Although adenovirus infections can occur at all ages, >80% of infections occur among children <4 years old. 1 A wide spectrum of clinical manifestations exist, ranging from self- limited respiratory or gastrointestinal disease to disseminated disease requiring extracorporeal membrane oxygenation (ECMO) therapy. 2, 3 Adenovirus in the neonatal period carries great significance because mortality rates exceed 50% for adenoviral pneumonia 3 and 85% for disseminated disease. 3–6 In a cohort of 37 neonates with adenovirus infection over a 17-year period, 22% required ECMO support; all 8 infants had disseminated disease, with 6 infants eventually succumbing to their illness (75%). 1 We present the clinical course of 2 neonates who were diagnosed with disseminated adenovirus with rapid clinical deterioration because of virus-associated hemophagocytic lymphohistiocytosis (HLH). Both developed pediatric acute respiratory distress syndrome and cardiovascular failure, ultimately succumbing to their illness despite prolonged ECMO support. These cases represent the potential consequences of an uncontrolled and ineffective immune response during a neonatal adenoviral infection. This case report was referred to our institutional review board, which deemed that case reports are not research. Health Insurance Portability and Accountability Act documents were received and signed by each family, and families were contacted by telephone by the principal investigator to confirm their knowledge of the plan to report the cases devoid of protected health information. CASE 1 A 10-day-old girl presented to the emergency department (ED) with a fever of 103°F and irritability. She was born by spontaneous vaginal delivery at 39 weeks’ gestation to a group B streptococcus–positive mother. The mother received appropriate antibiotics, and the infant was discharged on the third day of life. An ED examination was unremarkable, and significant laboratories included a total white blood cell count of 22.5 × Neonatal Adenovirus Infection Complicated by Hemophagocytic Lymphohistiocytosis Syndrome Nina Censoplano, DO, a Stephen Gorga, MD, b Kate Waldeck, MD, b Terri Stillwell, MD, MD, c Raja Rabah-Hammad, MD, d Heidi Flori, MD b Two infants with disseminated adenoviral infections are described. Both these infants had a similar clinical course and were also diagnosed with secondary hemophagocytic lymphohistiocytosis (HLH). Previous reports of immunocompromised adults with adenovirus-associated HLH are in the literature; however, this is the first report that we are aware of with this pathology occurring in infants. These cases are used to demonstrate the importance of thinking about HLH in patients who are diagnosed with adenovirus and exhibit prolonged fevers that are unresponsive to antimicrobial agents with hepatosplenomegaly and cytopenias. abstract To cite: Censoplano N, Gorga S, Waldeck K, et al. Neonatal Adenovirus Infection Complicated by Hemophagocytic Lymphohistiocytosis Syndrome. Pediatrics. 2018;141(s5):e20172061 Divisions of b Critical Care Medicine and c Infectious Disease, and d Department of Pathology, C.S. Mott Children’s Hospital, Medical School, University of Michigan, Ann Arbor, Michigan; and a Division of Cardiology, University of Colorado Anschutz Medical Campus, Children's Hospital of Colorado, Aurora, Colorado Drs Censoplano, Gorga, and Waldeck drafted the initial manuscript and reviewed and revised the manuscript; Dr Rabah-Hammad was responsible for formulating the figures and captions as well as editing the manuscript; Drs Stillwell and Flori critically reviewed the manuscript; and all authors approved the final manuscript as submitted and agree to be accountable for all aspects of the work. DOI: https://doi.org/10.1542/peds.2017-2061 Accepted for publication Oct 10, 2017 Address correspondence to Nina Censoplano, DO, The Heart Institute, Department of Pediatrics, University of Colorado at Denver School of Medicine, Children's Hospital Colorado, 13123 East 16th Ave, B-100, Aurora, CO 80045. E-mail: nina.censoplano@ childrenshospital.org PEDIATRICS (ISSN Numbers: Print, 0031-4005; Online, 1098-4275). Copyright © 2018 by the American Academy of Pediatrics FINANCIAL DISCLOSURE: The authors have indicated they have no financial relationships relevant to this article to disclose. FUNDING: No external funding. POTENTIAL CONFLICT OF INTEREST: The authors have indicated they have no potential conflicts of interest to disclose. PEDIATRICS Volume 141, number s5, April 2018:e20172061 CASE REPORT by guest on June 3, 2020 www.aappublications.org/news Downloaded from