Liver micro-abscesses in a preterm infant presenting with eosinophilic colitis

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ESPR132
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History: A preterm 33 4/7-week appropriate for dates female infant with birth weight of 2.22kg was admitted on day of life (DOL) 4 with abdominal distension, multiple episodes of bilious emesis, hematochezia and leukocytosis. She delivered vaginally to a 26-year old G3P2 mother following spontaneous rupture of membranes. Pregnancy was complicated with possible fetal intestinal obstruction based on prenatal imaging. Infant transitioned well after birth, stable initially in room air before requiring CPAP support for apnea of prematurity. Given prenatal concern for intestinal obstruction, infant had an abdominal x-ray and an ultrasound at referring hospital that were normal. She had normal abdominal examination and stooled with no emesis. Enteral feeds of maternal breast milk were started on DOL 1 and advanced before she was made NPO on DOL 4 due to multiple bilious emesis and hematochezia. Abdominal x-ray demonstrated gaseous distention but no pneumatosis. Antibiotics were started for sepsis concern while she was transferred for further evaluation; frank bloody stools mixed with mucus were noted on admission. Of note, family history was significant for a 14-month-old sibling diagnosed with milk protein allergy who had bloody stools.

Physical Examination: On admission, infant was noted to be well grown and appropriate for gestational age. Initial and subsequent vital signs were within normal limits except for one episode of elevated temperature up-to 38oC on DOL 5. Cardiorespiratory examination remained unremarkable with no respiratory distress, normal peripheral pulses and blood pressures, good perfusion and no murmurs. Initially her abdomen was distended and tender to palpation but with normoactive bowel sounds.

Laboratory & Diagnostic Imaging: Initial and subsequent serial abdominal x-rays revealed non-specific bowel gas fullness, with no pneumatosis. Infant had a normal upper GI study, while a contrast enema ruled out distal obstruction. An abdominal ultrasound with Doppler (Image 1) revealed hypoechoic, hypovascular anterior hepatic lesions concerning for microabscesses with scattered loops of bowel wall thickening in rectosigmoid region but no pneumatosis, pneumoperitoneum, or portal venous gas. An abdominal CT scan with contrast showed irregular hypodensities in anterior liver concerning (Image 2) for infectious etiology with mild mucosal hyperenhancement and bowel wall thickening in rectosigmoid region suggestive of infection or inflammation. Infant had leukocytosis and eosinophilia with a peak WBC of 44,000 on DOL 10 and an eosinophil count of >20% for four consecutive days (DOL 6 to 10). Anemia developed due to persistent hematochezia, requiring transfusion, with lowest hemoglobin of 8.9 g/dL. Clotting profile was normal with platelet count of 128,000. There was no transaminitis but infant developed direct hyperbilirubinemia (peak conjugated bilirubin 2.4 mg/dL) with peak GGTP of 178 U/L. Alpha-1 antitrypsin phenotype showed a normal PI*MM variant while Vitamin E and D levels were within normal limits. Echocardiogram showed a patent foramen ovale and cranial ultrasound was normal.

Final Diagnosis: Her abdominal tenderness and distention resolved within 48 hours of admission, but hematochezia persisted for several days while she remained NPO and on parenteral nutrition. Given bloody stools but normal abdominal examination, persistent leukocytosis and eosinophilia, plus a strong family history for milk allergy a diagnosis of transient eosinophilic colitis was entertained. As the initial hepatic findings on imaging were suggestive of infectious etiology along with radiographic evidence of inflammation in the rectosigmoid colon, it was also speculated that infant likely had infectious embolization from this region to the liver. She was empirically treated with broad spectrum antibiotics for 10 days including anaerobic coverage despite negative blood and urine cultures. Repeat abdominal ultrasound a week after the initial study showed resolution of the hepatic findings. Elemental formula feeds (20 kcal/oz) were started on DOL 13 and advanced to full volume over a week. Given possibility of milk protein allergy, maternal breast milk was reintroduced on DOL 24 after mother maintained a dairy free diet for a couple of weeks. After a 40-day hospital stay in NICU, infant was discharged home on maternal breast milk with complete resolution of hematochezia, leukocytosis and eosinophilia.

Discussion: This case represents the challenges that arise in diagnosing and treating eosinophilic colitis in premature infants as well as the risk of secondary systemic infections as shown in our patient. The immature enterohepatic and immune systems likely contributed to the unique complication of liver microabscesses. Damage to mucosal barrier at the inflamed rectosigmoid region may have promoted bacterial translocation causing seeding of the liver. In our patient, a definitive diagnosis via an intestinal biopsy was precluded due to limitations of prematurity. Furthermore, preterm infants often present with nonspecific and sometimes atypical clinical signs and symptoms, making a swift, definitive diagnosis of uncommon conditions like eosinophilic colitis more challenging. A better understanding of this complex gastrointestinal condition could lead to more focused management strategies especially in preterm infants, eliminating unnecessary diagnostic tests and prolonged courses of antibiotics.  

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Associated Sessions

University of Pittsburgh
UPMC- Children's Hospital of Pittsburgh
UPMC- Children's Hospital of Pittsburgh
UPMC Children's Hospital of Pittsburgh

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