title: Tumor necrosis factor receptor-associated periodic syndrome managed with the couple canakinumab-alendronate.
abstract: Management of tumor necrosis factor receptor-associated periodic syndrome (TRAPS) is puzzling, and therapeutic choices can be complicated, due to both wide genetic heterogeneity and protean clinical phenotype. We report on a 35-year-old female who was diagnosed with TRAPS, after finding the V95M mutation on the TNFRSF1A gene; who was treated in order with etanercept, anakinra, and canakinumab (150 mg/every 8 weeks by subcutaneous injection, then increased to 150 mg every 4 weeks); and who started therapy with oral alendronate (70 mg/weekly) to control her osteoporosis. Alendronate combined with canakinumab led to the optimal clinical control of all TRAPS manifestations and normalization of inflammatory markers. Further studies should be performed to clarify bisphosphonates' role in the scenery of autoinflammatory disorders.
authors.affiliation: Interdisciplinary Department of Medicine, Rheumatology Unit, Policlinico of Bari, Bari, Italy.
authors.forename: Giuseppe
authors.initials: G
authors.lastname: Lopalco
authors.forename: Donato
authors.initials: D
authors.lastname: Rigante
authors.forename: Antonio
authors.initials: A
authors.lastname: Vitale
authors.forename: Bruno
authors.initials: B
authors.lastname: Frediani
authors.forename: Florenzo
authors.initials: F
authors.lastname: Iannone
authors.forename: Luca
authors.initials: L
authors.lastname: Cantarini
mesh_terms: D000328:Adult; D019386:Alendronate / Q000008:administration & dosage*; D000911:Antibodies, Monoclonal / Q000008:administration & dosage*; D061067:Antibodies, Monoclonal, Humanized; D004359:Drug Therapy, Combination; D000068800:Etanercept / Q000008:administration & dosage; D005260:Female; D005334:Fever / Q000175:diagnosis* / Q000628:therapy*; D056660:Hereditary Autoinflammatory Diseases / Q000175:diagnosis* / Q000628:therapy*; D006801:Humans; D007074:Immunoglobulin G / Q000737:chemistry; D007249:Inflammation / Q000378:metabolism; D053590:Interleukin 1 Receptor Antagonist Protein / Q000008:administration & dosage; D009154:Mutation; D010024:Osteoporosis / Q000188:drug therapy; D010641:Phenotype; D047888:Receptors, Tumor Necrosis Factor, Type I / Q000235:genetics* / Q000378:metabolism*; D012008:Recurrence
chemical_list: D000911:Antibodies, Monoclonal; D061067:Antibodies, Monoclonal, Humanized; D007074:Immunoglobulin G; D053590:Interleukin 1 Receptor Antagonist Protein; D047888:Receptors, Tumor Necrosis Factor, Type I; C506235:TNFRSF1A protein, human; C541220:canakinumab; D000068800:Etanercept; D019386:Alendronate