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Indian Journal of Thoracic and... Jan 2022Parosteal osteosarcoma represents a low-grade, well-differentiated type of cortical surface osteosarcoma with a relatively better prognosis. Systemic metastasis is rare...
Parosteal osteosarcoma represents a low-grade, well-differentiated type of cortical surface osteosarcoma with a relatively better prognosis. Systemic metastasis is rare overall and occurs in 10-15% of patients only. Delayed recurrences (>5 years) of parosteal osteosarcoma are even rarer and only few isolated case reports of local recurrence with systemic metastasis occurring beyond 10 years exist in the literature. We present here a case of femoral parosteal osteosarcoma, successfully treated previously, that recurred with high-grade transformation locally and with lung metastasis after 12 years. The patient was managed by lung wedge resection followed by type A1 rotationplasty. Histopathology report confirmed evidence of high-grade transformation (de-differentiation) both locally and at the metastatic site, in view of strong CDK4 and MDM2 nuclear immuno-expression. The present report highlights the importance of long-term follow-up, even for a seemingly less aggressive osteosarcoma variant with identification of a novel pattern of lung metastasis.
PubMed: 34898877
DOI: 10.1007/s12055-021-01214-0 -
Sarcoma 2022Parosteal osteosarcoma (PAOS) is a surface osteosarcoma. Treatment options include wide excision and endoprosthetic or allograft. However, due to the low local...
BACKGROUND
Parosteal osteosarcoma (PAOS) is a surface osteosarcoma. Treatment options include wide excision and endoprosthetic or allograft. However, due to the low local recurrence and metastasis rate, when it appears in the posterior surface of the distal femur, the lesion can be managed with hemicortical wide resection and biological reconstruction with hemicortical allograft. The purpose of this study is to evaluate the oncological and functional outcomes of patients with parosteal osteosarcoma (PAOS) of the posterior cortex of the distal femur who underwent biological reconstruction after hemicortical resection.
METHODS
Eleven patients who underwent wide tumor resection and defect reconstruction of the posterior surface of the distal femur using hemicortical allograft were retrospectively studied. Local recurrence, metastasis, complications, and the functional outcome using the Musculoskeletal Tumor Society (MSTS) scoring system were evaluated.
RESULTS
The average postoperative follow-up period was 53.64 months (range, 30 to 84 months). At the latest follow-up, all patients had no evidence of disease without metastases. One patient with local recurrence underwent revision surgery with fibula autograft reconstruction. The mean MSTS score was 93.45 ± 3.56.
CONCLUSIONS
Treatment of patients with PAOS of the posterior aspect of the distal femur with hemicortical resection and allograft reconstruction has satisfactory oncological and functional outcome and low complication rates.
PubMed: 35692235
DOI: 10.1155/2022/5153924 -
Case Reports in Oncology 2021Osteosarcoma is the most common primary bone cancer in all age groups. Metastasis mostly occurs with high-grade tumors disseminating to the lungs and other bones. Spread...
Osteosarcoma is the most common primary bone cancer in all age groups. Metastasis mostly occurs with high-grade tumors disseminating to the lungs and other bones. Spread to the pancreas is rare and undocumented in the low-grade subtypes. Additionally, it is uncommon for the disease course of low-grade subtypes to involve multiple relapses. We present a 35-year-old woman with parosteal osteosarcoma who has experienced an atypical metastasis to the pancreas as well as multiple local and pulmonary relapses. The lesion was identified incidentally on routine imaging, and the patient underwent resection. We compare our case to the other reports of pancreatic metastasis in the literature. Despite being especially rare, clinicians ought to be aware of pancreatic metastasis of osteosarcoma. Furthermore, despite parosteal osteosarcoma's less aggressive disease course, it can uncommonly lead to multiple relapses. We present a rare case exemplifying these phenomena in the prognostically favorable histologic subtype of parosteal osteosarcoma.
PubMed: 34188664
DOI: 10.1159/000516033.2021.1.test -
Journal of Structural Biology Jun 2024Osteosarcoma (OS) is the most common malignant primary bone tumor in humans and occurs in various subtypes. Tumor formation happens through malignant osteoblasts...
Osteosarcoma (OS) is the most common malignant primary bone tumor in humans and occurs in various subtypes. Tumor formation happens through malignant osteoblasts producing immature bone. In the present paper we studied two different subtypes of osteosarcoma, from one individual with conventional OS with massive sclerosis and one individual with parosteal OS, based on a multimodal approach including small angle x-ray scattering (SAXS), wide angle x-ray diffraction (WAXS), backscattered electron imaging (BEI) and Raman spectroscopy. It was found that both tumors showed reduced mineral particle sizes and degree of orientation of the collagen-mineral composite in the affected areas, alongside with a decreased crystallinity. Distinct differences between the tumor material from the two individuals were found in the degree of mineralization. Further differences were observed in the carbonate to phosphate ratio, which is related to the degree of carbonate substitution in bone mineral and indicative of the turnover rate. The contraction of the c-axis of the bone mineral crystals proved to be a further, very sensitive parameter, potentially indicative of malignancy.
