Published online Feb 27, 2024. doi: 10.4240/wjgs.v16.i2.429
Peer-review started: October 17, 2023
First decision: November 1, 2023
Revised: November 29, 2024
Accepted: January 15, 2024
Article in press: January 15, 2024
Published online: February 27, 2024
Processing time: 131 Days and 8.7 Hours
Behcet’s disease (BD), a chronic vasculitic disorder affecting multiple organs, is characterized by recurrent oral and genital ulcers, arthritis, vasculitis, and intes
To evaluate the postoperative clinical course of intestinal BD and determine factors associated with its recurrence.
Data from patients who underwent surgical treatment for intestinal BD between January 2010 and August 2021 were retrospectively reviewed. Patients’ demo
We analyzed 39 surgeries in 31 patients. The mean patient age was 45.1 years, and the mean interval between the diagnosis of intestinal BD and surgical treatment was 4.9 years (range 1.0-8.0 years). The most common indication for surgery was medical intractability (n = 16, 41.0%), followed by fistula or abscess (n = 11, 28.2%). Laparoscopic approaches were used in 19 patients (48.7%), and 5 patients (12.8%) underwent emergency surgeries. The most common surgical procedure was ileocecal resection (n = 18, 46.2%), followed by right colectomy (n = 11, 28.2%). A diverting stoma was created in only one patient (2.6%). During a mean follow-up period of 45 (range 8-72) months, eight cases (20.5%) of recurrence in five patients required reoperation. The interval between operations was 12.1 months (range 6.3-17.8 mo). Four patients (10.3%) experienced recurrence within 1 year postoperatively, and all eight recurrences occurred within 2 years of the initial surgery. The reoperation rates at 1 and 3 years were 10.3% and 20.5%, respectively. A redo ileocolic anastomosis was performed in all recurrent cases. In multivariate Cox regression analysis, emergency surgery [hazard ratio (HR) 9.357, 95% confidence interval (CI): 1.608-54.453, P = 0.013] and elevated C-reactive protein (CRP) levels (HR 1.154, 95%CI: 1.002–1.328, P = 0.047), but not medication use, were predictors of recurrence.
Surgical resection is a feasible treatment option for complicated BD. Reoperation is associated with severe inflammatory conditions, reflected by increased CRP levels and the requirement for emergency surgery.
Core Tip: Behcet's disease (BD), a chronic vasculitic disorder impacting multiple organs, often necessitates surgical intervention due to medical intractability, fistulas, or abscesses. This study examined 31 patients who underwent surgery for intestinal BD. Recurrence occurred in 20.5% of cases, with all instances happening within two years after surgery. Emergency surgery and elevated C-reactive protein (CRP) levels were predictive of recurrence. This suggests that surgical resection is a viable option for complicated BD, but reoperation is more likely in patients with severe inflammation as indicated by elevated CRP levels and the requirement for emergency surgery.
- Citation: Park MY, Yoon YS, Park JH, Lee JL, Yu CS. Short- and long-term outcomes of surgical treatment in patients with intestinal Behcet’s disease. World J Gastrointest Surg 2024; 16(2): 429-437
- URL: https://www.wjgnet.com/1948-9366/full/v16/i2/429.htm
- DOI: https://dx.doi.org/10.4240/wjgs.v16.i2.429
Behcet’s disease (BD) is a chronic multisystemic vasculitic disorder affecting the arteries and veins[1,2]. It manifests as recurrent oral and genital ulcers, skin and ocular lesions, arthritis, vasculitis, neurologic lesions, or intestinal ulcers[1,2]. The prevalence of BD is higher in East Asia and the Mediterranean than in the United Kingdom and North America[3,4].
