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PubMed1990

Nonfunctioning paraganglioma of the liver, gallbladder and common bile duct.

Paragangliomas are rare extra-adrenal neoplasms of neural crest origin. Although most of these lesions are of retroperitoneal origin, paragangliomas have been reported at a host of remote sites, including the urinary bladder, larynx, orbit and lung. In our review of the literature, we discovered only solitary case reports of such neoplasms involving the gallbladder and hepatic duct. We report a case of paraganglioma simultaneously involving the liver, gallbladder and common bile duct, as well as celiac and portal lymph nodes. Our findings add paraganglioma to the list of differential diagnoses for hypervascular portacaval space lesions. Furthermore, the radiographic appearance of this unusual tumor may be sufficiently distinctive to suggest the correct histologic diagnosis preoperatively.

Cholangiopancreatography, Endoscopic Retrograde↗

PubMed1987

Localization and treatment of familial malignant nonfunctional paraganglioma with iodine-131 MIBG: report of two cases.

Two cases of familial, malignant, nonfunctional paraganglioma are reported. Uptake of iodine-131 metaiodobenzylguanidine ([131I]MIBG) by the tumors and metastases was demonstrated. In the first case, with multicentric and locally invasive disease, [131I]MIBG correctly localized a right carotid body paraganglioma which had been missed arteriographically. In the second case, with widespread, symptomatic metastatic disease, a therapeutic dose of [131I]MIBG produced palliation of bone pain after the failure of radio- and chemotherapy. Uptake of [131I]MIBG by paragangliomas does not correlate with catecholamine secretory activity. Iodine-131 MIBG should be considered as a therapeutic option in unresectable, malignant paragangliomas which take up this radiopharmaceutical.

3-Iodobenzylguanidine↗

PubMedDate not supplied

[Paraganglioma (chemodectoma) of the larynx. Presentation of 2 cases].

Two rare cases of laryngeal paraganglioma are reported. The first patient was a 14-year-old girl, that had a new laterocervical paraganglioma, 4 years after the excision of a laryngeal tumor. The other patient was a 33-year-old man. In both the cases, the diagnosis was made on histopathological ground and the tumors displayed the typical histological pattern of paragangliomas: epithelioid tumor cell arranged in nests (Zell-ballen) in a vascular stroma. Neoplastic cells were found to contain argyrophil granules and chromogranin. Laryngeal paragangliomas reported in the literature are 73, they generally are supraglottic, show no sex predilection and prefer the V-VII decades of age.

Adolescent↗

PubMed1989

A review of the histology, ultrastructure, immunohistology, and molecular biology of extra-adrenal paragangliomas.

This review summarizes our understanding of extra-adrenal paragangliomas, a subject that has evolved considerably during the past several years. Our object was to review the anatomical, histologic, and biological features of normal and neoplastic glands, with emphasis on immunohistologic studies, and briefly discuss the potential application of nucleic acid hybridization. Since it is difficult to predict clinical outcome for patients with paragangliomas, we have emphasized the differences between benign and malignant paragangliomas, concentrating on recent results obtained using immunohistologic techniques. These studies have emphasized the critical importance of the identification, by immunohistologic means, of two distinct cell populations, chief cells (type I) and sustentacular cells (type II). The relationship between these two cell populations, stable in normal glands and benign tumors, is progressively lost in tumors of increasing degrees of malignancy, sustentacular cells being absent from the most progressively metastasizing paragangliomas.

Adrenal Gland Neoplasms↗

PubMed1989

[Laryngeal paraganglioma].

A case is reported of laryngeal paraganglioma observed among 49,959 patients. A comparison is made of the characteristics of paragangliomas in general to those of laryngeal paraganglioma, which are more common in males; the case presented is that of a woman. Microscopic dissection failed to reveal the paraganglia in the trajectories of the superior and inferior laryngeal nerves, but they were found in histological sections of these nerves and their images were equal to those of the tumor studied. The trajectory of these nerves was investigated in preparations of the Embrioteca de Bellaterra (Prof. Doménech Mateu) and the clumps of paraganglioma cells from which these tumors proceed have been found; we emphasize the concept of neural crest pathology to which they pertain.

Adult↗

PubMedDate not supplied

[Paragangliomas of the chiasma and sellar area].

The article describes five cases of rare cerebral tumors, paragangliomas of the chiasma-sellar area. Their clinical course characteristically begins with the appearance of endocrine disorders manifested by progressive hypophyseal insufficiency. Analysis of the clinical and X-ray signs of paragangliomas of the chiasma-sellar region show these to be similar to adenomas of the hypophysis. A specific feature of paragangliomas of the chiasma-sellar area is their rich vascularization, which is demonstrated by angiography and verified in all cases during operation. The rich vascularization of paragangliomas of the chiasma-sellar area calls for their rapid and complete removal so as to reduce the loss of blood and provide reliable hemostasis.

