Systemic Inflammatory Syndrome Associated with a Case of Jugular Paraganglioma

Sokabe, Ayuko; Mizooka, Masafumi; Sakemi, Rinne; Kobayashi, Tomoki; Kishikawa, Nobusuke; Yokobayashi, Kenichi; Kanno, Keishi; Tazuma, Susumu

doi:10.2169/internalmedicine.55.4655

Cited by 7 publications

(6 citation statements)

References 17 publications

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“…(13,14) The classic signaling of this cytokine starts with the binding to its membrane receptor (IL-6R), which leads to the activation of the intracellular signaling pathway of the Janus kinases. Subsequently, signal transduction-activated transcription factors translocate into the nucleus and gene expression is induced, as in the case of pro-opiomelanocortin (POMC), (15,16) which then generates ACTH by cleavage. In this way, it is possible to explain ACTH-dependent hypercortisolism, which was one of the biochemical alterations presented by the reported patient.…”

Section: Discussionmentioning

confidence: 99%

Pheochromocytoma presenting as fever of unknown origin, a case report

et al. 2020

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Introduction: Pheochromocytoma is a generally benign neoplasm derived from chromaffin cells of the adrenal medulla. It is characterized by the production of large amounts of catecholamines and also by the capacity to secrete bioactive peptides such as cytokines, mainly interleukin-1 (IL-1), interleukin-6 (IL-6) and TNF alpha.Case presentation: 24-year-old man, who consulted for fever, myalgia, and choluria. His laboratory tests were compatible with a systemic inflammatory response without infectious or autoimmune causes. However, a fluorodeoxyglucose positron emission tomography (FDGPET) revealed a left adrenal mass, without extra-adrenal lesions. On admission, increased levels of differentiated urine methanephrines, elevated baseline cortisol, non-suppressed adrenocorticotrophic hormone (ACTH), and positive low dose dexamethasone suppression test for cortisol were found. With suspicion of catecholamine and ACTH-producing pheochromocytoma, a tumor resection was performed, which conspicuously resolved all alterations of the inflammatory response. The histologic findings confirmed a pheochromocytoma, but the immunostaining for ACTH was negative. A literature review and the comparison of the findings with other reported cases allowed inferring that this was a case of interleukin-producing pheochromocytoma.Conclusion: Pheochromocytoma may be a cause of febrile syndrome, with IL-6 being the main mediator, which explains the manifestationsof systemic inflammation and ACTH-mediated hypercortisolism.

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Section: Discussionmentioning

confidence: 99%

Pheochromocytoma presenting as fever of unknown origin, a case report

et al. 2020

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“…However, the literature reports only limited cases of IL-6 over-production in paraganglioma. Sokabe et al reported a case in a patient with the systemic inflammatory syndrome, having increased IL-6 level due to a jugular paraganglioma, without any detectable hormone secretion including catecholamines (20). Another case report in the literature presented the coexistence of Castleman disease and pheochromocytoma possibly by linking up to the expression of IL-6 by the tumor mass (17).…”

Section: Discussionmentioning

confidence: 99%

Retroperitoneal Castleman Disease Mimicking Paraganglioma in a Patient with Klinefelter Syndrome: A Case Report

Hepşen¹,

Özbek²,

Sencar³

et al. 2019

Turk J Endocrinol Metab

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“…Mass effect on facial nerve can lead to subclinical facial asymmetry, laterality of proprioception in the orofacial muscles and TMJ dysfunction [6]. Paraganglioma rarely secrete various neuropeptide hormones, such as adrenocorticotropic hormone, and inflammatory cytokines, such as interleukin-6 (IL-6) inducing acute inflammatory reaction and pain [7].…”

Section: Discussionmentioning

confidence: 99%

A Case of Jugular Paraganglioma Presented as Temporomandibular Joint Disorder

Azimova¹

2017

JNSK

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Background: Jugular paraganlioma is a slow-growing tumor originating from the paraganglion cells. The diagnosis of a jugular paraganglioma may be delayed due to its atypical symptoms. We report a case of jugular paraganglioma presented as temporomandibular joint (TMJ) disorder.Clinical presentation: 29-years old woman with preliminary diagnosis probable atypical face pain. She experienced her pain for the first time 2 months prior to reference. Pain intensity was seven to eight out of ten points on Visual Analogue Scale, simple and combined analgetics and NSAIDs were ineffective. Pain was dull, aching, localized in the right temporal area with irradiation to the ipsilateral maxillar and periauricular areas. Pain was more intensive in the nighttime. Temporomandibular joint movement (maximum unassisted and assisted opening, right to left movements) also increased pain. Significant dilatation of right foramen jugularis with its right side destruction was detected by computed tomography with contrast. The radiological findings were consistent with those of a jugular paraganglioma. Conclusion:This case confirms that in refractory headache and TMJ pain other reasons should be considered with rigorous investigation including neuroimaging.

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Systemic Inflammatory Syndrome Associated with a Case of Jugular Paraganglioma

Cited by 7 publications

References 17 publications

Pheochromocytoma presenting as fever of unknown origin, a case report

Pheochromocytoma presenting as fever of unknown origin, a case report

Retroperitoneal Castleman Disease Mimicking Paraganglioma in a Patient with Klinefelter Syndrome: A Case Report

A Case of Jugular Paraganglioma Presented as Temporomandibular Joint Disorder

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