Knowledge of the different anatomical variations in root canal system of dedicious dentition will improve the practice of the pediatric dentists. The teeth with C-shaped root canal configurations are definitely a problem in endodontic treatment. Dentists who are specialists of endodontics must have adequate knowledge about various root canal morphologies of primary tooth that have a tendency for rapid progression of dental caries to achieve a technically satisfactory outcome. This report presents an extraordinary case of unusual tooth morphology involving the mandibular first primary molar with a C-shaped configuration which has not yet been reported.