'Constipation' Lands Patient in Surgery for Unusual Anatomy

— What presented as a partial bowel obstruction was something much more rare

In an "exceptionally" rare case, an infant with abdominal distention, vomiting, and constipation was found to have a second colon, researchers reported.

The child presented to the emergency department but was transferred to a specialist pediatric surgical center, where imaging suggested total duplication of the colon and rectum, Lynn Sinitsky, MBBS, of the West Hertfordshire Teaching Hospitals NHS Trust in Watford in the U.K., and colleagues reported in BMJ Case Reportsopens in a new tab or window.

"I think our key learning was that he had had multiple interactions with healthcare professionals over his short life, but everyone just attributed it to straightforward constipation without stopping to think, why? Is there something else going on?" Sinitsky told MedPage Today.

The boy's parents said in the report that "from an early age, we felt there were signs that something was not quite right" -- including having green stools for 4 to 5 months.

"Despite repeatedly raising this, we were consistently reassured that green stools were within the range of normal," they wrote in an accompanying note. "In hindsight, this proved to be a significant indicator that an underlying issue was present."

Initially the emergency team suspected a partial bowel obstruction, and transferred the patient to the specialty surgical center. There, specialists used a "step-wise" approach and a variety of radiological modalities "to really understand his gut," Sinitsky said.

That included a CT scan with 3D reconstruction, which revealed "two separate colonic lumens descending into a single anal canal" but no other anomalies.

This was confirmed on laparotomy, which revealed that the duplicated colon was "heavily impacted with fecal material and shared a common wall and a single mesentery with the native colon."

"Given the extent of the duplication and the shared mesenteric blood supply, any resection of the duplicate colon would jeopardize the perfusion of the entire colon," they wrote. Instead, they decided to join the two colons by dividing the common wall between them.

Duplications in the alimentary canal can occur anywhere between the mouth and the anus, and are seen in about 1 in 4,500 births, the researchers reported. Males and females are equally affected, they noted, and more than 80% of duplications present before age 2.

Duplications of the colon account for 13% to 15% of alimentary canal duplications, and a total colonic duplication, "is an extremely rare presentation in infancy," with only a few cases having been reported in the U.K., they wrote.

"Given its rarity and wide spectrum of clinical manifestations, it poses a diagnostic and therapeutic challenge," they noted.

Nonetheless, 1 year later, the child is "thriving well," Sinitsky told MedPage Today. He made a quick recovery, tolerated enteral feeds, and was discharged home on day 7 after surgery. A repeat barium enema at 6 weeks showed normal colonic anatomy with a degree of residual colonic dilatation, they wrote, noting they were able to reverse the ileostomy at 10 weeks after surgery.

After that, the child was passing soft stool daily. "The patient continues to have a normal bowel habit and intermittently needs stool softeners due to constipation," they added. "There is no further concern regarding bowel habit, abdominal distention, appetite, or growth."

"Based on the progress so far, the long-term functional outcomes are anticipated to be favorable," they wrote.

When managing constipation in children under age 1, especially with this degree of abdominal distention, and how healthcare professionals work together, "makes a huge difference," Sinitsky said.

"We all worked collaboratively to get a good understanding of this infant's history, his journey, and then at the tertiary center, they worked very collaboratively with radiology to get the right imaging to understand what was happening so they could actually plan surgery appropriately," she said.