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The management of vagal paragangliomas is extremely challenging. Treatment of these lesions must be tailored individually for each patient. The best treatment modality depends on the patient's age and health and the size and extent of the tumor. This article discusses clinical presentation, multicentric and malignant vagal paragangliomas, evaluation, embolization, surgical management, and special considerations in the management of vagal paragangliomas.
Laryngeal paragangliomas are classified as supraglottic and infraglottic. This article defines each type of paraganglioma, discusses the clinical features and diagnoses, and covers the surgical management. This article also addresses sinonasal paragangliomas, including their clinical features, diagnosis, and treatment.
Paragangliomas are uncommon laryngeal tumors. A comprehensive review of the world literature disclosed the use of a variety of terms to designate the same disease as a result of the historical uncertainty of its origin. Important features of these neoplasms not previously reported include the high mortality rate in patients presenting with laryngeal pain and the need for longer follow-up periods to judge biological aggressiveness. An additional patient is presented to illustrate the diagnostic and therapeutic challenges inherent in the management of laryngeal paragangliomas. Radiotherapy has not been found to be beneficial, and surgery remains the treatment of choice for patients with laryngeal paragangliomas.
A case of primary intrathyroidal paraganglioma is reported, and the light microscopic and immunohistochemical findings are described. Primary paragangliomas of the thyroid region are extremely uncommon and are therefore often confused clinically and histopathologically with more common intrathyroidal mass lesions. The diagnostic difficulties are underscored by the present case, which was misdiagnosed twice, firstly as a medullary thyroid carcinoma and secondly as a follicular thyroid carcinoma. Immunohistochemistry may be very helpful in arriving at the correct diagnosis. The case was further complicated by a second neck mass contralateral to the original thyroid nodule, which was interpreted as consistent with metastasis. The second lesions was proved angiographically and histologically to be a carotid body paraganglioma.
We report a 49-year-old man with a paraganglioma involving the nasopharynx, which according to the literature is the fourth case in Japan and the twentieth in the world. The initial histopathologic findings of the biopsy specimen from the nasopharyngeal tumor suspected a carcinoid tumor. However, the final histopathologic diagnosis from the surgically resected specimen was paraganglioma, determined by electron microscopy and using several stains, such as Grimelius and Neuron-specific enolase (NSE) stains. We wish to add this case of "primary" nasopharyngeal paraganglioma to the literature, and to report the pathologic findings and clinical course of this unusual case.
Nonchromaffin paragangliomas are unusual tumours arising from widely distributed paraganglionic tissues probably of neural crest origin. In the head and neck region they are usually seen as carotid body or jugulotympanic tumours. Other rarely reported sites in the head and neck region are the orbit, nose and larynx. This report deals with a case of sinonasal paraganglioma which was initially treated with surgery and radiotherapy. Twenty two years later the tumour recurred and showed a rapid growth due to malignant transformation which we believe is late effect of radiotherapy. The clinical features, histopathology and role of radiotherapy in sinonasal paragangliomas together with a review of the medical literature have been discussed.
An unusual case of malignant paraganglioma that manifested itself as pain in the temporomandibular joint and trismus and later manifested itself as a rapidly growing neck mass is described. Computed tomography revealed a huge mass with upward and downward extension in the infratemporal fossa. Radiotherapy was completely ineffective. At autopsy, a malignant paraganglioma with multiple metastases was found, but the site of origin could not be determined because of the size and extent. One hundred and ninety-one cases of paraganglioma of the head and neck published in the Japanese literature were analyzed with regard to their site, clinical, radiographic and microscopic features as well as treatment and results.
Extraadrenal paragangliomas involving the spinal cord are not common and usually take the compression of the cauda equine. Two cases of paraganglioma of the cauda equina with a different presentation are reported, and the clinical and histopathology findings of this tumor, as well as diagnosis, treatment and prognosis are review. We stress the importance of the high tumor vascularization form of intradural that can make impossible achieve a complete resection. Laminotomy and intraoperatory echography are very useful in the approach to intradural tumors, such as paraganglioma.
