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Introduction: Anti-glomerular basement membrane (anti-GBM) glomerulonephritis is associated with severe kidney failure and high morbidity, and is exceedingly rare in children. The few pediatric studies published about this condition report dependency of kidney replacement therapy at presentation, and partial or complete response in kidney function in a minority of cases. Case Presentation: We describe the case of a 14-year-old girl with crescentic glomerulonephritis based on double positive anti-GBM and myeloperoxidase (MPO-ANCA) antibodies with a mitigated clinical course presenting with fatigue, anemia and an estimated glomerular filtration rate range between 34-42 ml/min/1.73m². Response to treatment with daily therapeutic plasma exchanges, corticosteroids and oral cyclophosphamide was prompt. Conclusion: This ultrarare presentation highlights the need to consider determining anti-GBM antibodies and/or obtaining a kidney biopsy even in children with less severe presentations of unexplained glomerulonephritis, and underlines the clinical treatment dilemma in this disease for children due to the potential long-term sequalae.

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