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      Retroperitoneal non-functioning paraganglioma: A difficult tumour to diagnose and treat

      case-report

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          Highlights

          • •

            We report a case of non-functional retroperitoneal paraganglioma.

          • •

            Non-functioning retroperitoneal paragangliomas are rare and difficult to diagnose.

          • •

            Surgical excision is recommended given potential malignant transformation.

          • •

            Lifelong follow up is recommended as recurrence and metastasis are common.

          Abstract

          Paragangliomas are rare neoplasms arising from cells of the primitive neural crest. These tumours are often difficult to diagnose and treat. We report a case of a 42 year old female presenting with abdominal pain who had a retroperitoneal tumour situated at the aortic bifurcation. Serum catecholamine levels were normal. Complete resection of the tumour was performed. The histological examination and immunohistochemical analyses concluded the diagnosis of an organ of Zuckerkandl paraganglioma.

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          The diagnosis and medical management of advanced neuroendocrine tumors.

          Gregory Kaltsas, Robert B. Grossman, G Michael Besser (2004)
          Neuroendocrine tumors (NETs) constitute a heterogeneous group of neoplasms that originate from endocrine glands such as the pituitary, the parathyroids, and the (neuroendocrine) adrenal, as well as endocrine islets within glandular tissue (thyroid or pancreatic) and cells dispersed between exocrine cells, such as endocrine cells of the digestive (gastroenteropancreatic) and respiratory tracts. Conventionally, NETs may present with a wide variety of functional or nonfunctional endocrine syndromes and may be familial and have other associated tumors. Assessment of specific or general tumor markers offers high sensitivity in establishing the diagnosis and can also have prognostic significance. Imaging modalities include endoscopic ultrasonography, computed tomography and magnetic resonance imaging, and particularly, scintigraphy with somatostatin analogs and metaiodobenzylguanidine. Successful treatment of disseminated NETs requires a multimodal approach; radical tumor surgery may be curative but is rarely possible. Well-differentiated and slow-growing gastroenteropancreatic tumors should be treated with somatostatin analogs or alpha-interferon, with chemotherapy being reserved for poorly differentiated and progressive tumors. Therapy with radionuclides may be used for tumors exhibiting uptake to a diagnostic scan, either after surgery to eradicate microscopic residual disease or later if conventional treatment or biotherapy fails. Maintenance of the quality of life should be a priority, particularly because patients with disseminated disease may experience prolonged survival.
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            Benign paragangliomas: clinical presentation and treatment outcomes in 236 patients.

            Mitchell G. Thompson, Lance D. Erickson, M Ebersold … (2001)
            Paragangliomas are rare tumors that arise from extraadrenal chromaffin cells. We examined the clinical characteristics, location, treatment, and outcome of 236 patients (141 females, 60%) with 297 benign paragangliomas evaluated at the Mayo Clinic during 1978-1998. The mean age (+/-SD) at diagnosis was 47 +/- 16 yr. Of the 297 paragangliomas, 205 were in the head and neck region, and 92 were below the neck. Paragangliomas were discovered and diagnosed incidentally on imaging studies in 9% of patients. Biochemical screening was performed in 128 patients; 40 patients (17% of the total and 31% of those screened) had hyperfunctional tumors. Of the 40 patients with tumoral catecholamine excess, 38 had documented hypertension. In patients identified with catecholamine-secreting paragangliomas, the sensitivities achieved by measurements in the 24-h urine collection were 74% for total metanephrines, 84% for norepinephrine, 18% for dopamine, and 14% for epinephrine. Multiple imaging modalities were used for tumor localization. The false negative rates were 0% for magnetic resonance imaging, 5.8% for computed tomography, 3.4% for angiography, 10.7% for ultrasonography, and 39% for radioactive iodine-labeled metaiodobenzylguanidine scintigraphy. Of 192 patients (81.4%) with follow-up data (mean, 43.9 months; range, 0.5-240), operative cure was achieved in 133 (69%). Of the 59 patients without cure, 23 had persistent disease, 5 had recurrent disease, 16 had multiple persistent synchronous tumors, and 15 subsequently developed metachronous tumors. In conclusion, most paragangliomas are nonhypersecretory and located in the head and neck region. Magnetic resonance imaging was associated with the lowest false negative rate, and metaiodobenzylguanidine was the least sensitive imaging study. A significant proportion of patients (31%) has persistent or recurrent disease, and long-term follow-up is important.
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              Extra-adrenal pheochromocytoma.

