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Bilateral hydroureteronephrosis in a patient with central diabetes insipidus is a rare condition. This rare presentation poses unique diagnostic and therapeutic challenges. Herein, we report a case of diabetes insipidus associated with bilateral hydroureteronephrosis and discuss its clinical and radiographic findings with a special focus on the urological aspects. Dramatic improvement, both clinically and radiologically, was seen after the administration of desmopressin therapy.