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1/41. Management of incidentally discovered cervical paragangliomas: report of two cases and review of current issues.

    Paragangliomas of the head and neck are uncommon neoplasms arising from the extra-adrenal paraganglia and include carotid body and glomus vagale tumors. These lesions may be discovered incidentally by imaging studies performed to evaluate carotid atherosclerotic occlusive disease. Incidental paragangliomas of the head and neck may be smaller than those discovered due to symptoms. Although surgical resection remains the definitive treatment for head and neck paragangliomas, important issues of management arise when such lesions are discovered. Two recent cases are reported. epidemiology, pathophysiology, diagnostic evaluation, and issues of management of head and neck paragangliomas are discussed.
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2/41. Cushing's syndrome associated with a chemodectoma and a carcinoid tumour.

    We present a case of Cushing's syndrome where 111In-octreotide scanning provided evidence for the presence of two neuroendocrine tumours. Uptake in the right neck corresponded to a chemodectoma, but there was no change in the clinical condition or fall in ACTH levels following surgical resection. Uptake in the left chest was assumed to relate to a bronchial carcinoid, but a tumour could not initially be localized on magnetic resonance imaging (MRI), spiral CT scanning or on selective venous sampling. A 1 cm bronchial carcinoid tumour was identified post-mortem which immunostained for ACTH. This case demonstrates that 111ln-octreotide scanning is a useful technique for identifying the source of ectopic ACTH production in difficult cases of Cushing's syndrome. Reliance should not be placed solely on standard imaging techniques to localize the tumour prior to surgery. Although rare, the possibility of a non-ACTH secreting neuroendocrine tumour should also be considered in patients with ectopic ACTH syndrome, who have positive 111In-octreotide scans.
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3/41. Malignant carotid body tumors: report of three cases.

    The aim of the current report was to describe 3 cases of malignant carotid body tumor (CBT) and to review the literature. My clinical records of 87 CBTs in 81 patients (6 bilateral) were reviewed, 79 of which were operated on. Three malignant cases were found. The first was in a 40-year-old man who presented with pulmonary metastases 6 years after resection of a CBT. He was treated with chemotherapy and interferon, but died with disseminated disease 2 years later. The second case was in a 56-year-old woman who had a 5-cm, fixed, hard mass in the upper aspect of the neck and a paralysis of the left vocal cord. This lesion was completely resected, and a shunt and reconstruction with a saphenous vein graft were performed. pathology revealed a malignant chemodectoma with invasion to 2 of the 5 lymph nodes removed. radiotherapy (50 Gy) was given after the operation. She is well and free of disease 68 months after the resection. The third case was in a 61-year-old woman who presented with an 8-cm nontender, hard, immobile mass in the left upper neck that displaced the left wall of the oropharynx toward the midline. A carotid arteriogram showed a CBT. On computed tomography, the tumor extended to the infratemporal fossa with no bone involvement. The lesion was embolized with a 40% reduction in vascularity. At surgical exploration, the tumor involved the sternocleidomastoid muscle and the lymph nodes at levels II and III, and the internal carotid artery could not be dissected free at the skull base, so only a partial resection was performed. This patient was lost to follow-up. These 3 cases are in agreement with the literature. Locoregional control is usually obtained with complete primary tumor resection and lymphadenectomy and eventual radiotherapy. Surgery with radiotherapy seems to be effective for isolated metastases. Current multidisciplinary treatments have been unsuccessful in controlling disseminated disease.
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4/41. Nasal paraganglioma. A case report.

    Nasal paraganglioma. A case report. Nonchromaffin paragangliomas or chemodectomas arise in paraganglia distributed in various parts of the body. The jugular bulb, the vagal body and the bifurcation of the carotid artery are the most common sites of origin of paraganglioma in the head and neck region. Paragangliomas in the nose and paranasal sinuses are extremely rare and very few cases of definite paraganglioma arising primarily in the nose or paranasal sinuses have been reported. The paraganglioma is a slow-growing tumour that produces nasal obstruction, profuse epistaxis and facial swelling. Complete excision of the glomus tumour is normally curative. We report a case of nasal paraganglioma and discuss the diagnosis and therapy.
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5/41. radiation therapy, an important mode of treatment for head and neck chemodectomas.

    Between 1970 and 1990, 22 patients with 44 chemodectomas in the head and neck region were seen at the netherlands Cancer Institute in Amsterdam. All patients were treated with radiation therapy (17 patients with radiation therapy only and 5 in combination with surgery). One patient was treated two times with an interval of 12 years at each side of the neck. Standard dose was 50 Gy in 25 fractions over 5 weeks. A radiation portal arrangement with oblique fields with paired wedges was used most frequently. The follow-up period ranged from 1 year to 20 years. Two recurrences at 2 and 9 years after treatment were observed. The actuarial local control rate was 88% at 10 years follow-up. Comparison of the results of surgery and radiotherapy demonstrates that radiation therapy is an effective treatment modality without mutilation or severe late morbidity for chemodectomas in the head and neck region.
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6/41. Familial paraganglioma.