Adenoma↗

PubMed1988

A comparison of paraganglioma, carcinoid tumor, and small-cell carcinoma of the larynx.

Laryngeal paraganglioma, carcinoid tumor, and small-cell carcinoma are rare. Histologically they are similar to analogous tumors in other locations but may be difficult to identify in small biopsy specimens. We compared the light microscopic, histochemical, immunohistochemical, and electron microscopic features of two laryngeal paragangliomas, one carcinoid tumor, and six small-cell carcinomas. The paraganglioma chief cells stained with Grimelius stain and for chromogranin and neuron-specific enolase. The carcinoid tumor cells stained with Grimelius stain and for chromogranin, serotonin, neuron-specific enolase, and keratin. The small-cell carcinoma cells stained for keratin and neuron-specific enolase. The patients with paragangliomas and carcinoid tumor remain healthy through 20 months of follow-up. Four of the patients with small-cell carcinomas have died. Distinction between these tumors is warranted by differing histologic appearances, staining characteristics, and biologic behavior.

Adult↗

PubMedDate not supplied

Cervical paraganglioma with intranuclear vacuoles in a fine needle aspirate.

In a 56-year-old man who presented with a cervical mass, a fine needle aspiration specimen was suggestive of a papillary thyroid adenocarcinoma because of the presence of prominent intranuclear vacuoles in rare cells. Operation and subsequent examination of the mass removed from the bifurcation of the carotid artery clearly identified a paraganglioma. This case emphasizes the following points: aspirates of cervical paragangliomas are difficult to interpret, especially when scanty; intranuclear vacuoles are found in cells of a variety of cervical lesions, including paragangliomas, and should not in themselves define the diagnosis of papillary thyroid adenocarcinoma; and aspiration of cervical paragangliomas is probably contraindicated (but may be done unwittingly from time to time).

Biopsy, Needle↗

PubMed1987

[Pedunculated polypoid gangliocytic paraganglioma of the afferent jejunal loop of a Billroth II stomach].

A mobile pedunculated polypoid tumor was endoscopically removed from the afferent jejunal loop after gastrojejunostomy of a 54-year-old patient with anamnestic evidence of intestinal bleeding. Histologically epithelial carcinoid-like as well as mesenchymal paraganglioma- and ganglioneuroma-like patterns are mixed in varying portions, characteristic for gangliocytic paraganglioma. Immunohistochemically, serotonin, neuron-specific enolase, cytokeratin, vimentin S-100 protein and neurofilament were demonstrable. Gangliocytic paragangliomas are almost exclusively observed in the second portion of the duodenum, especially around the papilla Vateri and only two have previously been reported in the jejunum. The histogenesis of the tumors is unclear, but they may probably be either hamartomas, hyperplastic or neoplastic proliferations of so called endodermal-neuroectodermal complexes. Although gangliocytic paragangliomas contain a carcinoid-like component, they behave in a benign fashion, and metastases or recidives have not been noticed. Tumors with a pedicle may be endoscopically removed without complications.

Diagnosis, Differential↗

PubMed1983

[Benign vagal paragangliomas (glomus tumors) in the upper neck. I].

A survey of parasympathic paraganglionic tissue and paragangliomas originating in the head and neck points out that their anatomy, histology and ultrastructures are better known than their physiology and the events of a hormonal activity. The terminology of the paragangliomas is actually confused which is reflected by the insecurity in the biologic evaluation of these structures. By means of three casuistic reports of typical localized vagal paragangliomas all clinical features were represented including possible hormonal effects. Excision is the method of choice in treatment. The surgical technique essentially differs from operations of other paragangliomas in head and neck. The description of prognosis also takes into consideration the criteria of malignancy.

Adult↗

PubMedDate not supplied

Fine needle aspiration biopsy cytology of paragangliomas. Cytologic, light microscopic and ultrastructural studies of three cases.

Paragangliomas are uncommon tumors arising in paraganglion tissue, which is especially well represented in the head and neck. In our hospital three head and neck masses recently proved to be paragangliomas on aspiratory biopsy cytology, with subsequent histologic confirmation. One was an apparently benign carotid body tumor, while the others were metastatic nasopharyngeal paragangliomas. The latter two tumors were clearly malignant, with roentgenographically proven pulmonary metastases in one and widespread lymph node involvement in the other. Histologically all were vascular tumors composed of large cells with abundant cytoplasm and pleomorphic, sometimes bizarre nuclei. These characteristic chief cells were clearly evident on cytology, with pleomorphic nuclei, prominent nucleoli, clumped chromatin and generally eosinophilic, finely granular or reticular cytoplasm. However, the distinction between benign and malignant tumors depended on the presence of mitoses or a tumor diathesis. Cytology seems to be able to provide a diagnosis of paraganglioma and, in these cases at least, an estimation of the biologic behavior.

