Response of an aggressive periosteal aneurysmal bone cyst (ABC) of the radius to denosumab therapy

Pauli, Chantal; Fuchs, Bruno; Pfirrmann, Christian W. A.; Bridge, Julia A.; Höfer, Silvia; Bode, Beata

doi:10.1186/1477-7819-12-17

Cited by 42 publications

(48 citation statements)

References 22 publications

(26 reference statements)

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“…[2] Denosumab is been recently implemented in the treatment of ABC, which reduces the formation of osteoclastic giant cells by selectively binding to the Rankl receptor. [9] About 26-56% of ABC tends to reoccur within the 1 st year of surgical treatment. Reoccurrence is mainly attributed to the inadequate removal of the tumor due to under exposure of the surgical field or difficulty in accessing the extension of the tumor.…”

Section: Discussionmentioning

confidence: 99%

Giant aneurysmal bone cyst of mandible: A case report and literature review

Hiremath¹,

Gaikwad²,

Charanthimath³

et al. 2015

JMRPS

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Aneurysmal bone cysts are infrequent in the craniofacial region. These cysts are non-neoplastic, expansile, and osteolytic in nature. Since these cysts are not lined by epithelium, they are considered as pseudocysts. Clinicopathological features and biological behavior of these cysts are enigmatic as they mimic various other pathogenetic lesions and two third of these cysts are associated with other bone pathology. Aim of the present paper was to report a case of aneurysmal bone cyst in 40-year-old female patient encountered in left mandibular posterior region which was surgically treated with hemimandibulectomy. The post-operative period was uneventful. Furthermore, emphasis was given to discuss its etiopathogenesis, clinicopathological variants, and other pathologies associated with this cyst.

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Section: Discussionmentioning

confidence: 99%

Giant aneurysmal bone cyst of mandible: A case report and literature review

Hiremath¹,

Gaikwad²,

Charanthimath³

et al. 2015

JMRPS

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“…In den letzten Jahren ist auch der Einsatz von Denosumab zur Behandlung aggressiver, lokal fortgeschrittener AKZ beschrieben worden [14,17,18]. Denosumab ist ein humaner, monoklonaler Antikör-per, der sich an den Receptor-activatorof-nuclear-factor-kappa-B(RANK)-Liganden bindet und ihn inhibiert [18].…”

Section: Denosumab-therapieunclassified

“…Bisher sind die Ergebnisse der Denosumab-Therapie bei 4 Patienten in der Literatur beschrieben [14,17,18]. Zwei Tumoren waren im Bereich der Halswirbelsäule lokalisiert, ein Tumor im Os sacrum und ein Tumor im Radius.…”

Section: Abb 2 8 Diaphysenersatz Im Bereich Der Proximalen Tibia Mitunclassified

“…Die 3 ersten Läsionen hatten eine enge Labebeziehung zum Spinalkanal und den umliegenden Nervenwurzeln, die vierte Läsion wies eine enge Lagebeziehung zum N. radialis auf. Bei allen 4 Tumoren wurde eine deutliche Größenregredienz der AKZ innerhalb von 1-4 Monaten nach Einleitung der Denosumab-Therapie beschrieben [14,17,18]. Eine operative Behandlung nach Abschluss der DenosumabTherapie konnte allerdings nur bei dem Patienten mit der AKZ des Radius durchgeführt werden [17].…”

Section: Abb 2 8 Diaphysenersatz Im Bereich Der Proximalen Tibia Mitunclassified

“…Bei allen 4 Tumoren wurde eine deutliche Größenregredienz der AKZ innerhalb von 1-4 Monaten nach Einleitung der Denosumab-Therapie beschrieben [14,17,18]. Eine operative Behandlung nach Abschluss der DenosumabTherapie konnte allerdings nur bei dem Patienten mit der AKZ des Radius durchgeführt werden [17]. Als Nebenwirkung trat bei einem Patienten eine asymptomatische Hypokalziämie auf [14].…”

