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1/11. paraganglioma of the thyroid: two cases that clarify and expand the clinical spectrum.

    BACKGROUND: Paragangliomas (PGs) can, on rare occasions, arise within the thyroid parenchyma presumably from displaced laryngeal paraganglia. On the basis of a limited number of reported cases, thyroid PGs invariably affect women, they are always benign, and they are usually mistaken for some other more common thyroid lesion. methods: We describe the histopathologic features, immunohistochemical findings, and clinical characteristics of two thyroid PGs. RESULTS: One tumor was incidentally discovered in a 55-year-old man during evaluation of a carotid bruit. The other tumor aggressively invaded the trachea and esophagus of a 52-year-old woman with a presumed long-standing nodular goiter. In both cases, the initial pathologic evaluation suggested medullary thyroid carcinoma. Both patients are alive without recurrent disease after surgical resection. CONCLUSIONS: These cases emphasize the need to consider PG in the differential diagnosis of neuroendocrine thyroid tumors, even in those tumors involving men or behaving in a locally aggressive fashion. Failure to do so carries grave implications regarding patient prognosis and management.
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2/11. Clear-cell odontogenic carcinoma with pulmonary metastases resembling pulmonary meningothelial-like nodules.

    Clear-cell odontogenic carcinoma (CCOC) is a rare neoplasm with malignant potential and unknown cytogenetic alterations. We describe the case of a 43-year-old woman who presented with an unusual odontogenic epithelial tumor. Histologically, the tumor was composed of clear-cell areas and exhibited a squamous pattern with little nuclear pleomorphism similar to benign squamous odontogenic tumor. Multiple small pulmonary nodules occurring 3 years after primary surgical treatment histologically closely resembled benign minute pulmonary meningothelial-like nodules (MPMN) with clear-cell features. comparative genomic hybridization (CGH) and immunohistochemistry, performed as diagnostic adjuncts, revealed in the odontogenic tumor and the pulmonary lesions a very similar pattern of chromosomal aberrations (loss of 9, gains of 14q, 19 and 20 in both, and additional loss of 6 in the odontogenic tumor) and the same pattern of expression (positive for cytokeratin 5, 6, 8, 19 and negative for cytokeratin 18, epithelial membrane antigen, and vimentin), differing from that of MPMN. These findings confirmed the final diagnosis of metastasizing CCOC with partial squamous differentiation, substantiated the unfavorable prognosis of the clear-cell component, and highlighted the diagnostic impact of CGH and immunohistochemistry for classification of these morphologically peculiar pulmonary CCOC metastases.
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3/11. Carney triad: case report and molecular analysis of gastric tumor.

    gastrointestinal stromal tumors (GISTs) are the most common mesenchymal tumors of the GI tract. Most of them are thought to be sporadic, but some arise in the settings of neurofibromatosis type I (NF-1) and the Carney triad. The Carney triad is a syndrome of unknown etiology, occurring predominantly in young females, comprising gastrointestinal stromal tumors, pulmonary chondromas, and extra-adrenal paragangliomas. GISTs of the Carney triad involve predominantly the body and the antrum of the stomach, are generally multifocal, and have a better prognosis than sporadic GISTs. We describe the clinical and pathological features of a case of Carney triad that featured multiple gastric GISTs, mediastinal paraganglioma, and esophageal leiomyoma. Ten years after gastric resection, the patient developed liver and peritoneal metastasis and was treated with Imatinib mesylate for 6 months with no change in the lesions. The molecular analysis of the GIST, the first reported in a gastric tumor from the triad, showed a wild-type KIT and PDGFRA genes.
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4/11. Metastatic renal cell carcinoma to the temporal bone: case report.

    We report an extremely rare case of metastasic renal cell carcinoma to the temporal bone which presented initially as a jugulotympanic paraganglioma. The clinical and radiological appearances were misleading.Investigations of concomitant high blood pressure revealed a tumour of the right kidney. biopsy of the mastoid mass was histologically compatible with a metastasis from a clear cell renal carcinoma.The patient underwent a radical nephrectomy and local external radiotherapy to the head. He also received adjuvant treatment with interferon-_ and interleukin 2. The clinical presentation, the radiological and histological features, the patterns of spread, the treatment options and the prognosis of these tumours are discussed. A review of the literature confirms the extremely unusual occurrence of this localisation.
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5/11. Aorticopulmonary paraganglioma (aortic body tumor): report of a case.

