Cancer of the Small Intestine in Patients with Crohn's Disease

Case Reports

Case 1 involved a 36-year-old female. She was diagnosed with Crohn's disease at 23 years of age. She suffered from diabetes as a complication, and her family history included a younger sister who also suffered from Crohn's disease. At 25 years of age, she experienced ileus due to stricture, for which she underwent surgery. Because she was diabetic, intestinal resection was avoided, while strictureplasty and bypass were performed. The patient demonstrated good progress after the surgery and visited the hospital once a year. Eleven years after the first operation, she began to suffer from nausea and abdominal pain. A computed tomography (CT) scan was performed, which led to a diagnosis of hypertrophy of the small intestinal wall (Figure 1A). A small bowel series of X-rays was performed, which resulted in a diagnosis of expansion of the oral side and the strictures that frequently occur in the small intestine inside the pelvis (Figure 2A). Treatment administered via an ileus tube was performed for two weeks. The patient's subjective symptoms, such as nausea and abdominal pain, improved; however, no improvement was observed in the expansion of the small intestine on an X-ray examination. Therefore, a second operation was performed. The findings of laparotomy revealed advanced adhesion between the intestines, as well as the presence of nodules in Douglas' pouch of the peritoneum. Since these findings were quite different from those observed during regular laparotomy in patients with Crohn's disease, an intraoperative pathological diagnosis was made by evaluating pieces of the intestinal resection tissue and nodules. The results showed well-to-moderately differentiated adenocarcinoma. Measurement of the levels of tumor markers during surgery revealed a clearly high carbohydrate antigen 19-9 (CA19-9) level of 962 U/ml (Table I). The final pathological diagnosis indicated the presence of cancerous lesions in two locations: in a region that was excluded during the bypass procedure and in the stricture on the oral side of the bypass (Figure 3A and B). After the operation, treatment with FOLFOX (levofolinate, fluouracil, oxaliplatin) and bevacizumab was commenced. Following the initiation of chemotherapy, the CA19-9 level returned to normal for a short period. However, in the sixth month after the operation, a CT scan revealed a pelvic tumor, thought to be a site of recurrence, that coincided with an increased in the CA19-9 level (Table I). Chemotherapy was subsequently administered as treatment for recurring large intestine cancer; however, an increase in the number of pelvic tumors could not be suppressed, resulting in the patient's death one year and six months after the operation.

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Figure 1.

A computed tomography (CT) image obtained before surgery in case 1 (A), and case 2 (B). Both images show hypertrophy of the small intestinal wall near the lesion (arrow).

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Figure 2.

Results of the small bowel X-ray series in case 1(A), and case 2(B). Both patients display multiple strictures (arrow). It is difficult to distinguish Crohn's disease from the cancer.

The second patient was a 45-year-old male. The disease presented as abdominal pain and melena at 22 years of age; therefore, he had been suffering from the disease for 23 years. At 23 years of age, the patient underwent appendectomy due to appendicitis, and at that time was diagnosed with Crohn's disease. At 25 years of age, ileocecal resection was performed due to stenosis of the ileum. The patient's progress after the operation was favorable, and 13 years passed without any treatment. At 38 years of age, the patient developed an aching sensation in the right side of the abdomen. For the next seven years, he received medical therapy, primarily centered on prednisolone. Since ileus had also appeared at that time, the patient was admitted to this hospital. The CT findings were similar those of case 1, which lead to a diagnosis of hypertrophy of the small intestinal wall (Figure 1B). A small bowel series of X-rays revealed an anastomotic site in the region of the previous surgery with multiple areas of stenosis on the oral side (Figure 2B). A fistula was also found near the anastomotic region, and the intestinal tract on the oral side had expanded. During colonoscopy, we were unable to insert the scope because the stenosis on the oral side was too severe. For this reason, we determined that the areas of stenosis were due to recurrence of Crohn's disease in the anastomotic region, and we performed surgery. The area around the anastomotic region was very hard, and nodules were found in the peritoneum. It was determined that the patient's symptoms were caused by advanced cancer; therefore, an intraoperative pathological diagnosis was made. The results indicated the presence of well-differentiated adenocarcinoma. We performed right hemicolectomy including the area of stenosis. The final pathological diagnosis was a cancerous lesion at the stricture on the oral side of the anastomosis formed during the previous operation (Figure 3C). After surgery, we administered chemotherapy with FOLFOX and bevacizumab. To date, there has been no obvious recurrence on imaging examinations.

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Figure 3.

Resected specimens in case 1. Cancer (arrows) was found in the region excluded during the previous bypass operation (A) and the stricture lesion (B) on the oral side of the bypass. Findings of the resected specimen in case 2 (C). Cancer (arrow) was found in the lesion in the stricture on the oral side of the anastomosis (triangle) created during the previous operation.