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1/56. Immature hepatic tumor of bimodal differentiation in a young adult patient: a novel lesion expressing beta-catenin and mimicking a distinct phase of hepatogenesis.

    PATIENT AND methods: A large right-sided liver tumor was incidentally discovered and totally resected in a 21-year-old female patient without chronic liver disease. Histopathologic examination revealed that this malignant tumor does not fit any of the known types of primary liver tumors reported thus far. RESULTS: The lesion chiefly consisted of numerous solid nests composed of immature epithelial cells disclosing a marker profile typical for hepatocytes and strongly expressing beta-catenin. These nests were tightly associated with abnormal duct-like profiles exhibiting features of bile duct cells. CONCLUSIONS: The finding of numerous neoplastic hepatobiliary units ('liverlets') suggests that this tumor may mimic a distinct developmental phase of hepatogenesis close to ductal plate formation.
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2/56. A case of undifferentiated (embryonal) sarcoma of the liver: fine needle aspiration cytology diagnosis by one cell type.

    We present the first case in bulgaria of cytologically verified undifferentiated (embryonal) sarcoma of the liver. A 10-year-old girl was admitted to the hospital with abdominal pain, extreme hepatomegaly, one-month fever, anemia and highly elevated erythrocyte sedimentation rate (95 mm). A large formation was found in the right liver lobe on ultrasound examination, which was polycystic on CT scan. Neither bone marrow abnormalities nor elevation of serum alpha-fetoprotein was found. In the scanty specimen obtained by fine-needle aspiration biopsy under ultrasound control a characteristic prevalence of polygonal cells within mesenchyme tissue was found. The cells had eccentric hyperchromic nuclei which were pushed to the periphery by large clusters of PAS-positive, diastase-resistant granules. vimentin and alpha 1-antitrypsyn were expressed immunohistochemically. The cells are pathognomonic of undifferentiated (embryonal) sarcoma of the liver. After preoperative chemotherapy with ifosfamide, vincristine and actinomycin D (CEVAIE, CWS--96) resection of the tumor including IV, V, VI and VII liver segments was performed. A 30 x 30-cm soft gray-whitish tumor with fine capsule was found. The cytological diagnosis was histologically verified. The patient had a mechanical subileus episode after the 6th course of chemotherapy. One year after the surgery she is in good condition. The histogenesis of this rare tumor is discussed regarding its unique cytology, immunohistochemical constellation and ultrastructure. A literature review is presented. The case indicates that the unique cell appearance in embryonal sarcoma of the liver provides a correct diagnosis even in a scanty specimen.
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3/56. Lectin-reactive alpha-fetoprotein (AFP-L3%) curability and prediction of clinical course after treatment of non-seminomatous germ cell tumors.

    OBJECTIVE: Alpha-fetoprotein (AFP) is an important tumor marker for non-seminomatous germ cell tumors (NSGCTs) that greatly affects diagnosis and the evaluations of therapy and therapeutic policy. However, it is sometimes very difficult to make the distinction between tumors and falsely elevated AFP levels due to benign liver disease. We assessed the usefulness of lectin-reactive alpha-fetoprotein (AFP-L3%), which has been reported to be superior to total AFP in both sensitivity and specificity in hepatocellular carcinomas, for the evaluation of predictions of clinical courses after the treatment of NSGCTs. methods: Frozen sera of 25 tumor-bearing patients with testicular cancers whose AFP levels were 5.0 ng/ml or higher were used. The total AFP levels and the ratio of L3 fraction to total AFP (AFP-L3%) were measured by liquid-phase binding assay (LBA). RESULTS: The total AFP levels were 6.3-14 907 ng/ml (median: 105.9 ng/ml). The median AFP-L3% was 69.9% (range;: 1.1-88.1%). Except for one patient, 24 patients (96.0%) with evident disease showed high levels of AFP-L3% of >50%, regardless of their total AFP levels. In nine patients whose sera were sequentially measured, AFP-L3% was considered highly useful for the detection of residual tumors (n = 2) and recurrence (n = 1) and for the exclusion of false-positive cases (n = 1). CONCLUSIONS: When the total AFP level increases slightly (e.g. to 5-20 ng/ml), a measurement of AFP-L3% may provide additional useful information for monitoring NSGCT patients and in distinguishing falsely elevated AFP.
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4/56. Ossifying pleomorphic adenoma of the maxillary antrum.

    An unusual case of pleomorphic adenoma with exuberant bone formation, occurring in the maxillary antrum of a 21-year-old male and showing repeated recurrence, is reported. In contrast to the endochondral ossification in the previous reported cases of pleomorphic adenoma, direct deposition of osteoid by metaplastic myoepithelial cells is suggested to explain the bone formation. The difficulties of distinguishing this tumour from osteosarcoma during intraoperative diagnosis are discussed.
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5/56. Primitive neuroectodermal tumors after prophylactic central nervous system irradiation in children. association with an activated K-ras gene.

