What presented as a partial bowel obstruction was something much rarer, according to British surgeons, who found that an infant with abdominal distention, vomiting and constipation, actually had a second colon, reports MedPage Today.
The child had initially presented to the emergency department but was transferred to a specialist paediatric surgical centre, where imaging suggested total duplication of the colon and rectum, Lynn Sinitsky, MBBS, of the West Hertfordshire Teaching Hospitals NHS Trust in England, and colleagues reported in BMJ Case Reports.
“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 four to five 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 centre. 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 faecal 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 jeopardise 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 one in 4 500 births, the researchers reported. Males and females are equally affected, they noted, and more than 80% of duplications present before the age of two.
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 United Kingdom, they wrote.
“Given its rarity and wide spectrum of clinical manifestations, it poses a diagnostic and therapeutic challenge,” they noted.
Nonetheless, one year later, the child is thriving, Sinitsky told MedPage Today. He made a quick recovery, tolerated enteral feeds, and was discharged home on day seven after surgery.
A repeat barium enema at six weeks showed normal colonic anatomy with a degree of residual colonic dilatation, and they were able to reverse the ileostomy at 10 weeks after surgery.
After that, the child was passing soft stool daily. “He 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 favourable,” they wrote.
When managing constipation in children under 12 months, 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 centre, 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.
Study details
Total colonic and rectal duplication in an infant: a multidisciplinary and multicentre diagnostic and surgical challenge
Shiv Hadani, Mohamed Ayman Atwa, Bashar Aldeiri et al.
Published in BMJ Case Reports in Volume 19 Issue 10
Abstract
A term infant, delivered by emergency Caesarean section, presented to the emergency department in a district hospital with abdominal distension exacerbated by vomiting and constipation. Examination revealed a distended, tense but non-tender abdomen and with an abdominal plain film radiograph, the clinical picture suggested partial bowel obstruction. The infant was transferred to a specialist paediatric surgical centre, and further radiological investigations suggested total colonic duplication. At surgery, complete colonic and rectal duplication was found, and complete division of the common wall was performed. The infant is currently thriving well and does not report any gastrointestinal symptoms. In this case report, we highlight the importance of multimodality imaging and multidisciplinary collaboration between paediatricians, radiologists and paediatric surgeons in the diagnosis and management of rare congenital anomalies.
MedPage Today article – ‘Constipation’ Lands Patient in Surgery for Unusual Anatomy (Open access)