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1/49. MR appearance of paraganglioma of the cauda equina. case reports.

    PURPOSE: To investigate the value of MR imaging for preoperative diagnosis of paraganglioma of the cauda equina. MATERIAL AND methods: A retrospective review of 2 cases of paraganglioma of the cauda equina examined with MR imaging was undertaken. Features assessed included the homogeneity of the lesions, presence or absence of serpiginous flow void and thin hypointense margins. RESULTS: In case 1, the tumor was hyperintense on the postcontrast examination and serpiginous flow void suggested vessels in the upper pole of the tumor. In case 2, the tumor was encapsulated by a thin hypointense margin on both T1- and T2-weighted images, which suggested hemosiderin. CONCLUSION: The MR appearance may be of great value in the preoperative diagnosis of paraganglioma of the cauda equina.
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2/49. Surgical resection of jugulare foramen tumors by juxtacondylar approach without facial nerve transposition.

    PRESENTATION: Jugulare foramen tumors (JFT) remain a difficult challenge especially in the forms extending extradurally and invading the petrous bone. In the standard technique, facial nerve function is placed at risks because of its transposition. We report on 31 extradural (N = 11) or intra-extra dural (N = 20) JFT resected surgically using the juxtacondylar approach alone or in combination with the infratemporal approach and without facial nerve transposition. RESULTS: The juxtacondylar approach permits the opening of the JF on its posteroinferior aspect and thus reduces the extent of petrous bone drilling. In tumors strictly located in the JF (N = 11), no petrous bone drilling is necessary and the facial nerve is never exposed. In tumors extending into the petrous bone (N = 20), the facial nerve was never transposed and moreover was kept in its bony canal in 15 cases. In only 5 cases, was the fallopian canal opened as the tumor was invading its bony wall. Similarly hearing function, when pre-operatively intact was always preserved and a better preservation of the lower cranial nerves could be achieved. Whatever the tumoral extent along the petrosal carotid artery, a radical resection could be realized in 30 cases. CONCLUSION: Therefore, we consider the juxtacondylar approach a useful adjunct to increase the possibilities of resection of JFT; it allows a better preservation of the neurovascular structures, especially the facial nerve which is kept in place and moreover kept in its bony canal when it is not invaded by the tumor.
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3/49. A case of Cushing's syndrome in ACTH-secreting mediastinal paraganglioma.

    Paragangliomas are unusual neuroendocrine cell tumors arising from paraganglia, of which ACTH-secreting cases in the mediastinum are extremely rare. A 51-year-old woman was admitted for generalized edema and weakness which began 5 months ago. Chest X-ray and CT scan revealed a tumor mass in the anterior mediastinum. The plasma cortisol and ACTH levels were very high. Other sources secreting ACTH, except mediastinal mass, were not found. Surgical excision of mediastinal mass and left supraclavicular lymph node was performed. The postoperative microscopic finding and immunohistochemical staining revealed organoid tumor cell nests (zellballen) and S-100 protein positive sustentacular cells which are characteristics of paraganglioma. This was thus a case of Cushing's syndrome resulting from ectopic ACTH production in anterior mediastinal paraganglioma.
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4/49. A diagnostic and therapeutic approach to paragangliomas of the larynx.

    BACKGROUND: Differentiating paragangliomas from moderately differentiated neuroendocrine carcinoma in the larynx is a difficult management problem. As the biological behavior of these 2 entities is different, we developed an algorithm for the preoperative diagnosis and treatment of this disease. DESIGN: The sample case from which the algorithm was developed consisted of a 69-year-old man who was transferred to us after tracheostomy and an attempt at biopsy for airway obstruction secondary to a vascular mass. biopsy resulted in substantial bleeding. Flexible laryngoscopy showed a vascular mass of the supraglottis. A computed tomographic scan showed 2 vascular masses at the carotid bifurcation and in the larynx. An arteriogram confirmed synchronous vascular tumors. RESULTS: The arteriogram showed the superior thyroid artery to be the major feeder vessel to this mass, a situation commonly seen in paragangliomas but not other neuroendocrine tumors. The presence of synchronous lesions and a vascular mass based on the superior thyroid artery helped differentiate paraganglioma from the other neuroendocrine tumors. As the biological behavior of paragangliomas is relatively benign, we performed a conservative supraglottic laryngectomy and excision of the carotid body tumor. Histologic diagnosis and immunohistochemical analysis confirmed the diagnosis of paraganglioma. CONCLUSIONS: The vascular nature of neuroendocrine tumors prevents preoperative pathological diagnosis. Radiologic features demonstrating a vascular mass with a dominant feeder vessel by the superior or inferior thyroid artery may help in the clinical diagnosis of paragangliomas of the larynx. Since paragangliomas are rarely malignant, a conservative surgical procedure should suffice.
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5/49. Tracheal paraganglioma: a diagnostic dilemma culminating in a complex airway management problem.

