Mineral and Bone Consequences of High Dose Denosumab Therapy to Treat an Aneurysmal Bone Cyst, a Child Case Report
Aneurysmal bone cysts (ABCs) are rare benign pseudotumoral bone lesions with potential aggressive behavior due to the extensive destruction of surrounding bone. Traditionally, these tumors were treated with open surgery, but there is more and more a shift to less invasive procedures. In particular,...
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doaj-5ecff5de04874af9b45e5ddafa758ddc2021-07-16T08:34:21ZengFrontiers Media S.A.Frontiers in Endocrinology1664-23922021-07-011210.3389/fendo.2021.698963698963Mineral and Bone Consequences of High Dose Denosumab Therapy to Treat an Aneurysmal Bone Cyst, a Child Case ReportGiulia Del Sindaco0Giulia Del Sindaco1Giulia Del Sindaco2Pablo Berlanga3Laurence Brugières4Eric Thebault5Giovanna Mantovani6Giovanna Mantovani7Philippe Wicart8Philippe Wicart9Agnès Linglart10Agnès Linglart11Agnès Linglart12Endocrinology Unit, Fondazione IRCCS Ca’ Granda Ospedale Maggiore Policlinico, ERN BOND, Milan, ItalyDepartment of Clinical Sciences and Community Health, University of Milan, Milan, ItalyAP-HP, Service d’endocrinologie et diabète de l’enfant, ERN BOND, ERN for rare endocrine disorders, et Plateforme d’expertise des maladies rares, Hôpital Bicêtre Paris Saclay, Le Kremlin-Bicêtre, FranceDepartment of Pediatric and Adolescent Oncology, Gustave Roussy Cancer Center, Paris-Saclay University, Villejuif, FranceDepartment of Pediatric and Adolescent Oncology, Gustave Roussy Cancer Center, Paris-Saclay University, Villejuif, FranceDepartment of Pediatric and Adolescent Oncology, Gustave Roussy Cancer Center, Paris-Saclay University, Villejuif, FranceEndocrinology Unit, Fondazione IRCCS Ca’ Granda Ospedale Maggiore Policlinico, ERN BOND, Milan, ItalyDepartment of Clinical Sciences and Community Health, University of Milan, Milan, ItalyAP-HP, Centre de Référence des maladies rares du métabolisme du Calcium et du Phosphate, filière OSCAR, Paris, FranceAP-HP, Department of Pediatric Orthopedic Surgery, Necker - Enfants Malades University Hospital, Paris, France. Paris Descartes University, Paris, FranceAP-HP, Service d’endocrinologie et diabète de l’enfant, ERN BOND, ERN for rare endocrine disorders, et Plateforme d’expertise des maladies rares, Hôpital Bicêtre Paris Saclay, Le Kremlin-Bicêtre, FranceAP-HP, Centre de Référence des maladies rares du métabolisme du Calcium et du Phosphate, filière OSCAR, Paris, FranceUniversité de Paris Saclay, INSERM, U1185, Le Kremlin-Bicêtre, FranceAneurysmal bone cysts (ABCs) are rare benign pseudotumoral bone lesions with potential aggressive behavior due to the extensive destruction of surrounding bone. Traditionally, these tumors were treated with open surgery, but there is more and more a shift to less invasive procedures. In particular, treatment for spinal ABCs is generally unsatisfactory due to the risk of morbidity, neurological impairment and recurrence, and there is a need for innovative therapies. Denosumab has been reported as a useful treatment in giant cell tumors of bone (GCTB), so its efficacy has been tested also in other fibro-osseus lesions affecting children and adolescents, such as spinal aneurysmal bone cysts. The pediatric literature is limited to case reports and small series, all of which highlight the efficacy of this treatment on lesions growth and associated bone pain. Some of these reports have already reported well known side effects associated with denosumab, such as hypocalcemia at the beginning of the treatment, and rebound hypercalcemia at the discontinuation. The latter seems to be more frequent in children and adolescents than in adults, probably due to the higher baseline bone turnover in children. In addition, the use of denosumab in young patients could affect both bone modeling and remodeling, even if the consequences on the growing skeleton have not been reported in detail. Here we describe the case of a spinal ABC diagnosed in an 8-year old young boy which was not accessible to surgery but responded favorably to denosumab. Our aim is to describe the rapid changes in mineral and bone homeostasis in this patient, that required advice from the experts of the European Reference Network (ERN) for rare bone and endocrine diseases.https://www.frontiersin.org/articles/10.3389/fendo.2021.698963/fulldenosumabaneurysmal bone cysthypercalcemiabisphosphonatebone modeling |
