Role of Denosumab in Endoscopic Endonasal Treatment for Juvenile Clival Giant Cell Tumor: A Case Report and Review of the Literature

Inoue, Akihiro; Ohnishi, Takanori; Kohno, Shohei; Nishikawa, Masahiro; Nishida, Naoya; Ohue, Shiro

doi:10.1016/j.wneu.2016.04.054

Cited by 25 publications

(36 citation statements)

References 23 publications

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“…Based on the experience in our patient and other similar reports [3, 16–19, 36] where denosumab was effective in reducing GCT size and brought to elimination of 90% or more giant cells we would now use denosumab upfront in order to reduce tumor size in similar situations. However, such an approach is only feasible, if surgery can be postponed and local aggressiveness of the tumor does not urge for acute surgical intervention.…”

Section: Resultsmentioning

confidence: 88%

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Denosumab treatment for progressive skull base giant cell tumor of bone in a 14 year old female – a case report and literature review

et al. 2017

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BackgroundGiant cell tumor of bone (GCT) is a rare primary bone tumor, which can metastasize and undergo malignant transformation. The standard treatment of GCT is surgery. In patients with unresectable or metastatic disease, additional therapeutic options are available. These include blocking of the receptor activator of NF-kappa B ligand (RANKL) signaling pathway, which plays a role in the pathogenesis of GCT of bone, via the anti-RANKL monoclonal antibody denosumab.Case PresentationHerein we report on a female teenager who presented in a very poor clinical condition (cachexia, diplopia, strabismus, dysphonia with palsy of cranial nerves V, VI, VIII, IX, X, XI and XII) due to progressive disease, after incomplete resection and adjuvant radiotherapy, of a GCT which affected the cervical spine (C1 and C2) as well as the skull base; and who had an impressive clinical response to denosumab therapy. To the best of our knowledge, this is the youngest patient ever reported with a skull base tumor treated with denosumab.ConclusionIn situations when surgery can be postponed and local aggressiveness of the tumor does not urge for acute surgical intervention, upfront use of denosumab in order to reduce the tumor size might be considered. Principally, the goal of denosumab therapy is to reduce tumor size as much as possible, with the ultimate goal to make local surgery (or as in our case re-surgery) amenable. However, improvement in quality of life, as demonstrated in our patient, is also an important aspect of such targeted therapies.Electronic supplementary materialThe online version of this article (doi:10.1186/s13052-017-0353-0) contains supplementary material, which is available to authorized users.

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Section: Resultsmentioning

confidence: 88%

“…The first and the only report describing beneficial effects of denosumab in the treatment of the skull base GCT was published recently by Inoue and colleagues, describing a 16 years-old male with a relapsed GCT of the skull base treated with denosumab after failure of the surgery, resulting in marked reduction in tumor size [36]. …”

Section: Discussionmentioning

confidence: 99%

Denosumab treatment for progressive skull base giant cell tumor of bone in a 14 year old female – a case report and literature review

et al. 2017

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“…be useful for recurrent or unresectable skull GCTs. 13,16,30,40 A phase II trial of RANKL by Branstetter et al 5 demonstrated a decrease of ≥ 90% giant cells and a reduction in stromal cells. In summary, we advised reoperation plus radiotherapy (dose ≥ 45 Gy) as an optimal choice for radiosensitive and resectable recurrent lesions and chemical therapy with Adriamycin/denosumab/bisphosphonate as salvage treatment for unresectable and radioresistant lesions; however, future studies are still needed.…”

Section: Proposed Treatment Algorithmmentioning

confidence: 99%

Surgical management and long-term outcomes of intracranial giant cell tumors: a single-institution experience with a systematic review

