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PubMed1992

[Alveolar rhabdomyosarcoma with massive bone marrow involvement and 2;13 chromosome translocation].

We report a 9-year-old girl who had massive bone marrow infiltration of tumor cells at the onset of alveolar rhabdomyosarcoma. She was admitted to a surgical hospital because of abdominal pain and tumor in the buttock. Computerized tomographic scans of the pelvis revealed an abnormal mass. She was referred to our department. Hematological examination showed pancytopenia. Blood chemistry revealed hypercalcemia and hyperuricemia. Bone marrow was occupied with 100% tumor cells. Chromosome analysis of tumor cells in bone marrow revealed a specific translocation, t(2;13) (q37;q14). This finding enabled the diagnosis of a disseminated alveolar rhabdomyosarcoma to be established. She was treated with vincristine, cyclophosphamide and epirubicin, achieved a complete remission, but died of relapse 3 months after diagnosis.

Bone Marrow Diseases↗
PubMed2005

Rhabdomyosarcoma of the middle ear and mastoid: a case report and review of the literature.

We report a case of rhabdomyosarcoma of the middle ear and mastoid in a 3-year-old boy. The patient was treated according to Intergroup Rhabdomyosarcoma Study IV protocol (chemo- and radiotherapy), and he experienced a complete remission. However 7 months after the completion of treatment, he experienced a recurrence at the primary site that spread to the brain. Despite treatment, the patient died of progressive metastasis to the lung 4 months later.

Child, Preschool↗
PubMedDate not supplied

TRAIL-induced cytotoxicity in a melphalan-resistant rhabdomyosarcoma cell line via activation of caspase-2.

The prognosis for patients with chemo-refractory rhabdomyosarcoma remains poor. The tumour necrosis factor-related apoptosis-inducing ligand (TRAIL) is a hopeful candidate for new strategies in chemotherapy. The effects of TRAIL and melphalan (Mel) in the rhabdomyosarcoma cell line TE-671 were investigated by colorimetric caspase assays and flow cytometry. TRAIL induced the activation of caspases-2, -3 and -8, but not the activation of caspase-9, in the Mel-resistant TE-671 cells. Inhibition of caspase-2 with the caspase-2 inhibitor z-VDVAD-fmk significantly down-regulated the TRAIL-induced caspase-3 activation, as well as the TRAIL-induced cytotoxicity. When TE-671 cells were treated with a combination of Mel and TRAIL, a significant synergism of drug-induced cytotoxicity was obtained. The inhibition of caspase-2 could completely abolish caspase-3 activation, suggesting that TRAIL sensitises TE-671 cells for Mel-induced cytotoxicity via a caspase-2- and -3-dependent mechanism. In conclusion, it was shown, for the first time, that TRAIL could sensitise Mel-resistant tumour cells to melphalan.

Antineoplastic Combined Chemotherapy Protocols↗
PubMed1991

Immunocytochemistry in fine needle aspirates of small cell-, round-, blue-cell malignant tumors of childhood (neuroblastoma, nephroblastoma, lymphoma, Ewing's sarcoma, rhabdomyosarcoma).

Immunocytochemistry with the use of monoclonal antibodies to vimentin, keratin, desmin, neurofilaments and leukocyte common antigen (LCA) was applied to 53 fine needle aspirates of small cell-, round-, blue-cell malignant tumors of childhood (neuroblastomas, nephroblastomas, non-Hodgkin's lymphomas, Ewing's sarcomas and rhabdomyosarcomas). The results revealed the following immunophenotypes: neuroblastomas were positive for neurofilaments. Nephroblastomas contained vimentin (in blastema cells), keratin (in epithelial and blastema cells) Non-Hodgkin's lymphomas were vimentin and LCA positive, keratin negative. Ewing's sarcomas displayed multidirectional differentiation. Rhabdomyosarcomas were desmin and vimentin positive. It is concluded that immunocytochemistry in fine needle aspirates should be used as an objective evidence to support morphologic differential diagnosis of small cell-, round-, blue cell malignant tumors of childhood.