Intestinal BD is a subtype of BD predominantly characterized by gastrointestinal symptoms, such as abdominal pain, hematochezia, diarrhea, abdominal masses, and intestinal ulcerations[5]. The incidence of gastrointestinal involvement varies across different countries, ranging from 0% to 60%[6,7]. Intestinal BD is more frequent in East Asia (including Korea and Japan) than in Mediterranean countries[6,7]. However, the appropriate treatments for this disorder are yet to be established. Intestinal BD is primarily treated empirically due to the scarcity of well-designed studies on this topic, the rarity of the disease, and the heterogeneity of disease patterns[8]. Medical treatment of BD entails using corticosteroids, 5-aminosalicylic acid, sulfasalazine, azathioprine, and 6-mercaptopurine. However, intestinal BD often requires surgical treatment because of poor response to medical treatment and high frequency of complications, such as intestinal perforation, fistula, or bleeding[9,10]. Some patients with intestinal BD experience frequent recurrences despite surgical treatment and require repeated surgeries[11]. Although surgical treatment is performed in many patients with intestinal BD, its clinical efficacy and long-term outcomes still need to be fully understood.
In this study, we aimed to evaluate the postoperative clinical course of intestinal BD and determine factors associated with reoperations due to recurrences.
We conducted a retrospective review of data from patients with intestinal BD who underwent surgical resection between January 2010 and August 2021 at Asan Medical Center, Seoul, South Korea. Intestinal BD was diagnosed by gastroenterologists based on colonoscopic criteria and clinical manifestations through a modified Delphi process[5]. Patients were classified as having definite, probable, or suspected intestinal BD. Patients with any evidence of other gastrointestinal diseases, such as Crohn’s disease, intestinal tuberculosis, or ischemic enteritis, during the follow-up period were excluded. Patient demographics and clinical characteristics, including age, sex, BD symptoms, medical treatments, laboratory findings, and indications for surgery, were compared. Collected surgical data included surgery type (open vs laparoscopy), surgical procedure, number of emergency surgeries, number of cases with a diverting stoma, expected blood loss, and duration of the surgery. The postoperative course was evaluated based on postoperative complications, mortality within 30 days of surgery, and reoperation related to the recurrence of intestinal BD symptoms. The study protocol was approved by the institutional review board of Asan Medical Center (approval No. 2022-0238).
Recurrence was defined as a relapse of intestinal BD symptoms, reappearance of ulcers at the anastomosis site, and newly developed fistula or abscess near the anastomosis associated with the reappearance of ulcers. If there were only other BD symptoms such as oral ulcer and genital ulcer other than the intestinal BD symptoms, they were excluded from recurrence. Reoperation was defined as a surgical intervention related to intestinal BD during the follow-up period after the initial surgery.
The primary outcome of this study was the recurrence rate of intestinal BD after surgical treatment. The secondary outcomes were factors associated with recurrence after the initial surgery.
Categorical variables are expressed as numbers and percentages and were compared using the chi-square test. Continuous variables are expressed as medians with interquartile ranges (IQRs) or mean ± SD values and were compared using the Student’s t-test. Cumulative recurrence rates were calculated using the Kaplan-Meier method, and factors associated with recurrence were compared using log-rank tests. All statistical analyses were performed using SPSS for Windows (version 25.0; SPSS Inc., Chicago, IL, United States), with P value of < 0.05 considered statistically significant.
A total of 39 surgeries in 31 patients with intestinal BD were performed between January 2010 and August 2021. The demographics and clinical characteristics of the patients are summarized in Table 1. The median age at the time of diagnosis of BD was 36 (IQR 29-49) years. The median age at the time of diagnosis of intestinal BD was 38 (IQR 30-49) years, and the median age at the time of initial surgery was 49 (IQR 31-57) years. The median interval between intestinal BD diagnosis and surgical treatment was 3 (IQR 1-8) years. The proportions of male and female patients were equivalent (51.3% vs 48.7%, respectively). The most common symptom of systemic BD was oral ulcers (35.9%), and the most common indication for surgical treatment of intestinal BD was medical intractability and the development of perforation or fistula (41.0% each). Patients who underwent surgery for intestinal BD had mildly elevated preoperative C-reactive protein (CRP) levels and erythrocyte sedimentation rates.
