Myopericytoma of the patella with local recurrence and patellectomy: A case report.

INTRODUCTION AND IMPORTANCE: Myopericytomas are tumors originating from perivascular myoid cells and exhibiting a wide range of histologic growth patterns. They rarely occur in bones, and no case of myopericytoma in the patella has been reported so far. CASE PRESENTATION: A 74-year-old male presented with a chief complaint of pain in the left knee. Magnetic resonance imaging revealed bone tumor and osteolytic lesions of the patella. The patient underwent bone tumor curettage and filling of the cavity with artificial bone. However, as the tumor reoccurred, a patellectomy was performed. The patient regained premorbid functional status after surgery. Additionally, there was no radiological evidence of recurrence of the lesion 3 years after patellectomy. CLINICAL DISCUSSION: Myopericytoma of the patella is very rare. However, it should be considered for the differential diagnosis of lytic lesions of the bone. Although surgery is curative, patellectomy may be necessary for recurrent cases. CONCLUSION: In conclusion, we report the first case of patellar myopericytoma. Although patellar myopericytoma might be rare, it should be considered for the differential diagnosis of lytic lesions of the bone. Surgery is curative; however, patellectomy may be necessary in recurrent cases.

Synergistic VSV Virotherapy and Carbon Nanotube Photothermal Therapy for Osteosarcoma in Murine Models.

BACKGROUND/AIM: Wide resection is usually performed for malignant bone and soft tissue tumors, but there is often functional impairment of the affected limb. In this study, we performed virotherapy with the vesicular stomatitis virus (VSV) and photothermal therapy using carbon nanotubes (CNTs) in combination for osteosarcoma, followed by marginal excision. The possibility of local treatment of the primary tumor was then assessed. MATERIALS AND METHODS: LM-8 cells (1×107) were subcutaneously implanted into 5-week-old mice to generate an in vivo osteosarcoma mouse model. Marginectomy was performed. Four groups with six mice each were created: VSV+SWCNTs group, VSV group, SWCNTs group, and an untreated group. Tumor margin resection was performed 2 weeks after tumor cell transplantation. The primary tumor volume, local recurrence, distant metastasis, and survival rate were evaluated. RESULTS: The combination of VSV virotherapy and CNTs photothermal therapy resulted in shrinkage of the primary tumor and reduced local recurrence after marginectomy. There was no significant difference in distant metastasis or survival rate for all groups. CONCLUSION: Combining virotherapy with VSV and CNTs photothermal therapy is useful for local treatment of osteosarcoma in murine models, possibly allowing for smaller tumor resection margins.

Functional outcome of campanacci grade 3 giant cell tumours of distal radius after resection and reconstruction with ulnar translocation technique: a case series.

Reconstruction after en-bloc resection can be challenging in cases of Campanacci grade 3 giant cell tumour of the bone (GCTB) of the distal radius. Here, the authors examined the outcomes of patients with Campanacci grade 3 GCTBs of the distal radius who underwent wrist arthrodesis and reconstruction with ulnar translocation. Material and methods: This case series was a retrospective single-centre study. Clinical assessments regarding the functional status and complications were follow-up. The functional results were evaluated using the Musculoskeletal Tumor Society (MSTS) and Disability of Arm, Shoulder, and Hand (DASH) Score. Paired t-tests were used to compare the MSTS and DASH scores separately before and after the operation. Statistical differences were considered significant at P less than 0.05. Results: Seven patients were included in this study. The mean follow-up period was 14.43±8.08 months. The average length of tumour resection was 9.78±2.88 cm. The average range of motion of the involved forearm was 82.66° of supination and 81.54° of pronation. The average MSTS score was 11.71±2.21 before and 25.14±2.41 after the operation (P<0.05). The average DASH score on admission was 40.14±14.66, which decreased to 9.02±4.23 after the operation (P<0.05). Of the seven cases, one case had a recurrence, and one patient had radioulnar synostosis. Conclusion: Wrist arthrodesis combined with ulnar translocation can be considered a simple and effective reconstruction method with preservation of function after en-bloc resection of Campanacci grade 3 GCTB of the distal radius. It provides good functional outcomes with low complication rates.

Oncolytic Effect of a Recombinant Vesicular Stomatitis Virus Encoding a Tumor-suppressor MicroRNA in an Osteosarcoma Mouse Model.