    Paragangliomas are unusual tumors that are sometimes familial. We treated a family who exhibited multiple head and neck paragangliomas (HNPGs) and pheochromocytomas. The purpose was to determine the clinical characteristics of paragangliomas with familial history and to define a better standardized proceeding in the management of these tumors. patients diagnosed with head and neck paragangliomas and identified retrospectively through clinical otolaryngology practices were given a medical and family history questionnaire. We studied a family who exhibited familial paragangliomas. This relationship was examined by reviewing the medical records of family members with verified tumors, carrying out neck computed tomography or magnetic resonance imaging on their relatives to look for tumors that had been unrecognized in the past. All patients underwent a complete head and neck examination. The initial evaluation usually included CT and/or MRI. Computed tomography and magnetic resonance imaging contributed additional information about tumor extension. angiography was performed in every patient with carotid body tumor, with one undergoing therapeutic embolization to reduce the tumor size. Eleven tumors were identified in four patients with a familial history. Familial disease was initially determined by pedigree analysis. Four patients with a median age of 31 years (range: 25-42) underwent surgery. Median follow-up was 5 years (range 2-14); carotid angiography provided essential mainstays for the definite diagnosis. All patients underwent successful surgical resection of the tumor after the appropriate preoperative preparation. There were no perioperative deaths or hemiplegia. Three patients had bilaterality carotid body paragangliomas. One patient had three paragangliomas, and two patients had bilateral carotid body paragangliomas associated with pheochromocytoma. Clinically functioning tumors and malignant tumors were not identified, and none of the patients died after surgery. During follow-up, none of the patients developed recurrence or metastatic disease. The carotid body paraganglioma (CBPG) and glomus vagale manifested as asymptomatic neck masses. The clinical pheochromocytomas typically present with uncontrolled hypertension. In conclusion, paragangliomas are rare, with multicentricity being more common in patients with a familial history. In patients with familial paragangliomas, high-resolution computed tomography and magnetic resonance imaging are recommended for early screening and contributed additional information about the tumor extension and definitive treatment. Early surgery is recommended to minimize major risks.
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7/41. An apparently sporadic paraganglioma with an SDHB gene germline mutation presenting at age 68 years.

    Paragangliomas (PGLs) are rare tumours arising from parasympathetic-associated paraganglia (particularly of the head and neck) or from sympathetic-associated paraganglia such as in the adrenal medulla when they are termed phaeochromocytomas and at extra-adrenal sites in the abdomen and thorax. Recent reports have found frequent germline mutations of VHL, RET, SDHB or SDHD not only in familial cases but also in apparently sporadic cases of phaeochromocytoma. These germline mutations are particularly likely to be found if multifocal disease is present or if the phaeochromocytoma or PGL occurs at a young age. We report a germline splice site mutation in SDHB in a patient presenting with an incidental, apparently sporadic, abdominal sympathetic PGL at 68 years of age.
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8/41. Nonfunctional paraganglioma of the liver.

    Paragangliomas occurring in the liver are rare. We report a patient with a history of nonfunctional paragangliomas in the head and neck who developed liver metastases years later. We discuss findings on cross-sectional imaging modalities, including magnetic resonance imaging (MRI), which demonstrate the hypervascular nature of the liver masses.
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9/41. Bone metastases from head and neck paragangliomas: uncommon MR findings in an uncommon condition--report of three cases.

    Malignancy (defined as metastatic disease) has been reported in nearly 5% of head and neck paragangliomas. Metastases preferentially target the lymph nodes, lung, liver, or bone. We describe three patients with multiple silent bone metastases exhibiting a fatty halo at MRI that coexisted with expansive bone lesions. In all cases, (123)I-metaiodobenzylguanidine ((123)MIBG) scintigraphy showed no abnormal tracer uptake, whereas (111)In-pentetreotide scintigraphy visualized a few silent bone metastases. These findings indicate that MRI should be included in the staging of paraganglioma patients with risk factors for malignancy.
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10/41. A case of familial paraganglioma syndrome type 4 caused by a mutation in the SDHB gene.

    BACKGROUND: A 40-year-old man was referred to our clinic with recurrent paragangliomas. He had undergone resection of a paraganglioma superior to the right adrenal gland at 19 years of age, resection of two para-aortic paragangliomas at 39 years of age, and resection of a paraganglioma in the interatrial septum at 40 years. The patient's mother had died at age 39 years of metastases from a carotid body tumor. INVESTIGATIONS: MRI and CT scanning, 131I-labeled metaiodobenzylguanidine scanning, and genetic testing for a mutation in the succinate dehydrogenase complex, subunit B gene. diagnosis: Familial paraganglioma syndrome type 4 caused by a mutation in the succinate dehydrogenase complex, subunit B gene. MANAGEMENT: The patient underwent two surgical procedures in our clinic. The first was to remove two para-aortic paragangliomas, and the second to remove a paraganglioma that involved both atria. The patient is at high risk for malignant disease and should undergo an annual monitoring program that consists of physical examination and measurement of his blood pressure and levels of urinary catecholamines and metanephrines. If these procedures suggest a recurrence of paraganglioma, 123I-labeled metaiodobenzylguanidine scanning should be performed. As he might develop nonfunctional tumors, however, he should also undergo CT scanning, MRI scanning, or both, of the neck, thorax, abdomen, and pelvis every 6-12 months. genetic testing has been offered to family members.
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