P1.31: Salvage therapy with Infliximab for anastomotic ulcers- a potential treatment for a challenging complication

Abstract

Background:Children with short bowel syndrome (SBS) may require bowel lengthening procedures such as the Serial Transverse Enteroplasty (STEP) procedure. STEP may be associated with severe complications such as anastomotic and staple line ulcers with gastrointestinal (GI) bleeding. These complications pose a therapeutic challenge and are often refractory to medical therapy. We present a child with recurrent GI bleeds from staple line ulcers responsive to treatment with Infliximab (IFX). Case:An 11 year old male with SBS and intestinal failure secondary to gastroschisis with midgut atresia underwent STEP procedures at 4 months of age and again at 4 years. He developed recurrent GI bleeds and was found to have multiple ulcers along staple lines on repeat endoscopies. Initial treatment included mesalamine (65mg/kg/day), oral budesonide (9mg), pantoprazole, omega-3, and cycling antibiotics with no improvement. Due to failure of all treatment attempts by 8 years old, he was started on IFX (6.5mg/kg/dose at week 0, 2, and 6 weeks then every 8 weeks). IFX level at dose #4 was <0.035ug/mL, anti-IFX antibodies <2AU/mL. Almost complete mucosal healing was found on endoscopy after dose #4. At infusion #6, anti-IFX antibodies were 70AU/mL, the child developed a rash and fever during the infusion and IFX was stopped. Other bioplogic therapies attempted included Adalimumab and Golimumab, not effective in preventing recurrent GI bleeding. The failure of medical therapies led to massive small bowel resection with removal of all areas involved with the previous STEP procedures. The most proximal staples were left in situ. Despite resection, new ulcers and GI bleeding occurred at the remaining staple site and in the duodenum. As a result IFX was reintroduced with methylprednisolone for premedication. The patient received IFX (10mg/kg/dose, week 0 and 2), repeat induction at week 4 due to low levels of IFX <0.035ug/mL. Follow up endoscopy showed excellent mucosal healing after 3 doses of IFX. Due to high anti-IFX antibodies, 190AU/mL, IFX was held at dose #4. Conclusion:Anastomotic and staple line ulcers post-STEP procedures were responsive to treatment with IFX in this case. Development of anti-IFX antibody can be a barrier for a successful outcome and should be monitored. The response to IFX suggests a potential role for innate immunity and TNF alpha in the pathogenesis of anastomotic ulcers.