ESOPHAGITIS AND PERINATAL CYTOMEGALOVIRUS INFECTION

A 6-week-old male infant presented to hospital with a large upper gastrointestinal hemorrhage. Endoscopic biopsies of his esophagus and duodenum showed cytomegalovirus (CMV) inclusions and CMV early antigen was detected by immunohistochemistry. There were no other signs of congenital CMV infection. This case adds to the clinical spectrum of perinatal CMV infection and may be more common than is currently recognized. Gastrointestinal involvement is an unusual manifestation of congenital and perinatal cytomegalovirus (CMV) infection, and esophageal disease is particularly uncommon. We describe an infant with an upper gastrointestinal hemorrhage caused by esophagitis attributed to perinatally acquired CMV infection. Case presentation. A 6-week-old boy was hospitalized after an acute onset of hematemesis and tachypnea. He was born after an uncomplicated full term pregnancy by spontaneous vaginal delivery with a birth weight of 2850 g. The perinatal course was uneventful, and the infant was thriving while being exclusively breast-fed. There was no history of fever, cough, vomiting, diarrhea, cracked or bleeding nipples in the mother, hematochezia or melena. His parents were awakened by the infant’s crying. He was found with bright red blood on his sleeper, mouth and nares. He was tachypneic and was brought to the hospital. On physical examination the infant was afebrile with a heart rate of 150/min, respiratory rate 90/min and an oxygen saturation of 96% in room air. His weight, length and head circumference were at the 50th percentile. There was dried blood in his nares. Respiratory examination showed intercostal and subcostal retractions with no grunting or nasal flaring. He had diminished breath sounds on the right with bronchial breathing. The remainder of his physical examination was normal. Laboratory investigations revealed a hemoglobin of 84 g/l, platelet count of 410 × 10 9/l and leukocyte count 9.1 × 10 9/l with 46% polymorphonuclear leukocytes, 4% band forms and 30% lymphocytes. Coagulation studies, transaminase, renal function, bilirubin and albumin values were normal. Chest radiography showed bilateral airspace disease predominantly in a central, posterior and basilar distribution. A nasogastric aspirate revealed coffee-ground material, which was negative for hemosiderin-laden macrophages. Bronchoscopy showed normal anatomy with no vascular anomalies or sites of bleeding. Bronchoalveolar lavage specimens had no organisms on a Gram-stained smear, and cultures were negative. No fungal elements were seen, and fungal species were not grown on culture. The esophagus appeared grossly congested without ulcerations or active bleeding. An upper gastrointestinal endoscopy performed 6 days later revealed a grossly normal appearing esophagus, stomach and duodenum. However, biopsy specimens from the esophagus and duodenum showed CMV inclusions, and the immunohistochemistry was positive for CMV early antigen. At the time of the receipt of the pathologic report, the infant was well with no recurrent gross or occult blood loss or feeding difficulties. It was elected not to treat him with ganciclovir or antireflux medications. At 15 months of age the child is well with growth parameters at the 50th percentile, and he is developmentally normal. Serology from the child and both parents was moderately positive for CMV IgG by enzyme-linked immunosorbent assay (Meridian Diagnostic Institute, Cincinnati, OH). Urine for CMV and studies for CMV antigenemia were not obtained during his initial presentation. HIV serology from the infant and parents was negative. Ophthalmologic examination showed no evidence of chorioretinitis, audiologic tests were normal and a computerized tomography scan of the brain showed no intracranial calcifications. Discussion. Congenital and perinatal CMV infection has a prevalence of 0.5 to 2.2%, making it the most common congenital/perinatally acquired viral infection. 1, 2 Only 5 to 10% of affected infants are symptomatic at birth, and the outcomes are highly variable. Gastrointestinal involvement is not usually a manifestation of congenital or perinatal CMV infection, although there have been reports of CMV-related enterocolitis in this population. 3, 4 CMV involvement of the esophagus is well-known in immunosuppressed children such as those with hematologic malignancies, those with HIV infection and those undergoing bone marrow and organ transplantation, but it is uncommon in immunocompetent children. 5 There has been one report of CMV esophagitis in a 3-week-old infant with vomiting and feeding intolerance. 6 That infant, however, had other stigmata of congenital CMV infection including hepatosplenomegaly and severe gastroesophageal reflux requiring surgical antireflux procedures. Treatment with ganciclovir was associated with clinical and histologic improvement. There has also been a report of an immunocompetent adult who developed acute erosive esophagitis associated with CMV infection acquired after receiving a blood transfusion and undergoing a splenectomy after trauma. 7 Our case is unusual with respect to the manner of presentation, lack of other manifestations of congenital CMV infection and spontaneous improvement. This case adds to the clinical spectrum of perinatal CMV infection and may represent a more common scenario than is currently recognized.

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