A case of pelvic giant cell tumor of bone, complete remission with denosumab: long duration of response

Abstract

Although giant cell tumor of bone has been considered as a disease with benign course, it can lead to bone destruction and serious morbidity. A 19-year-old case was presented with hip pain. There was a recurrence after 9 months of curative surgical resection and zoledronic acid use, and as surgical morbidity would be high, antiosteoclastic receptor activator of nuclear factor kappa B ligand inhibitor denosumab treatment was administered. She had a complete remission after 18 months of denosumab treatment. The important point in the present case is that it has been followed up without recurrence after around 42 months of denosumab use and 11 months of follow-up after the cessation of drug. In recurrent cases in which nonmetastatic surgery is not suitable, the use of denosumab decreases tumor progression. The duration of use in unresectable and advanced cases still remains unclear.

Denosumab for Effective Tumor Size Reduction in Patients With Giant Cell Tumors of the Bone: A Systematic Review and Meta-Analysis

“…A critical review of these published studies identified 60 studies that met the inclusion criteria for the present meta-analysis (Table 1). 11,12,44,60 -116 The www.clinicaltrials.gov website showed 7 ongoing studies of denosumab in the treatment of GCTB. The majority of the studies examined for this meta-analysis were case reports, case series studies, and nonrandomized, uncontrolled, open-label phase II studies (Figure 2).…”

“…Most participants in the studies analyzed were from case series studies, followed by the nonrandomized, uncontrolled, openlabel phase II studies, and then case reports. 90 Tsukamoto et al 92 Matcuk et al 106 Bilgetekin et al 62 Xará-Leite et al 65 Marinova et al 68 Kinoshita et al 70 Osaka et al 71 Liu et al 74 Wu et al 84 Law et al 85 Satcher et al 87 Yonezawa et al 89 Bardakhchyan et al 91 (continued) Menon et al 95 Inoue et al 97 Cavalcante et al 99 Yamagishi et al 100 Kajiwara et al 101 Nakazawa et al Setsu et al 103 Aponte-Tinao et al 104 Park et al 105 Vaishya et al 107 Gossai et al 109 Watanabe et al 110 Mattei et al 111 Hakozaki et al 112 Aghaloo et al 113 Rossi et al 114 Akaike Abbreviation: SD, standard deviation a The majority of study participants were women, and the median age was 30. There was no significant difference in the duration of treatment and follow-up time.…”

“…120 Statistical significance for the appearance of the GCTB on different parts of the body was only observed in the lower limbs (P ¼ .014), trunk and lung (P ¼ .002) in case series studies, and in the upper limb (P ¼ .037) in case reports in this investigation. The duration of treatment Bilgetekin et al 62 (2019) Xará-Leite et al 65 (2019) Kinoshita et al 70 2019Osaka et al 71 2019Marinova et al 68 (2018) Liu et al 74 (2018) Wu et al 84 (2018) Law et al 85 (2018) Satcher et al 87 von Borstel et al 100 (2017) Bardakhchy an et al 91 (2017) Tsukamoto et al 92 (2017) Menon et al 95 2017Inoue et al 97 (2016) Cavalcante et al 99 (2016) Yamagishi et al 100 (2016) Kajiwara et al 101 (2016) Nakazawaet al 102 (2016) Setsu et al 103 2016Aponte-Tinao et al 104 2015Park et al 105 (2015) Matcuk et al 106 2015Vaishya et al 107 2015Gossai et al 109 with denosumab did not differ significantly among the reviewed studies. As was also concluded in a previously published review, optimal treatment duration remains unclear.…”

Denosumab induction and Zoledronic acid maintenance therapy for recurrent unresectable giant cell tumour of the distal tibia: A case report with sustained tumour control after drug withdrawal

“…Although the occasional use of ZA as a salvage treatment in DM non-responders has been reported [50] , the position of ZA in the treatment of GCTB next to DM remains unexplored. The reverse sequence has been studied, but ZA was most often ineffective as a first-line option, while DM allowed for long-term disease control [51] , [52] , [53] .…”