---

Introduction: Therapeutic use of D-penicillamine is associated with several side effects. Proteinuria is the most common side effect of it, often due to membranous glomerulopathy. In this article we have reported a case of D-penicillamine induced focal segmental glomerulonephritis with systemic sclerosis. Case: In This article, we have presented a patient who had been under treatment with D-penicillamine and complicated by nephrotic range proteinuria resulting from focal segmental glomerulonephritis. Proteinuria was resolved one month after discontinuation of D-penicillamine without any additional treatment. She had no finding in favour of renal crisis of scleroderma. Conclusion: Such this pathologic form of renal involvment (FSGN) resulting from D-penicillamine has not been reported in the literature yet.