PubMed: 38871094
DOI: 10.1016/j.jsb.2024.108106 -
International Journal of Surgical... 2020Osteosarcoma arising from cortical surface is classified into parosteal, periosteal and high-grade surface osteosarcoma. Along the spectrum, parosteal osteosarcoma...
Osteosarcoma arising from cortical surface is classified into parosteal, periosteal and high-grade surface osteosarcoma. Along the spectrum, parosteal osteosarcoma occupies the well-differentiated end. It is a relatively rare disease entity, comprised only 4% of all osteosarcomas and barely reported in the literature. The objective of this study is to describe cases of parosteal osteosarcoma as well as a variety of treatment options amenable to such entity. Six cases of parosteal osteosarcoma were identified based on histopathological reports in a tertiary referral hospital in Jakarta, Indonesia between January 2001 and December 2019. The mean age was 29.8 years old; four of them (66.7%) were male. Distal end of femur was the most commonly involved bone (five cases, 83.3%). The patients were treated with wide excision followed by several different reconstruction methods: replacement with endoprosthesis, extracorporeal irradiation, knee arthrodesis, or prophylactic fixation. One of our patients presented with dedifferentiated component, and therefore was treated by limb ablation. While two cases died of pulmonary metastasis, other patients reported fair to excellent functional outcome.
Topics: Adolescent; Adult; Chemoradiotherapy, Adjuvant; Female; Femoral Neoplasms; Follow-Up Studies; Humans; Indonesia; Male; Middle Aged; Osteosarcoma, Juxtacortical; Rare Diseases; Plastic Surgery Procedures; Young Adult
PubMed: 32550023
DOI: 10.1155/2020/4807612 -
Case Reports in Oncology 2021Osteosarcoma is the most common malignant primary bone tumor that occurs most frequently in the second decade of life but rarely in patients over 40 years of age. The...
Osteosarcoma is the most common malignant primary bone tumor that occurs most frequently in the second decade of life but rarely in patients over 40 years of age. The most common primary sites of osteosarcoma are the distal femur followed by proximal tibia and proximal humerus, and involvement of the wrist is extremely rare. Moreover, dedifferentiated osteosarcoma is also a rare condition that progresses to high-grade osteosarcoma from low-grade osteosarcoma, usually central low-grade osteosarcoma or parosteal osteosarcoma that bears MDM2 and/or CDK4 gene amplifications. We herein report an extremely rare case of dedifferentiated osteosarcoma arising in the distal ulna of an adult over 40 years of age. The patient was a 46-year-old man with a 2-month history of pain in his left swollen wrist. The initial radiological findings suggested a benign bone tumor in the distal ulna, and the lesion was marginally excised at the nearby hospital. Although the pathological diagnosis at the nearby hospital suggested a benign cartilaginous tumor, the tumor recurred in an aggressive manner 8 months after the initial surgery. The patient was referred to our hospital, and an incisional biopsy showed a high-grade osteosarcoma. The primary tumor was retrospectively re-evaluated at our hospital and diagnosed as low-grade osteosarcoma. Since neoadjuvant chemotherapy failed to shrink the tumor, the patient had to undergo below the elbow amputation to cure the disease. Although the tumor was negative for MDM2 nor CDK4, the definitive diagnosis of dedifferentiated osteosarcoma was made according to the clinical course and the histological findings. Lung metastases were found 10 months after the amputation, which were successfully treated by neoadjuvant chemotherapy and surgery. The patient has been doing well with no evidence of disease for 1 year and 6 months. Surprisingly, the literature review revealed that many low-grade osteosarcomas of the distal ulna progressed to high-grade dedifferentiated osteosarcomas. One should bear in mind that the diagnosis and treatment for bone-forming tumors of the distal ulna should be made very carefully because, although rare, it is possible that the tumor may initially appear as a benign or low-grade malignant tumor and may progress to high-grade osteosarcoma.
PubMed: 34703440
DOI: 10.1159/000518266 -
BMC Cancer Jun 2024Immunodeficient murine models are usually used as the preclinical models of osteosarcoma. Such models do not effectively simulate the process of tumorigenesis and...
Immunodeficient murine models are usually used as the preclinical models of osteosarcoma. Such models do not effectively simulate the process of tumorigenesis and metastasis. Establishing a suitable animal model for understanding the mechanism of osteosarcoma and the clinical translation is indispensable. The UMR-106 cell suspension was injected into the marrow cavity of Balb/C nude mice. Tumor masses were harvested from nude mice and sectioned. The tumor fragments were transplanted into the marrow cavities of SD rats immunosuppressed with cyclosporine A. Through muti-rounds selection in SD rats, we constructed orthotopic osteosarcoma animal models using rats with intact immune systems. The primary tumor cells were cultured in-vitro to obtain the immune-tolerant cell line. VX2 tumor fragments were transplanted into the distal femur and parosteal radius of New Zealand white rabbit to construct orthotopic osteosarcoma animal models in rabbits. The rate of tumor formation in SD rats (P1 generation) was 30%. After four rounds of selection and six rounds of acclimatization in SD rats with intact immune systems, we obtained immune-tolerant cell lines and established the orthotopic osteosarcoma model of the distal femur in SD rats. Micro-CT images confirmed tumor-driven osteolysis and the bone destruction process. Moreover, the orthotopic model was also established in New Zealand white rabbits by implanting VX2 tumor fragments into rabbit radii and femurs. We constructed orthotopic osteosarcoma animal models in rats with intact immune systems through muti-rounds in-vivo selection and the rabbit osteosarcoma model.
