Short-term and long-term prognostic value of histological response and intensified chemotherapy in osteosarcoma: a retrospective reanalysis of the BO06 trial

Objectives Cure rate models accounting for cured and uncured patients, provide additional insights into long and short-term survival. We aim to evaluate the prognostic value of histological response and chemotherapy intensification on the cure fraction and progression-free survival (PFS) for the unc...

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Main Authors: Jakob Anninga, Hans Gelderblom, Marta Fiocco, Nan van Geloven, Eni Musta
Format: Article
Language:English
Published: BMJ Publishing Group 2022-05-01
Series:BMJ Open
Online Access:https://bmjopen.bmj.com/content/12/5/e052941.full
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author Jakob Anninga
Hans Gelderblom
Marta Fiocco
Nan van Geloven
Eni Musta
author_facet Jakob Anninga
Hans Gelderblom
Marta Fiocco
Nan van Geloven
Eni Musta
author_sort Jakob Anninga
collection DOAJ
description Objectives Cure rate models accounting for cured and uncured patients, provide additional insights into long and short-term survival. We aim to evaluate the prognostic value of histological response and chemotherapy intensification on the cure fraction and progression-free survival (PFS) for the uncured patients.Design Retrospective analysis of a randomised controlled trial, MRC BO06 (EORTC 80931).Setting Population-based study but proposed methodology can be applied to other trial designs.Participants A total of 497 patients with resectable highgrade osteosarcoma, of which 118 were excluded because chemotherapy was not started, histological response was not reported, abnormal dose was reported or had disease progression during treatment.Intervention(s) Two regimens with the same anticipated cumulative dose (doxorubicin 6×75 mg/m2/week; cisplatin 6×100 mg/m2/week) over different time schedules: every 3 weeks in regimen-C and every 2 weeks in regimen-DI.Primary and secondary outcome measures The primary outcome is PFS computed from end of treatment because cure, if it occurs, may happen at any time during treatment. A mixture cure model is used to study the effect of histological response and intensified chemotherapy on the cure status and PFS for the uncured patients.Results Histological response is a strong prognostic factor for the cure status (OR 3.00, 95% CI 1.75 to 5.17), but it has no clear effect on PFS for the uncured patients (HR 0.78, –95% CI 0.53 to 1.16). The cure fractions are 55% (46%–63%) and 29% (22%–35%), respectively, among patients with good and poor histological response (GR, PR). The intensified regimen was associated with a higher cure fraction among PR (OR 1.90, 95% CI 0.93 to 3.89), with no evidence of effect for GR (OR 0.78, 95% CI 0.38 to 1.59).Conclusions Accounting for cured patients is valuable in distinguishing the covariate effects on cure and PFS. Estimating cure chances based on these prognostic factors is relevant for counselling patients and can have an impact on treatment decisions.Trial registration number ISRCTN86294690.
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spelling doaj-art-58a5d42cdf1540f396f44f0a5c694eaf2025-08-20T02:16:56ZengBMJ Publishing GroupBMJ Open2044-60552022-05-0112510.1136/bmjopen-2021-052941Short-term and long-term prognostic value of histological response and intensified chemotherapy in osteosarcoma: a retrospective reanalysis of the BO06 trialJakob Anninga0Hans Gelderblom1Marta Fiocco2Nan van Geloven3Eni Musta4Department of Solid Tumors, Princess Máxima Center for Pediatric Oncology, Utrecht, Netherlands1 Department of Medical Oncology, Leiden Universitair Medisch Centrum, Leiden, Zuid-Holland, The Netherlands6 Department of Biomedical Data Sciences, Section of Medical Statistics and Bioinformatics, Leiden Universitair Medisch Centrum, Leiden, Zuid-Holland, The NetherlandsDepartment of Biomedical Data Sciences, Leiden University Medical Center, Leiden, The NetherlandsKorteweg-de Vries Institute for Mathematics, University of Amsterdam, Amsterdam, The NetherlandsObjectives Cure rate models accounting for cured and uncured patients, provide additional insights into long and short-term survival. We aim to evaluate the prognostic value of histological response and chemotherapy intensification on the cure fraction and progression-free survival (PFS) for the uncured patients.Design Retrospective analysis of a randomised controlled trial, MRC BO06 (EORTC 80931).Setting Population-based study but proposed methodology can be applied to other trial designs.Participants A total of 497 patients with resectable highgrade osteosarcoma, of which 118 were excluded because chemotherapy was not started, histological response was not reported, abnormal dose was reported or had disease progression during treatment.Intervention(s) Two regimens with the same anticipated cumulative dose (doxorubicin 6×75 mg/m2/week; cisplatin 6×100 mg/m2/week) over different time schedules: every 3 weeks in regimen-C and every 2 weeks in regimen-DI.Primary and secondary outcome measures The primary outcome is PFS computed from end of treatment because cure, if it occurs, may happen at any time during treatment. A mixture cure model is used to study the effect of histological response and intensified chemotherapy on the cure status and PFS for the uncured patients.Results Histological response is a strong prognostic factor for the cure status (OR 3.00, 95% CI 1.75 to 5.17), but it has no clear effect on PFS for the uncured patients (HR 0.78, –95% CI 0.53 to 1.16). The cure fractions are 55% (46%–63%) and 29% (22%–35%), respectively, among patients with good and poor histological response (GR, PR). The intensified regimen was associated with a higher cure fraction among PR (OR 1.90, 95% CI 0.93 to 3.89), with no evidence of effect for GR (OR 0.78, 95% CI 0.38 to 1.59).Conclusions Accounting for cured patients is valuable in distinguishing the covariate effects on cure and PFS. Estimating cure chances based on these prognostic factors is relevant for counselling patients and can have an impact on treatment decisions.Trial registration number ISRCTN86294690.https://bmjopen.bmj.com/content/12/5/e052941.full
spellingShingle Jakob Anninga
Hans Gelderblom
Marta Fiocco
Nan van Geloven
Eni Musta
Short-term and long-term prognostic value of histological response and intensified chemotherapy in osteosarcoma: a retrospective reanalysis of the BO06 trial
BMJ Open
title Short-term and long-term prognostic value of histological response and intensified chemotherapy in osteosarcoma: a retrospective reanalysis of the BO06 trial
title_full Short-term and long-term prognostic value of histological response and intensified chemotherapy in osteosarcoma: a retrospective reanalysis of the BO06 trial
title_fullStr Short-term and long-term prognostic value of histological response and intensified chemotherapy in osteosarcoma: a retrospective reanalysis of the BO06 trial
title_full_unstemmed Short-term and long-term prognostic value of histological response and intensified chemotherapy in osteosarcoma: a retrospective reanalysis of the BO06 trial
title_short Short-term and long-term prognostic value of histological response and intensified chemotherapy in osteosarcoma: a retrospective reanalysis of the BO06 trial
title_sort short term and long term prognostic value of histological response and intensified chemotherapy in osteosarcoma a retrospective reanalysis of the bo06 trial
url https://bmjopen.bmj.com/content/12/5/e052941.full
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