    Three patients had supratentorial malignant brain tumors 7 to 9 years after prophylactic central nervous system (CNS) treatment for acute lymphocytic leukemia or malignant T-cell lymphoma. Therapy was administered at the age of 3 to 8 years and included cranial irradiation (total dose, 1800 to 2400 cGy) and intrathecal methotrexate. The brain tumors had histologic and immunohistochemical features of primitive neuroectodermal tumors (PNET), including neuroblastic rosettes, rhythmic arrangement of tumor cells, and immunohistochemical expression of glial, and in one patient neuronal, marker proteins. Using polymerase chain reaction-mediated dna amplification from paraffin-embedded tissues and subsequent dna sequence analysis, an activating point mutation was detected in the K-ras protooncogene in one tumor. This mutation was a G to A transition in position 2 of codon 12, substituting aspartate (GAT) for glycine (GGT). This type of mutation has not been observed before in human brain tumors, but it is frequent in radiation-induced murine lymphomas. These observations suggest that PNET can be induced after completion of the embryonal and fetal development of the human CNS. Oncogene-activating point mutations may represent a pathogenetic mechanism involved in the genesis of radiation-induced brain tumors.
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ranking = 6569.0202159937
keywords = rosette
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6/56. The utility of ancillary studies in pediatric FNA cytology.

    We evaluated the diagnostic contribution of adjunct studies performed on aspirated material in the work-up of pediatric fine-needle aspiration (FNA) biopsies. Ancillary studies were performed on 54 of 136 (39.7%) pediatric FNA biopsies during a 5-year period. In 23 (16.9%) cases, immunocytochemical (ICC) studies, consisting of immunoperoxidase staining of direct smears and/or cell blocks or flow cytometric immunophenotyping, were performed. The studies were adequate in 14 cases (60.9%), suboptimal in five cases (21.7%), and inadequate in four cases (17.4%). Of the adequate and suboptimal cases, the ICC data helped to narrow the differential diagnosis or classify the disease process in eight cases (42.1%), confirmed cytologic impression in nine cases (47.4%), and gave contradictory results in two cases (10.5%). Adequate material for electron microscopy (EM) was obtained in 14/19 cases (73.7%). Ultrastructural studies were diagnostic, or helped classify the disease process in five cases (35.7%), confirmed the cytologic impression in four cases (28.6%), helped exclude diagnostic considerations in three cases (21.4%), and were judged to be non-contributory in two cases (14.3%). Cytogenetic studies revealed six of seven cases (all neoplasms) to have abnormal karyotypes. Special stains for organisms performed on smears from 25 cases including Ziehl-Neelsen, Gomori methenamine silver (GMS), Gram, and Warthin-Starry (WS) were negative except for 1/16 GMS and 4/9 Gram stains. In summary, we found that with appropriate case selection, ancillary studies performed on aspirated material can provide useful information in pediatric FNA cytology.
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7/56. Melanotic neuroectodermal tumour as a predominant component of an immature testicular teratoma. Case report with immunohistochemical investigations.

    A case of melanotic neuroectodermal tumour (MNT), or so-called retinal anlage tumour, as a predominant component of an immature testicular teratoma is presented. The patient was a 17-year-old man who furthermore had a mature mediastinal teratoma. The MNT was composed mainly of two cell types: small immature neuroblast-like cells and large columnar or cuboidal epithelial-like cells with or without melanin granules. The tumour cells were arranged in solid formations, nests, cords, alveolar and pseudoglandular structures with cleft-like or glomeruloid-like spaces. Myogenic differentiation was found in minor foci. immunohistochemistry showed both neuroepithelial and mesenchymal features with positive staining reaction for neuron-specific enolase (NSE), S-100 protein (S-100), melanoma antigen (HMB45), cytokeratin and vimentin. vimentin, desmin and actin were present in the myoid cells. To the best of our knowledge this is the first reported case of MNT originating in the testis. As this tumour component occurred in an immature teratoma, neuroectodermal differentiation of germ cell origin is considered most likely.
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8/56. Embryonal carcinoma arising in Turner's syndrome.

    risk of malignant transformation in a dysgenetic gonad is high. This is a report of a 23 year-old female who presented with symptoms and signs of ovarian neoplasm which was histologically confirmed as embryonal carcinoma. She had features of Turner's syndrome and was found to have XO chromosomal constitution. Embryonal carcinoma arising in a dysgenetic gonad is uncommon. It is even rarer when it arises in a patient with pure 45 XO. The following is a case report which highlights the role of chemotherapy and surgery in the management. It includes a literature review on the clinical features, genetic variants and malignant transformation in dysgenetic gonad. The role of prophylactic removal of dysgenetic gonad is discussed.
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keywords = formation
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9/56. A case of simultaneous bilateral nonseminomatous testicular tumors in persistent mullerian duct syndrome.

    Persistent mullerian duct syndrome is characteristically associated with unilateral or bilateral cryptorchidism. Like other undescended testes, these gonads are at an increased risk of malignant transformation. We report a case of synchronous bilateral mixed germ cell tumors in the cryptorchid testes of a patient with the persistent mullerian duct syndrome.
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10/56. Myelodysplastic syndrome (RARS) with i(12p) abnormality in a patient 10 months after diagnosis and successful treatment of a mediastinal germ cell tumor (MGCT).

    We report on a 21-year-old man with a mediastinal germ cell tumor (MGCT) who developed a myelodysplastic syndrome (MDS) (refractory anemia with ringed sideroblasts, RARS) 10 months after the start of successful treatment with cisplatin, etoposide, ifosfamide, and paclitaxel. A very rare early occurrence of a therapy-related MDS was suspected. cytogenetic analysis of the bone marrow revealed an aberrant karyotype, showing a deletion in 12p, an isochromosome 5p, as well as gain of an isochromosome 12p. Isochromosome 12p is a specific aberration frequently observed in MGCT. It also was described in patients who developed hematological transformation of a mediastinal germ cell tumor. In this report the association between mediastinal germ cell tumors and hematological malignancies including the possibility of a common genetic origin is discussed.
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