Infliximab treatment in pachydermoperiostosis: a rare disease without an effective therapeutic option.
Hypertrophic osteoarthropathy (HOA) is characterized by periostitis of tubular bones, thickened skin, and digital clubbing. Its pathogenesis is unknown but an inflammatory factor and increased bone remodeling have been implicated. It is a very rare disease, usually diagnosed late with few therapeutic options. Bone and joint pains are secondary to periostitis and are usually difficult to control. Tumor necrosis factor-alpha is a cytokine that induces other inflammatory cytokine production, has an osteoclastogenic effect in different rheumatic diseases and probably also has an important role in periostitis and the systemic inflammatory manifestations in HOA. We describe the case of a patient with the primary form of HOA, who had refractory bone pain and arthritis that responded partially to infliximab treatment.
da Costa
Fernanda Vicente
FV
Rheumatology Division, University Hospital, Federal University of Santa Catarina, Florianópolis, Brazil.
de Magalhães Souza Fialho
Sonia Cristina
SC
Zimmermann
Adriana F
AF
Neves
Fabrício S
FS
Werner de Castro
Gláucio Ricardo
GR
Pereira
Ivânio Alves
IA
D000328:Adult; D000893:Anti-Inflammatory Agents / Q000627:therapeutic use*; D000911:Antibodies, Monoclonal / Q000627:therapeutic use*; D006801:Humans; D000069285:Infliximab; D008297:Male; D010004:Osteoarthropathy, Primary Hypertrophic / Q000188:drug therapy*; D035583:Rare Diseases; D014409:Tumor Necrosis Factor-alpha / Q000037:antagonists & inhibitors*
D000893:Anti-Inflammatory Agents; D000911:Antibodies, Monoclonal; D014409:Tumor Necrosis Factor-alpha; D000069285:Infliximab