Cases (n = 39) | |
Age (yr) at the time of diagnosis of BD, median (IQR) | 36 (29-49) |
Age (yr) at the time of diagnosis of intestinal BD, median (IQR) | 38 (30-49) |
Age (yr) at the time of surgery, median (IQR) | 49 (31-57) |
Interval (yr) between intestinal BD diagnosis and surgical treatment, median (IQR) | 3 (1-8) |
Sex, n (%) | |
Male | 20 (51.3) |
Female | 19 (48.7) |
Symptoms and signs of BD, n (%) | |
Oral ulcer | 14 (35.9) |
Genital ulcer | 7 (18.0) |
Ocular lesion | 1 (2.6) |
Skin lesion | 0 (0.0) |
Arthritis | 0 (0.0) |
Vascular lesion | 1 (2.6) |
Neurologic lesion | 0 (0.0) |
Location of ulceration, n (%) | |
Ileocecal | 35 (89.7) |
Ascending colon | 3 (7.7) |
Rectum | 1 (2.6) |
Medications, n (%) | |
Steroids | 28 (71.8) |
Colchicine | 31 (79.5) |
5-ASA or sulfasalazine | 27 (69.2) |
Azathioprine or 6-MP | 23 (59.0) |
TNF-α inhibitor | 15 (38.5) |
Antibiotics | 13 (33.3) |
Indication for operation, n (%) | |
Medical intractability | 16 (41.0) |
Perforation or fistula | 16 (41.0) |
Stricture | 4 (10.3) |
Bleeding | 3 (7.7) |
Preoperative CRP (mg/L), median (IQR) | 3.9 (0.92-6.41) |
Preoperative ESR (mm/h), median (IQR) | 30.0 (15.0-49.5) |
Preoperative neutrophil (%), median (IQR) | 66.1 (59.0-73.8) |
Preoperative lymphocyte (%), median (IQR) | 18.1 (11.8-27.2) |
Preoperative procalcitonin, median (IQR) | 0.0 (0.0-0.1) |
Preoperative albumin (g/dL), median (IQR) | 3.0 (2.6-3.3) |
The evaluated surgical data are presented in Table 2. Emergency surgeries were performed in five patients. The laparoscopic and open surgery rates were similar (51.3% vs 48.7%, respectively). The most common surgical procedure was ileocecal resection (48.7%), followed by right hemicolectomy (28.2%). A diverting stoma was created in only one case. The median duration of surgery was 116 (IQR 95-740) min.
Cases (n = 39) | |
Emergency surgery, n (%) | 5 (12.8) |
Surgery type, n (%) | |
Open surgery | 20 (51.3) |
Laparoscopy | 19 (48.7) |
Surgical procedure, n (%) | |
SB segmental resection | 3 (7.7) |
Ileocecal resection | 19 (48.7) |
Right hemicolectomy | 11 (28.2) |
Other | 6 (15.4) |
Diverting stoma, n (%) | 1 (2.6) |
Expected blood loss, n (%) | |
Minimal | 14 (35.9) |
10-100 mL | 16 (41.0) |
> 100 mL | 1 (2.6) |
NA | 8 (20.5) |
Duration of surgery (min), median (IQR) | 116 (95-740) |
Data on the postoperative course are presented in Table 3. Five patients experienced post-operative complications, and no case of mortality was recorded. Eight (20.5%) cases and five patients required reoperation due to recurrence, all of which were performed within 2 years after the previous surgery. In five (12.8%) of these eight cases, reoperation was performed within 1 year after the previous surgery (Figure 1A). The cumulative recurrence rates after the initial surgery are 7.7% in 1 year, 12.8% in 2 years, 17.9% in 3 years and 20.5% in 4 years (Figure 1B). The most common causes of recurrence were medical intractability and the presence of perforation or fistula. A diverting stoma was created in one of the eight patients, and a redo anastomosis was performed in the other seven patients. The median interval between the previous surgery and reoperation due to recurrence was 12 (IQR 6.25-17.75) months.
