SDHB-negative jugular foramen paraganglioma manifesting malignant progression with pseudo-hypoxia related atypical uptake of [18F]-FDG: A case report.
World Neurosurg. 2018 Mar 03;:
Authors: Ohtake M, Tateishi K, Murata H, Nagashima Y, Yamanaka S, Yamamoto T
Abstract
BACKGROUND: Paragangliomas are generally benign, slow-growing tumors. However, approximately 10-20% are malignant, characterized by distant metastasis. Recently, a germline mutation in succinate dehydrogenase B subunit (SDHB) has been shown to be associated with malignant behavior in paraganglioma. Herein, we present a case of SDHB-negative malignant paraganglioma of the jugular foramen with pseudo-hypoxic microenvironment and unique imaging features on [18F]-fluoro-2-deoxy-d-glucose positron emission tomography ([18F]-FDG PET), and discuss the significance of SDHB immunohistochemistry and the potential of [18F]-FDG PET for clinical management.
CASE DESCRIPTION: A 55-year-old woman was diagnosed with jugular foramen paraganglioma. Initial surgical resection was performed; however, follow-up [18F]-FDG PET indicated multiple uptake regions throughout the body. Biopsies for multiple recurrent lesions revealed consistent pathological features, suggesting distant metastasis. Immuno-histochemical analysis revealed lack of SDHB immunostaining in all specimens. Pseudo-hypoxic markers, including hypoxia-inducible factor-1α and downstream glycolysis enzymes, were strongly expressed. [18F]-FDG PET demonstrated increased uptake in the lesions, and the patient died 3 years after initial metastasis.
CONCLUSION: In patients with head and neck paraganglioma without SDHB expression, close follow-up should be considered because of the risk for metastasis. In such cases, [18F]-FDG PET might be useful to detect metastasis due to atypical accumulation from pseudo-hypoxia induced glycolysis.
PMID: 29510284 [PubMed - as supplied by publisher]
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