The objective of this study was to report a case of a hydronephrotic patient with Strongyloides stercoralis infection, with discharge of rhabditoid larvae exclusively in urine. In 2013, a 72-yr-old male patient, hypertensive, obese, and diagnosed with hydronephrosis secondary to renal calculi, reported lumbar pain, polyuria, polaciuria, and dysuria, as well as frequent urinary tract infections. The microscopic analysis of urine sediment showed the presence of S. stercoralis rabditoid larvae. However, parasitological examinations by Baermann–Moraes, agar plate culture, and spontaneous sedimentation performed with 3 fecal samples on alternate days had negative results. The patient was treated with albendazole and to date has shown negative results in both parasitological and urine tests. This report deals with the unusual finding of S. stercoralis in a urine sample of an immunocompetent individual and absence of disseminated infection, but with hydronephrosis. Patients with nephropathies from S. stercoralis-endemic areas should be monitored periodically, as early detection may prevent the worsening of symptoms and renal failure.