Ontology type: schema:ScholarlyArticle Open Access: True
2015-12
AUTHORSMathilde Penel-Page, Isabelle Ray-Coquard, Julie Larcade, Magali Girodet, Laure Bouclier, Muriel Rogasik, Nadège Corradini, Natacha Entz-Werle, Laurence Brugieres, Julien Domont, Cyril Lervat, Sophie Piperno-Neumann, Helène Pacquement, Jacques-Olivier Bay, Jean-Claude Gentet, Antoine Thyss, Loic Chaigneau, Bérangère Narciso, Helène Cornille, Jean-Yves Blay, Perrine Marec-Bérard
ABSTRACTBACKGROUND: The objective of this study is to explore the off-label use of targeted therapies (TTs) for patients with osteosarcoma registered within the French Sarcoma Group--Bone Tumor Study Group (GSF-GETO) national registry. METHODS: All patients with an osteosarcoma, registered between January 1, 2009 and July 15, 2013 were analyzed. RESULTS: Twenty-nine patients with refractory relapsed osteosarcomas received 33 treatment lines of TTs. The median age at the beginning of treatment was 19 years (range 9-72). The median number of previous lines of chemotherapy was 3 (range 1-8). Before inclusion, 3 patients were in second complete remission, 26 were in progression for metastatic relapse. Twenty-three patients received sirolimus (in combination with cyclophosphamide for 18); 5, sunitinib; 4, sorafenib; and one, pazopanib. Stable disease was observed for 45.5% of patients (95% Confidence Interval (CI) [20-52.8]). The median Progression-Free Survival (PFS) was 3 months (95% CI [2-5.4]) for patients treated by sirolimus and 1.8 months (95% CI [1.3-2.8]) for patients receiving multi-targeted tyrosine kinase inhibitors; 6-month PFS 15%. The median Overall Survival (OS) was 6.8 months (95% CI [4.7-12.1]), and one-year OS was 24%. In a multivariate analysis, PFS was superior for patients receiving sirolimus compared to other TTs (Hazard Ratio (HR) = 2.7, 95% CI [1.05-7.1]). No toxic death was reported. Grade 3 and 4 toxicities were observed in 27 and 6% of cases respectively. CONCLUSION: Off-label TTs, especially sirolimus, reported benefit in the treatment of refractory osteosarcomas with an acceptable toxicity profile, including in pediatric population. More... »
PAGES854
http://scigraph.springernature.com/pub.10.1186/s12885-015-1894-5
DOIhttp://dx.doi.org/10.1186/s12885-015-1894-5
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PUBMEDhttps://www.ncbi.nlm.nih.gov/pubmed/26541413
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