RESUMO
BACKGROUND: Hemophagocytic lymphohistiocytosis (HLH) is a life-threatening, aggressive syndrome. It can be primary, which involves genetic mutation with an early presentation, or secondary to infections, malignancies, etc., due to absence of immune downregulation. It is a very rare condition in newborns. Dengue is a potential virus causing HLH, but, in newborns, there are only few case reports and limited clinical literature. OBSERVATION: Herein, in this report, we highlight a case of neonatal HLH, triggered by perinatal dengue. The neonate manifested clinically within the first week of life, the earliest reported timeline so far in the literature. CONCLUSION: HLH should be excluded in neonates especially when multisystem involvement cannot be explained by sepsis alone.