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PubMed2003

A case report of an obesity hypoventilation syndrome associated with obstructive sleep apnea due to a carotid body paraganglioma.

BACKGROUND AND PURPOSE: To the best of our knowledge, the association between an obstructive sleep apnea syndrome (OSAS) due to a neck mass and an obesity hypoventilation syndrome (OHS) has not been reported. PATIENTS AND METHODS: We report the case of a patient with obesity hypoventilation syndrome (OHS) in whom OSAS caused by a carotid body paraganglioma contributed to recurrent bouts of severe alveolar hypoventilation. RESULTS AND CONCLUSIONS: The complete surgical excision of the paraganglioma resulted in the cure of the OSAS and contributed to a clear improvement of the clinical symptoms of OHS.

Aged↗

PubMed2003

A pitfall of metaiodobenzylguanidine scan: paraganglioma in close proximity to adrenocortical adenoma.

We report a potential pitfall of 123I-metaiodobenzylguanine (MIBG) scan. Magnetic resonance imaging performed for other reasons, showed 2.5 cm tumor in the left adrenal gland. On questioning patient had episodic palpitations, flushing and hypertension suggestive of pheochromocytoma. Urinary metanephrine level was of borderline value but serum chromogranin A level was clearly elevated. 123I-MIBG scan showed accumulation of the tracer in the upper left abdomen and the finding was suspected to be intra-adrenal pheochromocytoma. During operation two separate tumors, adrenocortical adenoma in the left adrenal gland, and another smaller, extra-adrenal paraganglioma locating very close to the adenoma, were found. Thus the positive MIBG finding was caused by a paraganglioma with the concurrent presentation of nonfunctioning adrenocortical adenoma.

3-Iodobenzylguanidine↗

PubMed1997

Prognostic Factors Including Proliferation Markers Ki-67, bax, and bcl-2 in Temporal Bone Paraganglioma.

Valuable criteria with which to predict the clinical behavior of the temporal bone paraganglioma or the response to treatment are lacking. The analysis of markers of cell proliferation is a possibility to estimate the prognosis. Extensive patient data on 40 temporal bone paragangliomas were gathered over the years and correlated with the data obtained by staining histologic sections with bcl-2, bax, and MIB I markers of cellular proliferation. The immunohistochemistry was in all cases negative for bcl-2, positive for bax, and for Ki-67 positive in 20% of tumors. The scores for Ki-67 did not correlate with the majority of clinical parameters, except for treatment modality, preoperative hearing loss, and cranial nerve involvement. The tendency toward poorer hearing and a higher incidence of preoperative lower cranial nerve palsies was demonstrated in patients with higher Ki-67 scores. Furthermore, the higher rate of subtotal tumor removals in these patients reveals technical difficulties in accomplishing a radical removal, although the incidence of residual tumors was thus not affected. In view of the present information obtained with proliferation markers, the site of tumor origin still remains the most predictive variable for the course of the disease.

Journal Article↗

PubMed1993

Preoperative embolization of paragangliomas (glomus tumors) of the head and neck: histopathologic and clinical features.

Forty-eight surgical specimens were examined histologically and the case histories reviewed to determine the histopathologic features of embolized paragangliomas (glomus tumors) in relation to the time interval between embolization and surgery. Different degrees of thrombus formation and of multinucleated foreign body giant cells occurred during the first 7 days after embolization; thereafter, glant cells with active phagocytosis, fragmentation of embolic material, and partial revascularization were observed. Only one third of the tumor vessels were embolized. Complete obliteration of 40% of embolized vessels occurred more than 2 months postembolization. The histologic changes induced by embolization in paragangliomas of the head and neck may be classified in four stages. Histologically, the best time for surgery is within 8 days from embolization; surgery performed more than 8 days following embolization, however, is not compromised by revascularization.

Journal Article↗

PubMed1997

Arterial and venous paragangliomas: the value of a preoperative pacemaker and a multidisciplinary approach.

Non-chromaffin paragangliomas are very rare tumours. They arise from chemoreceptor cells at multiple sites throughout the body. They are usually very vascular and most arise in intimate contact with vital structures making their excision a surgical challenge. We describe in this paper four cases of paragangliomas in the carotid body and the jugular bulb. Their presentation was unusual. Two cases underwent excisional surgery and two had radiation treatment because they were not fit for surgery. We had to use a temporary pacemaker for preoperative preparation of the carotid body because of an associated hypersensitive carotid sinus syndrome. The clinical presentation and the surgical management will be described in this paper.

