Clinical characteristics and prognoses of six patients with multicentric giant cell tumor of the bone

Abstract

Multicentric giant cell tumor of the bone (MGCT) is a rare entity whose radiographic, pathological and biological features remain confusing. We retrospectively reviewed six patients (1 male, 5 female; average age, 22.33 years) treated for confirmed MGCT between 2001 and 2015. The patients' clinical information, images from radiographs (n = 14), CT (n = 13), MRI (n = 8), bone scintigraphy (n = 1) and PET-CT (n = 2), as well as histologic features, treatment and prognosis were analyzed. A total of 17 lesions were detected: 4 around the knee joint, 3 in the greater trochanter and head of the femur, 5 in the small bones of the feet, and 2 in flat bones. All these lesions occurred in an ipsilateral extremity. One patient had Paget's disease. On radiographs and CT, 12 lesions exhibited sclerotic margins or patchy sclerosis, 8 showed cortical discontinuity, and 5 showed soft tissue masses. On histopathology, 8 lesions showed signs of sarcomatous transformation and one had transformed into osteosarcoma. Ten lesions in 4 patients were initially treated with surgery, and 3 showed local recurrence. Seven lesions in 3 patients were treated with denosumab. All the patients are currently stable without metastasis. These results suggest MGCT tends to occur in uncommon sites with sclerosis. Because these lesions can be aggressive, patients should be carefully monitored for the recurrence or formation of other lesions, especially in an ipsilateral extremity.

Pre-operative denosumab is associated with higher risk of local recurrence in giant cell tumor of bone: a systematic review and meta-analysis

“…A significant decrease in enhancement in enhanced CT value suggested the decrease of blood supply after denosumab administration [22]. On MRI, marrow replacement by tissue with different signal intensity were reported [18]. For 18F-FDG PET/CT, studies found 94-96% cases underwent denosumab treatment had decreased SUVmax values [10,29].…”

“…2). Surgical choice was taken into account by comparing cases underwent curettage with denosumab and curettage alone in 4 studies [9,[16][17][18](OR: 2.13, 95% CI: 0.95 to 4.76, p = 0.07, I 2 = 29%)( Fig. 3), though the result was not statistically significant.…”

“…3), though the result was not statistically significant. Seven studies [9,[15][16][17][18][19][20] reported outcome on primary lesions, which also suggest significantly higher risk of local recurrence in the denosumab group (OR: 1.91, 95% CI: 1.17 to 3.09, p = 0.009, I 2 = 36%)( Fig. 4).…”

Pre-operative denosumab is associated with higher risk of local recurrence in giant cell tumor of bone: a systematic review and meta-analysis

“…1.16 to 4.85, p=0.02, I 2 =57%)(figure2) . Surgical choice was taken into account by comparing cases underwent curettage with denosumab and curettage alone in 4 studies [9,[16][17][18](OR: 2.13, 95% CI: 0.95 to 4.76, p=0.07, I 2 =29%)( Figure 3), though the result was not statistically significant. 7 studies [9,[15][16][17][18][19][20] reported outcome on primary lesions, which also suggest significantly higher risk of local recurrence in the denosumab group(OR: 1.91, 95% CI: 1.17 to 3.09, p=0.009, I 2 =36%)( Figure 4).…”

“…On MRI, marrow replacement by tissue with different signal intensity were reported. [18]For 18F-FDG PET/CT, studies found 94%-96% cases 8 underwent denosumab treatment had decreased SUVmax values [10,29]. Ueda et al [11] used CT/MRI and 18FDG-PET/PET-CT to assess objective response of giant cell tumor of bone after denosumab treatment and found a 35%/82%/71% response rate according to RECIST/EORTC/Choi Criteria respectively, suggesting a robust radiographic response after denosumab treatment.…”