Biopsy, Needle↗
PubMed1992

Rhabdomyosarcoma spheroids with central proliferation and differentiation.

A novel type of multicellular spheroids was established and characterized with regard to growth behavior, proliferation, and differentiation. The spheroids were grown from clonal rat rhabdomyosarcoma cells using the spinner flask technique. The cell aggregates showed several unique properties that were different from those observed in most of the spheroids investigated to date. These properties include a non-Gompertzian volume growth; the coexistence of undifferentiated mononuclear cells and of differentiated, myotube-like giant cells with numerous nuclei; a relatively homogeneous intraspheroidal distribution of proliferating mononuclear cells with thymidine labeling even in the center of spheroids greater than 1 mm; the absence of necrosis in such large spheroids, and the accumulation of myotube-like cells in the center of these spheroids instead. There was a decrease in the overall proliferative activity of the mononuclear cells with increasing proportion of giant cells in the rhabdomyosarcoma spheroids.

Animals↗
PubMed1992

Rhabdomyosarcoma of the petrous ridge. CT and MR imaging in an atypical case with multiple cranial nerve palsy.

The CT and MR findings are reported in a case of biopsy proven rhabdomyosarcoma of the skull base. The tumor presumably originated in a pneumatized petrous ridge and had an atypical presentation of multiple cranial nerve palsy. The lesion exhibited a soft tissue density and a nonexpansile bone destruction on unenhanced CT. On MR imaging the lesion showed homogeneous intermediate signal intensity on T1 weighted images and a high signal intensity on proton density and T2 weighted images. The scanty literature on CT and MR features of rhabdomyosarcoma of the head and neck is reviewed.

Child, Preschool↗
PubMed1991

[A case of chest wall rhabdomyosarcoma].

A 56-year-old man was admitted to our hospital with right chest pain. Chest X-ray, CT scan and MRI revealed a chest wall tumor and enlarged mediastinal lymph nodes. Percutaneous lung biopsy was performed, and the pathological diagnosis of pleomorphic rhabdomyosarcoma was obtained. The only significant abnormal laboratory finding was elevation of serum NSE (24.5 ng/ml). Although chemotherapy (VAC-ADM) and radiation therapy were performed, the patient died about 7 months after admission. To our knowledge, only 17 cases of chest wall rhabdomyosarcoma have been reported in Japan.

Antineoplastic Combined Chemotherapy Protocols↗
PubMedDate not supplied

Unusual leukemic presentation of rhabdomyosarcoma: report of two cases with immunological, ultrastructural and cytogenetical studies.

BACKGROUND: Bone marrow infiltration occurs rarely at presentation of rhabdomyosarcoma (RMS) or other childhood solid tumors. This possibility leads to misdiagnosis of leukemia and incorrect therapies might be administered. METHODS: We report two patients presenting with diffuse bone marrows involvement by neoplastic cells. Initial studies were not consistent with a diagnosis of leukemia and the cases were further studied extensively by indirect immunofluorescence, immunocytochemistry, electron microscopy and cytogenetics. RESULTS: In both cases blast cells were large, poorly differentiated, with immunological reactivity to the anti-desmin antibody. Ultrastructural findings of muscular features and chromosomal translocation t(2;13) (q37;q14) further confirmed the diagnosis of rhabdomyosarcoma of the alveolar subtype. This was then confirmed histologically in one patient. CONCLUSION: This study stresses the utility of analyzing cases of morphologically undifferentiated marrow blast cells by various techniques, as well as investigating for different types of both hematological and solid neoplasms.

Adolescent↗
PubMedDate not supplied

Clinical, ultrasonographic and CT evaluation of orbital rhabdomyosarcomas with management.

Fourteen cases of orbital rhabdomyosarcoma have been studied. Clinical presentation was by way of short history, fast growing proptosis, inflammatory signs and epistaxis. Computerised tomography showed only a mass lesion without bony involvement and was very useful in follow up. Ultrasonography revealed pseudo-encapsulation of the tumour which is postulated as possibly specific of Rhabdomyosarcoma due to its fast growth, cellular content and oedema fluid. A survival rate of 21% over a 3 year period has been achieved by combined mode therapy consisting of surgery, radiation and chemotherapy.

