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PubMed2004

High level of beta-hCG simulating pregnancy in recurrent osteosarcoma: case report and review of literature.

PURPOSE: A high serum level of beta human chorionic gonadotropin (hCG) normally indicates pregnancy in healthy women. We were confused by this finding in one of our patients. This 18-year-old girl presented with amenorrhoea of 1-month duration, a positive pregnancy test and a high beta-hCG serum level although taking contraceptives. Pregnancy was excluded by ultrasound. Three years previously, she had had an osteosarcoma of the humerus. The tumour initially had been wide resected and had shown a good response to neoadjuvant chemotherapy with COSS-96-protocol. METHODS: We reviewed the original histological result and the literature about possible similar findings. We analysed therapeutic options and the value of beta-hCG levels as a therapy monitor. RESULTS: During examination we detected a recurrent osteosarcoma of the left humerus. The local relapse evidently expressed beta-hCG which, retrospectively, could only sparsely be shown in the primary resectate. After intralesional surgery, chemotherapy and radiotherapy levels of beta-hCG normalised. CONCLUSION: Osteosarcoma very rarely is able to produce a paraneoplastic syndrome by high levels of beta-hCG. This may well be of diagnostic value and offer an additional monitoring tool. It can indicate tumour recurrence and dedifferentiation.

Adolescent↗
PubMed2000

Biodistribution of 99mTc-glutathione in mice with osteosarcoma: effect of gamma irradiation on tumour uptake.

The aim of this study was to determine the efficacy of 99mTc-glutathione (GSH) in scintigraphic demonstration of osteosarcoma tumour in mice and the effect of gamma irradiation of tumour on tumour uptake of 99mTc-GSH. The biodistribution of 99mTc-GSH was studied in 30 Balb C mice 3 weeks after isotransplanting osteosarcoma OTS-64 in their thighs. The mice were injected with 400 microCi of 99mTc-GSH in 0.1 ml through the tail vein. They were equally divided into two groups. In the second group the tumours were subjected to gamma irradiation for 10 min (20 Gy). The mice in both groups were killed at 1, 3 and 6 h. Scintigrams were obtained at each time point. The organs, tumours, some muscle and some blood were removed, weighed and assayed for radioactivity. Tumour, liver and muscle sections were also obtained for gross autoradiographic studies. The tumours were well visualized on scintigrams. The tumour uptake values as a function of time after injection were 3.27+/-0.80, 1.53+/-0.69, and 1.51+/-0.55 for the control and 5.18+/-1.28, 0.399+/-0.120, and 1.67+/-1.05%/g for the irradiated groups at 1, 3 and 6 h, respectively. The tumor-to-muscle concentration ratios were 34.03+/-12.2, 21.4+/-11.3 and 18.7+/-11.4 for the control and 18.8+/-7.2, 3.63+/-1.9, and 24.1+/-9.0 for the irradiated groups, respectively. The gross autoradiographic images of tumour sections indicated focal sites of increased uptake within tumour tissue, indicating the presence of necrotic areas. In conclusion, 99mTc-GSH accumulated in osteosarcoma and resulted in high tumour-to-other tissue concentration ratios in mice. The increase in uptake values after tumour irradiation might be a result of increased demand of tumour cells for GSH attributable to its well-known biological function as a reducing agent in addition to increased blood flow and capillary permeability in malignant tissues.

Animals↗
PubMed2004

Optimizing the surgical management of lung nodules in children with osteosarcoma: thoracoscopy for biopsies, thoracotomy for resections.

BACKGROUND: The goal of this study was to assess the role of thoracoscopy (TS) and thoracotomy (TT) in the management of lung nodules in children with osteosarcoma. METHODS: Charts of 16 osteosarcoma patients undergoing surgery for lung nodules were retrospectively analyzed for a correlation between nodule localization at CT scan, findings at surgery, and pathology. RESULTS: Fourteen TSs were performed in 10 children, eight of which were converted: two for technical problems, and six for inconsistency between CT scan and intraoperative findings. In three converted cases, TT allowed detection of more nodules than CT scan and/or TS. Eight TTs were performed as primary intention in seven children, in one as secondary surgery after a previous TS. In three cases, TT detected more nodules than CT scan. Overall, TT detected more nodules than CT scan in seven of 16 cases (sensitivity, 56.2%), six of whom had a predicted bilateral involvement. Neoplastic tissue was present in lung samples of all but three patients (86.4%). CONCLUSIONS: Lung nodules in osteosarcoma patients are usually metastases. CT scan is unreliable in detecting all the nodules, especially in the case of predicted bilateral involvement. If excision of all metastases is considered the goal of surgery, a TT approach should be chosen in patients with more than one thoracic nodule.