Biopsy, Needle↗

PubMed1980

Intrathoracic paravertebral malignant paraganglioma.

This article reviews the clinical and pathologic features of intrathoracic paravertebral paragangliomas. Including the present case, there have been 31 tumors reported in the English literature; the average age of patients was 29 years, with a sex distribution of 20 men and 11 women. Fifteen patients (48%) had symptoms related to excess secretion of catecholamines; the remaining 16 tumors were clinically nonfunctional. Seven patients (22%) had multiple paragangliomas. Complete surgical resection was attempted in 25 patients, 13 were alive with no evidence of tumor an average of 2.2 years later. Tumor was locally invasive in five patients, with involvement of the vertebral canal and symptoms of spinal cord compression. Malignant behavior with distant metastases was observed in two patients. As shown by the present case, the Grimelius stain is a useful diagnostic technique for demonstrating cytoplasmic argyrophilia of neoplastic chief cells. Electron microscopy demonstrated neurosecretory granules (average core diameter, 100 nm). "Light" and "dark" chief cell types were inconspicuous. Due to important clinical and pathologic differences, paravertebral paragangliomas should be distinguished from similar tumors occurring in the anterosuperior mediastinum (aorticopulmonary paragangliomas).

Adult↗

PubMedDate not supplied

[Flow-cytometric study of familial paragangliomas of the carotid body].

Paragangliomas of the carotid body with an autosomal dominant genetic transmission have been described in a familial presentation in 7-10% of all the cases observed. In less than 10% malignancy is confirmed by secondary metastatic localization rather than by typical histological features of malignant diseases, such as vascular or perineural invasion, nuclear pleomorphism, mitotic activity. The purpose of the study is to present a familial group of paragangliomas of the carotid boy and a flow cytometric analysis of tumor content DNA and to discuss the prognostic value of the results. The paragangliomas of these related patients (father and daughter) are diploid, without regional lymph node or distant metastases; clinical and cytometric findings support a good prognosis even if an accurate follow up of such neuroendocrine tumors is nonetheless mandatory. Considering data in Literature, the Authors also propose a prognostic classification of paragangliomas as follows: noninvasive (capsulated, without vascular or perineural infiltration, diploid); locally invasive (histological signs such as vascular or perineural infiltration, nuclear pleomorphism, abnormal mitoses etc, diploid); potentially malignant (histological signs, non-diploid, expression of few antigens) and malignant (regional or distant metastases).

Adolescent↗

PubMed1994

[Scintigraphy with iodine-131 metaiodobenzylguanidine in the study of paraganglioma. Comparison between benign and malignant tumors].

In order to evaluate 131I metaiodobenzylguanidine (MIBG) uptake in patients with benign or malignant paragangliomas, 28 patients (14 men and 14 women, mean age 37 +/- 10 years) with benign (no. = 15; group 1) or malignant (no. = 13; group 2) tumors underwent MIBG scintigraphy. A total of 110 lesions (20 benign and 90 malignant) were studied. In all patients histologic disease confirmation was obtained. MIBG uptake was quantified on 48 hours' images (Kodak NMC-1 films) using a photographic densitometer. The optical densities of tumor and adjacent or contralateral normal tissue were used to calculate the MIBG uptake intensity ratio for each lesion. In group 1, all patients exhibited 1 or 2 lesions with abnormal MIBG uptake. In group 2, all patients had 1 to 28 abnormal foci of MIBG uptake. In the patients with 2 or more lesions, the average MIBG uptake intensity ratio was calculated. MIBG uptake intensity ratio was significantly higher in malignant than in benign paragangliomas (5.2 +/- 2.4 vs 2.9 +/- 1.4, p < 0.01). Since MIBG uptake in paragangliomas reflects the intratumoral concentration of catecholamines, higher MIBG uptake in malignant lesions suggests a greater amount of stored catecholamines in these tumors. In conclusion, 131I MIBG scintigraphy may be useful to distinguish benign from malignant paragangliomas.

3-Iodobenzylguanidine↗

PubMed1994

Immunohistochemical demonstration of chromogranin A, chromogranin B, and secretogranin II in extra-adrenal paragangliomas.