Section: Abb 2 8 Diaphysenersatz Im Bereich Der Proximalen Tibia Mitunclassified

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Neue Operationsverfahren bei Knochentumoren

et al. 2014

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Primary bone neoplasms can be classified into benign, locally/aggressive and rarely metastasizing and malignant tumors. Patients with benign tumors usually undergo surgical treatment in cases of local symptoms, mainly consisting of pain or functional deficits due to compression of important anatomical structures, such as nerves or blood vessels. Locally/aggressive and rarely metastasizing tumors exhibit an infiltrative growth pattern, so that surgical treatment is necessary to prevent further destruction of bone leading to local instability. Finally, the surgical treatment of malignant tumors is, with few exceptions, considered to be a prerequisite for long-term survival, either alone or in combination with systemic chemotherapy. Whereas the main objective of surgery in the treatment of benign tumors is relief of local symptoms with a minimum amount of damage to healthy tissue and minimizing the risk of local recurrence while ensuring bone stability in locally aggressive and rarely metastasizing tumors, the primary goal in the operative treatment of bone sarcomas is the resection of the tumor with clear surgical margins followed by defect reconstruction and the preservation of function. This review examines the current developments in the surgical treatment of primary bone neoplasms with respect to the management of the tumors and novel reconstructive options.

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Use of Denosumab in Children With Osteoclast Bone Dysplasias: Report of Three Cases

et al. 2019

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Denosumab has been used successfully to treat disease‐associated osteoclast overactivity, including giant cell tumor of bone. Given its mechanism of action, denosumab is a potent potential treatment of other osteoclast bone dysplasias including central giant cell granuloma (CGCG), aneurysmal bone cyst (ABC), and cherubism. Relatively little is known about the safety and efficacy of denosumab in patients with these conditions, especially in children. We report on 3 pediatric patients treated with denosumab over a 3‐year period at UCLA Medical Center (Los Angeles and Santa Monica, CA, USA): a 12‐year‐old with recurrent ABC of the pelvis, a 14‐year‐old with CGCG of the mandible, and a 12‐year‐old with cherubism. All were started on a 1‐year course of 15 doses 120 mg s.c., given monthly with two loading doses on day 8 and 15. All patients demonstrated rapid and pronounced clinical improvement while on denosumab, including a significant reduction in pain and sclerosis of lytic lesions on radiographs. Within 1 month of initiating therapy, 2 patients experienced hypocalcemia (Common Terminology Criteria for Adverse Events [CTCAE] grade 2) and hypophosphatemia, with 1 patient experiencing symptoms. One patient went on to experience symptomatic rebound hypercalcemia (CTCAE grade 4) 5 months after completing therapy, requiring bisphosphonates and calcitonin. For the second patient, we developed a schedule to wean denosumab involving the progressive lengthening of time between doses from 1 to 4 months in 1‐month increments before cessation. We found that denosumab therapy results in significant clinical and radiographic improvement for pediatric patients with nonresectable ABC, CGCG, and cherubism. Problems with serum calcium may be more common in younger patients, with symptomatic and protracted rebound hypercalcemia after cessation of therapy the most significant. We present a potential solution to this problem with progressive spacing of doses. Potential serious adverse events from alterations in calcium homeostasis should be explored in prospective clinical trials. © 2019 The Authors. JBMR Plus published by Wiley Periodicals, Inc. on behalf of American Society for Bone and Mineral Research.

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Response of an aggressive periosteal aneurysmal bone cyst (ABC) of the radius to denosumab therapy

Cited by 42 publications

References 22 publications

Giant aneurysmal bone cyst of mandible: A case report and literature review

Giant aneurysmal bone cyst of mandible: A case report and literature review

Neue Operationsverfahren bei Knochentumoren

Use of Denosumab in Children With Osteoclast Bone Dysplasias: Report of Three Cases

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