An autopsy-verified case of the generalized cytomegalic inclusion disease occurring in a premature male patient was presented, and its clinicopathologic characteristics were discussed with a brief literature review. The lesions were found to be systemic and showed typical cytomegalic inclusion cells having large acidophilic intranuclear and/or intracytoplasmic inclusions in the lungs, liver and kidneys with prominent extramedullary hemopoiesis in the liver, spleen, and kidneys.