Adolescent↗
PubMed1991

Reduced metastatic ability of in vitro differentiated human rhabdomyosarcoma cells.

We studied the human embryonal rhabdomyosarcoma cell line RD and 8 derivatives obtained in our laboratory either by cell cloning or by culturing in vitro cells from tumors or secondaries grown in nude mice. The expression of desmin and of the embryonic isoform of myosin and the formation of multinuclear myotube-like structures were studied as specific markers of myogenic differentiation. During continuous growth, each derivative contained a proportion (ranging from 5 to 80% among derivatives) of desmin-positive cells and a small number of myosin-positive or multinuclear elements. Cells from continuous cultures were injected intravenously in nude mice, producing lung and kidney/adrenal nodules. No correlation was found between the proportion of cells expressing desmin and metastatic capacity. When cultures were grown in differentiation medium (Dulbecco's minimal essential medium + 2% horse serum) some derivatives (designated type A) showed a strong increase in the proportion of myosin-positive cells, while others (type B) showed no increase. In vitro differentiation significantly reduced the metastatic ability of type A cells, while no modification was observed in type B after growth in differentiation medium. The proliferative ability of type A and type B cells grown in differentiation medium did not correlate with the proportion of myosin-positive cells, and extensive formation of multinuclear myotubes was never observed. It was concluded that reduction of experimental metastatic ability was mediated by events related to late, though not necessarily terminal, differentiation of rhabdomyosarcoma cells.

Animals↗
PubMed1991

Molecular definition in a somatic cell hybrid of a specific 2:13 translocation breakpoint in childhood rhabdomyosarcoma.

A consistent, balanced, reciprocal chromosomal translocation t(2:13) (q35:q14) has been identified in alveolar rhabdomyosarcoma. Somatic cell hybrids have been constructed between rhabdomyosarcoma cell lines carrying the (2:13) translocation and mouse 3T3 cells. One hybrid cell line was shown to have retained the derivative (13:2) chromosome, but segregated the normal chromosome 13 and the derivative (2:13) chromosome. Using available DNA probes from human chromosome 13 we find that the loci for retinoblastoma, esterase D, p7D2, pG24E6.8 and pG14E1.9 lie distally to the 13q14 breakpoint, whereas those for p7F12, pHU10 and pG2E3.1 all lie proximally. Thus we have defined a region of 13q14 of approximately 28mB which contains the breakpoint associated with this rearrangement.

Animals↗
PubMed1991

Primary rhabdomyosarcoma of the ileum in an adult.

We report a case of primary rhabdomyosarcoma of the ileum occurring in a 45-year-old man. The rhabdomyoblastic nature was immunohistochemically confirmed by positivity with anti-myoglobin and anti-striated actin antisera. We discuss the differential diagnosis with similar tumors. To our knowledge, ileal rhabdomyosarcomas have not been previously reported in adults.

Humans↗
PubMed1991

Breast metastases after bone marrow transplantation for rhabdomyosarcoma.

Two adolescent females with metastatic alveolar rhabdomyosarcoma underwent intensive chemoradiotherapy and bone marrow transplantation, one autologous and one allogeneic. Recurrence was detected in the breasts of both patients, the first at autopsy and the other as the only site of treatment failure. Breast masses after bone marrow transplantation should be thoroughly investigated for metastatic disease in patients with rhabdomyosarcoma.

Adolescent↗
PubMed1990

12-O-tetradecanoylphorbol-13-acetate-induced differentiation of a human rhabdomyosarcoma cell line.

