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Extraintestinal strongyloidiasis is rare in patients without immunosuppression. We describe the first case of chylous ascites as a result of strongyloidiasis. Alcohol cessation, diuretic therapy and repeated paracentesis did not lead to improvement of refractory chylous ascites with positive nucleic acid amplification test indicative of Strongyloides stercoralis infection. Only after therapy with ivermectin, could diuretics be withdrawn. Although direct proof of S. stercoralis was not possible by microscopy, successfully treated strongyloidiasis was confirmed in this patient by the positive and negative nucleic acid amplification tests from ascites before and after treatment together with the clinical improvement only after antiparasitic therapy.