Esophageal Crohn's disease treated successfully with adalimumab.

To the Editor: A 38-year-old woman with a longstanding history of colitis, initially diagnosed as ulcerative colitis, was treated with sulfasalazine. Recently she had been treated with balsalazide and later with infliximab for severe attacks unresponsive to 5-ASA products, while the patient refused treatment with steroids. Despite good initial response, the patient experienced an attenuated response to infliximab and was later treated with IV cyclosporine (CsA) with excellent response and maintained with oral 6-mercaptopurine (6-MP; 50 mg po daily) and oral CsA. Four months later she presented with odynophagia, dysphagia, and weight loss. The patient underwent an upper gastrointestinal endoscopy that revealed the presence of extensive esophageal ulcerations in the midesophagus (Fig. 1A). Biopsies revealed acute and chronic inflammation with extensive ulcerations but no granulomas. Viral pathogens such as CMV or HSV and fungal organisms were not identified. Concomitant flexible sigmoidoscopy revealed patchy inflammatory changes with deep ulcerations in the rectosigmoid, and biopsies revealed acute and chronic inflammation, crypt architectural distortion, Paneth cell metaplasia, and several foci of ulceration but no granulomas or viral inclusions. In the absence of an infectious etiology a diagnosis of esophageal Crohn’s disease (CD) was made. The CsA was discontinued. The patient was treated with an adalimumab loading dose of 80 mg subcutaneously, followed by 40 mg every 2 weeks. Her symptoms dramatically improved within 1 week, and she remains well on adalimumab maintenance treatment and 6-MP. A repeat upper gastrointestinal endoscopy showed complete healing of the esophageal ulcerations (Fig. 1B), and biopsies showed no significant pathologic changes. Esophageal involvement in CD is infrequent. Decker and colleagues reported on 20 cases of esophageal CD between 1976 and 1998 at Mayo Clinic Rochester.1 Similar to our case, extraesophageal CD preceded or was found at the same time as the diagnosis of esophageal CD in all cases.1 Endoscopic findings included ulcerations or erosions and rarely strictures or fistula, whereas histology showed chronic active inflammation and ulcerations but no granulomata.1 Anti-TNF treatment has revolutionalized the management of CD, particularly when refractory to conventional treatment.2 Infliximab, a chimeric anti-TNF that was the first biologic treatment approved for the treatment of CD, is effective for the induction and maintenance of remission in inflammatory and fistulizing CD.2 Previous reports have also indicated that infliximab is effective for the treatment of esophageal CD.3–8 We have also successfully treated several patients with infliximab for esophageal CD (authors’ unpublished observations). Adalimumab, a human anti-TNF antibody, has also been reported as an effective treatment for both induction and maintenance of remission for CD,2 and in the current study we report also on the successful use of adalimumab as treatment for esophageal CD. We chose adalimimab since our patient had been previously treated with infliximab and developed an attenuated response to that treatment.9,10 In the pre-anti-TNF treatment era, treatment for esophageal CD included steroids and/or immunomodulators such as 6-MP or azathioprine.1 Although esophageal CD is rare, antiTNF agents, including adalimumab, appear to be effective, particularly for those patients whose CD is refractory to conventional treatments.