The effect of 12-O-tetradecanoyl phorbol-13-acetate (TPA) on proliferation and differentiation of the human embryonal rhabdomyosarcoma cell line RD was investigated. The proliferation of RD cells is drastically and reversibly inhibited by 100 nM TPA. The effect is evident after 24 h of treatment and is maximal after 50-70 h. The reduction of proliferation in treated cells is followed by increased expression of differentiative characters such as a large increase in muscle myosin expression and in the binding of 125I-alpha-bungarotoxin. Moreover TPA induces the appearance of myotube-like structures, which contain bundles of thick and thin myofilaments along with Z bodies. The described effects are not observed if the TPA-containing medium is replaced daily, thus suggesting that these effects might be related to substances secreted by treated cells. The phosphorylation of three proteins is significantly stimulated by TPA within minutes of its administration to RD cells. Although with a different pattern, the stimulation of protein phosphorylation is still clearly detectable after 6 days of incubation with TPA. These results on human rhabdomyosarcoma cells are, to our knowledge, the first evidence for a growth-inhibiting and a differentiative effect of TPA on a solid tumor of mesodermal origin.

Cell Differentiation↗
PubMed1990

Morphological and molecular characterization of spontaneous myogenic differentiation in a human rhabdomyosarcoma cell line.

Skeletal muscle differentiation consists of an ordered withdrawal of committed cells from the cell cycle and their fusion to form multinucleated myotubes. To determine if differentiation of malignant myoblasts parallels that of normal skeletal muscle, a cell line (Rh28) was established from an alveolar rhabdomyosarcoma. Rh28 displays a constant population doubling time of 45-55 h until passage 60, when the doubling time progressively increases until proliferation ceases. Loss of proliferative capacity is associated with morphological evidence of differentiation to multinucleated myotubes, fusion, and the expression of numerous muscle-specific genes. In contrast to normal myogenic differentiation, multinucleated cells continue to synthesize DNA and express abundant c-myc transcripts. These observations suggest synchronous replication and possible arrest in the G2-phase of the cell cycle, since there was no evidence of mitotic activity in differentiated cells. Terminal differentiation of early passage Rh28 cells was induced in the presence of 10% dialyzed fetal calf serum but not by medium containing 2% undialyzed serum, suggesting a role for low molecular weight growth factors in this process. Our data indicate that the Rh28 cell line may be of value in elucidating the relationship between oncogenic transformation and differentiation in rhabdomyosarcoma.

Cell Differentiation↗
PubMed1990

[A case of paratesticular rhabdomyosarcoma].

A case of paratesticular rhabdomyosarcoma in an 18-year-old male is reported. The patient was admitted for swelling of the right scrotum and right inguinal pain. Malignant tumor of the right testis was suspected and a high inguinal orchiectomy was performed. The pathological diagnosis was paratesticular rhabdomyosarcoma. Following retroperitoneal lymph node extirpation, chemotherapy, so-called CYVADIC therapy, consisting of cyclophosphamide, vincristine, adriamycin and dacarbazine was employed in 3 regions. The patient is in good health without recurrence 16 months after the surgery.

Adolescent↗
PubMedDate not supplied

[A case of rhabdomyosarcoma of the parotid region].

The present paper reports a case of rhabdomyosarcoma in a 4-year-old patient, the rapidity with which it spread and the intensity of its extension bringing the case under observation. It is, in fact, a case of embryonal rhabdomyosarcoma localized in the right parotid area with an extension to the temporal fossa, to the epi-oro-hypopharynx as far as the lateral vertebral region with C5-C6 involvement and penetrating the medium cranialis fossa. Initially the child showed signs of tissue growth at the right parotid site, originating without symptoms and rapidly increasing in volume. As exploratory surgery revealed that it would be impossible to achieve radical removal of the growth, the authors opted for radio-chemotherapy treatment. This type of approach, however, gives short lived results, in this case evidenced by recurrences within six months and leading to the patient's death after just eight months. It is important to stress the rapid evolution of this neoplasm, its tendency to recur and metastasize as well as its poor prognosis in general.

Child, Preschool↗
PubMedDate not supplied

[Case of rhabdomyosarcoma of the maxillary bone].

The paper describes a case of embryonal rhabdomyosarcoma of the maxillary antrum which, according to the Intergroup Rhabdomyosarcoma Study (IRS) protocol, was treated with a protocol which included cycles of chemotherapy and radiotherapy.

Adult↗