Aim: To describe a 4-year-old male child who presented bilateral gluteal lipodystrophy following intramuscular amikacin injections. Background: Intramuscular injection of various drugs can lead to localized lipodystrophy. These include insulin, corticosteroids, benzathine penicillin, methotrexate, aminoglycosides, human growth hormone, iron dextran, vasopressin, and DPT vaccine. The localized lipodystrophy following multiple intramuscular amikacin injections was not well described in pediatric population. Case description: We report a 4-year-old male who received multiple amikacin injections over bilateral gluteal region for 6 months and now presented with localized lipodystrophy over bilateral gluteal regions. Details of history avoided further unnecessary investigation, and parents were counseled about the nature of the lesion and no further active treatment. Conclusion: Intramuscular injection of amikacin can lead to localized lipodystrophy in children.