collection |
DOAJ |
language |
English |
format |
Article |
sources |
DOAJ |
author |
Giulia Del Sindaco Giulia Del Sindaco Giulia Del Sindaco Pablo Berlanga Laurence Brugières Eric Thebault Giovanna Mantovani Giovanna Mantovani Philippe Wicart Philippe Wicart Agnès Linglart Agnès Linglart Agnès Linglart |
spellingShingle |
Giulia Del Sindaco Giulia Del Sindaco Giulia Del Sindaco Pablo Berlanga Laurence Brugières Eric Thebault Giovanna Mantovani Giovanna Mantovani Philippe Wicart Philippe Wicart Agnès Linglart Agnès Linglart Agnès Linglart Mineral and Bone Consequences of High Dose Denosumab Therapy to Treat an Aneurysmal Bone Cyst, a Child Case Report Frontiers in Endocrinology denosumab aneurysmal bone cyst hypercalcemia bisphosphonate bone modeling |
author_facet |
Giulia Del Sindaco Giulia Del Sindaco Giulia Del Sindaco Pablo Berlanga Laurence Brugières Eric Thebault Giovanna Mantovani Giovanna Mantovani Philippe Wicart Philippe Wicart Agnès Linglart Agnès Linglart Agnès Linglart |
author_sort |
Giulia Del Sindaco |
title |
Mineral and Bone Consequences of High Dose Denosumab Therapy to Treat an Aneurysmal Bone Cyst, a Child Case Report |
title_short |
Mineral and Bone Consequences of High Dose Denosumab Therapy to Treat an Aneurysmal Bone Cyst, a Child Case Report |
title_full |
Mineral and Bone Consequences of High Dose Denosumab Therapy to Treat an Aneurysmal Bone Cyst, a Child Case Report |
title_fullStr |
Mineral and Bone Consequences of High Dose Denosumab Therapy to Treat an Aneurysmal Bone Cyst, a Child Case Report |
title_full_unstemmed |
Mineral and Bone Consequences of High Dose Denosumab Therapy to Treat an Aneurysmal Bone Cyst, a Child Case Report |
title_sort |
mineral and bone consequences of high dose denosumab therapy to treat an aneurysmal bone cyst, a child case report |
publisher |
Frontiers Media S.A. |
series |
Frontiers in Endocrinology |
issn |
1664-2392 |
publishDate |
2021-07-01 |
description |
Aneurysmal bone cysts (ABCs) are rare benign pseudotumoral bone lesions with potential aggressive behavior due to the extensive destruction of surrounding bone. Traditionally, these tumors were treated with open surgery, but there is more and more a shift to less invasive procedures. In particular, treatment for spinal ABCs is generally unsatisfactory due to the risk of morbidity, neurological impairment and recurrence, and there is a need for innovative therapies. Denosumab has been reported as a useful treatment in giant cell tumors of bone (GCTB), so its efficacy has been tested also in other fibro-osseus lesions affecting children and adolescents, such as spinal aneurysmal bone cysts. The pediatric literature is limited to case reports and small series, all of which highlight the efficacy of this treatment on lesions growth and associated bone pain. Some of these reports have already reported well known side effects associated with denosumab, such as hypocalcemia at the beginning of the treatment, and rebound hypercalcemia at the discontinuation. The latter seems to be more frequent in children and adolescents than in adults, probably due to the higher baseline bone turnover in children. In addition, the use of denosumab in young patients could affect both bone modeling and remodeling, even if the consequences on the growing skeleton have not been reported in detail. Here we describe the case of a spinal ABC diagnosed in an 8-year old young boy which was not accessible to surgery but responded favorably to denosumab. Our aim is to describe the rapid changes in mineral and bone homeostasis in this patient, that required advice from the experts of the European Reference Network (ERN) for rare bone and endocrine diseases. |
topic |
denosumab aneurysmal bone cyst hypercalcemia bisphosphonate bone modeling |
url |
https://www.frontiersin.org/articles/10.3389/fendo.2021.698963/full |
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