    A 41-year-old man presented to his general practitioner (GP) with a wheeze and dyspnoea on exercise. asthma was diagnosed and treatment with inhaled corticosteroid and a beta(2)-agonist commenced. Despite this, his condition deteriorated over three weeks culminating in stridor, requiring emergency admission to hospital. Nasendoscopy revealed a polypoidal lesion in the upper trachea, acting like a ball valve. A local anaesthetic tracheostomy, secured his airway. Subsequent direct laryngoscopy allowed avulsion of the lesion. Alternative methods of airway management are discussed. histology revealed a paraganglioma. The aetiology of paragangliomas is described and a literature review of tracheal paraganglioma is presented. Post-operative recovery was unremarkable. However, tumour recurrence occurred at nine months. A subsequent revision tracheostomy and laser resection has ensured disease-free survival, one year later. We recommend that acute onset wheeze, refractive to appropriate therapy, should be referred for urgent examination of the upper aerodigestive tract.
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6/49. Clinics in diagnostic imaging (68). Intradural extramedullary spinal paraganglioma.

    Paragangliomas rarely present as spine tumours. The correct diagnosis is generally not suspected pre-operatively and initial imaging is often non-specific. A 36-year-old man with low back pain, and progressive leg numbness and weakness, was found to have an expansile intradural extramedullary spinal tumour on radiographs and magnetic resonance imaging. Surgery revealed a paraganglioma. The features of spinal paraganglioma and differential diagnosis of intradural extramedullary tumours are discussed.
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7/49. Primary paraganglioma strictly confined to the liver and mimicking hepatocellular carcinoma: an immunohistochemical and in situ hybridization study.

    We describe a case of primary nonfunctioning paraganglioma that, unlike any other previously reported case, was strictly confined to the liver and must therefore have arisen on liver parenchyma. An asymptomatic 46-year-old man was referred to us for laparotomy and a right hemihepatectomy after a preoperative diagnosis of fibrolamellar hepatocellular carcinoma, based on a fine-needle biopsy. An 8-cm resiliently firm, pale gray nodule with a large central area of fibrosis and a thin fibrous capsule was resected. The polygonal eosinophilic tumor cells containing round nuclei lacking nucleoli were arranged in small nests set in a vascularly rich stroma. At immunohistochemistry neoplastic cells were strongly positive for chromogranin a, neuron-specific enolase, synaptophysin, and IGF-II protein; they were negative for keratin, S-100 protein, CD10, vimentin, and smooth muscle actin. in situ hybridization confirmed that, as in other sites, liver paraganglioma can express IGF-II gene. Conversely (and unlike hepatocellular carcinomas), the neoplastic cells did not express albumin mRNA, which was detected only in surrounding hepatocytes. The clinical course was benign and the patient is well and free of neoplastic disease 9 years after surgery. knowledge of the entity should avoid possible confusion with hepatocellular carcinoma, especially of the fibrolamellar variety.
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8/49. Variable hemodynamic fluctuations during resection of multicentric extraadrenal pheochromocytomas.