Paragangliomas of the urinary bladder are very rare. To date, there are no reliable methods for predicting their clinical behavior, so long-term follow-up is required. We describe a paraganglioma of the urinary bladder in a 32-year-old male who presented with painless gross hematuria. Abdominal ultrasonography revealed a protruding mass with hematoma over the right lateral wall of the urinary bladder. The tumor was not completely resected by transurethral resection of bladder tumor (TURBT) due to intermingling with the bladder wall. Follow-up I131-metaiodobenzylguanidine was performed 3 weeks after surgery and suspected incomplete resection lesions were noted. Histologic examination of the tumor indicated paraganglioma of the urinary bladder. We also provide a brief review of the literature for comparison.
A case of paraganglioma arising within a parathyroid gland is reported. The lesion was an incidental finding in a block dissection of neck performed for squamous carcinoma of the pharynx. A well-circumscribed lesion, exhibiting the characteristic pathological features of a paraganglioma, was embedded within the right inferior parathyroid gland. Due to its location, the chief histological differential diagnosis was an unusual variant of parathyroid adenoma. Immunohistochemistry and electron microscopy assisted in reaching a diagnosis. This, as far as we are aware, is the first reported case of a paraganglioma of the parathyroid gland.
A paraganglioma is a neuroendocrine neoplasm that originates from the paraganglion cells of the parasympathetic system. The average age of presentation is in the fifth decade. We report a case of laryngeal paraganglioma in a five-year-old child, the youngest case ever recorded. The features of paraganglioma which differentiate it from other tumours are also discussed.
Sixty patients with glomus jugulare tumour, seen at St. Bartholomew's Hospital, London, over forty years, have been reviewed. The term 'jugulo-tympanic paraganglioma' is suggested as a more accurate name for these tumours. The following simple classification is recommended: Group I tumours (tympanic paragangliomas) are those with or without VIIth nerve palsy, but with no other cranial nerve involvement. Group II tumours (jugular paragangliomas) are those with involvement of any cranial nerve other than the VIIth. The method and results of treatment by radiotherapy are discussed. It is concluded that surgery and radiotherapy give comparable results in Group I tumours, but radiotherapy gives superior results in Group II tumours.
Paraganglioma (pheochromocytoma) is probably the most fascinating of all tumors as it can present with a wide range of clinical manifestations. Paraganglioma of the urinary bladder is one of the rare tumors and constitute less than 10% of all bladder tumors. The common presentation of paraganglioma of the urinary bladder is painless haematuria, headache, palpitation and anxiety. Malignancy is uncommon and no histological feature is characteristic of malignancy. Invasion of adjacent organs or metastasis to the lymph nodes is the only criteria to suggest malignancy. In the presence of proven metastasis radical cystectomy with pelvic lymphadenectomy is recommended. In the present case the tumor was arising from anterior wall of the bladder away from the bladder neck and that in a large capacity bladder it was possible to get a wide margin for partial cystectomy along with bilateral pelvic lymphadenectomy without violating the oncological principles. At a follow up of two years there is no evidence of recurrence of tumor.
Paragangliomas (glomus tumours) are benign, hypervascular tumours which in general are treated by surgical excision. The indication for treatment of these often slow-growing tumours needs additional criteria for predicting tumour progressiveness. For this reason the nuclear DNA content of 99 paragangliomas, 65 of them originating from patients with a positive family history, was analysed by flow cytometry. Unequivocal evidence of DNA aneuploidy was found in 37% of these clinically and histologically benign tumours, the average duration of follow up amounting to at least 10 years. The DNA index of the aneuploid tumours ranged from 0.90 to 2.03. No correlation was found between DNA ploidy and familiality or between DNA content and clinical criteria indicative of tumour progression, which means that DNA ploidy of these tumours cannot serve as a predictor for an expected growth pattern or familiality. DNA aneuploidy in hereditary and sporadic paragangliomas is not clinically related to malignancy, but indicates that these tumours are true neoplasias cytogenetically.
