Surgical Management and Denosumab for Aneurysmal Bone Cysts of the Spine in an Australian Tertiary Paediatric Centre.

Autor: Vanderniet JA; Institute of Endocrinology and Diabetes, The Children's Hospital at Westmead, Locked Bag 4001, Westmead, Sydney, NSW, 2145, Australia. joel.vanderniet@health.nsw.gov.au., Tsinas D; Department of Orthopaedics, The Children's Hospital at Westmead, Sydney, NSW, Australia., Wall CL; Institute of Endocrinology and Diabetes, The Children's Hospital at Westmead, Locked Bag 4001, Westmead, Sydney, NSW, 2145, Australia., Girgis CM; Faculty of Medicine and Health, University of Sydney, Sydney, NSW, Australia.; Department of Endocrinology, Royal North Shore Hospital, Sydney, NSW, Australia.; Department of Diabetes and Endocrinology, Westmead Hospital, Sydney, NSW, Australia., London K; Department of Nuclear Medicine, The Children's Hospital at Westmead, Sydney, NSW, Australia., Keane C; Department of Orthopaedics, The Children's Hospital at Westmead, Sydney, NSW, Australia., Briody J; Department of Nuclear Medicine, The Children's Hospital at Westmead, Sydney, NSW, Australia., Hibbert S; Department of Dentistry, The Children's Hospital at Westmead, Sydney, NSW, Australia.; Department of Paediatric Dentistry, Westmead Centre for Oral Health, Sydney, NSW, Australia., Poon M; Institute of Endocrinology and Diabetes, The Children's Hospital at Westmead, Locked Bag 4001, Westmead, Sydney, NSW, 2145, Australia., Padhye B; Faculty of Medicine and Health, University of Sydney, Sydney, NSW, Australia.; Cancer Centre for Children, The Children's Hospital at Westmead, Sydney, NSW, Australia., Biggin A; Institute of Endocrinology and Diabetes, The Children's Hospital at Westmead, Locked Bag 4001, Westmead, Sydney, NSW, 2145, Australia.; Faculty of Medicine and Health, University of Sydney, Sydney, NSW, Australia., Dalla-Pozza L; Faculty of Medicine and Health, University of Sydney, Sydney, NSW, Australia.; Cancer Centre for Children, The Children's Hospital at Westmead, Sydney, NSW, Australia., Gray RJ; Department of Orthopaedics, The Children's Hospital at Westmead, Sydney, NSW, Australia., Munns CF; Child Health Research Centre, The University of Queensland, Brisbane, QLD, Australia.; Mayne Academy of Paediatrics, The University of Queensland, Brisbane, QLD, Australia.
Jazyk: angličtina
Zdroj: Calcified tissue international [Calcif Tissue Int] 2023 May; Vol. 112 (5), pp. 592-602. Date of Electronic Publication: 2023 Feb 21.
DOI: 10.1007/s00223-023-01068-1
Abstrakt: Aneurysmal bone cysts (ABC) are rare osteolytic, benign but often locally aggressive tumours of the long bones or vertebrae. For spinal ABC, surgical management, embolisation or sclerotherapy alone often carry high morbidity and/or high recurrence rates. Interruption of receptor activator of nuclear factor-kappa B ligand (RANKL) signalling holds promise as an effective therapeutic strategy for these tumours. We aimed to review the approach to surgical management and evaluate the efficacy and safety of denosumab for ABC of the spine in children. Retrospective review of 7 patients treated with denosumab using a standardised protocol for ABC of the spine in a tertiary paediatric centre. Surgical intervention was only conducted if there was spinal instability or significant neurological impairment. Denosumab 70 mg/m 2 was given 4-weekly for at least 6 months, followed by 2 doses of zoledronate 0.025 mg/kg, aiming to prevent rebound hypercalcaemia. All patients achieved stability of the spine and resolution of neurological impairment, if present. Six patients achieved metabolic remission and have ceased denosumab without recurrence to date; the other showed clinical and radiological improvement without complete metabolic remission. Three patients developed symptomatic hypercalcaemia 5-7 months after cessation of denosumab, requiring additional bisphosphonate treatment. We present our algorithm for the surgical and medical management of paediatric spinal ABC. Denosumab produced a radiological and metabolic response in all patients, with complete remission in most. Follow-up time was not long enough to evaluate the endurance of response after cessation in some patients. Incidence of rebound hypercalcaemia in this paediatric cohort was high, prompting a change to our protocol.
(© 2023. Crown.)
Databáze: MEDLINE
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