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PubMed1987

Extra-adrenal paraganglioma and pulmonary chondroma: a case report and review of the literature.

This report describes the findings of an extra-adrenal paraganglioma and chondroma of the lung in an elderly man. Tissue analysis of the paraganglioma showed a high level of catecholamines, as well as the presence of somatostatin and small amounts of other peptide hormones. Immunoperoxidase staining demonstrated the distribution of peptide hormones in the tumor. This case illustrates two important points. First, it confirms the ability of paraganglionic tissue to secrete peptide hormones. Secondary, it adds yet another case to the unusual group of patients described by Carney (Carney JA: Cancer 43:374-382, 1979).

Aged↗

PubMed1988

Functioning metastases of a nonfunctioning paraganglioma.

A case of nonfunctioning paraganglioma, initially qualified as benign, is described. Two years following extirpation, functioning bone and lung metastases were manifested by hypertensive crises and increased levels of vanilmandelic acid and catecholamines in urine. The criteria for benign and malignant paragangliomas are discussed with regard to functional transformation of the tumor described in this case. It is concluded that the phenotypic heterogeneity of the primary tumor cells could explain the difference in biological behavior of the primary tumor and its metastases.

Adult↗

PubMed1992

Immunocytochemical demonstration of IGF-II immunoreactivity in human phaeochromocytoma and extra-adrenal abdominal paraganglioma.

Insulin-like growth factor (IGF)-II immunoreactivity was demonstrated in 14 adrenal glands, six retroperitoneal paraganglia, 18 phaeochromocytomas, and six extra-adrenal abdominal paragangliomas through the use of a monoclonal antibody against rat IGF-II which cross-reacts with human IGF-II. A subpopulation of parenchymal cells in normal adrenal medulla and in retroperitoneal paraganglia was positively immunolabelled. Eighteen cases with phaeochromocytoma including two malignant and four multiple endocrine neoplasias all contained numerous IGF-II-immunoreactive tumour cells. All six extra-adrenal abdominal paragangliomas contained numerous immunoreactive tumour cells. Almost all the other normal human adult tissues examined failed to immunolabel. These results indicate that IGF-II-like immunoreactivity is widely expressed in normal and neoplastic human paraganglionic tissues, although its biological significance in these tissues has not been determined.

Abdominal Neoplasms↗

PubMed1995

Melanotic paraganglioma of the posterior mediastinum.

A melanotic paraganglioma occurred in a 57-year-old woman, located in the left paravertebral space of the upper mediastinum. It was totally resected. During a 5 year follow up period neither tumour recurrence nor metastasis were observed. Histological examination of the tumour revealed a paraganglioma with monomorphous chief cell like elements which were arranged in a "zellballen" pattern. Immunohistochemical results also were in accordance with the diagnosis since neuron-specific enolase, chromogranin and synaptophysin were found in tumour cells whereas keratin was not. Additionally, neurosecretory granules were found in tumour cells during electron microscopy. A peculiar feature of the tumour was its strong pigmentation due to melanin located within the tumour cells and tumour associated melanophages. The simultaneous expression of functional properties of two different neural crest derived cells in one tumour stresses the close relationship between all neural crest elements and is in accordance with the observation of other melanotic, non-melanomatous tumours.

Cytoplasmic Granules↗

PubMed1981

Spontaneous neurite outgrowth and vasoactive intestinal peptide-like immunoreactivity of cultures of human paraganglioma cells from the glomus jugulare.

The chief cells of paraganglionic tissues have morphological and functional similarities to adrenal chromaffin cells, and both cell types are derived from the neural crest. In the present investigation cells from two glomus jugulare paragangliomas were studied in culture. Approximately 50% of the cells from one tumor, and 7% from the other spontaneously formed neurite-like processes. Numerous granular and agranular synaptic-like vesicles also appeared in the process-forming cells. In contrast to findings with normal and neoplastic adrenal chromaffin cells, addition of nerve growth factor (NGF) to the culture medium had no major effects on proportion of cells with processes. Dexamethasone caused only a small decrease in process length. Culturing of the tumors also appeared to promote production of material with VIP-like immunoreactivity. It is concluded that the phenotype of paraganglioma as well as pheochromocytoma cells may be altered in vitro. Responsiveness to specific factors such as NGF or steroids, however, may vary for related tumor cell types in different anatomic locations.

Axons↗

PubMed1985

A case of spinal paraganglioma.

The neuroradiologic finding of a paraganglioma in the vertebral canal is described here for the first time. These tumors occur more frequently than was previously presumed. Similarities with paragangliomas in the craniocervical region are apparent on angiograms. We consider angiography to be indicated when, in association with a spinal tumor, urinary biogenic amine levels are elevated. When necessary, embolization can be performed after angiography.

