Endocrine Journal
Online ISSN : 1348-4540
Print ISSN : 0918-8959
ISSN-L : 0918-8959
Syndrome of inappropriate secretion of thyrotropin associated with thymoma-related peripheral nerve hyperexcitability
Kenji OhbaKentaro ShirakawaYuta OkawaHiroyuki IwakiHideyuki MatsunagaShingo SuzukiAkio MatsushitaHiroshi MoritaShigekazu SasakiYutaka OkiHirotoshi Nakamura
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2011 Volume 58 Issue 7 Pages 597-602


Syndrome of inappropriate secretion of thyrotropin (SITSH) is a clinical state of inappropriately elevated secretion of thyrotropin (TSH) in the presence of elevated free thyroid hormones. Peripheral nerve hyperexcitability (PNH) is a rare disorder characterized by muscle twitching at rest. No relation between them is known. A 49-year-old man was referred to our hospital because of elevated serum free thyroxine (2.6 ng/dL; normal range, 0.9-1.7) and normal TSH (2.7 mIU/L; normal range, 0.5-5.0). Genetic analysis revealed no mutations of the thyroid hormone receptor β gene. Magnetic resonance imaging visualized no pituitary adenoma. He complained of appetite loss, weight loss, myokymia, paraesthesia, hyperhydrosis and insomnia. Chest X ray and computed tomography (CT) scan showed a mediastinal tumor diagnosed as a thymoma by CT-guided biopsy. Electromyography disclosed fasciculations and myokymic discharges. Nerve conduction studies showed prolonged after-discharges following evoked compound muscle action potential. The patient was diagnosed with thymoma-associated PNH based on neurological manifestations and neurophysiological findings, and was treated with pulse therapy with methylprednisolone after thymectomy. Interestingly, the SITSH state became less prominent as his neurological manifestations improved. This is the first case of SITSH possibly caused by thymoma-associated PNH.

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