Twelve sympathetic and 14 parasympathetic extra-adrenal paragangliomas were investigated immunohistochemically with antibodies against chromogranin A, chromogranin B, and secretogranin II. In sympathetic paragangliomas chromogranin A was found in 12/12 and chromogranin B in 11/12 tumors in almost all chief cells (the remaining tumor was focally chromogranin B positive), whereas secretogranin II was immunolocalized in the majority of chief cells in 5/12, in a focal distribution in 3/12, and only in a few scattered tumor cells in 3/12 cases. One case showed no secretogranin II immunoreactivity. In parasympathetic paragangliomas both chromogranin B and secretogranin II immunoreactivity was demonstrated in the majority of chief cells of all 14 tumors investigated. Chromogranin A showed a strong immunostaining in 2/14 cases; in 12 tumors chromogranin A was found in only a few chief cells or was completely absent. It is concluded that sympathetic and parasympathetic paragangliomas show a divergent expression of chromogranins/secretogranins that apparently reflects the different histogenetic origins of these tumors.

Adolescent↗

PubMed1993

[New histopathologic data of prognostic value in extra-adrenal paragangliomas. Study of 9 cases].

BACKGROUND: The biological behavior of paragangliomas is difficult to evaluate by classic histological criteria thus justifying the use of immunohistochemical markers as prognostic factors. METHODS: Nine extra-adrenal paragangliomas (three jugulo-tympanic, four carotid-body tumors, and two retroperitoneal) were studied by conventional histological criteria, and also by chromogranin A and neuron-specific enolase (NSE) immunohistochemical staining for the study of chief cells, and S-100 as a marker of sustentacular cells. The rate of cell proliferation was studied by the proliferating cell nuclear antigen (PCNA). The correlation between these parameters and the clinical evolution of the neoplasms, which were classified as benign, locally aggressive, and malignant (with metastasis), were also analyzed. RESULTS: The atypia and the mitotic rate did not correlate with the behavior of the tumor. Less immunostaining with the anti-S-100 and anti-chromogranin A antibodies was observed in the malignant paragangliomas and in those which were locally aggressive. In the benign tumors the proliferative rate (PCNA) oscillated between 0.7% and 3.7%, and 40 or less PCNA positive cells were counted in 10 high-power field (HPF) (40x). In malignant and locally aggressive tumors the proliferative rate was 5% or more, with 60 or more cells that were positive for PCNA being found in 10 HPF. CONCLUSIONS: The histopathologic signs implying worse prognosis in extra-adrenal paragangliomas are a decrease in chromogranin A and S-100 immunoreactivity and a rate of cell proliferation of 5% or greater, or a number of cells stained for proliferating cell nuclear antigen greater than 50 in 10 high-power field.

Adult↗

PubMed1993

[Functioning paraganglioma].

Pheochromocytomas and functioning paragangliomas are rare tumors arising from indifferent cells of the primitive neural crest. These tumors produce clinical symptoms (pheochromocytoma syndrome) related to catecholamine production. The pheochromocytoma syndrome, constituted by arterial hypertension, headache and sweating, is due in 80% of cases to adrenal pheochromocytomas and only in 20% of cases is due to extra-adrenal secreting paragangliomas. The diffused localization of the Paraganglion System and the common embryologic origin, can explain the occasional identification of multicentric tumors (10%), and the association with multiple endocrine neoplasms (MEN), especially in familial occurrence. Careful follow-up of patients, in order to detect the later development of multicentric or metachronous neoplasms is necessary. The Authors present their experience with 80 patients affected by neoplasms involving the adrenal medulla and the multicentric paraganglion system, surgically treated during the last 25 years. Eight patients (10%) were affected by functioning extra-adrenal paragangliomas: 7 patients (87.5%) had solitary extra-adrenal tumors (6 abdominal and 1 thoracic) with a classic pheochromocytoma syndrome, and 1 patient (12.5%) had a double asymptomatic abdominal paraganglioma.

Adult↗

PubMed1996

Paraganglioma of the cauda equina. A case report and review of the literature.

A 59-year-old man presented with clinical evidence of a primary tumor of the cauda equina region. It was well circumscribed and was completely removed by neurosurgery. Routine staining showed that it had structural similarities to an ependymoma, but immunohistochemistry with antisera to synaptophysin, NSE, chromogranin-A and PGP 9.5 proved it to be a neuroendocrine tumor, i.e. a paraganglioma. We propose the use of endocrine markers in cases with tumors of the cauda equina to differentiate a paraganglioma from an ependymoma. Paragangliomas appear to have a better clinical outcome than ependymomas. Recurrence after surgery for a paraganglioma in the cauda equina region, especially if it is encapsulated, is rarely encountered.

Biomarkers, Tumor↗