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A rare case of Goodpasture syndrome concomitant with bleeding jejunal Dieulafoy's lesion. An 81-year-old man was diagnosed with Goodpasture syndrome (GS) because he met the criteria of positive anti-GBM antibodies, rapid progressive glomerulonephritis and pulmonary hemorrhage. After starting plasmapheresis and steroid pulse therapy, he experienced tarry stool and contrast-enhanced CT revealed an aneurysmal finding in the jejunum. Paroral enteroscopy showed a jejunal Dieulafoy's lesion (DL) with gush-out hemorrhage. Hemostasis was successfully achieved by hemoclipping, and he then experienced no re-bleeding events. GS can present as a jejunal DL, and contrast-enhanced CT is useful for investigating the etiology and site of small intestinal bleeding, which can lead to smooth, effective endoscopic hemostasis.