

Patient 1, with severe Crohn's colitis, presented at 5 months with hematochezia and subsequent diarrhea, failure to thrive and elevated inflammatory markers. Colonoscopy revealed aphthous ulcerations in the rectosigmoid and cecum with granulomas. Investigation for immune deficiency and interleukin-10 defects was negative. Treatment with corticosteroids, exclusive enteral nutrition, sulfasalazine and infliximab was unsuccessful; he had an allergic rash on azathioprine. At the age of 14 months he fully responded (PUCAI=0) within 5 days to oral V&G following 6 months of chronically active disease (PUCAI 20-85). Over the next 14 months he received no maintenance treatment; he had 2 exacerbations which were successfully treated with 2 week courses of V&G. Seven months after completing the third course, he continues to be in complete clinical remission with no medications.

Patient 2 had intermittent hematochezia starting at 8 months of age. At 2.5 years she presented with bloody diarrhea along with elevated transaminases and GGT. Colonoscopy demonstrated pancolitis and liver biopsy was consistent with PSC. Investigation for immune deficiency and interleukin-10 defects was negative. She was refractory to 5-ASA. Over the next 7 months she received three courses of oral V&G with prompt and complete remission each time of both her colitis symptoms and her liver markers, including normalization of CRP. At last follow-up she is in complete clinical remission with normal transaminases and GGT without any maintenance treatment.

Conclusion

We have reported the first two cases of VEO-IBD successfully treated with only oral antibiotics, using intermittent courses of vancomycin and gentamicin. The treatment also resulted in normalization of liver enzymes in a patient who had concurrent PSC. Both antimicrobial and immunomodulatory effects, including stimulation of regulatory T cells, may play a role in the mechanism of action. As VEO-IBD is often difficult to treat, our findings represent a potential treatment and should be further investigated in controlled trials.