    A case of aorticopulmonary paraganglioma in a 57-year-old man is described. The tumor comprised nests of uniform cells in a fibrovascular stroma. Electron microscopy revealed abundant neurosecretory granules, and S-100 protein staining demonstrated scattered sustentacular cells at the periphery of typical zellballen. The findings in this case correlated with those of studies on the prognosis for extraadrenal paragangliomas.
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6/11. Carney's triad with recurrent gastric leiomyoblastoma.

    The triad of gastric leiomyoblastoma, extra-adrenal paraganglioma, and pulmonary chondroma was first described by Carney in 1977. Fewer than 30 patients with this syndrome have since been reported. In most patients the initial lesion has been a gastric leiomyoblastoma. The patient we describe had recurrences of a gastric leiomyoblastoma 15 and 17 years, respectively, after the initial gastric resection. Subsequently, multiple pulmonary lesions were excised and confirmed to be chondromas, but no evidence of a paraganglioma has yet been identified. Gastric leiomyoblastomas in patients with Carney's triad frequently recur; however, the prognosis is quite favorable in contrast to that of sporadic cases. Whenever a patient with one of the classical lesions of Carney's triad is encountered, the possibility of this unusual syndrome should be considered. The evaluation and follow-up of these patients should include a search for the other two components.
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7/11. neoplasms of the vagus nerve.

    A tumor of the vagus nerve often is discovered unexpectedly at operation. We report 36 of these rare neoplasms in 35 patients. The majority of the tumors presented as a mass in the upper cervical or parapharyngeal region. Usually the mass was asymptomatic. The following types and frequencies of neoplasms of the vagus nerve were noted: paragangliomas, 50%; neurilemmomas, 31%; neurofibromas, 14%; and neurofibrosarcomas, 6%. Surgical resection, with preservation of the vagus nerve when possible, is the treatment of choice. The clinical features, diagnosis, management, and prognosis of the tumors are presented. Special problems that occur with vagal neoplasms include postoperative dysfunction, catecholamine secretion, and intracranial or skull-base extension.
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8/11. Vagal body tumor: paraganglioma of the head and neck.

    Vagal body tumors (VBTs) are rare tumors of the paraganglion cells of the vagus nerve, usually occurring near the ganglion nodosum. They can be familial, multicentric, malignant, and sometimes hormonally active. The most accurate diagnosis is made by angiography. Primary treatment is surgical excision. radiation therapy may play a role in preoperative preparation or in palliation. The prognosis is good when the tumor is found early and completely resected. Lifelong follow-up of patients and their family members is recommended.
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9/11. Nonchromaffin paraganglioma of the retroperitoneum.

    We reviewed 37 cases described previously in the literature to provide a patient operated upon for a retroperitoneal nonchromaffin paraganglioma with qualified advice concerning followup examinations and prognosis. This type of tumor apparently grows slowly and metastasizes late. A radical operation is the treatment of choice and provides a good prognosis. We suggested that our patient undergo clinical followup and ultrasound investigations at 3 to 6-month intervals.
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10/11. Von Recklinghausen's disease with a malignant meningeal, cerebral and optic nerve tumour and bilateral vagal schwannomas. Possible mesenchymal histogenesis on light and electron microscopy.

    The clinical, histopathologic and fine structural features of multiple unusual tumours detected in a 20-year-old patient with von Recklinghausen's disease, who died within a year of onset of symptoms of a rapidly expanding intracranial tumour, are described. The tumour was found to involve the falx cerebri, the basal leptomeninges and dura mater, both olfactory and optic nerves, both frontal lobes, the right temporal lobe and middle cerebral peduncle, both middle cerebellar peduncles, and with a metastasis in a cervical node. On light and electron microscopy this tumour appeared to be a fibroblastic meningeal sarcoma with giant cells, mitotic figures, a rich reticulin matrix throughout, and tumour cells full of rough ER but without any glial filaments. Also very unusual was the involvement of both vagus nerves in their cervical and intrathoracic portions, by a schwannomatous benign tumour and with a non-chromaffin paraganglioma at its termination in the oesophagus. One of the few cutaneous "neurofibromas" was also schwannian, containing tumour cells with a basement membrane on electron microscopy. Most, if not all, of these tumours appeared mesenchymal in origin, more aggressive in behaviour and carrying a graver prognosis in von Recklinghausen's disease.
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