behet 's disease ( bd ) was first described by behet in 1937 as a chronic recurrent disease characterized by recurrent oral ulceration , genital ulceration and ocular involvement1 ) , additional involvement of many other organs has been reported including : joints , skin , blood vessels , the nervous system and gastrointestinal tract2 ) . reports on invasion into the gastrointestinal tract vary depending on country , however , it has been reported to be present in three to 60% of bd patients3 ) . for these patients , medical treatment is the primary approach to therapy , however , additional surgical treatment is necessary for cases with complications such as , perforation , intractable pain and hemorrhage . however , lesions frequently recur after surgical treatment and the incidence of more than one surgical procedure is high ; such cases may be untreatable and lead to death . recently , cases of bd with gastrointestinal involvement have been treated with tumor necrosis factor alpha ( tnf- ) antibody ( infliximab)4 , 5 ) , however , there is no prior case of such treatment reported in korea . we treated a patient with recurrent intestinal ulcer who underwent right hemicolectomy due to uncontrolled intestinal bleeding with infliximab . a 47 years old male patient was admitted to the hospital due to severe abdominal pain for five days . the past medical history was significant for recurrent oral and genital ulceration over the past 20 years . he underwent right hemicolectomy due to uncontrolled intestinal bleeding caused by intestinal behet 's ulceration 10 years previously . subsequently , he was treated by conventional medical therapy including corticosteroids , sulfasalazine and azathioprine . however , intestinal behet 's ulceration recurred at the anastomosis site and the patient required several subsequent hospitalizations due to repeated abdominal pain and intestinal bleeding . the physical examination showed an acutely ill patient with a blood pressure of 120/70 mmhg , a pulse rate of 94 times / min , a respiratory rate of 24 times / min and a temperature of 38.5. the abdomen was flat with mild tenderness ; the bowel sounds were normal . the laboratory test results showed a white blood cell count , 540/mm ; hemoglobin , 7.2 g / dl ; hematocrit , 28% ; platelet count , 47,000/mm ; total protein , 5.7 g / dl ; and erythrocyte sedimentation rate ( esr ) was increased to 60 mm / h and c - reactive protein ( crp ) was increased to 70 mg / l . the crohn 's disease activity index ( cdai)6 ) at the time of admission was 183 and the harvey - bradshaw index ( hbi)7 ) was 10 . the computed tomography showed a suspicious large deep ulcer with bowel wall thickening at the right hemicolectomy site but no obstruction or fistula was observed ( figure 1 ) . a colonoscopy showed the right hemicolectomy and a well demarcated large deep ulcer with a clear ulcer bed at the stomal site ( figure 2 ) . colonic biopsies showed ulceration with necrotic fibrin debris , an inflammatory infiltrate with neutrophils predominating but no granuloma ( figure 3 ) . because of the uncontrolled continuous abdominal pain , infliximab ( 5 mg / kg ) was started . there were no observed side effects from the infliximab ; two weeks after the treatment was started , the symptoms improved dramatically . after one month , the laboratory testing showed a white blood cell count , 4,470/mm ; hemoglobin , 12.3 g / dl ; hematocrit , 36.8% and a platelet count , 168,000/mm . the esr and crp were decreased to 11 mm / h and 6.7 mg / l , respectively and the cdai and hbi were decreased to 88 and 2 , respectively . follow up colonoscopy showed a well demarcated marginal ulcer with granulation tissue at the stomal site , however , the ulcer size was decreased compared to the prior study ( figure 4 ) . a second infliximab ( 5 mg / kg ) treatment was administered one month after the first . the symptoms continued to improve and the abdominal pain resolved completely four months after the second injection of infliximab . the crp and esr were maintained in the normal range and the value of cdai and hbi continued to decrease . when intestinal ulceration occurs in patients with bd it is found predominately in the ileocecal region , however , ulcers may be found throughout the entire colon . colonoscopy findings usually show deep punched out ulcerations with nodular margins and converging folds ; the histological findings show a non specific inflammation . intestinal bd may be associated with a significant morbidity and mortality which results from the high complication rate8 , 9 ) . although the exact pathogenesis of bd is not completely understood , it has been suggested that the disease is due to an abnormal immune response to environmental factors such as microbial agents in genetically susceptible individuals10 ) . the presence of autoantibodies , a decrease in the t4/t8 ratio and an increased number of circulating natural killer cells and + t cells also suggest an autoimmune etiology of disease10 , 11 ) . in patients with active bd an increase of tnf in the blood and an increase of + t cells producing tnf have been observed12 ) . improvement of symptoms has been reported after the administration of tnf blockers in patients unresponsive to conventional therapy13 ) . therefore , tnf has been considered to play an important role in the inflammation observed in bd patients and this may account for the response to infliximab . conventional medical therapeutic options include : corticosteroids , mesalamine , thalidomide , bowel rest , total parenteral nutrition and immunosuppressive agents such as azathioprine2 ) . however , surgery is generally required due to resistance to medications , frequent recurrence of lesions and complications such as intestinal perforation and fistula formation . according to cha et al.14 ) , the rate of surgery over five years has been reported to be approximately 47% . recurrent ulcers after surgery are commonly found near the anastomosis site , as in this case ; the ulcers appear as deep multiple aphthous ulcers or as cutaneous intestinal fistula . when these ulcers are present , the recurrence rate has been reported to be as high as 40~80%15 ) . in our case , 10 years prior to the admission , the patient meet the criteria for the diagnosis of bd recommended by international study group for behet 's disease16 ) . based on repeated oral and genital ulceration as well as a positive pathergy test , severe abdominal pain and uncontrolled intestinal bleeding , the patient was diagnosed with intestinal bd and underwent a right hemicolectomy . despite conventional medical treatment , he had recurrent intestinal ulceration and intractable abdominal pain , therefore , another treatment modality was required . infliximab has been effective for induction and maintenance treatment in patients with bd and has been particularly successful for treatment of cases with severe mucocutaneous17 ) , gastrointestinal4 , 5 ) and ocular18 ) manifestations of bd . for patients with intestinal bd , in cases treated with infliximab , the standard dose of infliximab has not yet been established , however , treatment according to the dose and protocol used for crohn 's disease5 ) or based on the symptoms of the patient4 , 19 ) have been reported . the disease activity of the patient was evaluated before and after treatment , the cdai and hbi were used for behet 's disease because there is no activity index established to date . in our case , infliximab ( 5 mg / kg ) was administered at 0 and 4 weeks and symptomatic complete remission was observed at the 12 month follow up , in addition , endoscopic improvement was also observed . this case illustrates that for patients with bd who fail to respond to conventional treatment , infliximab may be a safe and effective new therapeutic option .
behet 's disease ( bd ) is a chronic relapsing multisystem disease characterized by oral ulceration , genital ulceration and ocular lesions . 
 gastrointestinal involvement is rare , often difficult to treat and associated with a high mortality rate . 
 we treated a 47-year - old korean man with bd who had a recurrent intestinal ulcer with tumor necrosis factor  antibody ( infliximab ) ; he initially underwent right hemicolectomy due to uncontrolled intestinal bleeding . for patients with intestinal bd who fail to respond to conventional treatment 
 , infliximab may be a safe and effective new therapeutic option .