Angiography↗

PubMed1993

Paraganglioma of the cauda equina: magnetic resonance imaging.

We report the clinical and imaging features of a paraganglioma of the cauda equina. Raised CSF protein caused an unusual appearance of the spinal canal below the obstruction. No specific identifying features were encountered; paraganglioma is uncommon in this site and usually thought to be an ependymoma or a neuroma; histopathological examination is necessary for correct diagnosis.

Cauda Equina↗

PubMed1996

Cauda equina paraganglioma with subsequent intracranial and intraspinal metastases.

A case of cauda equina paraganglioma is described; subsequent intracranial and intraspinal metastases occurred after partial resection and adjunctive radiotherapy. Cerebrospinal fluid dissemination is a rare complication of spinal paragangliomas. Factors predictive of this unusual biological behaviour are discussed.

Biomarkers, Tumor↗

PubMed1991

Paragangliomas: neuroendocrine features and cytometric DNA distribution patterns. A clinico-pathological study of 22 cases.

Paragangliomas from 22 patients with extraadrenal tumours of this type were studied. Neuroendocrine features were examined using immunohistochemical techniques. Twenty-two antisera raised against neuroendocrine "markers", regulatory peptides, serotonin and intermediate filament proteins were studied in this group and cytometric DNA assessments were made by means of image cytometry. One normal and 5 hyperplastic carotid bodies were used as controls in the DNA cytometric investigations. Clinical and/or histopathological evidence of "malignancy" was present in 5 cases. The tumour cells showed heterogeneity with regard to their expression of different peptides, and the immunohistochemical analyses did not permit differentiation between benign and malignant paragangliomas. An euploid nuclear DNA distribution pattern was found in all controls and in 17 of the tumours; all except 1 were clinico-pathologically benign. An aneuploid DNA pattern was observed in 5 of the cases and some malignant features were present in 4 of these cases. DNA data may give further information apart from that obtained from the histopathological findings which may be of value in predicting the biological behaviour of this tumour type.

Adult↗

PubMed1985

Gastric epitheloid leiomyomas, pulmonary chondroma, non-functioning metastasizing extra-adrenal paraganglioma and myxoma: a variant of Carney's triad. Report of a patient.

A female suffering from a complete Carney's triad (two gastric epithelioid leiomyomas, recurrent and metastasizing laryngeal paraganglioma, and pulmonary chondroma or hamartoma), and from a myxoma is described. The time elapsed between the detection of the gastric tumors and the metastases of the paraganglioma was 19 years.

Chondroma↗

PubMed1999

Malignant paraganglioma metachronously recurring at short duration in different paraganglions: report of a case.

We report the rare case of a 57-year-old man with retroperitoneal malignant paraganglioma. He was referred to our hospital complaining of left lower abdominal dull pain. Computer tomography (CT), magnetic resonance imaging, and aortography showed a mass measuring 5 x 4 cm in size on the left side of the abdominal aorta below the renal artery. The resected tumor was confirmed histologically to be malignant paraganglioma. Irradiation of the resected area was performed. Four months after the operation, an abdominal CT scan showed a 5.5 x 3.0 cm mass on the right side of the aorta below the diaphragm. This tumor was thereafter also resected. Three months after the second operation, a soft subcutaneous nodule measuring 4.5 x 2.0 cm in size was palpable above the left clavicle and was visible on a CT scan. A resection was again performed. All tumors showed the same histological findings. This is the first case reported in the Japanese literature with such a short-term demonstration of multiple metachronous recurrences in different paraganglions.

Humans↗

PubMed2006

Aortico-pulmonary paraganglioma: case report and Japanese review.

Aortico-pulmonary paraganglioma (APPG) is a rare middle mediastinal tumor. We experienced a case of APPG in a 52-year-old man. Chest computed tomography and magnetic resonance imaging revealed a multi-cystic mass in the subaortic area. A left thoracotomy was performed without definitive preoperative diagnosis. The tumor was strongly adherent to the pericardium and the surrounding large vessels, but a complete resection was undertaken. Histological and immunohistochemical examination revealed that it was a malignant paraganglioma with a microscopically positive surgical margin. After radiotherapy of 50 Gy for the mediastinum, the patient almost recovered from his hoarseness by thyroplasty. We also reviewed nine Japanese cases of APPG reported previously. Though APPG is rare, we must consider that a middle mediastinal tumor may be APPG, and preoperative examination and preoperative planning are necessary to prevent massive bleeding and microscopic residual tumor.

Adult↗

PubMedDate not supplied

Primary functioning hepatic paraganglioma: a case report.

