A case of giant ileal duplication in an adult, successfully treated with laparoscope-assisted surgery
© Matsumoto; licensee Springer. 2015
Received: 4 November 2014
Accepted: 9 January 2015
Published: 30 January 2015
Alimentary tract duplication is a rare congenital malformation but can occur anywhere along the digestive tract. Most patients become symptomatic in early childhood, and only a few cases of adult patients have been reported in the literature. We herein report a unique case of a giant ileal duplication in an adult, which was successfully treated with laparoscope-assisted surgery. A 60-year-old male was admitted because of abdominal pain. Imaging studies revealed a well-defined cystic mass, measuring 15 cm, in the ileocecal region. We diagnosed it as a duplicated ileum and performed laparoscope-assisted surgery. The duplication was successfully resected with attached normal ileum, and there were no major complications in the postoperative course.
Duplication of the alimentary tract is a rare congenital malformation that can arise throughout the alimentary tract from the oral cavity to the anus . Most patients are diagnosed in their infancy or childhood. More than 80% of cases present before they are 2 years old as an acute abdomen or bowel obstruction, while a minority may remain asymptomatic until adulthood . We herein report a rare case of a giant ileal duplication in an adult, which was successfully treated with laparoscope-assisted surgery.
A 60-year-old male was admitted to our hospital with a complaint of right lower abdominal pain. The patient did not have same symptoms before, and he only had a medical history of asthma. The laboratory data on admission showed a slightly elevated WBC count of 9,300/μl and a CRP level of 1.0 mg/dl, but the tumor marker levels (CEA, CA19-9) were almost within the normal limits. Abdominal computed tomography (CT) demonstrated a 15-cm large cystic mass, like a dilated intestine, and fluid collection was observed inside.
Based on these findings, we diagnosed the cyst as an ileal duplication and decided to perform surgery in order to prevent a recurrence of abdominal pain and lethal complications, such as bleeding or perforation.
The postoperative period was uneventful without any asthma attack, and the patient was discharged on the 13th postoperative day.
Duplication of the alimentary tract is rare congenital anomaly observed in one out of 25,000 deliveries . These anomalies are usually present in childhood and occasionally in adults, and can be encountered anywhere throughout the gastrointestinal tract, from the mouth to the anus. The terminal ileum is thought to be the most frequently involved .
Although symptoms such as vomiting, constipation, abdominal pain, obstruction, an abdominal mass, and hemorrhage have been reported, duplication can be easily misdiagnosed as Meckel’s diverticulum, appendicitis, a choledochal cyst, or Crohn’s disease, especially when it occurs in adults. Prior to surgery, it is difficult to diagnose alimentary duplication because of the non-specificity of symptoms and the presentation . Indeed, only 11.2% of cases have been correctly diagnosed before surgery in Japan . Recently, the usefulness of capsule endoscopy  or double balloon endoscopy  for the detection of the condition has been reported, but the disease still cannot be detected in patients who have no communication to the normal intestine.
Surgical treatment is advocated after a diagnosis of ileal duplication in order to prevent potentially lethal complications such as perforation, volvulus, intussusception, bowel obstruction, and enteric bleeding. Heterotopic mucosa of gastric or pancreatic origin are sometimes seen, with a frequency of 17%–36% for gastric mucosa , and these are thought to result in perforation or bleeding [10,11]. The duplication itself is also thought to have malignant potential [5,12,13]. In our case, the specimen had a similar physiological architecture to the small bowel, with indicated villi, crypts, and a two-layered muscular wall. The tissue of gastric or pancreatic origin was not observed.
Recently, some reports have suggested that laparoscope-assisted surgery could be useful for making a diagnosis and treating this condition [14,15]. Sixteen cases of alimentary tract duplication in adults, treated with laparoscopic surgery, have been reported in the Japanese literature [16-28]. Only four cases were diagnosed before surgery, but an accurate intraoperative diagnosis could be made in the rest of the cases, and accurate resection was performed. Our case had the largest cyst in the reported cases.
In conclusion, it is difficult to diagnose alimentary duplications, and laparoscope-assisted surgery is thought to be beneficial for both making a diagnosis and performing accurate treatment.
Written informed consent was obtained from the patient for publication of this case report and any accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal.
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