Primary or secondary hemophagocytic lymphohistiocytosis (HLH) is characterized by immune activation and lifethreatening cytopenias. Causative relationship between a number of pathogens, autoimmune diseases and even hematologic malignancies with secondary HLH (sHLH) have been reported; however, correlation with myelodysplastic syndromes (MDS) is exceedingly unusual. HLH in COVID-19 positive patients has been described. Nevertheless, patients who have recovered after COVID-19 infection and test negative for the virus by RT-PCR may develop sHLH due to immune system dysregulation. We present here a rare case of sHLH manifestation after COVID-19 infection in a patient with a history of MDS. The recently revised H-score contributed to diagnosis.