An extra-adrenal pheochromocytoma is known as a paraganglioma. This report describes a patient with a rare primary functioning hepatic paraganglioma that resulted in hypertension. Computed tomography showed a highly vascular lesion located in segment 6 of the liver; it measured 6 x 5.5 cm. A right hemihepatectomy was subsequently performed; this was followed by an uneventful recovery and the disappearance of hypertension. The imaging characteristics and therapeutic principles of this rare tumor were gleaned from a review of the literature. Identification of this malignant tumor or possible recurrence is difficult, so longterm follow-up is recommended.

Adult↗

PubMed1990

Carboplatin in the treatment of metastatic carcinoid tumours and paraganglioma: a phase II study.

A total of 13 patients with metastatic carcinoid tumour, paraganglioma, or unclassified "apudoma" were treated with single-agent carboplatin at a dose of 400 mg/m2 given by intravenous infusion every 4 weeks, on the basis that this new agent shows high activity against small-cell lung cancer that also has "apudoma" characteristics. No objective tumour responses were seen. Overall, 2 patients achieved minor regression, 4/9 (44%) showed a reduction of greater than 50% in urinary 5-HIAA excretion and 8/13 (62%) reported symptomatic improvement. Treatment was well tolerated, with neutropenia and thrombocytopenia being the main toxicities. Carboplatin, like other cytotoxic agents, does not appear to have major activity against these tumours, although further studies in patients with metastatic paraganglioma are warranted.

Adult↗

PubMed1999

Visualisation of a paraganglioma by technetium-99m-sestamibi scintigraphy.

A 68-yr-old woman presented to our observation with multinodular goiter and a contiguous right laterocervical mass. In spite of ultrasound, technetium and iodine scan, CT and fine-needle biopsy, the precise origin of the mass remained uncertain. On additional multi-phase sestamibi scan, the neck region showed an early high uptake rapidly decreasing over time in the laterocervical mass, and a persistent inhomogeneous distribution in the thyroid gland. This behavior suggested that the laterocervical mass could derive from an anatomical structure other than the thyroid. Surgical exploration established the extrathyroid nature of the laterocervical mass and the histological examination confirmed that it was a typical paraganglioma. This finding is in keeping with a recent report of positive sestamibi uptake in a cervical paraganglioma, although our case showed a more rapid kinetic. This tumor should be therefore taken into consideration in the differential interpretation of focal sestamibi uptake.

Aged↗

PubMed2004

Long-term efficacy of doxazosin plus atenolol in the management of severe and sustained arterial hypertension and reversibility of the cardiac damage induced by chronic cathecolamine excess. A case report in a young girl with recurrent, functioning paraganglioma.

Herein we report on a young girl with recurrent, functioning paraganglioma of the organ of Zuckerkandl and severe and sustained arterial hypertension (systolic pressure >200, diastolic pressure >120 mmHg); with evidence of cardiac damage induced by chronic cathecolamine excess. She promptly and steadily improved after the institution of doxazosin (6 mg/day) plus atenolol (50 mg bid) treatment. This case demonstrates that a correct therapeutic strategy in the long-term management of patients with inoperable catecholamine-producing neuroendocrine tumors (pheochromocytomas and paragangliomas) can maintain arterial pressure in the normal range and reverse the cardiac damage induced by chronic cathecolamine excess.

3-Iodobenzylguanidine↗

PubMed2003

[ACTH-producing paraganglioma of the paranasal sinuses].

Paragangliomas are tumours of neuroectodermal origin and hormonally active in rare instances. They are very unusual in the paranasal sinuses. We report on the first case--as far as we know--of a recurrent nasal paraganglioma, which started to produce ACTH after a period of ten years and resulted in Cushing's syndrome. Resection of the tumour normalised ACTH and cortisol secretion.

ACTH Syndrome, Ectopic↗

PubMed2003

Paraganglioma of the nasal cavity: a case report.

We describe the case of a 72-year-old woman presenting with a 1-year history of recurrent epistaxis and unilateral progressive nasal obstruction with associated rhinolalia resulting from the presence of a tumor mass occupying two-thirds of the right nasal cavity. Histopathologically, neoplastic cells or "chief cells" were arranged in well-defined nests, which had the classic alveolar or so-called "zellballen" pattern. Immunohistochemical studies highlighted the presence of S-100 protein-positive sustentacular cells located at the periphery of the clusters of chief cells. The chief cells showed a diffuse and intense positivity for neuron-specific enolase and synaptophysin. A diagnosis of paraganglioma was made. The lesion was excised completely and the patient did not develop recurrences or distant metastases after 8 months of follow-up. Paragangliomas arising in the nasal cavity and paranasal sinuses are extremely rare tumors. We report on the clinical, histopathological and immunohistochemical findings of our case and review the cases previously described in the literature.

Aged↗