A case of pustulosis palmaris et plantaris (PPP) with hyperostosis was reported. The patient was a 30 year-old man, who had suffered PPP from 1977. On 23rd of July 1984, he was admitted to our hospital because he became to feel right gluteal pain in walking.The sclerotic changes were observed in sternocostoclavicular region and right ilium by roentgenograms and CT scan. Right iliosacral joint space was disappeared.Histological examination of left clavicula and right ilium revealed the findings such as the thickening of trabecular bone, the proliferation of osteoblasts, the fibrosis in medullary space and the inflammatory cell infiltration. Furthermore, the increased amount of osteoid formation was demonstrated in both osseous lesions by the osteoid stain (Rales' tetrachrome method).