Gastric and Duodenal Fistulas in Crohn’s Disease, a Surgical Challenge: Report of 5 Cases and a Review of the Literature

Background

Fistulas are a common complication of Crohn’s disease (CD), but a fistula with involvement of the stomach and duodenum is a rare condition affecting approximately 0.3–5% of CD patients [1]. This condition usually results from inflammation in other sites of the gastrointestinal tract. Management of these cases is complex and involves preoperative optimization and surgical treatment; however, there is controversy regarding the optimal surgical approach, which may include primary duodenal closure or more complex procedures such as duodenojejunostomy [2–5]. Postoperative maintenance therapy with anti-TNF agents is important to prevent recurrence, a topic that is not well discussed in published studies about this specific condition. Such controversies are justified by actual evidence that is based on case reports and case series. At the same time, larger studies clarifying the optimum pre-operative preparation, the ideal surgical procedure, and long-term maintenance therapy and outcomes are lacking. Here, we reviewed 855 medical records of CD patients treated in our service and found 4 cases of duodenal fistula and 1 case of gastric fistula. We describe each case and discuss aspects of management of this condition.

Case Reports

CASE 1:

A 70-year-old man with a 4-year history of CD presented with abdominal pain, nausea, diarrhea, and weight loss. Computed tomography (CT) enterography demonstrated thickening of the ileal wall, a transverse-ileal fistula, and an ileum-duodenal fistula (Figure 1). He underwent typhlectomy with primary ileocolic anastomosis and primary duodenal closure in 2 layers of transversal suture associated with an omental patch.

CASE 2:

A 65-year-old man who had been followed for CD for 18 years presented with abdominal pain, fever, and diarrhea. CT demonstrated pancolitis, and colonoscopy revealed stenosis of the sigmoid colon. He underwent an exploratory laparotomy with additional findings of fistula involving the transverse colon and third duodenal portion. A subtotal colectomy was performed, and a transverse section of the duodenum was covered with an omental patch using a linear stapler.

CASE 3:

A 33-year-old man had been followed for CD for 18 years. He had a past medical history of typhlectomy 4 years previously, and presented with abdominal pain, weight loss, and cutaneous fistulas. CT revealed an ileocolic-duodenal fistula and fistulous paths to the right psoas and right inferior quadrant skin. Upper digestive endoscopy (UDE) demonstrated a suspected duodenal fistula (Figure 2). He underwent ileocolonic anastomosis resection with terminal ileostomy and mucosal fistula of the transverse colon along with resection of cutaneous fistulas and primary duodenal closure, which was covered with an omental patch.

CASE 4:

A 47-year-old man, with an 8-year history of CD and a medical history of right ileocolectomy 5 years previously, presented with abdominal pain, fecaloid vomiting, and weight loss. CT demonstrated thickening of the ileotransverse anastomosis and fistula to the second duodenal portion. He underwent anastomosis resection, duodenal closure, and an omental patch. A presentation of postoperative duodenal dehiscence was treated surgically with duodenojejunostomy.

CASE 5:

A 47-year-old woman with CD that had been monitored for 5 years presented to our service with diarrhea, thin stools, weight loss, and fecaloid vomiting. CT demonstrated strictures involving the transverse and descending colon and a cologastric fistula involving the splenic flexure (Figure 3). UDE showed a cologastric fistula, and a nasoenteric catheter was placed. She underwent extended left colectomy and primary reconstruction as well as gastric fistula debridement with primary closure in 2 layers.

All patients had an impaired nutritional status and all of them underwent preoperative optimization with nutritional therapy and antibiotics. All patients received postoperative maintenance therapy with anti-TNF agents to prevent recurrence and were followed up closely with early endoscopic surveillance in the first year after resection. Except for the complications related to Case 4, there were no other complications in the short- or long-term followup. Details regarding each case are presented in Table 1.

Discussion

Gastric and duodenal fistulas are rare complications of CD and usually result from disease activity in the ileum, ascending and transverse colon, or ileocolic anastomosis. Symptoms are usually nonspecific, such as diarrhea and abdominal pain, but more specific symptoms can occur, including fecaloid vomiting and intestinal malabsorption, when long bowel segments are bypassed. Fistulas originating from intrinsic duodenal CD are extremely rare [2,3,5]. Klein described a prevalence of 0.5% of duodenal fistulas in CD patients [6]. Michelassi reported that 5% of 290 CD fistulas presented with gastroduodenal involvement [4]. Greenstein described gastric fistulas affecting 0.8% of patients with ileocolitis and 0.6% of patients with colitis [7]. In our study, a fistulizing pathway affecting the stomach and duodenum occurred in 0.58% of patients with CD, similar to other studies. The simultaneous infection of the stomach with Helicobacter pylori was not investigated in this study.

Wilk suggested that CD patients with previous ileocolic resection are at a higher risk for gastric and duodenal fistulas, and some authors argue that the omentum should be placed between the anastomosis and duodenum in ileocolectomy for CD to avoid this complication [1,5]. Gong found an increased rate of recurrence of coloduodenal fistula in patients whose fistula originated from a previous anastomosis [8].

Gastric and duodenal fistulas are not an absolute indication for surgery. A recent prospective cohort study showed that biological agents can effectively bring closure in a quarter of patients with internal fistula tracts [9].