BACKGROUND/AIM: Attempts have been made to enhance treatment with vesicular stomatitis virus (VSV) for osteosarcoma. We have previously shown that VSV incorporated with miRNA143 enhanced the antitumor effect at some doses; however, the range of the doses was narrow. This has not been evaluated in vivo, and the synergistic effect of this antitumor effect in animals is unknown. The purpose of the study was to evaluate the oncolytic effect of VSV-miRNA on osteosarcoma cells in vivo. MATERIALS AND METHODS: A novel oncolytic VSV was developed by incorporating the tumor-suppressor microRNA143 (rVSV-miR143). In order to compare the antitumor effects of administration methods (intravenous and intratumoral administration) of rVSV-miR143 with those of VSV, a comparative analysis of primary tumor volume, metastatic lesions and survival rate was performed in mouse models of osteosarcoma. RESULTS: Following intratumoral injection, rVSV-miR143 showed a significant reduction in primary tumor volume, but no significant difference was observed in metastatic lesions and survival rate compared to VSV. Following intravenous injection, rVSV-miR143 revealed no significant difference in primary tumor volume, metastatic lesion and survival rate compared to VSV. CONCLUSION: VSV incorporating tumor-suppressor miRNA143 demonstrated a slightly synergistic antitumor effect on osteosarcoma in vivo.

Incomplete Atypical Femoral Fracture Treated by Prophylactic Intramedullary Nail Fixation: A Case Series.

Long-term bisphosphonate use may be associated with atypical femoral fractures. In this report, we describe three cases of bisphosphonate-associated incomplete atypical femoral fracture, treated by prophylactic intramedullary nail fixation. Patients with long-term intake of bisphosphonates must be carefully monitored; atypical femoral fracture should be suspected in the presence of symptoms such as thigh pain. Its early identification is important to avoid a complete fracture and invasive surgery, and prophylactic fixation is recommended for incomplete atypical femoral fractures.

Utility of intraoperative magnetic resonance imaging for giant cell tumor of bone after denosumab treatment: a pilot study.

BACKGROUND: Giant cell tumor of bone (GCTB) is an intermediate but locally aggressive neoplasm. Current treatment of high-risk GCTB involves administration of denosumab, which inhibits bone destruction and promotes osteosclerosis. However, denosumab monotherapy is not a curative treatment for GCTB and surgical treatment remains required. Denosumab treatment complicates surgery, and the recurrence rate of GCTB is high (20%-30%). PURPOSE: To examine the utility of intraoperative magnetic resonance imaging (iMRI) for detection and reduction of residual tumor after denosumab treatment and to investigate the utility of iMRI, which is not yet widely used. MATERIAL AND METHODS: We enrolled five patients who received denosumab for a median period of eight months (range 6-12 months). Surgery was performed when the degree of osteosclerosis around the articular surface was deemed appropriate. We performed iMRI using a modified operation table to identify residual tumor after initial curettage and evaluated the rate of detection of residual tumor by iMRI, intraoperative and postoperative complications, exposure time of iMRI, and operation time. RESULTS: Suspected residual tumor tissue was identified in all five cases and was confirmed by histopathology after additional curettage. The rate of detection of residual tumor by iMRI was 100%. Residual tumor was located in sites which were difficult to remove due to osteosclerosis. The iMRI was performed safely and without trouble. During the median follow-up period of 10 months (range 6-24 months), no adverse events or recurrences occurred. CONCLUSION: Intraoperative MRI could contribute to the reduction of residual tumor tissue and it may prevent recurrence of GCTB after denosumab therapy.

Giant cell tumor of the cervical spine treated by carbon ion radiotherapy: A case report.

