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Excerpt
Esophageal fistula arising from Crohn's esophagitis is very rare, and its management has been particularly difficult. Infliximab has been shown to be efficacious in the treatment of enterocutaneous fistula. However, treatment of a perforating esophageal fistula with infliximab has not been reported.
On February 1, 2001, a 30-year-old man with history of Crohn's disease presented to The Brooklyn Hospital Center (New York, NY, U.S.A.) with dysphagia to liquids. In 1991, he had symptoms of abdominal pain and bloody diarrhea and was diagnosed with Crohn's colitis. In 1992, he was diagnosed with pyoderma gangrenosum of the left lower extremity and was treated with oral dapsone and a skin graft. In 1994, the patient reported dysphagia to both solids and liquids. An esophagogastroduodenoscopy revealed severe esophagitis with ulcerations, multiple fissures, and bridging mucosa spanning the entirety of the esophagus. Biopsies of the esophagus showed apthous ulceration of squamous mucosa and patchy acute and chronic esophagitis without evidence of granulomata or fungi. A diagnosis of Crohn's disease of the esophagus was made, and the patient was treated with oral mesalamine and metronidazole, which resulted in clinical improvement. However, a follow-up esophagram in 1995 demonstrated increased severity of the esophagitis. His dysphagia worsened, and he was prescribed nightly Rowasa enemas given orally and ciprofloxacin, which again resulted in symptomatic improvement. Follow-up esophagogastroduodenoscopy in 1996 confirmed that the esophageal inflammation had improved; however, many mucosal bridges, fissures, and ulcers remained. Several focal esophageal strictures were dilated with a through-the-scope balloon. The patient remained clinically stable for the next few years despite an esophagogastroduodenoscopy in 1999 that was essentially unchanged when compared with that performed in 1996.
On hospital admission, the patient was afebrile and stable. The pulmonary examination showed clear lung fields, except for mild dullness at the left lung base. The abdomen was benign. His white blood cell count was 30,300/mm3, and a chest x-ray revealed patchy infiltrate at the left lung base. Esophagogastroduodenoscopy showed severe diffuse fissures and bridging mucosa in the esophagus with focal strictures, as was noted in previous endoscopies. A gastrograffin esophagram and a chest computed tomography demonstrated a diffusely ulcerated esophagus and an esophagobronchial fistula at the level of the mid esophagus that was communicating with the left inferior lobar bronchus. A percutaneous gastrostomy was placed to optimize the patient's nutrition.
The patient took the first dose of 5 mg/kg infliximab while he was in the hospital in February 2001 and a second dose on day 11. Subsequent similar doses of infliximab were given as an outpatient on day 47 and on day 82. 6-Mercaptopurine at a dosage of 50 mg/d via a percutaneous endoscopic gastrostomy tube was initiated on March 2001; the infusions were well tolerated. The erythrocyte sedimentation rate remained elevated at 96 mm/h on June 16, 2001, but decreased to 11 mm/h by June 27th. A repeat barium esophagram on June 20, 2001, demonstrated a healing of the esophagobronchial fistula. Afterward, the patient restarted a soft oral diet and remained free of complications.
Infliximab is a chimeric monocloncal antibody directed against tumor necrosis factor-alfa. In randomized controlled trials, infliximab has been shown to be efficacious in treating moderate to severe Crohn's disease. 1,2 Recently, Heller et al. 3 reported successful infliximab treatment of a 74-year-old man with severe esophageal Crohn's disease. The patient presented with deep esophageal ulcerations and underwent treatment with concomitant steroids, metronidazole, and 6-mercaptopurine. Remarkably, there was significant resolution of the esophageal lesions 4 weeks after a single 5-mg/kg infusion. Steroids were tapered and discontinued at 6 months.
Present et al.
