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

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OBJECTIVE

Intracranial 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.

METHODS

The 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).

RESULTS

The 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.

CONCLUSIONS

GTR 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/)

ABBREVIATIONS EMA = epithelial membrane antigen; EMPT = estimated mean PFS time; GCRG = giant cell reparative granuloma; GCT = giant cell tumor; GKRS = Gamma Knife radiosurgery; GTR = gross-total resection; IMRT = intensity-modulated radiotherapy; KPS = Karnofsky Performance Scale; OS = overall survival; PFS = progression-free survival; PR = partial resection; STR = subtotal resection; TMJ = temporomandibular joint.

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Article Information

Correspondence Jun-Ting Zhang: Beijing Tiantan Hospital, Capital Medical University, Beijing, People’s Republic of China. zhangjunting2003@aliyun.com.

INCLUDE WHEN CITING Published online October 12, 2018; DOI: 10.3171/2018.4.JNS1849.

J.C.W. and D.L. contributed equally to this work.

Disclosures The authors report no conflict of interest concerning the materials or methods used in this study or the findings specified in this paper.

© AANS, except where prohibited by US copyright law.

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Figures

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    PRISMA flow diagram showing the inclusion and exclusion process for the analysis. Figure is available in color online only.

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    A and B: Preoperative CT scans showing a lesion located in the temporal bone expanding into the TMJ (A) and a sphenoid lesion involving the ethmoid bone (B) that presented as a sharply circumscribed, expansile, lytic bone lesion. C and D: H & E staining of GCTs revealing numerous multinucleated giant cells that lack atypical nuclei, scattered in a background of uniform mononuclear cells. Original magnification ×100 (C); ×400 (D). Figure is available in color online only.

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    Kaplan-Meier curve analysis (log-rank test) showing the different PFS rates among GTR, STR, and PR (A); between Ki-67 < 10% and Ki-67 ≥ 10% (B); between radiotherapy (RT) and non-RT (C); between RT with dose ≥ 45 Gy and < 45 Gy (D); and among image stages I, II, and III (E). Kaplan-Meier curve (log-rank test) showing OS and PFS of the pooled cohort (F); the different PFS times among GTR, STR, and PR (G); between RT and non-RT (H); between RT with a total dose ≥ 45 Gy and < 45 Gy (I); and the different PFS rates of these 4 treatment protocols (J). Patients with GTR plus RT, GTR alone, or non-GTR plus RT had significantly better PFS than patients with non-GTR alone. Figure is available in color online only.

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    A case of GCT in a 28-year-old man whose main complaint was tinnitus accompanying a headache for 12 months. Preoperative contrast MRI scans showing a lesion located in the temporal bone expanding into the TMJ: mixed iso- and hyperintensity accompanying extremely hypointense peripheral lesion on T2-weighted image (A) and heterogeneous enhancement on contrasted MR image (B). After GTR, there was no additional treatment for him. Contrasted MRI scans (C) showed complete resection without recurrence after 144 months.

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