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A 19-year-old woman with a 6-year history, consisting of frequent falls and weakness due to loss of strength in the lower extremities. During the last 10 hours, she presented a sudden episode of left hemiplegia, deviation of the labial commissure to the right, dysarthria and four emetic episodes. She was diagnosed with an ischemic stroke caused by a deficiency of the S-protein and, in addition, a muscular dystrophy. This case report may indicate a relationship between muscular dystrophy and ischemic stroke caused by a protein S deficiency