Adult↗

PubMed2004

Paraganglioma-like dermal melanocytic tumor: a unique entity distinct from cellular blue nevus, clear cell sarcoma, and cutaneous melanoma.

We are reporting a previously undescribed primary dermal melanocytic tumor identified by reviewing all dermal melanocytic tumors referred in consultation that did not qualify histologically as a previously described entity. From these cases, 8 were remarkably similar. We termed them "paraganglioma-like dermal melanocytic tumor" (PDMT) based on their nested growth pattern. This term is used descriptively and does not imply any histogenetic or biologic similarity to true paraganglioma. PDMT is primarily a tumor of the extremities of adult females (18-53 years, mean 35 years; males 2; females 6) which present as a dermal nodule (range, 0.5-4.2 cm; mean, 1.4 cm) composed of nests of clear to amphophilic oval cells separated by delicate fibrous strands. Nuclear atypia was mild and mitotic activity low (1-4 mitoses/10 HPF). Melanin was not obvious on light microscopy. Tumors expressed S-100 protein (8 of 8), Melan-A (4 of 8), HMB-45 (8 of 8), and microphthalmia transcription factor (8 of 8) and lacked pancytokeratin (8 of 8) and smooth muscle actin (8 of 8). FISH analysis of 5 cases revealed an intact EWS gene locus, supporting absence of the clear cell sarcoma 12;22 translocation. Follow-up information in 8 patients (range, 35-92 months; mean, 54 months) indicated that all were alive without disease. PDMT comprises a clinically and pathologically unique subtype of dermal melanocytic tumors. Our study suggests a benign course, although a lesion of low malignant potential cannot be excluded.

Adolescent↗

PubMed2006

Baroreflex failure: a rare complication of carotid paraganglioma surgery.

Multiple head and neck paragangliomas are a rare occurrence. We report a patient with removal of bilateral carotid paragangliomas who subsequently developed baroreflex failure 1 month after surgery. The pathology and physiology of this presentation is extremely interesting because it exhibits the complex homeostatic mechanisms involved in the maintenance of steady-state blood pressure. Although rare, it is important to be aware of baroreflex failure and its variable course. Most postsurgical cases occur within days of surgery but, as this report demonstrates, hypertensive urgency can occur weeks to months after surgery; and if not recognized early, it can lead to disastrous postoperative complications such as stroke and myocardial infarction.

Adult↗

PubMed2006

Surgical removal of jugular paragangliomas after stenting of the intratemporal internal carotid artery: a preliminary report.

OBJECTIVE: Jugular paragangliomas with extensive involvement of the internal carotid artery (ICA) represent a true challenge for surgeons, especially in the presence of inadequate collateral circulation through the circle of Willis. The aim of our study is to present a preliminary report of our experience with the surgical removal of three such cases using the stenting of the ICA as the method of choice for protecting and preserving the integrity of the artery. METHODS: This retrospective study was conducted at Gruppo Otologico, a private referral center for neurotology and skull base surgery. The subjects of our study are three cases of jugular paragangliomas with extensive involvement of the ICA and inadequate collateral circulation. These cases are the first three cases operated at our center after stenting of the intratemporal portion of the ICA. RESULTS: Complete surgical removal of the tumor, including the part involving the ICA, was achieved in all cases. Over a follow-up period of 22 to 30 months, no complications occurred and the patency of the stented arteries was preserved. CONCLUSION: Although more follow-up is still needed before establishing the exact long-term outcome of stenting the intratemporal ICA, our preliminary report shows that the stent has facilitated the complete surgical removal of the tumor, preserving the integrity of the ICA.

Adult↗

PubMed1986

Aggressive oncocytic neuroendocrine tumour ('oncocytic paraganglioma') of the cauda equina.