Adolescent↗
PubMed2004

Primary osteosarcoma of the lung: report of a case.

Osteosarcoma of the lung without an extrathoracic primary tumor is extremely rare, with only eight cases documented in the literature, to the best of our knowledge. We report a case of primary osteosarcoma of the lung found in an asymptomatic 74-year-old woman. Computed tomography showed a heterogeneous mass beside the aortic arch, and the patient underwent a left upper lobectomy. The pathology results confirmed a diagnosis of primary osteosarcoma.

Aged↗
PubMed2004

Skull osteosarcoma: illustrated review.

A case of conventional intramedullary osteosarcoma (epithelioid subtype) with 10 years of evolution and another of high grade surface osteosarcoma of the chondroblastic type, both in the skull, gave rise to several diagnostic and therapeutic difficulties. Key points concerning the definition, classification, imaging, clinical series data and treatment options available for skull osteosarcoma are highlighted.

Adult↗
PubMed2003

Proliferative activity of osteosarcoma cells: comparison of osteoblastic and nonosteoblastic regions.

There are two opinions regarding malignancy evaluation by location for osteoblastic osteosarcoma: One is that the nonosteoblastic region is undifferentiated and the degree of malignancy is high; the other is that the osteoblastic region sometimes shows a marked chemotherapy effect, so the degree of biological malignancy is higher. To clarify this point, we compared and examined the difference in growth ability at intratumoral locations within the same tumor using proliferating cell nuclear antigen (PCNA) immunohistochemical staining and argyrophilic nucleolar organizer regions (AgNORs) staining. The subjects were 10 patients with osteoblastic osteosarcoma at the distal region of the femur. Specimens obtained during surgery were stained with PCNA and AgNORs, after which about 2400 tumor cells were counted per field of view, respectively. In all patients, the nonosteoblastic region showed a higher value than the osteoblastic region for both the PCNA labeling index and AgNORs number. A positive correlation was observed between the PCNA labeling index and the AgNORs number. Both the PCNA-labeling index and AgNORs number in the metastatic foci were higher than in the primary lesions. There are differences in proliferative ability in the intratumoral locations within the same osteoblastic osteosarcoma. Moreover, there are differences in proliferative ability between the metastatic foci and the primary lesion.

Adolescent↗
PubMed2003

Osteosarcoma arising from desmoplastic fibroma treated 16 years earlier: a case report.

We present a rare case of malignant transformation of desmoplastic fibroma in a 37-year-old man. In 1984 we performed curettage and bone grafting for a bone tumor of the left distal femur. Histologically, we obtained a final diagnosis of desmoplastic fibroma. After this treatment, the patient had no particular symptoms but felt tension in the left knee in 2000 and consulted our department. We performed tumor curettage and bone grafting although malignant findings were recognized by imaging. The last pathology diagnosis was an osteosarcoma (in view of the formation of an osteoid). We performed caffeine-assisted chemotherapy and an additional wide excision. In the literature, two reports of a high-grade sarcoma developing in a desmoplastic fibroma have appeared. It is sometimes difficult to distinguish desmoplastic fibroma from low-grade fibrosarcoma or intraosseous-type osteosarcoma, although the radiographic findings and aspects of the resected specimen in 1984 were typical of a desmoplastic fibroma. Moreover, from a clinical standpoint, because the recurrence came 16 years after the initial operation, and lung metastasis was not seen, it is difficult to believe this case was malignant originally. This extremely rare lesion is believed to be a desmoplastic fibroma transformed to osteosarcoma.

Adult↗
PubMed2004

Periosteal osteosarcoma with secondary bone marrow involvement: a case report.

Periosteal osteosarcoma is an exceedingly rare type of chondroblastic osteosarcoma, showing a rather good prognosis, and secondary bone marrow involvement is unusual. However, there have been some reports describing periosteal sarcoma involving medullary bone. We encountered a patient, a 38-year-old man, who had a bone surface tumor in the left tibia. An X-ray showed an erosive cortical mass extraosseous portion, located in the diaphysis of the tibia. Other images revealed a thin cortex, periosteal reactions, coarse mineralization in the extraosseous portion, and bone marrow involvement. Grossly, surgical materials showed that the tumor mainly existed at the periosteal portion, only a part of the cortex was destroyed, and there was medullary involvement throughout. Histological examinations showed a predominantly chondroid component with malignant osteoid formation. On the basis of the histological macroscopic and microscopic findings, we made the diagnosis of periosteal osteosarcoma with secondary bone marrow involvement.