              R Whalen, A Althausen, G H Daniels (1991)
              Extra-adrenal pheochromocytomas may arise in any portion of the paraganglion system, although they most commonly occur below the diaphragm. The most common site of occurrence of extra-adrenal pheochromocytoma is the superior para-aortic region between the diaphragm and lower renal poles. Although the traditional teaching has been that 10% of all pheochromocytomas are at extra-adrenal sites, this may be an underestimation. Extra-adrenal pheochromocytomas probably represent at least 15% of adult and 30% of childhood pheochromocytomas. They may be malignant in up to 40% of the cases, although conflicting data add to the uncertainty of this point. Patients with tumors arising at extra-adrenal sites commonly present with headache, palpitations, sweating and hypertension. The diagnosis is most often confirmed by demonstrating increased catecholamine production, usually by measurement of urinary catecholamines and/or their metabolites. CT scanning is presently the imaging procedure of choice for localization. The roles of MRI and 131I-MIBG scintigraphy in the localization process are still being determined. Thorough preoperative pharmacological preparation, attentive intraoperative monitoring and aggressive surgical therapy all have an important role in achieving the safest and most successful outcome. Complete surgical excision is the treatment of choice for primary extra-adrenal pheochromocytoma as well as recurrent or metastatic disease. When residual tumor cannot be resected, medical therapy for symptomatic relief is preferred, since radiotherapy and chemotherapy have limited effectiveness. Extra-adrenal pheochromocytomas are more likely to recur and to metastasize than their adrenal counterparts, making lifelong followup with annual determinations of catecholamine production essential.
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                Author and article information

                Contributors
                Emma Gannan
                Journal
                Journal ID (nlm-ta): Int J Surg Case Rep
                Journal ID (iso-abbrev): Int J Surg Case Rep
                Title: International Journal of Surgery Case Reports
                Publisher: Elsevier
                ISSN (Electronic): 2210-2612
                Publication date PMC-release: 12 November 2015
                Publication date Collection: 2015
                Publication date (Electronic): 12 November 2015
                Volume: 17
                Pages: 133-135
                Affiliations
                [a ]Department of General Surgery, Ballarat Base Hospital, Drummond St North, Ballarat, VIC 3350, Australia
                [b ]Department of General Surgery, The Royal Melbourne Hospital, Grattan St, Parkville, VIC 3050, Australia
                [c ]Department of Pathology, Ballarat Base Hospital, Drummond St North, Ballarat, VIC 3350, Australia
                Author notes
                [* ]Corresponding author at: Department of General Surgery, The Royal Melbourne Hospital, Grattan Street, Parkville, VIC 3050, Australia. ecgannan@ 123456yahoo.com.au
                Article
                Publisher ID: S2210-2612(15)00484-8
                DOI: 10.1016/j.ijscr.2015.11.004
                PMC ID: 4701798
                PubMed ID: 26624503
                SO-VID: bb6fd8cd-3270-415a-825d-11ec5a325d1f
                Copyright © © 2015 The Authors
                License:

                This is an open access article under the CC BY-NC-ND license (http://creativecommons.org/licenses/by-nc-nd/4.0/).

                History
                Date received : 15 July 2015
                Date revision received : 8 November 2015
                Date accepted : 8 November 2015
                Categories
                Subject: Case Report

                Keywords: ct, computed tomography,nse, neuron-specific enolase,mibg, metaiodobenzylguanidine,paraganglioma,organ of zuckerkandl,neuroendocrine tumour
                Data availability:
                Keywords: ct, computed tomography, nse, neuron-specific enolase, mibg, metaiodobenzylguanidine, paraganglioma, organ of zuckerkandl, neuroendocrine tumour

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