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A Japanese girl, 2 years, 8 months of age, with palmoplantar keratosis and dendritic corneal opacities, showed increased tyrosine levels in the plasma, urine, and cerebrospinal fluid. The mental and physical growth was riot retarded. The hepatorenal functions were within normal limits. Electron microscopically, the epidermal keratinocytes showed increased tonofibrils and no structures suggestive of tyrosine crystals. Cytosol and mitochondrial tyrosine aminotransferase (TAT) activities of the liver were greatly decreased, while p -hydroxyphenyl pyruvate oxidase ( p -HPPO) activity was not decreased. The plasma tyrosine levels were controlled for 3 years with low phenylalanine-tyrosine diet.

Thirty-one patients with Bowen's disease were studied in a restricted district of Namikata and its neighborhood of Ehime, Japan. Nineteen were alive and 12 were dead; the youngest living patient was 52 years of age. Invasive skin cancers were found in 10 patients and internal malignancies in 10, including 7 patients with pulmonary cancers. Palmoplantar keratosis was present in 25 patients and raindrop-type pigment anomalies in 15. Neutron activation analysis of hair showed only slightly higher arsenic values in patients with Bowen's disease than those in normal controls, though the differences were statistically significant at p less than 0.05. A possible arsenic exposure 43 years previously was considered responsible for the occurrence of neoplasms, though the arsenic route and amount were not determined. Bowen's disease started within 10 years, invasive skin cancers after 20 years, and pulmonary cancers after 30 years following the suspected arsenic exposure.