title: Long-term Effects of Neridronate in Adults with Osteogenesis Imperfecta: An Observational Three-Year Italian Study.
abstract: The aim of this study was to assess the long-term efficacy and safety of i.v. neridronate in the treatment of osteogenesis imperfecta (OI). One hundred and fourteen patients affected by OI were included in the study. Neridronate was administered by i.v. infusion at the dosage of 2 mg/kg, up to a maximum of 100 mg at three-month intervals for 3 years. Dual X-ray absorptiometry of the lumbar spine, hip, and ultradistal and proximal radius were evaluated every 6 months. Blood calcium, phosphate, albumin, fasting urinary calcium/creatinine ratio, total serum alkaline phosphatase, and bone alkaline phosphatase were obtained at baseline and every 3 months. The mean lumbar spine and total hip BMD significantly increased from baseline to any time point (p < 0.001). The mean ultradistal radius BMD significantly increased from baseline only at month 18 (p = 0.026), 30 (p = 0.046), and 36 (p = 0.013), respectively. The mean proximal radius BMD did not change during the whole observation. The levels of bone turnover markers significantly decreased from baseline to any post-baseline observation time. The study was not able to find any statistically significant effect on fracture risk (p = 0.185). The percentage of patients with fractures was unaltered during treatment as compared to the 3-year period before treatment. The most common AEs were fragility fractures, back pain, arthralgia, fever, and joint sprain. An acute phase reaction was reported in 26 (22.8%) patients. None of the reported SAEs were considered as treatment-related. Long-term treatment with i.v. neridronate has positive effects on BMD and bone turnover markers with a good safety profile, although no significant effect on the risk of fracture was observed.
authors.affiliation: Rheumatology Unit, Department of Medicine, University of Verona, p.le L Scuro 2, 37100, Verona, Italy.
authors.forename: O
authors.initials: O
authors.lastname: Viapiana
authors.affiliation: Rheumatology Unit, Department of Medicine, University of Verona, p.le L Scuro 2, 37100, Verona, Italy. luca.idolazzi@univr.it.
authors.forename: L
authors.initials: L
authors.lastname: Idolazzi
authors.affiliation: Rheumatology Unit, Department of Medicine, University of Verona, p.le L Scuro 2, 37100, Verona, Italy.
authors.forename: A
authors.initials: A
authors.lastname: Fassio
authors.affiliation: Rheumatology Unit, Department of Medicine, University of Verona, p.le L Scuro 2, 37100, Verona, Italy.
authors.forename: G
authors.initials: G
authors.lastname: Orsolini
authors.affiliation: Rheumatology Unit, Department of Medicine, University of Verona, p.le L Scuro 2, 37100, Verona, Italy.
authors.forename: M
authors.initials: M
authors.lastname: Rossini
authors.affiliation: Rheumatology Unit, Department of Medicine, University of Verona, p.le L Scuro 2, 37100, Verona, Italy.
authors.forename: G
authors.initials: G
authors.lastname: Adami
authors.affiliation: Internal Medicine, Department of Medicine, University of Verona, p.le L Scuro 2, 37100, Verona, Italy.
authors.forename: F
authors.initials: F
authors.lastname: Bertoldo
authors.affiliation: Rheumatology Unit, Department of Medicine, University of Verona, p.le L Scuro 2, 37100, Verona, Italy.
authors.forename: D
authors.initials: D
authors.lastname: Gatti
mesh_terms: D015502:Absorptiometry, Photon / Q000379:methods; D000469:Alkaline Phosphatase / Q000097:blood; D015519:Bone Density / Q000187:drug effects*; D001842:Bone and Bones / Q000187:drug effects; D004164:Diphosphonates / Q000627:therapeutic use*; D005260:Female; D050723:Fractures, Bone / Q000188:drug therapy; D006801:Humans; D007558:Italy; D008297:Male; D010013:Osteogenesis Imperfecta / Q000188:drug therapy*; D013995:Time; D016896:Treatment Outcome
chemical_list: D004164:Diphosphonates; C053389:6-amino-1-hydroxyhexane-1,1-diphosphonate; D000469:Alkaline Phosphatase
keywords: Neridronate; Osteogenesis imperfecta; Safety; Tolerability