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Introduction: Drug-induced immune thrombocytopenia is considered extremely rare, as a cause of neonatal thrombocytopenia (PLT 0.000/μL). Diagnosis of drug-induced immune thrombocytopenia is considered as definite when all four George criteria are met: 1. Drug administration preceded thrombocytopenia. Recovery was complete and sustained after drug discontinuation 2. Other drugs administered prior to thrombocytopenia were continued or reintroduced after discontinuation of the suspected drug 3. Other etiologies of thrombocytopenia excluded 4. Re-exposure to the drug resulted in recurrent thrombocytopenia Objectives: We describe an interesting case of early onset, severe ampicilline-induced immune thrombocytopenia, in newborn twins. Material - Method: Monochorionic- diamniotic female twin neonates (A) and (B) (GA: 36 weeks) Results: Twins were born after an uncomplicated pregnancy via cesarean section (due to previous one). Perinatal history was unremarkable. Administration of empirical treatment with ampicillin and amikasin intravenously. On 2nd day of life (DOL), they exhibited severe thrombocytopenia (table 1), without hemorrhagic diathesis. Coagulation tests of (A) twin were prolonged (day 3). Laboratory screening for infection (CRP, blood culture), TORCH, perinatal asphyxia, necrotizing enterocolitis, neonatal alloimmune thrombocytopenia and disseminated intravascular coagulation, was negative. Drug-induced immune thrombocytopenia is an allergic reaction type II and in our case was related to ampicillin. Antibiotics were discontinued at 2nd DOL. Platelet transfusions were administered in both twins and fresh frozen plasma in (A) twin. On 5th DOL, platelet count was normal as well as coagulation tests of (A) twin, and no recurrence of thrombocytopenia was observed. Conclusions: Drug-induced neonatal immune thrombocytopenia is acute and severe. Clinical suspicion is important for diagnosis, which is probable in our case. [ABSTRACT FROM AUTHOR]