INTRODUCTION: Giant cell tumor (GCT) of the bone is a benign-malignant intermediate tumor with locally destructive growth and a relatively high local recurrence rate. Neurological symptoms may develop in patients with GCT of the spine, and surgical treatment is prioritized in cases where resection is possible. However, the local recurrence rate of GCT of the bone is higher than that of GCT at other sites owing to the associated surgical challenges, and treatment is often difficult. No study to date has reported long-term remission of recurrent tumors for more than 5 years by treatment with carbon ion beam radiotherapy after resection of GCT of the cervical spine. PATIENT CONCERNS: A 14-year-old boy who experienced recurrence after surgery for GCT of the cervical spine. DIAGNOSIS: The patient presented with cervical pain, and computed tomography revealed a mass of the C2 vertebral body. He underwent surgery for tumor resection and autologous bone grafting, and the final pathological diagnosis was GCT. The transplanted bone exhibited gradual progression of resorption, and recurrent tumors were observed on computed tomography and magnetic resonance imaging 1 year and 4 months after surgery. INTERVENTIONS: The patient was started on denosumab at 15 years of age and received carbon ion beam therapy with 70.4 Gy administered in 32 sessions over 7 weeks. OUTCOMES: No progressive tumor growth was observed, there were no neurological symptoms such as paralysis or pain were noted, and the patient was in remission for 5 years after irradiation. CONCLUSION: These findings suggest that carbon ion radiotherapy is a safe and effective therapeutic option for patients with recurrent GCT of the cervical spine.

Tuberculosis of the knee masquerading as pigmented villonodular synovitis: Report of two cases.

INTRODUCTION AND IMPORTANCE: The manifestation of tuberculous mimicry is challenging to diagnose, especially in extrapulmonary Tuberculosis (TB). Pigmented villonodular synovitis (PVNS) may mimic tuberculous arthritis of the knee joint. In young, otherwise healthy-appearing individuals, tuberculous arthritis and PVNS in the knee joint could present as monoarticular involvement, with painful swelling of extended duration and limited movement. The core therapies for tuberculous arthritis and PVNS are different. CASE PRESENTATION: There are two patients. First, male 25 years old presented with a painful mass at the left knee of 3-years duration. The second patient was 14 years old boy who presented with a painful mass at his left knee for ten months before being admitted to the hospital. From the physical examination, plain x-ray, and MRI, both patients are highly suggestive of PVNS. However, the histopathology result and microbial culture showed a tuberculous-specific process. CLINICAL DISCUSSION: Because of its clinical manifestations, X-ray, and MRI features, patients were initially suspected of having the diffuse type of PVNS. An improper diagnosis may be due to atypical clinical presentation, wide use of antibiotics, the low specificity of diagnostic tools, and most of all, the clinician not prioritizing the possibility of tuberculous arthritis. Open biopsy and the result of the microbial culture establish the definitive diagnosis of knee tuberculosis. CONCLUSION: In conclusion, the clinical and radiologic findings in TB gonitis and PVNS are occasionally similar. Therefore TB gonitis should be highly suspicious, especially in endemic areas. Histopathology results and mycobacterial culture need to determine the diagnosis.

Extraskeletal Ewing sarcoma attached to the ulnar nerve: A case report.

INTRODUCTION: Extraskeletal Ewing sarcoma (EES) of the extremity is uncommon, and only a small number of reported cases have been devoted to the upper-extremity. PRESENTATION OF CASE: A 65-year-old woman presented with a recurrent EES, a highly malignant tumor, involving the ulnar nerve at the right elbow region which was initially suspected as a benign soft tissue tumor, schwannoma, thus marginal excision had been performed. Due to its malignant behaviour, we treated the recurrent lesion with wide excision and reconstruction combined with chemotherapy. Histological evaluation revealed a monotonous small round cells appearance. DISCUSSION: EES of the extremity involving the ulnar nerve is fairly uncommon. The tumor was often smaller in the adult than in the child population which was consistent with the present case, thus may mimic a benign tumor. Because of the overlapping histopathological features of EES with other tumors, other investigations such as immunohistochemistry and cytogenetic studies must be performed to allow definitive diagnosis. The result of our study was negative for the EWSR1-FLI-1 and CIC-DUX4 fusion gene, however, other less frequent translocations could be found in this case which does not exclude the diagnosis of Ewing sarcoma family. CONCLUSION: Few cases of EES involving the ulnar nerve have been previously reported. The correct diagnosis of EES involving the ulnar nerve has become particularly important in order to enable the initiation of comprehensive management that have the potential to reduce disease progression and the avoidance of improper and potentially harmful surgical therapy.

Granulocyte Colony-stimulating Factor-induced Aortitis with Lung Injury, Splenomegaly, and a Rash During Treatment for Recurrent Extraosseous Mucinous Chondrosarcoma.