    PURPOSE: To report the perioperative management and the serious hemodynamic fluctuations during manipulation of an organ of Zuckerkandl tumour in a patient undergoing resection of multicentric extraadrenal pheochromocytomas. CLINICAL FINDINGS: A 28-yr-old man who had undergone at age 12 a laparotomy for excision of an extraadrenal pheochromocytoma complained of paroxysmal headache, occasional sweating and palpitations. The arterial blood pressure (BP) was 200/100 mmHg. A 24-hr-urine collection showed catecholamines 5076 microg x 24 hr(-1) (normal < 25 microg x 24 hr(-1)). Computed tomography of the abdomen revealed two retroperitoneal masses, one adjacent to the lower pole of the right kidney and a second larger mass located at the aortic bifurcation in the region of the organ of Zuckerkandl. The patient was scheduled for excision of multiple extraadrenal pheochromocytomas. He was prepared preoperatively for two weeks with prazosin 1 mg po q six hours and propranolol 10 mg tid. Manipulation of the infrarenal tumour was uneventful but manipulation of the Zuckerkandl tumour resulted in severe hypertensive episodes with BP ranging from 200/100 to 320/120 mmHg. Surgery was interrupted temporarily; the hypertensive crisis was controlled by the infusion of sodium nitroprusside and by iv phentolamine and esmolol. CONCLUSION: In a patient undergoing resection of recurrent multicentric extraadrenal pheochromoctyomas, severe hypertensive episodes occurred during manipulation of one tumour but not during manipulation of the other. This may be attributed to inadequate preparation of the patient, difficult surgical dissection of the large Zuckerkandl pheochromocytoma, and/or secondary to an excessive and different pattern of release of catecholamines during manipulation of the Zuckerkandl tumour.
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9/49. Abdominal and pelvic extra-adrenal paraganglioma: a review of literature and a report on 7 cases.

    INTRODUCTION: Extra-adrenal paraganglioma (pheochromocytoma) is a rare tumor. Herein we describe the clinical and pathological findings in patients with paragangliomas of the urinary bladder, seminal vesicle and retroperitoneum. methods: Between January 1994 and January 2001, extra-adrenal paragangliomas were diagnosed in 7 patients: 3 males and 4 females. The mean age of our patients was 32 /- 15.9 years. We reviewed the clinical data. Urinary metanephrines and vanillyl mandelic acid and blood catecholamine levels were estimated in 4 cases. CT scan and/or MRI were used in the imaging of all cases. 123I-MIBG was used in only 1 patient, who harbored multiple tumors. All the patients but one underwent surgical treatment. RESULTS: The definitive diagnosis was made by histopathological examination of the removed tumors and was confirmed in all cases by the immunohistochemical stains of chromogranin a and S100 protein. There was metastasis in the pelvic lymph nodes in 1 patient. Follow-up ranged from 3 to 82 months (mean = 37.9 /- 25.8). The catecholamine level was elevated in 3 patients under basal conditions and during endoscopic resection of the tumor in a fourth patient. In all cases, the catecholamine level was normalized after surgery. There was no recurrence or metastasis in any case following surgery. CONCLUSION: Pre-operative diagnosis of nonfunctioning bladder paraganglioma is difficult, but the tumors should be suspected in patients who have hypertension, hematuria or mass effects due to the tumor growth in the pelvis and/or retroperitoneum. Six of the seven cases reported here were found in the usual locations: 3 in the urinary bladder, 2 in the renal hilum and 1 in the organ of Zuckerkandl. One patient had multiple tumors, including a paraganglioma of the seminal vesicles. Resection is the treatment of choice, and in the case of urinary bladder paraganglioma should include total cystectomy. In patients with unresectable multiple tumors, medical therapy may be used to control hypertension.
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10/49. Extra adrenal retroperitoneal paraganglioma.

    We herein report a case of a 45-year-old Saudi lady not diabetic nor hypertensive who presented to the emergency room with a one day history of severe central and lower abdominal pain. On examination, she was hemodynamically stable and abdominal examination showed tenderness in the lower abdomen. Her hematological and biochemical investigations were normal. Computed tomography of the abdomen showed an 8 x 7 cm retroperitoneal mass located at the aortic bifurcation. The patient had exploratory laparotomy and complete excision of the mass. The histopathological study showed a paraganglioma. The patient had an uneventful postoperative period and follow up.
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