OBJECTIVE: To date there is no uniformly effective treatment for either chronic recurrent multifocal osteomyelitis (CRMO) or synovitis, acne, pustulosis, hyperostosis and osteitis (SAPHO) syndrome. We report on our clinical experience of using biologic therapy to treat children with these conditions. METHODS: Retrospective descriptive case series of four children with refractory disease treated with biologics. Disease activity was assessed at predetermined time points (T = 0, T = 6 weeks and T = 12 months after the start of biologic therapy, and at latest follow-up) using a combination of clinical examination and radiological findings: a 10 cm pain and physician visual analogue scale; the Childhood Health Assessment Questionnaire as an assessment of disability; and changes in markers of systemic inflammation. RESULTS: There was an initial improvement in all parameters assessed for all three children treated with TNF-alpha blockade, although the third case had to discontinue the therapy due to a suspected (but unconfirmed) fungal skin infection. Anakinra treatment alleviated the symptoms in the fourth patient at 6 weeks, but there was no sustained response to treatment at 1-year follow-up. CONCLUSION: We present our preliminary experience of using biological therapies to treat children with CRMO and SAPHO in conjunction with other immunosuppression. Further studies are needed to establish the role of these therapies in refractory CRMO and SAPHO.
OBJECTIVE: To date there is no uniformly effective treatment for either chronic recurrent multifocal osteomyelitis (CRMO) or synovitis, acne, pustulosis, hyperostosis and osteitis (SAPHO) syndrome. We report on our clinical experience of using biologic therapy to treat children with these conditions. METHODS: Retrospective descriptive case series of four children with refractory disease treated with biologics. Disease activity was assessed at predetermined time points (T = 0, T = 6 weeks and T = 12 months after the start of biologic therapy, and at latest follow-up) using a combination of clinical examination and radiological findings: a 10 cm pain and physician visual analogue scale; the Childhood Health Assessment Questionnaire as an assessment of disability; and changes in markers of systemic inflammation. RESULTS: There was an initial improvement in all parameters assessed for all three children treated with TNF-alpha blockade, although the third case had to discontinue the therapy due to a suspected (but unconfirmed) fungal skin infection. Anakinra treatment alleviated the symptoms in the fourth patient at 6 weeks, but there was no sustained response to treatment at 1-year follow-up. CONCLUSION: We present our preliminary experience of using biological therapies to treat children with CRMO and SAPHO in conjunction with other immunosuppression. Further studies are needed to establish the role of these therapies in refractory CRMO and SAPHO.
Authors: Rena C Zuo; Daniella M Schwartz; Chyi-Chia Richard Lee; Milan J Anadkat; Edward W Cowen; Haley B Naik Journal: J Am Acad Dermatol Date: 2014-08-12 Impact factor: 11.527
Authors: Elena Tronconi; Angela Miniaci; Michelangelo Baldazzi; Laura Greco; Andrea Pession Journal: Rheumatol Int Date: 2017-11-11 Impact factor: 2.631
Authors: Suzanne L Cassel; John R Janczy; Xinyu Bing; Shruti P Wilson; Alicia K Olivier; Jesse E Otero; Yoichiro Iwakura; Dmitry M Shayakhmetov; Alexander G Bassuk; Yousef Abu-Amer; Kim A Brogden; Trudy L Burns; Fayyaz S Sutterwala; Polly J Ferguson Journal: Proc Natl Acad Sci U S A Date: 2014-01-06 Impact factor: 11.205
Authors: Yongdong Zhao; Eveline Y Wu; Melissa S Oliver; Ashley M Cooper; Matthew L Basiaga; Sheetal S Vora; Tzielan C Lee; Emily Fox; Gil Amarilyo; Sara M Stern; Jeffrey A Dvergsten; Kathleen A Haines; Kelly A Rouster-Stevens; Karen B Onel; Julie Cherian; Jonathan S Hausmann; Paivi Miettunen; Tania Cellucci; Farzana Nuruzzaman; Angela Taneja; Karyl S Barron; Matthew C Hollander; Sivia K Lapidus; Suzanne C Li; Seza Ozen; Hermann Girschick; Ronald M Laxer; Fatma Dedeoglu; Christian M Hedrich; Polly J Ferguson Journal: Arthritis Care Res (Hoboken) Date: 2018-07-12 Impact factor: 4.794