We herein report a case of aortitis induced by granulocyte colony-stimulating factor (G-CSF) that coincided with lung injury, splenomegaly, and cutaneous manifestations during treatment for recurrent extraosseous mucinous chondrosarcoma. Computed tomography revealed large-vessel vasculitis, splenomegaly, and pulmonary interstitial changes. Treatment with prednisolone was successful. Because sarcoma is a rare disease, this case is valuable for showing clinicians that G-CSF preparations could cause aortitis regardless of the patient's underlying diseases or therapeutic pharmacological backgrounds.

Development of an Oncolytic Recombinant Vesicular Stomatitis Virus Encoding a Tumor-suppressor MicroRNA.

BACKGROUND: Attempts have been made to enhance systemic therapy for osteosarcoma. In our previous study, the systemic administration of a vesicular stomatitis virus (VSV) improved the survival rates of mice with osteosarcoma but did not improve the long-term survival of the animals. MATERIALS AND METHODS: In the present study, we developed a novel oncolytic VSV by incorporating tumor-suppressor microRNA143 (rVSV-miR143) to compare the antitumor effects of various doses (10×10-4, 5×10-4, and 1×10-4 multiplicity of infection) of rVSV-miR143 with those of VSV in vitro. RESULTS: The cytotoxicity and migration-inhibitory effects of rVSV-miR143 on the osteosarcoma cells were significantly higher than those of VSV alone at a dose of 5×10-4 multiplicity of infection, indicating that rVSV-miRNA143 enhances the antitumor effect at certain doses. CONCLUSION: VSV incorporating tumor-suppressor miRNA143 demonstrated a synergistic antitumor effect on osteosarcoma cells in vitro.

Rapidly growing Fibro-osseous pseudotumor of the digit: A case report.

INTRODUCTION: Fibro-osseous pseudotumor of the digit is a rare benign lesion of subcutaneous tissue that typically arises in the parabone site of the proximal phalanx in young adult females. The lesion is histopathologically characterized by fibroblastic proliferation and osteoid formation. Good prognosis following complete surgical excision of the tumor has been reported, with a very low recurrence rate and no reports of malignant transformation. Despite its benign clinical behavior, the lesion can be mistaken for a malignant neoplasm, such as an extraskeletal or parosteal osteosarcoma, in case of rapid growth, thereby rendering the diagnosis challenging. PATIENT CONCERNS: We report the case of a 30-year-old right-handed male who presented to our hospital with a rapidly growing mass on the dorsal aspect of the right little finger. DIAGNOSIS: The patient was suspected to have soft tissue tumor of the little finger. The lesion could be considered a malignant tumor on the basis of clinical findings. INTERVENTIONS: The patient underwent surgery for exploration and excision of the mass. OUTCOMES: The excised mass was diagnosed to be fibro-osseous pseudotumor of the digit upon histological assessment. Postoperatively, the wound healed without complications. At postoperative 6 months, there were no signs or symptoms of recurrence, and the patient returned to his premorbid functional status. CONCLUSION: Following the detection of a soft tissue mass with clinicopathological features of pseudomalignancy in the digit, clinicians should consider fibro-osseous pseudotumor of the digit as a possible diagnosis, thereby avoiding unnecessary aggressive surgery.

Novel Near-Infrared Fluorescence-Guided Surgery With Vesicular Stomatitis Virus for Complete Surgical Resection of Osteosarcomas in Mice.

Attempts have been made to visualize tumor cells intraoperatively with fluorescence guidance. However, the clear demarcation and complete tumor resection have always been a challenging task. To address this, we have developed a novel fluorescence bioimaging system with vesicular stomatitis virus (VSV) incorporating Katushka, near-infrared fluorescent protein. VSV is tumor-specific owing to the deficiency of antiviral interferon signaling pathways in tumor cells. We aimed to evaluate the tumor specificity of the recombinant VSV-Katushka (rVSV-K) in osteosarcoma cells and to assess the feasibility of complete tumor resection by the rVSV-K fluorescence guidance. In in vitro experiments, mouse and human osteosarcoma cell lines and normal human mesenchymal stem cells were infected with rVSV-K and observed by fluorescence microscopy. Near-infrared fluorescence was observed only in osteosarcoma cells, even at a low-concentration of virus infections. In in vivo experiments, mouse osteosarcoma (LM8) cells were transplanted subcutaneously into the back of immune-competent mice to produce an osteosarcoma, which was then injected with rVSV-K. The areas emitting fluorescence were resected using a bioimaging system. The distance between the surgical and tumor margins of the fluorescence-guided resection with rVSV-K group was significantly larger than that of the non-guided resection groups. The local recurrence rate was significantly lower in the fluorescence-guided resection with rVSV-K group than in the non-guided resection groups. The distant metastasis rate and average survival rate were not significantly different between all groups. These results suggest that the rVSV-K is specific to osteosarcoma cells and enables complete tumor resection of osteosarcomas in mice. © 2019 Orthopaedic Research Society. Published by Wiley Periodicals, Inc. J Orthop Res.

