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Intrauterine limb gangrene with cerebral infarction is a rare condition and only few cases of this condition have been reported. The neonatal thrombosis needs to be distinguished from intrauterine arterial thrombosis as it is caused by catheterization of the umbilical artery or less commonly seen as complication of sepsis or coagulation disorders. The report describes the case of preterm infant with bilateral brachial artery thrombosis in utero and neonatal gangrene of the left forearm. The right forearm of the infant had a small patch of gangrene with irregular areas of edema and bluish discoloration, extending to the hand. The hand of infant was associated with thrombosis of left middle cerebral artery and acute infarction in left cerebral hemisphere. Both of the upper limbs in the patient were affected, including the left cerebral hemisphere; although in majority of the cases, the limbs are equally affected and the left side of the brain is preferentially involved. LMWH was administered in the present case, which significantly improved the upper right limb. Laboratory findings, including the coagulation markers within normal reference, ranges for the chronologic and gestational age. Further complications were prevented by early post-natal use of anticoagulants and prevention of infection; although the exact cause of this condition was not clear.