The study of biomarkers and their signaling pathways has led to the development of new therapeutic strategies in a number of disorders. The purpose of the present systematic review is to provide an overview of different biomarkers in preterm newborns with corticosteroid-induced hypertrophic cardiomyopathy (CCHC). Several pathophysiological biomarkers are presented and discussed with the aim of investigating their diagnostic and prognostic value, particularly in relation to the potential progression of the disease and/or mortality in adulthood. This investigative approach may not only provide pathophysiological information on this serious drug-induced adverse effect, but also suggest novel therapeutic approaches to be applied in controlling its harmful consequences.