title: Denosumab in patients with giant-cell tumor of bone in Norway: results from a nationwide cohort.
abstract: BACKGROUND
  Denosumab is a relatively new treatment option for patients with giant-cell tumor of bone (GCTB). The purpose of this study was to report the results for patients treated in Norway.
  
  
  METHODS
  Patients treated with denosumab for GCTB were identified from the clinical databases at the Norwegian sarcoma reference centers. Data were retrieved from the clinical databases and supplemented by retrospective review of patient records. Denosumab was given as a subcutaneous injection every 4 weeks with loading doses on day 8 and 15 in cycle 1.
  
  
  RESULTS
  Eighteen patients treated with denosumab for GCTB were identified. Denosumab was given for recurrent disease in seven cases and as first-line treatment in 11 patients, of which 6 received therapy as part of a neoadjuvant/adjuvant strategy and 5 for surgically unsalvageable primary tumor. Ten of 12 patients with unresectable disease are still on denosumab without progression with median treatment duration of 41 months (range 18-60). Two patients discontinued treatment due to osteonecrosis of the jaw and reduced compliance, respectively. In the adjuvant group, four patients experienced disease recurrence after stopping denosumab. In three of six patients, the extent of surgery was reduced due to neoadjuvant therapy. Seventeen of 18 patients underwent response evaluation with 18F-FDG PET/CT at median 4.7 weeks from starting denosumab. Median baseline SUVmaxwas 11.0 and median SUVmaxat evaluation was 4.9 (p < 0.001).
  
  
  CONCLUSIONS
  In a nationwide GCTB patient cohort, denosumab was an effective agent and durable responses were observed. Our results do not support the use of adjuvant therapy in routine clinical practice. 18F-FDG PET/CT could be a valuable tool for early response evaluation.
authors.affiliation: a Department of Oncology , Oslo University Hospital , Oslo , Norway.
authors.forename: Kjetil
authors.initials: K
authors.lastname: Boye
authors.affiliation: b Department of Oncology , Haukeland University Hospital , Bergen , Norway.
authors.forename: Nina Louise
authors.initials: NL
authors.lastname: Jebsen
authors.affiliation: c Department of Orthopaedic Surgery , Oslo University Hospital , Oslo , Norway.
authors.forename: Olga
authors.initials: O
authors.lastname: Zaikova
authors.affiliation: d Department of Oncology , St Olav University Hospital , Trondheim , Norway.
authors.forename: Heidi
authors.initials: H
authors.lastname: Knobel
authors.affiliation: e Department of Nuclear Medicine , Oslo University Hospital , Oslo , Norway.
authors.forename: Ayca M
authors.initials: AM
authors.lastname: Løndalen
authors.affiliation: b Department of Oncology , Haukeland University Hospital , Bergen , Norway.
authors.forename: Clement S
authors.initials: CS
authors.lastname: Trovik
authors.affiliation: b Department of Oncology , Haukeland University Hospital , Bergen , Norway.
authors.forename: Odd R
authors.initials: OR
authors.lastname: Monge
authors.affiliation: a Department of Oncology , Oslo University Hospital , Oslo , Norway.
authors.forename: Kirsten Sundby
authors.initials: KS
authors.lastname: Hall
mesh_terms: D000293:Adolescent; D000328:Adult; D050071:Bone Density Conservation Agents / Q000627:therapeutic use*; D001859:Bone Neoplasms / Q000188:drug therapy* / Q000453:epidemiology; D016208:Databases, Factual; D000069448:Denosumab / Q000627:therapeutic use*; D005260:Female; D018212:Giant Cell Tumor of Bone / Q000188:drug therapy* / Q000453:epidemiology; D006801:Humans; D007279:Injections, Subcutaneous; D008297:Male; D008875:Middle Aged; D009664:Norway / Q000453:epidemiology; D012189:Retrospective Studies; D016896:Treatment Outcome; D055815:Young Adult
chemical_list: D050071:Bone Density Conservation Agents; D000069448:Denosumab