Weng

Wang

et al. 2019

Journal of Neurosurgery

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OBJECTIVEIntracranial giant cell tumors (GCTs) are extremely rare neoplasms with dismal survival and recurrence rates. The authors aimed to confirm independent adverse factors for progression-free survival (PFS) and to propose an optimal treatment algorithm.METHODSThe authors reviewed the clinical data of 43 cases of intracranial GCTs in their series. They also reviewed 90 cases of previously reported GCTs in the English language between 1982 and 2017 using Ovid MEDLINE, Embase, PubMed, and Cochrane databases with keywords of “giant cell tumor” or “osteoclastoma” and “skull,” “skull base,” “temporal,” “frontal,” “sphenoid,” or “occipital.” These prior publication data were processed and used according to PRISMA (Preferred Reporting Items for Systematic Reviews and Meta-Analyses) guidelines. Aforementioned risk factors for the authors’ series and the pooled cases were evaluated in patients not lost to follow-up (m = 38 and n = 128, respectively).RESULTSThe authors’ cohort included 28 males and 15 females with a mean age of 30.5 years. Gross-total resection (GTR) was achieved in 15 (34.9%) patients. Fifteen patients (39.5%) who did not undergo GTR received postoperative radiotherapy with a mean total dose of 54.7 ± 4.1 Gy. After a mean follow-up of 71.3 months, 12 (31.6%) patients experienced recurrence, and 4 (10.5%) died of disease. The actuarial 5-year PFS and overall survival (OS) were 68.6% and 90.0% in the authors’ cohort, respectively. A multivariate Cox regression analysis verified that partial resection (HR 7.909, 95% CI 2.296–27.247, p = 0.001), no radiotherapy (HR 0.114, 95% CI 0.023–0.568, p = 0.008), and Ki-67 ≥ 10% (HR 7.816, 95% CI 1.584–38.575, p = 0.012) were independent adverse factors for PFS. Among the 90 cases in the literature, GTR was achieved in 49 (54.4%) cases. Radiotherapy was administered to 33 (36.7%) patients with a mean total dose of 47.1 ± 5.6 Gy. After a mean follow-up of 31.5 months, recurrence and death occurred in 17 (18.9%) and 5 (5.6%) cases, respectively. Among the pooled cases, the 5-year PFS and OS were 69.6% and 89.2%, respectively. A multivariate model demonstrated that partial resection (HR 4.792, 95% CI 2.909–7.893, p < 0.001) and no radiotherapy (HR 0.165, 95% CI 0.065–0.423, p < 0.001) were independent adverse factors for poor PFS.CONCLUSIONSGTR and radiotherapy were independent favorable factors for PFS of intracranial GCTs. Based on these findings, GTR alone or GTR plus radiotherapy was advocated as an optimal treatment; otherwise, partial resection plus radiotherapy with a dose ≥ 45 Gy, if tolerable, was a secondary alternative. Lack of randomized data of the study was stressed, and future studies with larger cohorts are necessary to verify these findings.Systematic review no.: CRD42018090878 (crd.york.ac.uk/PROSPERO/)

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“…In one of these reports, denosumab was given after tumor regrowth in combination with proton therapy [32]. In another, it was administered after early tumor regrowth was identified [33]. In yet another case, similar to the one we present, denosumab was given immediately after an incomplete surgical resection, with no evidence of recurrent disease at 7 months [34].…”

Section: Discussionmentioning

confidence: 67%

“…Neoadjuvant denosumab therapy for preoperative tumor downstaging has shown to decrease patient morbidity and frequency of surgical management [26,31]. Various case reports have identified regression of sellar lesions and a decrease in associated symptoms after initiating denosumab therapy [32][33][34]. In one of these reports, denosumab was given after tumor regrowth in combination with proton therapy [32].…”

Section: Discussionmentioning

confidence: 99%

Sphenoid Sinus Giant Cell Tumor: A Case Report and Literature Review

Pousti

Andera

Haugen

2020

ORL

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We describe a 27-year-old female with a giant cell tumor of her sphenoid sinus, presenting with nasal obstruction and cranial neuropathies. Both the surgical and subsequent medical management are reviewed. Additionally, we review the overall presentation, pathophysiology, and management of giant cell tumors of the skull base. Current treatment recommendations are presented.A 27-year-old female presented to our institution with a 1-month history of retro-orbital headache, neck pain, nasal obstruction, and blurred vision. CT head identified a sphenoid mass with extension into the sella, clivus, and ethmoid sinuses ( Fig. 1). MRI outlined a clival mass extending into the sphenoid sinus and cavernous sinus with a mass effect on the contents of the sella turcica (Fig. 2). An endoscopic endonasal biopsy was performed with pathology consistent with GCTb (Fig. 3).The patient then presented to the emergency department 5 days later with new-onset diplopia of the right eye, nasal drainage, and worsening headache. She was found to have a right-sided cranial nerve VI palsy. MRI showed invasion into the cavernous sinus with encroachment on the optic chiasm and internal carotid arteries bilaterally. ConclusionGiant cell tumors of the skull base are rare, benign, and locally aggressive, with high recurrence rates. The age of presentation is usually between 20 and 40 years with a predominance in females. The presenting clinical symptoms vary widely depending on tumor location. Hearing loss, headache, tinnitus, and dysfunction of the cranial nerves are the usual modes of presentation in skull base involvement. Surgical excision remains the treatment of choice, yet due to the possibility of incomplete resection and high recurrence rates, adjuvant therapy with denosumab may be necessary to improve local control. Our report, in combination with previous reports, provides evidence to support this as a potential new standard of care.

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Role of Denosumab in Endoscopic Endonasal Treatment for Juvenile Clival Giant Cell Tumor: A Case Report and Review of the Literature

Cited by 25 publications

References 23 publications

Denosumab treatment for progressive skull base giant cell tumor of bone in a 14 year old female – a case report and literature review

Denosumab treatment for progressive skull base giant cell tumor of bone in a 14 year old female – a case report and literature review

Surgical management and long-term outcomes of intracranial giant cell tumors: a single-institution experience with a systematic review

Sphenoid Sinus Giant Cell Tumor: A Case Report and Literature Review

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