Coexistence of giant cell tumor of tendon sheath and enchondroma in the middle phalanx of the little finger mimicking a malignant tumor: A case report.

Giant cell tumor of the tendon sheath is a type of slow-growing benign soft tissue tumor that typically arises from the synovium of the tendon sheath. Enchondroma is a benign bone tumor comprising of mature hyaline cartilage that centrally develops within the tubular bone. While giant cell tumor of the tendon sheath or enchondroma are common benign soft tissue and bone tumors, respectively the simultaneous occurrence of these tumors in the same region of the hand is exceedingly rare, and it can mimic a malignant tumor, thereby making the diagnosis more challenging. Herein, we report an unusual imaging presentation of the coexistence of these tumors in the middle phalanx of the little finger, which to the best of our knowledge has not been previously reported, and this initially present as a single intrinsic osseous lesion mimicking malignancy. The coexistence of these tumor types must be considered in the differential diagnosis of an intramedullary lytic lesion with a poor margin associated with a soft tissue mass of the fingers, and a meticulous preoperative magnetic resonance imaging investigation was required.

Conventional 99mTc-(hydroxy) methylene diphosphate remains useful to predict osteosarcoma response to neoadjuvant chemotherapy: Individual patient data and aggregate data meta-analyses.

BACKGROUND: The current standard of chemotherapy response evaluation holds the most important prognostic factor to be the histological assessment of the tumor necrosis of the excised lesion, but the major challenge is to find an early prognostic factor that will allow the adjuvant treatment regimen to be adjusted. The objective of this systematic review is to provide an up-to-date and unprecedented summary of the value of Technetium-methylene diphosphate or -hydroxymethylene diphosphate (Tc-MDP/HMDP) scintigraphy for the preoperative evaluation of osteosarcoma response to chemotherapy. METHODS: Studies evaluating the alteration ratio (percentage change of the Tc-99m -MDP/HMDP uptake between before and after neoadjuvant chemotherapy) to predict the histological response of osteosarcoma to chemotherapy were searched for in MEDLINE, EMBASE, and Web of Science. A meta-analysis of individual patient data (IPD) was performed to determine the optimal cut-off point from the receiver operating characteristic (ROC) curve. Additionally, aggregate data (AD) meta-analysis was performed to compare the value of Tc-MDP/HMDP scintigraphy with that of other quantitative modalities, such as dynamic magnetic resonance imaging (MRI), Tl scintigraphy, and F-FDG PET-CT. RESULTS: Seven studies with 154 patients were included for the IPD meta-analysis. The optimal cut-off point of the alteration ratio was 31.0%. Five studies with 123 patients were considered for the AD meta-analysis. The pooled sensitivity and specificity were 0.76 (95% CI, 0.63-0.86) and 0.89 (95% CI, 0.79-0.95), respectively. There was a significant difference between the good and poor responders in terms of the diagnostic odds ratio. The summary ROC curve demonstrated that the area under curve (AUC) was 0.892, indicating excellent diagnostic accuracy. CONCLUSION: Our findings have suggested that conventional Tc-MDP/HMDP scintigraphy remains as useful as recent quantitative modalities to predict the histological response of osteosarcoma to neoadjuvant chemotherapy.

A meta-analysis supports core needle biopsy by radiologists for better histological diagnosis in soft tissue and bone sarcomas.