Cases (n = 39) | |
Postoperative complications, n (%) | |
Anastomosis leakage | 1 (2.6) |
Intra-abdominal abscess | 0 (0.0) |
Fistula | 0 (0.0) |
Wound infection | 3 (7.7) |
Bleeding | 1 (2.6) |
Ileus | 0 (0.0) |
Mortality, n (%) | 0 (0.0) |
Clavien–Dindo classification, n (%) | |
1 | 2 (5.2) |
2 | 0 (0.0) |
3 | 3 (7.7) |
4 | 0 (0.0) |
Cause of reoperation, n (%) | |
Medical intractability | 4 (50.0) |
Perforation or fistula | 4 (50.0) |
Stricture | 0 (0.0) |
Bleeding | 0 (0.0) |
Reoperation procedure, n (%) | |
SB segmental resection | 0 (0.0) |
Redo anastomosis | 7 (87.5) |
Diverting stoma | 1 (12.5) |
Follow-up period (mo), median (IQR) | 42.0 (8.0-72.0) |
Time to recurrence (mo), median (IQR) | 12.0 (6.25-17.75) |
Univariate analyses demonstrated that elevated CRP levels and emergency surgeries were significantly associated with the recurrence of intestinal BD. In multivariate analysis, emergency surgery remained significantly associated with the recurrence of intestinal BD (Table 4).
Univariate analysis | Multivariate analysis | |||
HR (95%CI) | P value | HR (95%CI) | P value | |
Sex | 0.358 (0.084-1.518) | 0.163 | 2.372 (0.370-15.186) | 0.362 |
Age at the time of surgery | 0.979 (0.923-1.039) | 0.487 | ||
Use of steroid | 1.922 (0.386-9.566) | 0.425 | ||
Use of colchicine | 1.901 (0.232-15.552) | 0.549 | ||
Use of azathioprine or 6-MP | 0.919 (0.219-3.848) | 0.908 | ||
Use of 5-ASA or sulfasalazine | 1.195 (0.241-5.930) | 0.828 | ||
Use of TNF-α inhibitor | 1.485 (0.371-5.945) | 0.577 | ||
Use of antibiotics | 0.279 (0.034-2.267) | 0.232 | ||
Preoperative CRP level | 1.129 (1.031-1.236) | 0.009 | 0.988 (0.868-1.125) | 0.855 |
Preoperative ESR | 1.001 (0.978-1.025) | 0.925 | ||
Preoperative neutrophil | 0.973 (0.080-1.172) | 0.774 | ||
Preoperative lymphocyte | 0.898 (0.650-1.240) | 0.514 | ||
Preoperative procalcitonin | 28.659 (0.290-2832.482) | 0.152 | ||
Preoperative albumin level | 0.336 (0.081-1.394) | 0.133 | 0.220 (0.028-1.739) | 0.151 |
Emergency surgery | 12.216 (2.669-55.910) | 0.001 | 10.746 (1.486-77.686) | 0.019 |
Open surgery | 1.423 (0.340-5.961) | 0.629 | ||
Postoperative complication | 2.051 (0.248-16.962) | 0.505 |
Intestinal BD is characterized by deep ulcers, most commonly found in the ileocecal area or terminal ileum[12]. Many patients with intestinal BD require emergency surgeries because deep ulcers tend to penetrate the intestinal wall[13]. However, only a few studies have explored the long-term clinical outcomes and related prognostic factors in surgical patients with intestinal BD. Therefore, in this study, we aimed to investigate the long-term outcomes of surgical treatment for intestinal BD and identify the predictive factors for recurrence and reoperation.
