A Preterm Infant with Abdominal Distension and Bloody Stools

A 40-day-old, preterm, female infant was referred to a neonatal intensive care unit secondary to abdominal distension and bloody stools. The infant, born by vaginal delivery at 336/7 weeks of gestation secondary to maternal preeclampsia, weighed 1950 g. Both Caucasian parents had no chronic medical conditions. At the referring facility, she had 3 episodes (on days 3, 6, and 20 of life) of abdominal distension and bloody gastric aspirates and/or hematochezia. Enteral feeding (expressed breast milk), initiated at age 3 days, was stopped at multiple intervals. She was first diagnosed with necrotizing enterocolitis and received antibiotics for 7 days. Her symptoms were then attributed to cow milk protein allergy and her enteral feedings were changed sequentially to 2 different hypoallergenic formulas. Her symptoms recurred on day 40, prompting referral. At the referral hospital, physical examination noted mild abdominal tenderness. Laboratory evaluation showed a low hemoglobin (7.6 g/dL) and positive fecal occult blood. Abdominal radiography revealed bowel loop distension and bowel wall edema suggestive of necrotizing enterocolitis. A barium enema revealed bowel wall thickening in the distal ileum. An upper gastrointestinal (GI) series confirmed jejunal and ileal bowel wall thickening (Figure). Abdominal computed tomography angiography showed enhancement of bowel wall and dilatation of the superior mesenteric artery, the superior mesenteric vein, and the portal vein. These radiographic findings were suggestive of GI hemangioma.1Soukoulis I.W. Liang M.G. Fox V.L. Mulliken J.B. Alomari A.I. Fishman S.J. Gastrointestinal infantile hemangioma: presentation and management.J Pediatr Gastroenterol Nutr. 2015; 61: 415-420Crossref PubMed Scopus (25) Google Scholar, 2Drolet B.A. Pope E. Juern A.M. Sato T. Howell B. Puttgen K.B. et al.Gastrointestinal bleeding in infantile hemangioma: a complication of segmental, rather than multifocal, infantile hemangiomas.J Pediatr. 2012; 160 (1021.e3-6.e3)Abstract Full Text Full Text PDF PubMed Scopus (36) Google Scholar, 3Rao A.B. Pence J. Mirkin D.L. Diffuse infantile hemangiomatosis of the ileum presenting with multiple perforations: a case report and review of the literature.J Pediatr Surg. 2010; 45: 1890-1892Abstract Full Text Full Text PDF PubMed Scopus (10) Google Scholar GI hemangiomas are rare bowel tumors that are located exclusively in the midgut, in the distribution of the superior mesenteric artery.3Rao A.B. Pence J. Mirkin D.L. Diffuse infantile hemangiomatosis of the ileum presenting with multiple perforations: a case report and review of the literature.J Pediatr Surg. 2010; 45: 1890-1892Abstract Full Text Full Text PDF PubMed Scopus (10) Google Scholar Patients are mostly female, sometimes have associated cutaneous hemangiomas,1Soukoulis I.W. Liang M.G. Fox V.L. Mulliken J.B. Alomari A.I. Fishman S.J. Gastrointestinal infantile hemangioma: presentation and management.J Pediatr Gastroenterol Nutr. 2015; 61: 415-420Crossref PubMed Scopus (25) Google Scholar, 2Drolet B.A. Pope E. Juern A.M. Sato T. Howell B. Puttgen K.B. et al.Gastrointestinal bleeding in infantile hemangioma: a complication of segmental, rather than multifocal, infantile hemangiomas.J Pediatr. 2012; 160 (1021.e3-6.e3)Abstract Full Text Full Text PDF PubMed Scopus (36) Google Scholar, 3Rao A.B. Pence J. Mirkin D.L. Diffuse infantile hemangiomatosis of the ileum presenting with multiple perforations: a case report and review of the literature.J Pediatr Surg. 2010; 45: 1890-1892Abstract Full Text Full Text PDF PubMed Scopus (10) Google Scholar are frequently misdiagnosed with necrotizing enterocolitis or cow milk protein sensitivity,1Soukoulis I.W. Liang M.G. Fox V.L. Mulliken J.B. Alomari A.I. Fishman S.J. Gastrointestinal infantile hemangioma: presentation and management.J Pediatr Gastroenterol Nutr. 2015; 61: 415-420Crossref PubMed Scopus (25) Google Scholar and occasionally undergo laparotomy for diagnostic purposes.1Soukoulis I.W. Liang M.G. Fox V.L. Mulliken J.B. Alomari A.I. Fishman S.J. Gastrointestinal infantile hemangioma: presentation and management.J Pediatr Gastroenterol Nutr. 2015; 61: 415-420Crossref PubMed Scopus (25) Google Scholar, 4Morris G.A. Stratchko L. Sabri M. Intestinal hemangioma presenting as recurrent hematochezia in a 6-week-old male.J Ped Surg Case Rep. 2015; 23: 280-282Crossref Scopus (6) Google Scholar High-resolution ultrasound examination with Doppler, considered to be the least invasive radiographic modality, confirmed computed tomography angiography findings in this infant (Figure).1Soukoulis I.W. Liang M.G. Fox V.L. Mulliken J.B. Alomari A.I. Fishman S.J. Gastrointestinal infantile hemangioma: presentation and management.J Pediatr Gastroenterol Nutr. 2015; 61: 415-420Crossref PubMed Scopus (25) Google Scholar Propranolol is the suggested first line therapy.5Raphael M.F. Breur J.M. Vlasveld F.A. Elbert N.J. Liem Y.T. Kon M. et al.Treatment of infantile hemangiomas: therapeutic options in regard to side effects and adverse events—a review of the literature.Expert Opin Drug Saf. 2016; 15: 199-214Crossref PubMed Scopus (7) Google Scholar Our patient was placed on oral propranolol and the dose was titrated to 3 mg/kg/day with improvement in her abdominal symptoms. The duration of therapy is solely dictated by clinical symptoms and cessation of GI bleeding and can last for ≤9 months.1Soukoulis I.W. Liang M.G. Fox V.L. Mulliken J.B. Alomari A.I. Fishman S.J. Gastrointestinal infantile hemangioma: presentation and management.J Pediatr Gastroenterol Nutr. 2015; 61: 415-420Crossref PubMed Scopus (25) Google Scholar, 5Raphael M.F. Breur J.M. Vlasveld F.A. Elbert N.J. Liem Y.T. Kon M. et al.Treatment of infantile hemangiomas: therapeutic options in regard to side effects and adverse events—a review of the literature.Expert Opin Drug Saf. 2016; 15: 199-214Crossref PubMed Scopus (7) Google Scholar Occasionally, infants with persistent GI bleeding or perforation require bowel resection.1Soukoulis I.W. Liang M.G. Fox V.L. Mulliken J.B. Alomari A.I. Fishman S.J. Gastrointestinal infantile hemangioma: presentation and management.J Pediatr Gastroenterol Nutr. 2015; 61: 415-420Crossref PubMed Scopus (25) Google Scholar, 4Morris G.A. Stratchko L. Sabri M. Intestinal hemangioma presenting as recurrent hematochezia in a 6-week-old male.J Ped Surg Case Rep. 2015; 23: 280-282Crossref Scopus (6) Google Scholar Our patient was discharged home at 119 days on full enteral feeding and propranolol therapy. We thank Denise M. Abrams, MD, at the Vascular Anomalies Center (VAC) at Boston Children's Hospital for her help in this patient's evaluation and review of the case report.