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1. Abnormal production of tumor necrosis factor (TNF) -- alpha and clinical efficacy of the TNF inhibitor etanercept in a patient with PAPA syndrome [corrected].

Author

Cortis E, De Benedetti F, Insalaco A, Cioschi S, Muratori F, D'Urbano LE, and Ugazio AG

Subjects
Acne Vulgaris drug therapy, Acne Vulgaris genetics, Adaptor Proteins, Signal Transducing genetics, Adolescent, Adult, Arthritis drug therapy, Arthritis genetics, Child, Child, Preschool, Cytoskeletal Proteins genetics, Etanercept, Female, Humans, Male, Pyoderma drug therapy, Pyoderma genetics, Syndrome, Tumor Necrosis Factor-alpha biosynthesis, Acne Vulgaris complications, Anti-Inflammatory Agents, Non-Steroidal therapeutic use, Arthritis complications, Immunoglobulin G therapeutic use, Pyoderma complications, Receptors, Tumor Necrosis Factor therapeutic use, Tumor Necrosis Factor-alpha antagonists & inhibitors
Abstract

We report a family with pyogenic sterile arthritis, pyoderna and acne syndrome (PAPA). The proband presented several episodes of sterile pyogenic arthritis and became unresponsive to glucocorticoids. After treatment with the tumor necrosis factor inhibitor etanercept, the disease underwent rapid and sustained clinical remission. Production of tumor necrosis factor-alpha by mononuclear cells of the proband and of the affected relatives was abnormally elevated.

Published
2004
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