The emergence of resistant strains of mycobacterium tuberculosis (TB) to antituberculous drugs has compounded the management of the chronic infection. More than 90% of rifampicin (RIF)-resistant isolates are also isoniazid resistant; hence, rifampicin resistance (RR) is a surrogate marker for multidrug resistant TB (MDR-TB). Although there are limited reports of pediatric RR/MDR-TB in Nigeria, there had not been similar report in our hospital until now. A 2-year-old girl was admitted with 2-month history of fever, cough with dyspnea, and progressive weight loss. There was no known contact with adult who had chronic cough; the toddler and her parents have not been treated for TB in the past. Her chest X-ray showed nodular opacities, while gastric washout for GeneXpert MTB/RIF confirmed RIF-resistant TB. The parents declined screening for TB despite counseling. The patient was subsequently referred to a specialized center for the management of drug-resistant TB, but the parents failed to go for the treatment. Young children are at risk of developing TB disease and MDR/RR-TB, which is more complex to manage than drug-susceptible TB due to longer treatment duration, increased toxicity, as well as poor parental compliance to the demand of treatment.