BACKGROUND: Although surgical biopsy has historically been considered to be the standard diagnostic biopsy for soft tissue and bone sarcomas, recent literature suggests that percutaneous core needle biopsy yields similar results. Therefore, an evaluation of the exact diagnostic accuracy and associated influential variables of core needle biopsy that is based on a large data set would be useful. METHODS: We searched MEDLINE, Web of Science, and EMBASE to identify core needle biopsy studies for predicting final histological subtypes of musculoskeletal lesions. The diagnostic accuracies of core needle biopsy and of surgical biopsy were assessed and compared by using random-effect meta-analyses. The factors relevant to diagnostic accuracy were evaluated by meta-regression and subgroup analyses. RESULTS: We selected 32 studies comprising 7209 musculoskeletal lesions. The pooled proportion estimate for the diagnostic accuracy of core needle biopsy was 0.84 (95% confidential interval, CI: 0.81-0.87), which indicated an approximate 84% concordance between core needle biopsy results and final histological diagnoses. The findings of meta-regression and subgroup analyses suggested that radiologists were better core needle biopsy operators than surgeons. An additional meta-analysis for direct comparison between core needle biopsy and surgical biopsy demonstrated that diagnostic accuracy was significantly lower for core needle biopsy than for surgical (pooled odds ratio: 0.39, 95% CI: 0.20-0.76). CONCLUSION: Our results suggested that core needle biopsy should be performed by expert radiologists and that surgical biopsy should be performed if diagnosis following core needle biopsy does not match the clinical presentation and radiographic findings.

Effect of Continuous Compression Stimulation on Pressure-Pain Threshold and Muscle Spasms in Older Adults With Knee Osteoarthritis: A Randomized Trial.

OBJECTIVE: The purpose of this study was to assess the effect of continuous compression stimulation on pressure-pain threshold and muscle spasms in older adults with knee osteoarthritis. METHODS: Thirty-two older adults with knee osteoarthritis on outpatient visits were randomly divided into 2 groups. Those in the treatment group (n = 16) received 5-minute massage therapy (continuous compression stimulation), and those in the control group (n = 16) received sham massage therapy (touch without compression). Immediately before and after single-intervention sessions, the pressure-pain threshold, muscle spasm, and pain were quantified. RESULTS: The change in pain on walking in the treatment group exceeded 1.9 cm, corresponding to the minimum clinically important difference. In the treatment group, the pressure-pain threshold improved significantly for pain both at rest and while walking, but the improvement in muscle spasm was not significant. CONCLUSIONS: Massage therapy resulted in minimal clinically important changes for pain relief. There was an increase in the pressure-pain threshold in the older adults with knee osteoarthritis. We propose that the improvements in pain may be related to the medial thigh muscle rather than knee osteoarthritis.

Metastatic tumor cells detection and anti-metastatic potential with vesicular stomatitis virus in immunocompetent murine model of osteosarcoma.

Sarcomas are associated with a high incidence of lung metastasis, which leads to a high-risk of cancer death. This study was performed to explore the pre-clinical theranostic potential of a novel fully functional recombinant vesicular stomatitis virus carrying imaging gene Katushka (rVSV-K), as virotherapy and circulating tumor cells (CTCs) detection in the syngeneic mouse model of osteosarcoma with spontaneous pulmonary metastases. Recombinant VSV-K was generated and evaluated in vitro on human and murine osteosarcoma cells. Spontaneous osteosarcoma metastases were established in immune-competent mice by implanting subcutaneously syngeneic osteosarcoma LM8 cells. The vector was injected into the tumor-bearing mice via jugular vein either once or repeatedly. To assess effectiveness, primary tumor growth and development of lung metastasis as well as survival were evaluated. We found that rVSV-K efficiently replicated in and killed all osteosarcoma cell lines in time-dependent manners. Both single or repeated systemic injections of the virus did not inhibit the growth of the primary tumor, but the repeated administration could effectively suppress the development of lung metastases and was likely responsible for the observed increase in survival. Furthermore, we demonstrated, for the first time, that CTCs in blood samples from syngeneic osteosarcoma-bearing mice were successfully detected by utilizing rVSV-K ex vivo. Our results show that repeated systemic injections of rVSV-K are an effective anti-metastatic agent against osteosarcoma in immune-competent mice and this virus to be a useful tool for detection of osteosarcoma CTCs, suggesting that further development of future viral-based theranostic approach in patients with osteosarcoma is warranted. © 2018 Orthopaedic Research Society. Published by Wiley Periodicals, Inc. J Orthop Res 36:2562-2569, 2018.