Multiple atypical thymic carcinoids with paraneoplastic giant cell arteritis
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CASE REPORT
Multiple atypical thymic carcinoids with paraneoplastic giant cell arteritis Takashi Nakayama1 · Sayaka Katagiri1 · Takuma Kikkawa1 · Tetsuya Obara1 · Tatsuo Mori2 · Takahiro Kiriu3 Received: 4 July 2019 / Accepted: 10 October 2019 © The Japanese Association for Thoracic Surgery 2019
Abstract Multiple thymic carcinoids are rare, and giant cell arteritis (GCA) is one of the less recognized paraneoplastic diseases. The co-occurrence of these two diseases is therefore extremely rare. We report herein a patient with multiple atypical thymic carcinoids and asymptomatic paraneoplastic GCA. All the thymic carcinoids were diagnosed histopathologically as atypical thymic carcinoids with an intrathymic metastasis. Treatment consisted of a complete tumor resection followed by observation of the GCA without any adjuvant therapy. Subsequent positron emission tomography revealed a decrease in F-fludeoxyglucose accumulation in the systemic arteries. Based on these findings, paraneoplastic GCA was diagnosed. Thymic carcinoids rarely involve intrathymic metastasis or cause neopleonastic GCA. However, when they do, a complete tumor resection is the best option for management. Keywords Thymic carcinoid · Intrathymic metastasis · Paraneoplastic · Giant cell arteritis
Introduction
Case report
Multiple thymic carcinoids are rare, and it is sometimes difficult to determine if multiple tumors are metastatic or multicentric. Carcinoids are intimately associated with paraneoplastic disorders, especially cases of multiple endocrine neoplasia type 1 (MEN-1). Because paraneoplastic disorders mainly have the appearance of a hormonal disorder, giant cell arteritis (GCA) is often not recognized as a paraneoplastic disease associated with thymic carcinoids [1]. We report herein a patient with non-MEN-1, multiple atypical thymic carcinoids, and asymptomatic paraneoplastic GCA.
An 80-year-old, asymptomatic female patient underwent a chest X-ray during a routine health examination. She had no past medical history or family history of hereditary disease. An abnormal mass was detected on chest X-ray, and computed tomography (CT) revealed three tumors in the anterior mediastinum (Fig. 1a–c). Laboratory tests revealed only a moderately elevated erythrocyte sedimentation rate (ESR) [ESR:52mm/h (GCA’ criteria value > 50mm/h)], while C-reactive protein, collagen disease-related autoantibodies, and tumor markers were negative. A CT-guided needle biopsy of the largest tumor located in the lower right pole of the thymus was performed, and atypical thymic carcinoid was pathologically diagnosed. Positron emission tomography (PET)-CT visualized an abnormal clustering of three tumors and luminal abnormalities in the systemic arteries from the temporal to the femoral artery (Fig. 1d). Although the patient had no tenderness or engorgement of the temporal arteries, a biopsy of bilateral temporal artery was done, and GCA was pathologically diagnosed (Fig. 2). After confirming that there was no risk of vision loss or cerebrovascular dysfunction, a
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