An oncocytic neuroendocrine tumour ('oncocytic paraganglioma') of the cauda equina is reported. The tumour was predominantly intradural, with extension into and destruction of surrounding vertebral bone. The tumour had an organoid pattern, and the tumour cells had abundant non-argyrophilic eosinophilic cytoplasm. Immunocytochemical stains for neurone-specific enolase, S-100 protein, keratin and carcinoembryonic antigen were positive, but stains for glial fibrillary acidic protein were negative. On ultrastructural examination, there were numerous mitochondria and scattered 200 nm dense-core membrane-bound granules, that rarely clustered in small aggregates. Intermediate filaments were focally arranged in long compact bundles. The histogenesis of tumours reported as cauda equina paragangliomas is discussed.

Actin Cytoskeleton↗

PubMed1989

Malignant paraganglioma of the organ of Zuckerkandl. Initially diagnosed as renal cell carcinoma by biopsy of a right humeral fracture lesion.

Malignant paraganglioma of the organ of Zuckerkandl (malignant POZ) in a 38-year-old woman is reported. Ten months before her death, the patient suffered from right shoulder pain. By biopsy of a pathological right humeral fracture, a histological diagnosis of metastatic renal cell carcinoma was initially made. However, autopsy revealed a left retroperitoneal tumor, which was diagnosed as malignant POZ. Upon immunohistochemical examination, tumor cells showed definite positivity for neuronspecific enolase (NSE) and chromogranins, and neurosecretory granules were found electron microscopically. The skeletal system is the most common site for distant metastases of malignant POZ. We were unable to find any previous reports of malignant POZ which were first manifested as humeral metastases, as in the present case. As paraganglioma might be histologically confused with a large variety of neoplasms (e.g. renal cell carcinoma), differential diagnosis of this condition is discussed.

Adult↗

PubMed1987

Primary adenocarcinoma of the temporal bone mimicking paragangliomas: radiographic and clinical recognition.

Three cases of primary adenocarcinoma of the temporal bone which simulated a glomus jugulare tumor (on the basis of a thorough preoperative evaluation) are detailed. In each case, a vascular mass was seen beneath an intact tympanic membrane and angiography revealed the presence of a highly vascular mass centered on the jugular bulb. Computed tomography revealed erosive changes within the jugular fossa that were characteristic of a glomus jugulare tumor; in two cases, a significant posterior fossa extension was documented. The intraoperative findings failed to suggest a lesion different from paraganglioma in two of the cases; in the remaining case, the erosion of both bone and dura was diffuse and more suggestive of a malignant neoplasm. A discussion of the clinical behavior of adenocarcinomas of the middle ear is included in order to emphasize the importance of differentiating these lesions preoperatively from the more commonly encountered paragangliomas.

Adenocarcinoma, Papillary↗

PubMed2000

Bilateral carotid body paraganglioma: case report.

CONTEXT: Surgical treatment of carotid body paragangliomas is a challenge to the surgeon because of the large vascularization of the tumor, involvement of the carotid vessels and the close anatomical relationship with the cranial nerves. CASE REPORT: A 63-year-old patient was submitted to resection of two carotid body paraganglioma tumors found in the right-side and left-side carotid bodies at the bifurcation of the common carotid arteries. Two surgeries were performed at different times and neither of them presented any morbidity. Arteriography was fundamental for diagnosis of the small, asymptomatic tumor on the right side. DESIGN: Case Report

Carotid Arteries↗

PubMed2004

Anatomy involved in the jugular foramen approach for jugulotympanic paraganglioma resection.

The goal in paraganglioma resection is to allow adequate exposure to remove the lesion while preserving cranial nerve function. Knowledge of the anatomy of the jugular foramen is crucial to this endeavor. In this report the authors describe a jugular foramen approach for the resection of glomus jugulare tumors in cases in which rerouting of the facial nerve can be avoided. This approach provides adequate exposure of the jugular bulb for many jugulotympanic paragangliomas without increased risk of injury to the facial nerve. In addition, special circumstances surrounding intracranial and carotid artery involvement are briefly discussed.

Cranial Nerve Injuries↗

PubMed2001

[Anesthesia for carotid paraganglioma exeresis. Report of 3 cases].

This report of carotid paraganglioma excision in three patients discusses differential diagnosis, preoperative assessment, preoperative embolization of the tumor, monitoring of anesthesia including cerebral oximetry, and postoperative complications. We consider cerebral protection to be essential during carotid paraganglioma surgery. Such protection may be provided by drugs such as sodium thiopental and by temporarily shunting the internal carotid artery. Preoperative angiography is also important for evaluating retrograde circulation through Willis's polygon and to examine the arteries irrigating the tumor. Information thus obtained helps establish the need for presurgical embolization of the tumor, thereby possibly reducing the risks, such as obstructive hematoma that are inherent to the procedure. Finally, in our opinion, full monitoring should include cerebral oximetry so that possible complications can be detected and resolved.

