Background: Tuberculosis remains a worldwide health problem despite tremendous improvements in diagnosis and management. Globally, 6.7 million cases of tuberculosis were reported to WHO in 2017 and India ranked for highest annual incidence. Extra–pulmonary tuberculosis comprises 15–20 % total burden of the disease in which Genito–urinary tuberculosis is nearly 4 to10% in various studies. Genito–urinary tuberculosis may result in nephron loss, ureteric stricture, bladder contraction, prostatitis, epididymitis, orchitis and even functional loss of these organs. Simultaneous end organ failure of kidney and bladder is rare, because disease is usually detected early, well before bladder involvement. Here we come across two neglected cases of Genitourinary Tuberculosis presented with putty kidney and thimble bladder in same patient. Case details: 17 years old adolescent and 54 years old adult male, presented with history of intermittent ,mild, dull aching right flank pain and severe lower urinary tract symptoms since last two years. Presumptive diagnosis of genitourinary tuberculosis was made in view of prolong non– specific symptoms and multiple empirical course of antibiotic in suspicion of urinary tract infection. Disease Confirmed with TB PCR of urine .Category one Chemotherapy started. Imaging showed as small capacity bladder with right non–functioning kidney. Full details of disease and prognosis was explained to the patient . After completion of induction phase both patient underwent right nephrectomy. For small contracted bladder, one Patient opted augmentation ileo– cystoplasty and other cystectomy and ileal conduit. At 6 months follow up both the patients completed Anti–tubercular treatment course and doing well. Conclusion: Impact of untreated tuberculosis on the genitor–urinary tract is devastating. Because of, non–specific symptoms, prolong subclinical course and low yield of current available investigations, chemotherapy is delayed that leads to dysfunctional anatomical changes. Therefore, Surgery continues to play a major role to decrease morbidity as well as to restore functional integrity of genitourinary system.