Topics: Animals; Osteosarcoma; Rabbits; Rats; Disease Models, Animal; Bone Neoplasms; Cell Line, Tumor; Mice; Mice, Nude; Rats, Sprague-Dawley; X-Ray Microtomography; Mice, Inbred BALB C; Immunocompetence; Humans; Neoplasm Transplantation; Femur; Male
PubMed: 38849717
DOI: 10.1186/s12885-024-12361-z -
Malaysian Orthopaedic Journal Mar 2023Mediastinal germ cell tumours are a rare group of extragonadal germ cell tumours with less than 5% prevalence of all germ cell tumours. Primary mediastinal germ cell...
Mediastinal germ cell tumours are a rare group of extragonadal germ cell tumours with less than 5% prevalence of all germ cell tumours. Primary mediastinal germ cell tumours themselves account for 16-36% of the extragonadal germ cell tumours. Along the spectrum of osteosarcoma, parosteal osteosarcoma is a well-differentiated surface osteosarcoma with a prevalence of 4% of all osteosarcoma. As such synchronous primary parosteal osteosarcoma and primary mediastinal germ cell tumour are exceedingly rare. This leads to complexity in determining the most appropriate chemotherapy for two different types of tumours and its potential side effects of reduced immunity leading to potential secondary infection. Here we report a case of a 16-year-old boy who presented with synchronous primary osteosarcoma and primary mediastinal germ cell tumour, complicated with atypical mycobacterial infection post-operatively. Additionally, we discuss our choice of chemotherapy and the management of the atypical mycobacterial infection.
PubMed: 37064628
DOI: 10.5704/MOJ.2303.023 -
Orthopaedics & Traumatology, Surgery &... Jun 2022Proximity of tumor to the neurovascular bundle (NVB) makes achieving a wide margin difficult. In low-grade parosteal osteosarcoma (POS), it is not clear whether...
BACKGROUND
Proximity of tumor to the neurovascular bundle (NVB) makes achieving a wide margin difficult. In low-grade parosteal osteosarcoma (POS), it is not clear whether adherence of tumor to NVB increases the rate of local recurrence (LR). In this study, we evaluated whether tumor adherence to the NVB increases the risk of LR in low-grade POS of the distal femur?
HYPOTHESIS
We hypothesized that if the thin neurovascular barrier (the adventitia of the vessels and the epineurium of the nerve) prevents tumor penetration, the rate of LR should be comparable between the lesions with and without a continuous layer of healthy fatty tissue between the tumor and NVB.
MATERIALS AND METHODS
In a retrospective survey, 30 patients with low-grade POS of the posterior aspect of the distal femur were evaluated for the proximity of mass to NVB. Based on the proximal to distal T1 axial MRI sections, the tumors were divided into two groups including the tumors with an uninterrupted (group A) and interrupted (group B) rim of fatty tissue between the mass and NVB. The rate of LR was compared between the two study groups. The concordance of MRI in detecting NVB adherence was checked with pathology specimen.
RESULTS
Using MRI, we identified 16 cases in group A and 14 cases in group B. The MRI status of fatty rim was concordant with pathology specimen in 96.4% of cases. The mean follow-up period of the two groups was not statistically different (117±27.6 vs. 105.8±29.4 months, respectively, p=0.29). The other baseline characteristics of the two groups were statistically comparable, as well. The rate of LR was 12.5% (2 out of 16 patients) in group A and 14.3% (2 out of 14 patients) in group B (95% CI: 0.142-9.586, p=0.87). The 10-year recurrence-free survival was 87.5% for group A and 85.7% for group B (p=0.9).
DISCUSSION
The absence of a continuous rim of fatty tissue between the tumor and NVB in MRI does not increase the risk of LR in low-grade POS of the distal femur.
LEVEL OF EVIDENCE
IV.
Topics: Bone Neoplasms; Femur; Humans; Osteosarcoma; Osteosarcoma, Juxtacortical; Recurrence; Retrospective Studies
PubMed: 35093566
DOI: 10.1016/j.otsr.2022.103220 -
Radiology Case Reports Oct 2023Parosteal osteosarcomas (POS) are well-differentiated low-grade malignant sarcomas that are located on the surface of the bone. POS of the temporal bone is exceptionally...
Parosteal osteosarcomas (POS) are well-differentiated low-grade malignant sarcomas that are located on the surface of the bone. POS of the temporal bone is exceptionally rare, with less than a hand full of cases present in modern literature. Here, we report a POS of the temporal bone found incidentally and with an uncharacteristic presentation. We also review the unique imaging and histopathological findings of this entity and discuss why developing a broad differential diagnosis and proceeding with early intervention are considered imperative in this disease.
PubMed: 37577081
DOI: 10.1016/j.radcr.2023.07.038