Adult↗
PubMed2003

Inferior vena cava thrombus removal using hypothermic circulatory arrest in two patients with osteosarcoma.

In this case report, the technique of deep hypothermia and circulatory arrest (DHCA) is used to successfully extract extensive symptomatic tumor thrombus from the inferior vena cava (IVC) in two patients with osteosarcoma. This is the first description of the use of this modality for treatment of osteosarcoma involving the IVC. Osteosarcoma and the use of DHCA are discussed.

Adult↗
PubMed2002

Osteosarcoma after bone marrow transplantation for acute lymphoblastic leukemia.

A male patient initially diagnosed with acute lymphoblastic leukemia at age 9 years received chemotherapy (total body irradiation, 12 Gy) followed by allogeneic bone marrow transplantation. Since then, he had been in complete remission. Three years after the bone marrow transplantation, he complained of increasing pain in the right knee. Radiological and histological examinations led to a diagnosis of conventional osteosarcoma. We performed intensive chemotherapy and wide local excision of the osteosarcoma. Intensive chemotherapy was accomplished as planned, although recovery from myelosuppression was delayed during some cycles. Polymerase chain reaction-single-strand conformation polymorphism analysis revealed a p53 gene mutation in exon 7 in the tumor cells, but not in skin or blood cells. This is an extremely rare case of osteosarcoma after bone marrow transplantation.

Bone Marrow Transplantation↗
PubMed1993

Expression and nature of the alkaline phosphatase gene in cultured osteosarcoma cells.

The molecular mechanism for the differences in specific activity of alkaline phosphatase in six human osteosarcoma cell lines was investigated. Five of the lines expressed only the tissue-non-specific or liver/bone/kidney isoenzyme of alkaline phosphatase. The sixth line had the lowest levels of alkaline phosphatase and this was determined to be a mixture of liver/bone/kidney isoenzyme and at least one other form. The mRNA of liver/bone/kidney alkaline phosphatase was identified by Northern analysis in the three cell lines that expressed the largest amount of alkaline phosphatase catalytic activity. This mRNA was indistinguishable in size from that seen in control mRNA from normal kidney (2.5 kb). Southern analysis demonstrated that EcoRI or HindIII restriction fragment patterns and the intensity of the bands, of the liver/bone/kidney alkaline phosphatase gene in the osteosarcoma cell lines were identical to that of the control DNA from normal peripheral blood leukocytes. Thus, the gene coding for liver/bone/kidney alkaline phosphatase appears to be intact in all of these osteosarcoma cells and it is unlikely that rearrangement, deletion or amplification of the gene is responsible for its activation or inactivation. Slot blot analysis revealed varying amounts of the transcripts of the liver/bone/kidney isoenzyme in each of the cell lines. The best fit line of a plot of the log of the level of mRNA of alkaline phosphatase vs. the log of the specific activity of liver/bone/kidney alkaline phosphatase was constructed. This gave a Pearson correlation coefficient of 0.92 (P < 0.008), demonstrating a significant relationship between the two variables. It is likely that the regulation of alkaline phosphatase activity is at the transcriptional process rather than the translational or post-translational processes and that the specific activity of the enzyme may be controlled by the amount of steady-state mRNA of the liver/bone/kidney isoenzyme.

Alkaline Phosphatase↗
PubMed1985

Primary osteosarcoma of the uterine corpus. Report of a case and critical review of the literature.

Uterine osteosarcoma represents an extremely rare pure heterologous sarcoma of the uterus. In the present case osteosarcoma occurred primarily in the uterine cavity of a 56-year-old woman. The tumor was anaplastic and the presence of different proliferative patterns caused differential diagnostic problems with various malignant tumor types. The osteoid features of extracellular matrix were demonstrated by electron microscopy. Clinical and pathological features of previously reported cases of uterine osteosarcoma are critically reviewed.

Aged↗
PubMed1991

Usefulness of antikeratin immunoreactivity in osteosarcomas of the jaw.

