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Two cases of hypophosphatemic osteomalacia who have not been treated with vitamin D are presented. Case 1. A 27-year-old man was admitted to our hospital because of severe bow leg and bilateral knee pain. From infancy slightly bow leg was noted and his height increased slowly. He had lower leg pain from the age of 12 years. At 21 years old, osteotomy of his right tibia was performed to correct bow log deformity at the other hospital. Roentgenographic study discloses pseudofracture (Looser's zone) in right tibia, hyperostosis of cranial vault, ribs, clavicles, long and short tubular bones and features of sacro-iliitis.Laboratory investigation shows hypophosphatemia, hyperalkaline phosphatasemia and normal calcemia. We perfomed corrective osteotomy of bilateral tibiae. Histological examination of bone shows the accumulation of osteoid. Case 2. is a 31-year-old, elder sister of Case 1. Clinical and roentgenographic features of this case resembles those in Case 1, except that spondyloarthritic features are more prominet than in Case 1.