|Year : 2013 | Volume
| Issue : 3 | Page : 238-240
Intestinal Behcet's disease presenting with massive lower gastrointestinal bleeding
Firas W Obeidat1, Fatima N Obeidat2, Awni D Shahait1
1 Department of General Surgery, The University of Jordan, Amman, Jordan
2 Department of Pathology, The University of Jordan, Amman, Jordan
|Date of Web Publication||28-Mar-2014|
Fatima N Obeidat
735, Amman 11953
Source of Support: None, Conflict of Interest: None
Gastrointestinal (GI) involvement in Behcet's disease (BD) is variable, with the so called "Intestinal BD" being a rare presentation of this disease. Furthermore, its association with massive lower GI bleeding is considered a rare presentation. In this case presentation, we report case of a 27-year-old male, known to have BD who presented with massive lower GI bleeding, requiring empirical subtotal colectomy. Intestinal BD has variable presentation that may require urgent surgical management. It is also important to keep it in the differential diagnosis when dealing with patients presenting with lower GI bleeding with history of BD.
Keywords: Behcet′s disease, BD, colectomy, gastrointestinal bleeding, GI, ulcer
|How to cite this article:|
Obeidat FW, Obeidat FN, Shahait AD. Intestinal Behcet's disease presenting with massive lower gastrointestinal bleeding. Arch Int Surg 2013;3:238-40
|How to cite this URL:|
Obeidat FW, Obeidat FN, Shahait AD. Intestinal Behcet's disease presenting with massive lower gastrointestinal bleeding. Arch Int Surg [serial online] 2013 [cited 2019 Aug 24];3:238-40. Available from: http://www.archintsurg.org/text.asp?2013/3/3/238/129575
| Introduction|| |
Intestinal Behcet's disease (BD) is a rare manifestation of BD, a multisystemic inflammatory disorder of unknown etiology. There is a significant variation in its incidence according to the geographical location, being more common in Japan (50%-60%), and less common in Mediterranean area (0%-5%).  Its manifestation consists mainly of aphthoid or punched out ulcers, presenting in the ileocecal region.  Furthermore, intestinal BD presenting with massive lower gastrointestinal (GI) bleeding is considered a rare phenomenon, with few cases reported in the literature. Herein, we report a case of a 27-year-old male, known to have BD, presenting with massive lower GI bleeding, requiring surgical intervention.
| Case Report|| |
A 27-year-old male known to have BD diagnosed 3 years prior to presentation and was placed on prednisone 20 mg/day presented to us with lower GI bleeding. The patient presented to our hospital complaining of rectal bleeding and black tarry stool of 2 days duration. In addition, there was associated epigastric pain and vomiting but without hematemesis. There was also generalized weakness and dizziness. His vital signs were normal. Further clinical examination was unremarkable. His hemoglobin on admission was 11.4 g/dL.
Esophagogastroduodenoscopy showed huge penetrating gastric ulcer about 3 cm in diameter, with clean base located at the greater curvature without evidence of active bleeding, biopsies were not taken due to the risk of bleeding. Moreover, a colonoscopy was tried and it reached the proximal sigmoid colon only, due to presence of black tarry stool, without evidence of active bleeding. A diagnosis of lower GI bleeding in a patient with BD was made.
Few hours later, he developed attacks of massive lower GI bleeding, requiring transferring the patient to the intensive care unit and starting blood transfusion. In spite of aggressive resuscitation, his blood pressure continued to drop, reaching 80/50 mm Hg and failed to improve despite adequate attempt at resuscitation. His pulse rate was 110 beats/min, while his hemoglobin dropped to 9.7 g/dL. Following resuscitation, an urgent exploratory laparotomy was done. Intraoperatively, there were no gross pathologies seen. In addition, the whole colon was filled with blood, with no gross evidence of blood in the small intestine. Sigmoidoscopy was preformed and was normal indicating that source of bleeding was still proximal. On the basis of these findings, we decided to proceed with empirical subtotal colectomy with ileorectal anastamosis. A huge cecal ulcer was found measuring 6 × 4 cm [Figure 1]. Intraoperatively, he received 5 units packed red blood cells, 4 units of platelets, and 5 units of fresh frozen plasma. The postoperative hemoglobin was 11.8 g/dL which stabilized around this level. The postoperative course was smooth and the patient was discharged 12 days after the operation.