Adult↗

PubMed2002

[Carotid body paraganglioma: an uncommon localization. A case report].

Paraganglioma are rare lesions, which come from neural cells. They arise the most frequently from carotid body. The tumor is classically located in the carotid bifurcation, between internal and external carotid arteries. We report a case of carotid body paraganglioma which is uncommonly located under the carotid bifurcation.

Adult↗

PubMedDate not supplied

Carotid body paraganglioma and SDHD mutation in a Greek family.

BACKGROUND: Carotid body (CB) is a highly specialized paraganglion originating from the neural crest ectoderm. CB paraganglion can be caused either by a genetic predisposition (hereditary paraganglia) or by chronic hypoxic stimulation. Germline mutations in any of the following genes: SDHD, SDHC, SDHB, PGL2 or other unknown genes, can cause paragangliomas (PGLs). MATERIALS AND METHODS: We studied a Greek family in which the two daughters had carotid body paraganglioma, whereas both parents did not. RNA extraction, reverse transcriptase polymerase chain reaction and direct DNA sequencing were performed, in order to identify SDHD mutations in all four exons. RESULTS: Our results revealed the existence of the missense mutation Y114C, in exon-4 of the SDHD gene, in the unaffected father and both affected sisters. CONCLUSION: DNA testing was performed, for the first time in Greece, on patients with carotid body tumor. This marks a new geographical location, in the literature, for this mutation.

Adult↗

PubMed2005

[The jugulotympanic paragangliomas: 41 cases report].

OBJECTIVE: The jugulotympanic paragangliomas (JTP) represents the most frequent tumour of the middle ear but also of the temporal bone, after the acoustic neurinoma. The management of these vascular tumours remains uncleared. The purpose of this study was to report our experience about JTP in the CHU of Grenoble. MATERIALS AND METHODS: Retrospective study of 41 patients, between 1973 and 1996. Six stages A, 8 stages B and 27 stages C are reported in whom 20 cases (49%) presented an intracranial extension (classification of Fisch). There were 2 familial cases with multiple localisations, in particular carotid. All the patients were divided in 3 groups: surgery or radiation therapy in first intention, surgery followed by radiation therapy. RESULTS: A total tumor removal without recurrence was achieved by surgery in more than 95% of the cases with 6 years follow-up but was associated with significant morbidity (major cranial nerve injury). We noticed one death by laryngospasme (C2Di2 tumour operated by infratemporal A approach). A stabilization of the tumour was obtained with radiotherapy in first intention in 75% of the cases (5 years follow-up) but with a risk of radionecrosis. A revision surgery was necessary in 3 cases. CONCLUSION: The comparaison of our different therapeutic management, surgery (23), radiation therapy (16) or combined (2), encourage us to perform a radical surgery whenever possible. Because of the slow rate of growth, the radiotherapy is indicated for older patients, at risk for surgery or extensive tumors. The objectives of the radiation therapy are to obtain a tumoral stabilization with improvement of the symptoms and low morbidity. The management of this rare pathology must be multidisciplinary. The recent discoveries on genes encoding three succinate dehydrogenase subunits (SDHD, SDHB et SDHC) will allow a genetic detection of asymptomatic case and will define the procedures for their management, coordinated by a national network PGL.NET. A retrospective study could also study the real incidence of familial paragangliomas.

Adult↗

PubMed2006

[Combined treatment of embolization followed by surgical resection in carotid paraganglioma].

Carotid body tumors represent a relatively uncommon finding in vascular surgery. We present the case of a 62 years old man with a right carotid paraganglioma diagnosed by cervical ultrasound scanner, magnetic resonance imaging and carotid arteriography. We decided to treat the patient in two times. In a first approach the tumor was percutaneously embolized, later on in a second time the paraganglioma was surgically resected. The patient is doing well without echographic evidence of tumor 3 years after the operation. This modality of treatment, embolization followed by surgical excision is a useful and valuable alternative.

Brain Neoplasms↗