title: Denosumab for the Treatment of Mastocytosis-Related Osteoporosis: A Case Series.
abstract: The purpose of this study was to investigate the therapeutic effect of denosumab, an anti-RANKL monoclonal antibody for the treatment of bone loss in indolent systemic mastocytosis (ISM) patients intolerant to bisphosphonates. Four patients underwent upon informed consent a treatment with denosumab 60 mg administered subcutaneously every 6 months with the same regimen used for postmenopausal osteoporosis. Bone mineral density (BMD) was measured at lumbar and femoral sites at baseline and after 1 year. C-terminal telopeptide of collagen type I (CTX), bone alkaline phosphatase (bALP) and tryptase serum level were determined at baseline and after 12 months with fasting blood samples withdrawals. BMD increased significantly at both sites during the 12 months; all the patients had an important decrease of serum CTX and of lesser extent of bALP serum levels. After denosumab treatment, a decrease in serum tryptase level was observed in all the patients. No adverse events or new fractures occurred. Denosumab seems to be a valid alternative for the treatment of bone loss in ISM. RANKL might be of key importance in the pathogenesis of ISM bone involvement.
authors.affiliation: Rheumatology Section, Department of Medicine, University of Verona, Piazzale L. Scuro 10, 37134, Verona, Italy. giovanniorsolini@gmail.com.
authors.forename: Giovanni
authors.initials: G
authors.lastname: Orsolini
authors.affiliation: Rheumatology Section, Department of Medicine, University of Verona, Piazzale L. Scuro 10, 37134, Verona, Italy.
authors.forename: Irene
authors.initials: I
authors.lastname: Gavioli
authors.affiliation: Rheumatology Section, Department of Medicine, University of Verona, Piazzale L. Scuro 10, 37134, Verona, Italy.
authors.forename: Gaia
authors.initials: G
authors.lastname: Tripi
authors.affiliation: Rheumatology Section, Department of Medicine, University of Verona, Piazzale L. Scuro 10, 37134, Verona, Italy.
authors.forename: Ombretta
authors.initials: O
authors.lastname: Viapiana
authors.affiliation: Rheumatology Section, Department of Medicine, University of Verona, Piazzale L. Scuro 10, 37134, Verona, Italy.
authors.forename: Davide
authors.initials: D
authors.lastname: Gatti
authors.affiliation: Rheumatology Section, Department of Medicine, University of Verona, Piazzale L. Scuro 10, 37134, Verona, Italy.
authors.forename: Luca
authors.initials: L
authors.lastname: Idolazzi
authors.affiliation: Hematology Section, Department of Medicine, University of Verona, Verona, Italy.
authors.forename: Roberta
authors.initials: R
authors.lastname: Zanotti
authors.affiliation: Rheumatology Section, Department of Medicine, University of Verona, Piazzale L. Scuro 10, 37134, Verona, Italy.
authors.forename: Maurizio
authors.initials: M
authors.lastname: Rossini
mesh_terms: D000368:Aged; D000369:Aged, 80 and over; D015519:Bone Density / Q000187:drug effects*; D050071:Bone Density Conservation Agents / Q000627:therapeutic use*; D016723:Bone Remodeling / Q000187:drug effects*; D000069448:Denosumab / Q000627:therapeutic use*; D005260:Female; D050723:Fractures, Bone / Q000188:drug therapy; D006801:Humans; D008415:Mastocytosis / Q000150:complications* / Q000188:drug therapy; D008875:Middle Aged; D010024:Osteoporosis / Q000150:complications / Q000188:drug therapy*
chemical_list: D050071:Bone Density Conservation Agents; D000069448:Denosumab
keywords: Denosumab; Osteoporosis; RANKL; Systemic mastocytosis