|Figure 1: Huge cecal ulcer measuring 6 × 4 cm (surrounded by back arrowheads)|
Click here to view
The histopathologic examination showed multiple colonic ulcers with underlying heavy lymphocytic infiltration [Figure 2]. Some of the small submucosal blood vessels showed infiltration of their walls by lymphocytes; features consistent with lymphocytic vasculitis [Figure 3]. On follow-up, 8 months after the operation, he was doing well, with repeated upper endoscopy showing complete healing of the gastric ulcer after treatment with omeprazole, amoxicillin, and clarithromycin.
|Figure 2: Multiple ulcerations with underlying heavy lymphocytic infiltration|
Click here to view
|Figure 3: A submucosal small blood vessel showed lymphocytic infiltration of its wall; consistent with lymphocytic vasculitis|
Click here to view
| Discussion|| |
In 1940, Bechguard  described for the first time, GI involvement in BD. Although GI symptoms are common reaching 40% of patients, true involvement occurring as ulcers is rare with only 1% of all cases. , About 10% of these patients require surgical intervention at some stage.  Regarding the clinical manifestation, Wang et al., and Kasahara et al., reported that abdominal pain is the most common symptom (92%-95%) among intestinal BD patients requiring surgical intervention. , Moreover, only 17% of these patients presented with melena as our patient, while 50% of intestinal BD had complications as fistulization, perforation, and bleeding.  The most common location involved by intestinal BD is the ileocecal region.  These ulcers tend to be large, discrete, superficial with punched out edges, similar to what we found in our patient.
Surgical management is needed in some of these patients, especially those with complication. The most frequent procedure preformed is a right hemicolectomy with ileal resection, , with controversy regarding the length of normal adjacent bowel to the involved area to be resected.  In our case, small bowel was excluded from being the source of bleeding by gross examination introperatively. With normal preoperative sigmodescopy, a subtotal colectomy was considered appropriate and this was done. Based on this, many authors have recommended doing a wide resection to decrease the risk of recurrence or anastamosis leakage, occurring in 40%-87.5% at anastamosis site. ,
In this paper, we reported a case of a 27-year-old male, who was known to have BD for 3 years, presented with melena and progressed in the form of massive lower GI bleeding. This case report is a rare manifestation of BD with intestinal involvement with only few cases reported in the literature and probably the first one in Jordan. In resource poor setting like ours, surgical intervention should not be unduly delayed in patients that have massive lower GI hemorrhage.
| References|| |
|1.||Sakane T, Takeno M, Suzuki N, Inaba G. Behcet's disease. N Engl J Med 1999;341:1284-91. |
|2.||Fujita H, Kiriyama M, Kawamura T, Ii T, Takegawa S, Dohba S, et al. Massive hemorrhage in a patient with intestinal Behcet's disease: Report of a case. Surg Today 2002;32:378-82. |
|3.||Bechguard. A case of recurrent stomatitis pathos escorted to conjunctivitis of ulcers on the genitals of skin. Ugeskr Laeger 1940;102:1019-23. |
|4.||Kasahara Y, Tanaka S, Nishino M, Umemura H, Shiraha S, Kuyama T. Intestinal involvement in Behcet's disease: Review of 136 surgical cases in the Japanese literature. Dis Colon Rectum 1981;24:103-6. |
|5.||Masugi J, Matsui T, Fujimori T, Maeda S. A case of Behcet's disease with multiple longitudinal ulcers all over the colon. Am J Gastroenterol 1994;89:778-80. |
|6.||Altintas E, Senli MS, Polat A, Sezgin O. A case of Behcet's disease presenting with massive lower gastrointestinal bleeding. Turk J Gastroenterol 2009;20:57-61. |
|7.||Wang WB, Zhao YP, Cong L, Jing H, Liao Q, Zhang TP. Clinical characters of gastrointestinal lesions in intestinal Behcet's disease. Chin Med Sci J 2011;26:168-71. |
|8.||Bayraktar Y, Ozaslan E, Van Thiel DH. Gastrointestinal manifestations of Behcet's disease. J Clin Gastroenterol 2000;30:144-54. |
|9.||Sayek I, Aran O, Uzunalimoglu B, Hersek E. Intestinal Behcet's disease: Surgical experience in seven cases. Hepatogastroenterology 1991;38:81-3. |
|10.||Lee KS, Kim SJ, Lee BC, Yoon DS, Lee WJ, Chi HS. Surgical treatment of intestinal Behcet's disease. Yonsei Med J 1997;38:455-60. |
[Figure 1], [Figure 2], [Figure 3]