The management of osteosarcoma in children and adolescents in a resource-limited setting: quality improvement considerations to improve treatment outcomes

Abstract

Background The survival rates for children and adolescents with osteosarcoma in low-income countries are poor. Insufficient data regarding the challenges of managing osteosarcoma in resource-limited settings has been published. We evaluated the treatment of osteosarcoma in children and adolescents with the aim of improving the health system and management outcomes. Methods We sourced data on children under 18 years treated for osteosarcoma at the Uganda Cancer Institute between January 2016 and December 2020. Descriptive statistics and Kaplan-Meier survival analysis were used. Results Seventy-four osteosarcoma cases were identified, with a median age of 13 years (IQR 9.8–15). Referrals were made after a median of 28 days (range 1-147). Before appropriate referral, more than a quarter (26%) had undergone invasive procedures that could compromise tumour integrity and outcome. Half (50%) of the patients had metastatic disease at diagnosis, primarily to the lungs (n=43; 92%). Only 14 (33%) patients received neoadjuvant chemotherapy. Forty-three (58.1%) patients underwent limb amputation surgery, including 25 localized tumours and 18 patients with distant metastatic disease. No metastatectomies were performed. Adjuvant chemotherapy was delayed for longer than 21 days in 26 (61%) patients. No pathology reports described the status of resection margins or the degree of chemotherapy-induced necrosis. Twenty-six (35%) patients abandoned treatment, mainly due to pending radical surgery (n=18/26; 69%). Only 18% (n=13) were still alive; 46% (n=34) had died; and 37% (n=27) had an unknown status. The median overall survival was 1.1 years, and was significantly negatively affected by disease metastasis, timing of adjuvant therapy, and treatment abandonment. Conclusions Osteosarcoma outcomes for children and adolescents at the Uganda Cancer Institute are extremely poor. The quality of care can be improved by addressing delayed referrals, high rates of prior manipulative therapy, metastatic disease, treatment abandonment, surgical challenges, and delayed resumption of adjuvant chemotherapy. Keywords Osteosarcoma, Children, Adolescents, Low- and middle-income countries, Resource-limited setting, Uganda