Introduction: Mycobacterial infection is a potential infectious complication of solid organ transplantation, which rarely occurs, but presenting significant risks to patient and challenges in terms of treatment. Illustration Case: A 42 years old woman after a 12th year of kidney transplantation was hospitalized with the chief complaints of fever and lower right abdominal pain, and diagnosed of acute appendicitis. The patient then underwent surgical appendectomy. Histopathology examination of appendix tissue shown granulomatous inflammation and NTM PCR of appendix tissue was positive for M.fortuitum. When she came for outpatient’s routine control, she also complained of right leg swollen and redness and eventually a purulent wound. The PCR of ulcer was positive for M.tuberculosis. Patient received outpatient therapy azithromycin, moxifloxacine, ethambutol, and INH. The clinical improvement was achieved after two months treatment. Discussion: The prevalence of active tuberculosis among patients with kidney transplantation is vary considerably depending on geographical area. In endemic areas could rise to 15.2%. Interestingly, where M. tuberculosis is endemic, cases of M. tuberculosis infection is much higher than NTM. Immunosuppressive therapy after transplantation compromises cell-mediated immunity, leaving the patient at risk for opportunistic as well as routine community-acquired infections. Diagnosis of mycobacterial infection in kidney transplant recipients is a challenge for clinician. Management mycobacterial infection is complex due to long treatment period with multidrug regimens and interaction with immunosuppressive drugs, and also requires good compliance from the patient. Conclusion: Mycobacterial infection a major cause of morbidity in kidney transplant recipients. High index of suspicion with team collaboration is necessary for early detection and management, with expectation to provide good result.