A Rare Manifestation of a Presumed Non-Osteophilic Brain Neoplasm: Extensive Axial Skeletal Metastases From Glioblastoma With Primitive Neuronal Components

BackgroundGlioblastoma multiforme (GBM) is the most common malignant tumor of the central nervous system. GBM with primitive neuronal component (GBM-PNC) is an aggressive variant identified in 0.5% of GBMs. Extracranial metastasis from GBM-PNC is a rare and challenging situation.MethodsA special case of early-onset GBM with systemic bone metastasis was enrolled. Clinical data, including patient characteristics, disease course, and serial radiological images were retrieved and analyzed. Tumor tissues were obtained by surgical resections and were made into formalin-fixed paraffin-embedded sections. Histopathological examinations and genetic testing were performed for both the primary and metastatic tumor specimens.ResultsA 20-year-old man suffered from GBM with acute intratumoral hemorrhage of the left temporal lobe. He was treated by gross total resection and chemoradiotherapy following the Stupp protocol. Seven months later, he returned with a five-week history of progressive neck pain and unsteady gait. The radiographic examinations identified vertebral collapse at C4 and C6. Similar osteolytic lesions were also observed at the thoracolumbar spine, pelvic, and left femur. Anterior spondylectomy of C4 and C6 was performed. The resected vertebral bodies were infiltrated with greyish, soft, and ill-defined tumor tissue. One month later, he developed mechanical low-back pain and paraplegia caused by thoracolumbar metastases. Another spine surgery was performed, including T10 total en-bloc spondylectomy, T7-9, L2-3, and L5-S1 laminectomy. After the operation, the patient’s neurological function and spinal stability remained stable. However, he finally succumbed to the rapidly increased tumor burden and died 15 months from onset because of cachexia and multiple organ failure. In addition to typical GBM morphology, the histological examinations identified monomorphic small-round cells with positive immunohistochemical staining of synaptophysin and CD99, indicating the coexistence of PNC. The next-generation sequencing detected pathogenic mutations in TP53 and DNMT3A. Based on above findings, a confirmed diagnosis of systemic metastases from GBM-PNC (IDH-wild type, WHO grade IV) was made.ConclusionsThe present case highlights the occurrence and severity of extensive axial skeletal metastases from GBM-PNC. This rare variant of GBM requires aggressive multimodal treatment including surgery and chemoradiotherapy targeting PNC. The pathological screening of PNC is recommended in patients with early-onset GBM and intratumoral hemorrhage. Surgery for spinal metastasis is appropriate in patients with chemoradioresistance and relatively good general status, with the objectives of restoring spinal stability and relieving spinal cord compression.

Primary intraosseous meningioma of the vertebra: illustrative case

BACKGROUND Primary intraosseous meningiomas (PIMs) are rare, and PIMs of the vertebrae have not yet been reported. The authors report a case of primary meningioma arising from the vertebrae. OBSERVATIONS A 49-year-old man presented with lower back pain and numbness in both lower extremities. Lumbar spine magnetic resonance imaging revealed an L2 pathological fracture with epidural and paraspinal invasion. The patient had undergone a first palliative decompression and fixation surgery, and the diagnosis turned out to be a World Health Organization grade III anaplastic meningioma based on histopathology. The tumor had progressed after first operation and radiation therapy, and the patient was referred to the authors’ institute for excision. The patient had an uneventful postoperative course after a revisional total en bloc spondylectomy of L2. LESSONS The authors present a rare case of PIM of the vertebrae with epidural and paraspinal invasion. Careful preoperative assessment and surgical planning is crucial for successful patient management.

Hybrid therapy versus Total En Bloc Spondyectomy in the treatment of Solitary Radioresistant Spinal Metastases：A propensity score-matched analysis

Background Both hybrid therapy (HT) and total en bloc spondylectomy (TES)can lead to good results for solitary radioresistant metastatic spinal tumors with high-grade epidural spinal cord compression (ESCC). However, there is still a lack of comparative studies on the treatment efficacy of these two methods. Methods We retrospectively reviewed patients with the above-mentioned tumors between January 2012 and May 2019. A total of 157 patients underwent surgery, among whom 64 received HT, and 93 were treated with TES. Propensity score matching allowed the generation of best-matched pairs for the 2 categories (1:1 ratio). Local control rates and survival rates were estimated using the Kaplan–Meier method. Results All patients received a minimum of 2-year follow-up. The longest follow-up time was 88 months. The survival rates and local progression-free survival rates after HT were comparable with TES at 1 year (84.6% vs. 83.1%; 90.2% vs. 90%), 2 year (60.8% vs. 64.3%; 64.1% vs. 62.1%), and 5 year (18.8% vs. 24.1%; 24.4% vs. 28.4%). There were no significant differences in pain control, improvement of neurological status, spine stabilization restoration, and improvement in quality of life between groups. However, HT showed more advantages in shortening operative time and reducing intraoperative blood loss than TES. Conclusion Our results suggest that HT can obtain satisfactory results comparable to TES for solitary radioresistant metastatic spinal tumors with high-grade ESCC. In addition, compared with TES, HT has the advantages of shortening operative time and reducing perioperative complications. For solitary radioresistant metastatic spinal tumors with a high degree of ESCC, HT may be a promising treatment in the future.

