A Case of Prolonged Catatonia Caused by Sjögren’s Syndrome

Abstract

Sjögren’s syndrome (SS) is a chronic autoimmune disorder, often associated with some neuropsychiatric symptoms as well as systemic lupus erythematosus. Although catatonia is frequently reported in patients with systemic lupus erythematosus, it has been rarely reported in patients with SS. Herein, we present a case of SS with catatonia effectively and safely treated with modified electroconvulsive therapy (ECT). A 58-year-old woman showed prolonged catatonia and depressive mood along with pathologically dried eye and mouth. Based on physical findings and blood tests, she was diagnosed with SS. Because of the presence of pressure sores, we were unable to perform lumbar puncture for the diagnosis of abacterial encephalitis. Alternatively, single-photon emission computed tomography of her brain revealed multifocal hypoperfused areas in the parietotemporal region. Consequently, we performed ECT for the treatment of catatonia comorbid with SS. Following 20 sessions of ECT, the catatonia was improved without obvious adverse effects. One week after the last ECT, elevated levels of interleukin-6 were identified in the cerebral fluid. After receiving steroid pulse therapy, she has not experienced catatonia for more than 5 years. SS can cause catatonia, and ECT is a safe and effective option for the treatment of catatonia with SS.

Evidence-based consensus guidelines for the management of catatonia: Recommendations from the British Association for Psychopharmacology

“…However, it should be noted that there is a strong association between anti-NMDA receptor encephalitis and catatonia, with most patients with this form of autoimmune encephalitis experiencing catatonia at some point in their illness ( Espinola-Nadurille et al, 2016 ; Rogers et al, 2019 ). Other autoantibodies have also been identified in association with catatonia including anti-Hu antibodies, anti-myelin oligodendrocyte glycoprotein antibodies, antinuclear antibodies (ANA), antiphospholipid antibodies, anti-ribosomal P antibodies, anti-Ro antibodies, anti-Smith antibodies, double-stranded DNA antibodies, GABA-A receptor antibodies, GAD-65 antibodies, leucine-rich glioma-inactivated 1 antibodies, ribonucleoprotein antibodies and septin-7 antibodies ( Boeke et al, 2018 ; Chuck et al, 2022 ; Endres et al, 2020 ; Ferrafiat et al, 2021 ; Fujimori et al, 2021 ; Harmon et al, 2022 ; Hinson et al, 2022 ; Inagaki et al, 2020 ; Kusztal et al, 2014 ; Pettingill et al, 2015 ; Samra et al, 2020 ; Witek et al, 2018 ). However, the prevalence and pathogenicity of these antibodies in catatonia is unclear, although it is a rapidly expanding field ( Rogers et al, 2019 ).…”

Exploring the Psychiatric Manifestations of Primary Sjögren’s Syndrome: A Narrative Review

“…These conditions include various psychiatric disorders [ 11 ]. The comorbid psychiatric disorders include cognitive dysfunction [ 12 ], depression [ 13 ], bipolar mood disorder [ 14 ], psychosis [ 15 , 16 ], catatonia [ 15 , 17 ], and obsessive-compulsive disorder [ 11 , 18 ]. Cognitive dysfunction, often called “brain fog,” is the most common psychiatric manifestation reported by pSS patients.…”