The use of infliximab has been reported, with good results in cases of refractory duodenal ulcers, duodenal stenosis, and duodenal fistulas [10,11]. In a prospective study, Annunziata et al [12] demonstrated that among 19 patients with gastroduodenal CD, 72.7% of the patients treated with infliximab or adalimumab achieved mucosal healing 12 weeks after the initiation of treatment. Other reports [13,14] have also supported the efficacy of adalimumab treatment in closing internal fistulas.

The efficacy of ustekinumab has also been demonstrated, but evidence supporting its efficacy for enterocutaneous fistula is sparse. However, we did find 2 reports of upper enterocutaneous fistula closures that were successfully treated with ustekinumab [15,16].

Surgery is the most common treatment if biological agents are not as efficient as they are for perianal fistulas [17–19]. Resection of the involved segment and closure of duodenal defects are the basis for surgical treatment [2–4,20]. Michelassi et al [4] and Lee and Schraut [20] reported complication rates of 33.3% and 18.2% and mortality rates of 0% and 9.1% for surgical treatment of duodenal fistulas, respectively. Before a patient undergoes a laparotomy, it is very important to assess the patient’s general condition and nutritional status, as was performed in our cases, to achieve adequate preoperative optimization. This includes nutritional therapy with enteral and/or parenteral nutrition. Preoperative enteral nutrition has been shown, in a guideline review, to reduce the rate of postoperative complications in malnourished CD patients undergoing surgery [21].

When present, control of abdominal septic conditions with antibiotics and percutaneous drainage should be performed if needed, and surgical treatment should be postponed until better conditions are achieved. Preoperative abscess and low albumin levels are independent risk factors for intra-abdominal septic complications, as shown by a recent meta-analysis [22], and improvement of nutritional status and reduction of inflammation, marked by improvement of serum albumin and CPR levels, have been associated with fewer postoperative complications [23,24]. Specifically, regarding coloduodenal fistulas, Gong reported that preoperative optimization decreases major postoperative and intra-abdominal infectious complications, and also lowers the rates of temporary fecal diversion [8]. In these cases, no patient had pancreatic involvement. Two patients had increased amylase, but without symptoms or clinical diagnosis of pancreatitis.

Controversy exists about the ideal surgical procedure for duodenal closure, and some authors suggest that duodenal defects could be treated successfully in most cases with primary transverse closure followed by an omental patch when feasible [3,5,8,20] (Figure 4). Complication rates range from 18.2% for morbidity and 9.1% for mortality in an older series, as described by Lee [20]. A morbidity of 28–50% and zero mortality were reported in a recent series by Gong [8]. This is consistent with our findings. For larger defects (larger than 3 cm), and in older studies, some authors have reported good results with duodenojejunostomy (Figure 4) since there is no extensive jejunal disease, as seen in the reoperation in Case 4. Case 4 represents the first reported case in the literature using this technique as a bail-out strategy. A serosal patch with the jejunum is also an alternative [1,2,20]. The most recent and largest series reporting coloduodenal fistulas in CD was published by Gong and showed that an even larger defect could be treated with primary closure, and the duodenal defect size was not associated with a higher incidence of fistulas or stenosis. However, larger series are needed to confirm these findings [8]. Temporary duodenal decompression with a nasoduodenal catheter is an important strategy in postoperative care, and a double lumen enteral catheter can be used for simultaneous decompression and feeding [5,18,25].

Because of the rarity of duodenocolic fistula cases, the reported data in the literature with CD are limited [26].

Postoperative recurrence should be a concern after ileocolonic intestinal resection, and prophylactic treatment is recommended for patients with one or more risk factors, which include smoking, prior intestinal surgery, granulomas, myenteric plexitis in the resection specimen, and penetrating disease at the index surgery. All of our patients had at least one risk factor and were at high risk of recurrence. Thiopurines and anti-TNF agents are the drugs of choice for this purpose, and high-dose mesalazine and metronidazole can also be used. However, anti-TNF agents seem to be the most effective. At this point in time, no fixed strategy is recommended, and therapy decisions should be individualized [27,28]. Due to the lack of details and discussion regarding postoperative recurrence prophylaxis in the published reports of gastric and duodenal fistula in CD, all of our patients started receiving anti-TNF agents 3 to 6 weeks after surgery. Regardless of the prophylactic therapy indicated, close followup with clinical evaluation, biomarkers, and early endoscopic surveillance with ileocolonoscopy in the first year after surgery are essential to early recurrence detection and successful management [27–29].

A multidisciplinary approach in the management of patients with inflammatory bowel disease is essential, especially in these surgical situations, in which gastroenterologists, nutritionists, and surgeons must work together to seek the best therapy for patients with such a complex disease.

The authors clarify that our outpatient care center for patients with inflammatory bowel diseases is located at a public hospital in a city of 14 million inhabitants. Patients who are in consultation are referred by the national public health system. Patients who arrive for followup of the disease, in most cases, already present with a severe phenotype of the disease, sometimes without prior correct diagnosis and without adequate treatment. These patients also require time to be hospitalized and undergo tests.

Conclusions

Despite being a rare and complex complication of CD, gastric and duodenal fistula can be treated successfully with preoperative optimization, resection of the compromised intestinal segment followed by duodenal or gastric closure and covering by an omental patch, and adequate postoperative follow-up with endoscopic surveillance and prophylactic treatment to prevent postoperative recurrence.