A young child being diagnosed with cancer naturally generates a pretty melancholy reaction. Each cancer can be managed with a vast array of treatment options that are available either individually or as a combination, the final goal of which is total eradication of the condition in the affected individual. Since, most of these treatments are administered during the age of tooth formation, they may affect stages of odontogenesis. Most common treatment of childhood cancers includes--chemotherapy and radiotherapy. With recent advancements in cancer therapy additional treatment options like laser therapy, radiation in the form of brachytherapy or teletherapy, cryotherapy, thermochemotherapy, etc. are available. As treatment of childhood cancers starts at a very young age coinciding with dental development, a number of dental malformations have been reported in childhood cancer survivors. The most common ocular cancer in children is retinoblastoma. This is the first such reported case and unique one where microdontia has affected all the first premolars.