Biomechanics of artificial pedicle fixation in a 3D-printed prosthesis after total en bloc spondylectomy: a finite element analysis

Background This study compared the biomechanics of artificial pedicle fixation in spine reconstruction with a 3-dimensional (3D)-printed prosthesis after total en bloc spondylectomy (TES) by finite element analysis. Methods A thoracolumbar (T10–L2) finite element model was developed and validated. Two models of T12 TES were established in combination with different fixation methods: Model A consisted of long-segment posterior fixation (T10/11, L1/2) + 3D-printed prosthesis; and Model B consisted of Model A + two artificial pedicle fixation screws. The models were evaluated with an applied of 7.5 N·m and axial force of 200 N. We recorded and analyzed the following: (1) stiffness of the two fixation systems, (2) hardware stress in the two fixation systems, and (3) stress on the endplate adjacent to the 3D-printed prosthesis. Results The fixation strength of Model B was enhanced by the screws in the artificial pedicle, which was mainly manifested as an improvement in rotational stability. The stress transmission of the artificial pedicle fixation screws reduced the stress on the posterior rods and endplate adjacent to the 3D-printed prosthesis in all directions of motion, especially in rotation. Conclusions After TES, the posterior long-segment fixation combined with the anterior 3D printed prosthesis could maintain postoperative spinal stability, but adding artificial pedicle fixation increased the stability of the fixation system and reduced the risk of prosthesis subsidence and instrumentation failure.

The most appropriate titanium mesh cage size for anterior spinal reconstruction after single-level lumbar total en bloc spondylectomy: a finite element analysis and cadaveric validation study

Purpose There is little information available regarding the cage diameter that can provide the most rigid construct reconstruction after total en bloc spondylectomy (TES). The aim of this study was thus to determine the most appropriate titanium mesh cage diameter for reconstruction after spondylectomy. Methods A finite element model of the single level lumbar TES was created. Six models of titanium mesh cage with diameters of 1/3, 1/2, 2/3, 3/4, 4/5 of the caudad adjacent vertebra, and 1/1 of the cephalad vertebra were tested for construct stiffness. The peak von Mises stress (MPa) at the failure point and the site of failure were measured as outcomes. A cadaveric validation study also conducted to validate the finite element model. Results For axial loading, the maximum stress points were at the titanium mesh cage, with maximum stress of 44,598 MPa, 23,505 MPa, 23,778 MPa, and 16,598 MPa, 10,172 MPa, 10,805 MPa in the 1/3, 1/2, 2/3, 3/4, 4/5, and 1/1 diameter model, respectively. For torsional load, the maximum stress point in each of the cages was identified at the rod area of the spondylectomy site, with maximum stress of 390.9 MPa (failed at 4459 cycles), 141.35 MPa, 70.098 MPa, and 88.972 MPa, 42.249 MPa, 15.827 MPa, respectively. A cadaveric validation study results were coincided with the finite element model results. Conclusion The most appropriate mesh cage diameter for reconstruction is 1/1 the diameter of the lower endplate of the adjacent cephalad vertebra, due to its ability to withstand both axial and torsional stress. According to the difficulty of large size cage insertion, a cage diameter of more than half of the upper endplate of the caudad vertebrae is acceptable in term of withstand stress. A cage diameter of 1/3 is unacceptable for reconstruction after total en bloc spondylectomy.

Biomechanics of artificial pedicle fixation in a 3D-printed prosthesis after total en bloc spondylectomy: A finite element analysis

Background: This study compared the biomechanics of artificial pedicle fixation in spine reconstruction with a 3-dimensional (3D)-printed prosthesis after total en bloc spondylectomy (TES) by finite element analysis.Methods: A thoracolumbar (T10–L2) finite element model was developed and validated. Two models of T12 TES were established in combination with different fixation methods: Model A consisted of long-segment posterior fixation (T10/11, L1/2) + 3D-printed prosthesis; and Model B consisted of Model A + two artificial pedicle fixation screws. The models were evaluated with an applied of 7.5 N·m and axial force of 200 N. We recorded and analyzed the following: 1) stiffness of the two fixation systems; 2) hardware stress in the two fixation systems; and 3) stress on the endplate adjacent to the 3D-printed prosthesis.Results: The fixation strength of Model B was enhanced by the screws in the artificial pedicle, which was mainly manifested as an improvement in rotational stability. The stress transmission of the artificial pedicle fixation screws reduced the stress on the posterior rods and endplate adjacent to the 3D-printed prosthesis in all directions of motion, especially in rotation.Conclusions: After TES, the posterior long-segment fixation combined with the anterior 3D printed prosthesis could maintain postoperative spinal stability, but adding artificial pedicle fixation increased the stability of the fixation system and reduced the risk of prosthesis subsidence and instrumentation failure.