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04 November 2023 : Case report  Japan

[In Press] Hashimoto Encephalopathy Presenting with Acute Psychosis and Inappropriate Secretion of Antidiuretic Hormone: A Rare Case Responding to Steroid Therapy

Unusual clinical course, Challenging differential diagnosis

Masato Okitsu ORCID logo1ABDEF, Keizo Sugaya1AEF, Makoto Yoneda2B, Kazushi Takahashi1AE

Am J Case Rep In Press; DOI:   :: ID: 942297

Available online: 2023-11-04, In Press, Corrected Proof

Publication in the "In-Press" formula aims at speeding up the public availability of the pending manuscript while waiting for the final publication. The assigned DOI number is active and citable. The availability of the article in the Medline, PubMed and PMC databases as well as Web of Science will be obtained after the final publication according to the journal schedule


Hashimoto’s encephalopathy (HE) is an autoimmune encephalopathy that can involve various symptoms including psychosis. Syndrome of inappropriate secretion of antidiuretic hormone (SIADH) may be a complication in some neurological diseases. However, the simultaneous occurrence of subacute psychosis and SIADH as the manifestation of HE, observed in the present case, has rarely been reported.
A 72-year-old man was hospitalized with a 4-month history of abnormal behaviors, including talkativeness, stopping consumption of coffee and cigarettes, hoarding garbage, and sleep disorders. On physical examination, increased and incoherent speech with flight of idea and delusion were observed. The Mini-Mental State Examination score was 28/30. Laboratory findings included hyponatremia due to SIADH and a positive result for anti-thyroid and anti-NH2 terminal of alpha-enolase antibodies. Cerebrospinal fluid examination revealed only elevation of IL-6. Brain magnetic resonance imaging was unremarkable; however, (I-123)-iodoamphetamine single-photon emission computed tomography showed extensive hyperperfusion involving the brainstem and bilateral frontal and medial temporal lobes. Electroencephalography showed generalized slow waves, but there were no epileptiform discharges. After 2 courses of high-dose intravenous methylprednisolone followed by oral prednisolone, his symptoms improved. Based on the findings of clinical features and steroid responsiveness, he was diagnosed with HE. Oral prednisolone and antipsychotic drugs were decreased without a relapse and he was discharged to his home.
Although psychosis complicating SIADH is rare, HE should be considered in the differential diagnosis because of its treatment efficacy.

Keywords: Hyponatremia; Psychotic Disorders; Sleep Disorders, Intrinsic; Autoimmune Limbic Encephalitis; Hashimoto’s Encephalitis

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American Journal of Case Reports eISSN: 1941-5923
American Journal of Case Reports eISSN: 1941-5923