Anja Schnabel1,2, Ursula Range3,4, Gabriele Hahn3,4, Reinhard Berner3,4, Christian M Hedrich1,2. 1. From the Pediatric Rheumatology and Immunology Section, Department of Pediatrics, and Department of Radiology, and the Institute for Medical Informatics and Biometry, Universitätsklinikum Carl Gustav Carus, Technische Universität Dresden, Dresden, Germany. anja.schnabel@uniklinikum-dresden.de christian.hedrich@uniklinikum-dresden.de. 2. A. Schnabel, MD, MSc, Pediatric Rheumatology and Immunology Section, Department of Pediatrics, Universitätsklinikum Carl Gustav Carus, Technische Universität Dresden; U. Range, PhD, Institute for Medical Informatics and Biometry, Universitätsklinikum Carl Gustav Carus, Technische Universität Dresden; G. Hahn, MD, Department of Radiology, Universitätsklinikum Carl Gustav Carus, Technische Universität Dresden; R. Berner, MD, PhD, Pediatric Rheumatology and Immunology Section, Department of Pediatrics, Universitätsklinikum Carl Gustav Carus, Technische Universität Dresden; C.M. Hedrich, MD, PhD, Pediatric Rheumatology and Immunology Section, Department of Pediatrics, Universitätsklinikum Carl Gustav Carus, Technische Universität Dresden. anja.schnabel@uniklinikum-dresden.de christian.hedrich@uniklinikum-dresden.de. 3. From the Pediatric Rheumatology and Immunology Section, Department of Pediatrics, and Department of Radiology, and the Institute for Medical Informatics and Biometry, Universitätsklinikum Carl Gustav Carus, Technische Universität Dresden, Dresden, Germany. 4. A. Schnabel, MD, MSc, Pediatric Rheumatology and Immunology Section, Department of Pediatrics, Universitätsklinikum Carl Gustav Carus, Technische Universität Dresden; U. Range, PhD, Institute for Medical Informatics and Biometry, Universitätsklinikum Carl Gustav Carus, Technische Universität Dresden; G. Hahn, MD, Department of Radiology, Universitätsklinikum Carl Gustav Carus, Technische Universität Dresden; R. Berner, MD, PhD, Pediatric Rheumatology and Immunology Section, Department of Pediatrics, Universitätsklinikum Carl Gustav Carus, Technische Universität Dresden; C.M. Hedrich, MD, PhD, Pediatric Rheumatology and Immunology Section, Department of Pediatrics, Universitätsklinikum Carl Gustav Carus, Technische Universität Dresden.
Abstract
OBJECTIVE: The autoinflammatory bone disorder chronic nonbacterial osteomyelitis (CNO) covers a wide clinical spectrum, ranging from mild self-limited presentations to chronically active or recurrent courses, which are then referred to as chronic recurrent multifocal osteomyelitis (CRMO). Little is known about treatment options and longterm outcomes. We investigated treatment responses and outcomes in children with CNO. METHODS: A retrospective chart review was conducted in a tertiary referral center, covering 2004-2015. Disease activity was measured at 0, 3, 6, 12, and 24 months after treatment initiation, and at the last recorded visit. RESULTS: Fifty-six patients with CNO were identified; 44 had multifocal CNO. Fifty percent of patients relapsed after a median of 2.4 years, and as few as 40% remained relapse-free after 5 years. Nonsteroidal antiinflammatory drugs were used as first-line treatment in 55 patients, inducing remission after 3 months in all individuals with relapse rates of 50% after 2 years. Further treatment included corticosteroids (n = 23), tumor necrosis factor-α (TNF-α) inhibitors (n = 7), and bisphosphonates (n = 8). While 47% of patients with CNO relapsed within 1 year after corticosteroid therapy, favorable outcomes were achieved with TNF-α inhibitors or bisphosphonates (pamidronate). CONCLUSION: CNO is a chronic disease with favorable outcomes within the first year, but high relapse rates in longterm followup. Particularly, patients with CRMO with long-lasting, uncontrolled inflammation were at risk for the development of arthritis. Our findings underscore the importance of a timely diagnosis and treatment initiation. Prospective studies are warranted to establish evidence-based diagnostic and therapeutic approaches to CNO.
OBJECTIVE: The autoinflammatory bone disorder chronic nonbacterial osteomyelitis (CNO) covers a wide clinical spectrum, ranging from mild self-limited presentations to chronically active or recurrent courses, which are then referred to as chronic recurrent multifocal osteomyelitis (CRMO). Little is known about treatment options and longterm outcomes. We investigated treatment responses and outcomes in children with CNO. METHODS: A retrospective chart review was conducted in a tertiary referral center, covering 2004-2015. Disease activity was measured at 0, 3, 6, 12, and 24 months after treatment initiation, and at the last recorded visit. RESULTS: Fifty-six patients with CNO were identified; 44 had multifocal CNO. Fifty percent of patients relapsed after a median of 2.4 years, and as few as 40% remained relapse-free after 5 years. Nonsteroidal antiinflammatory drugs were used as first-line treatment in 55 patients, inducing remission after 3 months in all individuals with relapse rates of 50% after 2 years. Further treatment included corticosteroids (n = 23), tumor necrosis factor-α (TNF-α) inhibitors (n = 7), and bisphosphonates (n = 8). While 47% of patients with CNO relapsed within 1 year after corticosteroid therapy, favorable outcomes were achieved with TNF-α inhibitors or bisphosphonates (pamidronate). CONCLUSION: CNO is a chronic disease with favorable outcomes within the first year, but high relapse rates in longterm followup. Particularly, patients with CRMO with long-lasting, uncontrolled inflammation were at risk for the development of arthritis. Our findings underscore the importance of a timely diagnosis and treatment initiation. Prospective studies are warranted to establish evidence-based diagnostic and therapeutic approaches to CNO.
Authors: Elena Tronconi; Angela Miniaci; Michelangelo Baldazzi; Laura Greco; Andrea Pession Journal: Rheumatol Int Date: 2017-11-11 Impact factor: 2.631
Authors: Mikhail M Kostik; Olga L Kopchak; Irina A Chikova; Eugenia A Isupova; Alexander Y Mushkin Journal: Rheumatol Int Date: 2018-08-31 Impact factor: 2.631
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