Author(s):
Figueiredo, AS ; Oliveira, AL ; Caetano, A ; Moraes-Fontes, MF
Date: 2020
Persistent ID: http://hdl.handle.net/10400.17/3467
Origin: Repositório do Centro Hospitalar de Lisboa Central, EPE
Subject(s): Acquired Hyperostosis Syndrome / diagnosis*; Acquired Hyperostosis Syndrome / drug therapy*; Adult; Female; Male; HCC IMA; HCC DAUTOIM; Humans; Acquired Hyperostosis Syndrome / immunology; Antibodies, Monoclonal, Humanized / therapeutic use; Bone and Bones / diagnostic imaging*; Middle Aged; Radionuclide Imaging; Treatment Outcome; Tumor Necrosis Factor Inhibitors / therapeutic use*; Ustekinumab / therapeutic use
Description
SAPHO (synovitis, acne, pustulosis, hyperostosis and osteitis) syndrome is a heterogeneous condition combining osteoarticular and cutaneous manifestations. Conventional treatments are mostly ineffective. We hereby report two patients, the first with an aggressive form of disease and the second with an incomplete response to two different anti-TNF-α agents. Both were successfully treated with tocilizumab and ustekinumab, respectively, over a long period of time. A narrative review of a biological therapy in SAPHO syndrome yielded very little information on the specific use of these agents. We highlight the advantages of personalising therapy and describe emerging promising treatments for this disease.