The immunohistochemical typing of cytoplasmic intermediate filaments has proved helpful to the pathologist in classifying poorly differentiated malignant neoplasms. In general, identification of keratin-type intermediate filaments has been associated with epithelial histodifferentiation, but several exceptions to this generalization have been reported in the literature. A recent report identified false-positive immunostaining for keratin in osteosarcomas of the jaws that was attributed to cross-reactivity induced by enzyme digestion of the tissue specimens before immunostaining. Because the jaws are unique in the skeletal system because of their relatively high incidence of intraosseous epithelial neoplasms, false-positive immunoreactions for keratin could complicate differentiating sarcomatoid epithelial neoplasms from poorly differentiated osteosarcomas. To evaluate this possible pitfall in our laboratory, eight osteosarcomas of the jaws were evaluated for keratin immunostaining with polyclonal and monoclonal antibodies on tissue sections that had been enzymatically treated with protease. No immunostaining was demonstrated in these tumors. Repudiation of the usefulness of antikeratin immunohistochemistry for intraosseous jaw tumors was not confirmed with the procedures used in our laboratory.

Adult↗
PubMed1991

Statistical observation of osteosarcoma of the maxillofacial region in Japan. Analysis of 114 Japanese cases reported between 1930 and 1989.

In Japan 114 cases of osteosarcoma of the maxillofacial region have been reported during the past 60 years. The average age at the onset of the disease in this region was about one or two decades later than that of osteosarcoma of other regions. Females were affected more frequently (59.6%) than males (40.4%). The mandible was more frequently affected (58.8%) than the maxilla (41.2%). Swelling was the conspicuous symptom for osteosarcoma of the maxillofacial region. Most patients received surgical treatment with or without radiation and chemotherapy; however, the prognosis is poor, regardless of the type of treatment.

Adolescent↗
PubMed1984

Production of antibody to human osteosarcoma associated antigens by continuous human lymphoblastoid cell lines.

Human lymphoblastoid cell lines that produce specific antibody against human osteosarcoma associated plasma membrane antigens have been established by preselecting OSAA binding human B lymphocytes from an osteosarcoma patient followed by Epstein-Barr virus (EBV) transformation. Four cell lines ( Kla -1, Kla -2, Kla -3, Kla -4) were obtained and cloned. The antibodies were lambda light containing IgM. The specificities of the antibodies were confirmed by immunofluorescence assays using tumor cell lines of various histologic types. Positive immunofluorescence was observed with all human osteosarcoma cell lines tested except one but not with tumor cell lines of other histologic types.

Antibodies, Neoplasm↗
PubMed1991

Complete association of loss of heterozygosity of chromosomes 13 and 17 in osteosarcoma.

Mutations of the retinoblastoma (RB1) gene are not confined to retinoblastoma, but are also involved in the development of osteosarcoma. Structural aberrations within the RB1 gene have been studied in fresh samples of eleven cases of osteosarcoma. In five cases a rearrangement was detected, one of which was best explained as a partial duplication. The chromosomal mechanisms by which the nonmutated RB1 allele was lost appeared to be similar in frequency to those that have been reported for retinoblastoma. Loss of heterozygosity was observed for chromosomes 3, 11, 13, 17, and 22. However, when no loss of heterozygosity of chromosome 13 was detected, the other chromosomes retained their heterozygosity as well. A complete association of loss of heterozygosity of chromosomes 13 and 17 was observed. This can be taken as an indication of the involvement of another tumor suppressor gene at chromosome 17 in the initiation of osteosarcoma.

Adolescent↗
PubMed1991

Chromosome 13 alterations in osteosarcoma cell lines derived from a patient with previous retinoblastoma.

Various sublines of cells established from an osteosarcoma that developed in a patient (O.H.) with previous bilateral retinoblastoma were examined for different restriction fragment-length polymorphisms of chromosome 13q, as well as for rearrangements of the retinoblastoma gene using a cDNA probe. The independently established sublines were used to help separate primary and secondary events taking place in tumorigenesis of the osteosarcoma of this patient. Information from the present DNA analysis, taken together with data from cytogenetic and enzymatic studies on chromosome 13 in the cell lines, revealed both common and distinct genetic changes on chromosome 13q. The common changes may indicate the nature of the first and second mutational events in the development of the osteosarcoma. The first, constitutional cancer predisposing mutation seemed to be a base mutation or a small deletion/insertion, and the second event involved a deletion of a larger part of the long arm of chromosome 13. The distinct genetic changes included other deletion and duplication events of chromosome 13q. The existence of multiple sublines with different genetic constitutions provides improved possibilities for gaining insight into the nature of the genetic lesions leading to tumor formation, as these may reflect the clonal variation present in the primary tumor. We also demonstrate the difficulty of inferring from single tumor cell isolates to properties of the primary tumor.

Chromosome Aberrations↗