STUDY DESIGN: Case report. OBJECTIVES: To report on a case of paraganglioma presenting in an uncommon extradural thoracic localization. SETTING: Department of Neurosurgery, Florence, Italy. CASE REPORT: A 43-year-old woman with a thoracic lesion extending into the extradural space along four levels, T(1)-T(4), presented with sudden spastic incomplete paraplegia and paresthesia at the lower limbs. RESULTS: The neoplasm was surgically resected 'en bloc' and histological findings corresponded to paraganglioma. One year after surgery, the patient was walking without assistance, a T(3)-T(4) hypoesthesia was still present and an magnetic resonance imaging (MRI) study showed no signs of focal recurrence. CONCLUSIONS: The imaging features of thoracic paragangliomas may be misleading and an advanced malignant lesion could be primarily suspected; thus, a histological study is always needed. Total resection is the gold standard therapy. Owing to the risk of recurrence or multicentric growth, follow-up must be prolonged and accurate.
AIM: This study analyses the occurrence and distribution of neurotrophins and their receptors in some types of tumours of neural-crest derived cells. METHODS AND RESULTS: Light microscopy immunohistochemistry associated with quantitative image analysis was used to study the expression of neurotrophins (nerve growth factor, brain-derived neurotrophic factor and neurotrophin (NT)-3) and their cognate receptors (p75(LNGFR), TrkA, TrkB and TrkC) in histologically defined ganglioneuroma, phaeochromocytoma and paraganglioma. The material was fixed in 10% formaldehyde, paraffin-embedded and processed for indirect peroxidase immunohistochemistry using a battery of poly- and monoclonal antibodies to detect neurotrophins and their receptors, as well as some neuronal, endocrine and glial cell markers. A subpopulation of cells in phaeochromocytomas and ganglioneuromas expressed NT-3, but not other neurotrophins, while in paragangliomas no neurotrophins were detected. Regarding neurotrophin receptors, all tumours lacked p75(LNGFR), except for the ganglionic part of a case of mixed phaeochromocytoma, whereas they displayed TrkA (two of two ganglioneuromas, six of nine phaeochomocytomas and three of four paragangliomas). Furthermore, TrkC was regularly detected in a neuronal subpopulation in ganglioneuroma. Interestingly, the percentage of neurones expressing TrkA and TrkC was increased with respect to normal tissues in ganglioneuromas, as well as the percentage of the area occupied by TrkA-immunoreactive cells in the phaeochromocytomas. CONCLUSION: The pattern of expression of neurotrophins and neurotrophin receptors in the analysed tumours basically matches that of sympathetic neurones, adrenal chromaffin cells and paraganglionic cells, and suggests responsiveness of these cells to neurotrophins. Nevertheless, the function of TrkA and TrkC in regulating the biology of these tumours, if any, remains to be elucidated.
We report a rare case of pelvic malignant paraganglioma that was treated with surgery, combination chemotherapy and radiation. A 47-year-old man was diagnosed with pelvic malignant paraganglioma that had metastasised to the thoracic vertebrae. The pelvic mass, which was 6 cm in size, was on the posterior side of the bladder and had invaded the prostate, seminal vesicle and bladder neck. We resected the intrapelvic tumor and lymph nodes using cystoprostatectomy. Metastases to bilateral obturator lymph nodes and the right internal iliac lymph node were shown by pathology. Adjuvant therapies included six courses of the combination chemotherapy (cyclophosphamide, vincristine and dacarbazine), and 12 courses of VP-16 therapy. Radiation therapy was done for metastasis of the thoracic vertebrae. Local recurrence, progression of bone metastasis and new metastasis have not been detected since these treatments. The patient has been clinically stable during 20 months of follow-up. Chemotherapy of cyclophosphamide, vincristine and dacarbazine and VP-16 with radiation appears to be effective in treating advanced malignant paraganglioma.
Paragangliomas arising as primary neoplasms in the intracranial portion of the central nervous system are relatively uncommon and only have been rarely reported. We report a case of apparent primary paraganglioma arising in the left cerebellar hemisphere in an 11-year-old girl who presented with a 6-month history of headaches. The tumor was marked by a nested architectural pattern (zellballen), delimited by S-100-positive sustentacular cells. Morphologic and immunohistochemical features of the neoplasm are discussed and literature is reviewed regarding involvement of the central nervous system by paraganglioma.