Vincristine is used in combination with other agents in the treatment of paediatric malignancies. Cranial nerve palsies involving oculomotor are seen rarely with ptosis and ophthalmoplegias being the most common manifestations, usually associated with polyneuropathy. We herein describe a three year girl with Vincristine-induced isolated bilateral ptosis without associated polyneuropathy causing diagnostic dilemma. Complete recovery of ptosis was achieved following treatment with pyridoxine and pyridostigmine. To the best of our knowledge this is a first case of isolated bilateral ptosis of Vincristine induced neuropathy.The top journals are peer reviewed scholarly journals. These provide high quality, meticulously reviewed and rapid publication, to cater the insistent need of scientific community. These journals are indexed with all their citations noted. The top open access journals are indexed in MEDLINE, PUBMED, SCOPUS, COPERNICUS, CAS, EBSCO and ISI.