In this study, 8 of the 39 surgical cases (20.5%) of intestinal BD required reoperation due to recurrence during the follow-up period. The complicated recurrences that required reoperation were confirmed through CT, endoscopy, and physical examination. When no findings other than BD-related complications observed as a result of physical exami
A key finding of the present study is that the timing of surgery for intestinal BD may be an important prognostic factor for reoperation. In this study, patients underwent surgery approximately 3 years after being diagnosed with intestinal BD. Furthermore, > 80.0% of patients underwent surgery after the disease had become refractory to medical treatment or after complications, such as perforation, fistula, or obstruction had developed. As a result, > 12.0% of the patients required emergency surgery, which was identified as a significant prognostic factor in our multivariate analysis. A previous study reported that patients with intestinal BD who underwent surgery earlier exhibit better prognoses in terms of recurrence risk and reoperation rate than those of patients who underwent surgical treatment at a later stage[17]. In addition, the higher preoperative CRP level was significant with higher reoperation risk. Therefore, it can be expected that the more severe systemic inflammation which cause an increasing in CRP level before operation, the more affected the reoperation. In approximately 90% (35/39) of cases, intra-venous antibiotics were administered postoperatively. Most of the antibiotics administered were metronidazole and ciprofloxacin, and carbapenem was also administered. Since most of the patients were administered similar antibiotics, antibiotics would not have had significant effects on changes of patients’ inflammatory markers and risk of recurrence.
This study had several limitations. First, although our study included patients who were followed up for a sufficient period, the inclusion of more patients and surgeries is needed to make a definitive conclusion. Second, a selection bias might have been present because of the retrospective study design.
Surgical resection is a feasible treatment option for complicated BD, although this condition is associated with poor clinical courses and high reoperation rates. Furthermore, reoperation is associated with severe inflammatory conditions, as reflected by the requirement for emergency surgery or elevated CRP levels at the time of surgery. Therefore, a timely surgical treatment is essential to reduce the reoperation rate.
Behcet's disease (BD) is a chronic vasculitic disorder that impacts various organs, presenting with recurring oral and genital ulcers, arthritis, vasculitis, and intestinal ulcers. Despite the frequent occurrence of intestinal complications in BD, the effectiveness and long-term results of surgical interventions for intestinal BD are yet to be definitively established.
Despite the frequent occurrence of intestinal complications in BD, the effectiveness and long-term results of surgical interventions for intestinal BD are yet to be definitively established.
To assess the postoperative clinical outcomes of intestinal BD and identify factors associated with its recurrence.
A retrospective review of patients with intestinal Behcet's disease undergoing surgical resection at Asan Medical Center in Seoul, South Korea, between January 2010 and August 2021 was conducted. The study focused on patient demo
In a study involving 31 patients who underwent 39 surgeries for intestinal Behcet's disease, the mean patient age was 45.1 years, with a mean interval of 4.9 years between the diagnosis and surgical treatment. The primary indications for surgery were medical intractability (41.0%) and fistula or abscess (28.2%). Laparoscopic approaches were used in 48.7% of cases, and eight recurrences (20.5%) requiring reoperation were observed during a mean follow-up of 45 months, with a recurrence rate of 10.3% at 1 year and 20.5% at 3 years. Emergency surgery and elevated C-reactive protein levels were identified as predictors of recurrence in multivariate analysis.
Surgical resection is a viable treatment for complicated BD, despite its association with challenging clinical courses and elevated reoperation rates, emphasizing the importance of timely surgical intervention to mitigate reoperation risk, particularly in the presence of severe inflammatory conditions.
It is necessary to identify factors, including specific biomarkers, that may influence recurrence through a larger number of patients with intestinal BD and make efforts to reduce recurrence rates using this information.
Provenance and peer review: Invited article; Externally peer reviewed.
Peer-review model: Single blind
Specialty type: Surgery
Country/Territory of origin: South Korea
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P-Reviewer: Liang W, China; Xu JD, China S-Editor: Yan JP L-Editor: A P-Editor: Zheng XM
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