Infantile sigmoid colon stricture secondary to cytomegalovirus

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A 3‐month‐old girl was referred to the surgical service with a history of intermittent feed intolerance and abdominal distension, non‐bilious vomiting and fevers. The episodes began at 2 weeks of age, were associated with rectal bleeding and elevated inflammatory markers, and settled with bowel rest and courses of intravenous antibiotics. She was born at 34 weeks via Caesarean section, with a birthweight of 1445 g. She had persistent thrombocytopenia from birth, and her C‐reactive protein was persistently elevated from 2 weeks of age. No cause for the intrauterine growth retardation was identified; in particular her TORCH serology was negative, as was Guthrie card polymerase chain reaction (PCR) testing for cytomegalovirus (CMV). She developed recurrent fevers and abnormal liver function tests. At 10 weeks of age, PCR testing for CMV was positive on urine, stool and blood, as was CMV IgM antibody.
On examination, the baby was undernourished, with marked abdominal distension. An abdominal radiograph demonstrated generalized, grossly dilated bowel loops. A rectal biopsy showed a negative acetylcholinesterase reaction, and ganglions were seen in the submucosa, excluding Hirschsprung disease. An upper gastrointestinal contrast study confirmed normal intestinal rotation, with contrast passing freely through markedly dilated colonic loops. A subsequent lower gastrointestinal contrast study demonstrated retained contrast material within the dilated colon, with an area of severe narrowing in the distal sigmoid colon (Fig. 1).
At laparotomy, a massively dilated descending and proximal sigmoid colon was found, with a transition point in the distal sigmoid colon. The colon was opened at this point and a fibrotic stricture approximately 6 cm in length was noted (Fig. 2a,b). This was excised and submitted for histology. An end sigmoid colostomy was fashioned and the rectal stump was oversewn. The patient received IV gancyclovir for several weeks until PCR testing of the blood for CMV was negative. Nasogastric feeding was re‐established and she was discharged 4 weeks post‐operatively.
Histological examination of the resected specimen identified an inflammatory stricture with extensive mucosal ulceration. The floor of the ulcer was composed of granulation tissue, with a dense mixed inflammatory infiltrate covered by fibrinopurulent exudates. A very occasional intranuclear inclusion body characteristic of CMV was seen, confirmed by immunohistochemistry (Fig. 3).
CMV is a member of the herpes virus group and is the commonest congenital viral infection with an incidence of 0.2–2.2%.1 Infection in the neonate may be congenital, perinatal or post‐natal. Congenital infection is due to maternal primary CMV infection or reactivation of the virus. Perinatal infection may occur during delivery. Post‐natal infection occurs most often from infected breast milk or transfusion with CMV‐infected blood products.
Congenital CMV infection may produce a wide range of clinical symptoms affecting the central nervous system, liver, spleen and eyes, but gastrointestinal involvement is uncommon.2 However, any part of the gastrointestinal tract may be involved. The association between necrotizing enterocolitis and CMV inclusion bodies on pathologic examination was first reported in 1979.3 Since then, reported surgical presentations with neonatal CMV infection have included meconium peritonitis, intestinal perforation and colonic dysmotility.4 The mechanism remains unknown, with one hypothesis suggesting CMV infection induces intestinal inflammation and a vasculitis, leading to intestinal ischaemia and destruction of the myenteric plexus.4
CMV infection causing a neonatal colonic stricture has only been described three times before, all in premature or small for gestational age neonates, as in this patient.2 This case demonstrates that clinicians should be wary of the gastrointestinal complications of CMV infection in neonates and infants. In particular, symptoms of abdominal distension, feed intolerance or decreased stool frequency in a neonate with a history of CMV should prompt evaluation for intestinal strictures.
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