A novel approach to the management of a central giant cell granuloma with denosumab: A case report and review of current treatments

Gupta, Benjamin; Stanton, Norman; Coleman, Hedley; White, Curt M.; Singh, Jasvir

doi:10.1016/j.jcms.2015.04.011

Cited by 34 publications

(36 citation statements)

References 37 publications

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“…In the 3 patients described here, hypocalcemia and hypercalcemia were the primary complications noted. In the 22 other published cases of off-label denosumab use in pediatric and adult patients (Table 1), (7,(9)(10)(11)(12)(13)(14)(15)(16)(17)(18)(19)(20)(21) complications described include symptomatic and asymptomatic hyper-calcemia (2 patients, 1 with FD, 1 with CGCG), asymptomatic hypocalcemia (3 patients, 2 with FD, 1 with ABC), asymptomatic hypophosphatemia (3 patients with FD), extremity/ skeletal pain (1 patient with CGCG), and delayed wound healing (1 patient with CGCG). In the only other case of symptomatic hypercalcemia described previously, also in a pediatric patient, the hypercalcemia was preceded by hypophosphatemia and increased serum PTH upon initiation of therapy.…”

Section: Discussionmentioning

confidence: 99%

“…No clinical trials of denosumab have been published, and there are no ongoing clinical trials listed on Clinicaltrials.gov for these groups of patients. Cases have been reported for the use of denosumab in ABC, (7,(9)(10)(11)(12)(13) CGCG, (14)(15)(16)(17)(18) cherubism, (19) and FD (20,21) for 8, 10, 1, and 3 patients, respectively. Of these, 5 patients were <16 years old when they began treatment.…”

Section: Introductionmentioning

confidence: 99%

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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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Section: Discussionmentioning

confidence: 99%

Section: Introductionmentioning

confidence: 99%

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

et al. 2019

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“…Intralesional triamcinolone, alone or combined with denosumab, or interferon, was used to reverse the osteolytic effect of CGCG [4][5][6][7]. Other clinicians, however, have reported inaccurate information about treated cases of CGCG [8][9][10].…”

Section: Discussionmentioning

confidence: 99%

Idle gossip about validity of intralesional steroid injection in treating central giant cell granuloma

Alerraqi¹

2016

Horm Stud

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Treating central giant cell granuloma (CGCG) via intralesional injection of steroids was proven effective in some cases. Later, clinicians have preferred this treatment modality to surgical intervention for it avoids gnathic/bony disfigurement. Advocating this non-surgical therapy, several papers and reviews have recommended putting it into action as the first line of treatment, alone or combined with other treatment options, especially in treating aggressive CGCG. Other authors have reported inaccurate information to validate this approach. This paper scrutinizes, in retrospect, the infelicities about treating CGCG via intralesional injection of steroids and concludes that intralesional steroid injection is useful ONLY in treating non-aggressive CGCG and peripheral giant cell granulomas.

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“…At the time of the writing of this manuscript, denosumab had been used in a total of 11 cases reported in the English literature thus far. [20][21][22][23] Antiresorptive therapy is associated with the risk of osteonecrosis of the jaws (ONJs). [24] The frequency of ONJ in oncology patients receiving oncologic doses of bisphosphonate or denosumab is estimated at 1%-15%.…”

Section: Discussionmentioning

confidence: 99%

Denosumab Treatment for Aggressive Multiple Recurrent Familial Central Giant Cell Granulomas

Ottavainen

Rytkönen²,

Uusitalo³

et al. 2014

Journal of Oral and Maxillofacial Surgery

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Background: Aggressive familial giant-cell granulomas of the jaws can be severely deforming. Surgical and nonsurgical treatments may be associated with multiple recurrences. Denosumab, a new generation antiresorptive drug, is an osteoclast inhibitor, which may be particularly useful to manage such potentially disfiguring lesions. Materials and Methods: Two sisters, both with a history of multiple recurrent aggressive central giant-cell granuloma (CGCG)-like lesions in both jaws, were referred for management. All lesions were histologically consistent with the diagnosis of CGCG. The lesions were treated surgically with curettage and perilesional injection of triamcinolone. In particular, the older sister had four separate anatomic sites where some of her lesions had multiple recurrences necessitating three repeat procedures. A course of subcutaneous denosumab was administered following the last giant-cell granuloma removal in both sisters. Results: Bony healing was normal. No further recurrences were observed over 3.5 years of follow-up after denosumab therapy in either sister. Conclusions: In this small cohort comprising two sisters with multiple aggressive recurrent giant-cell granuloma lesions at multiple sites in the mouth, subcutaneous denosumab administration was associated with success over 3.5 years of follow-up. This report cautiously adds to the clinical experience in the use of denosumab for the treatment of recurrent aggressive familial CGCG lesions.

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A novel approach to the management of a central giant cell granuloma with denosumab: A case report and review of current treatments

Cited by 34 publications

References 37 publications

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

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

Idle gossip about validity of intralesional steroid injection in treating central giant cell granuloma

Denosumab Treatment for Aggressive Multiple Recurrent Familial Central Giant Cell Granulomas

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