Catatonia and Psychosis as Manifestations of Primary Sjögren’s Syndrome

Rosado, Steeve Neves; Silveira, Vera Maria Freitas da; Reis, Ana Isabel; Gordinho, André; Noronha, Carla

doi:10.12890/2018_000855

Cited by 9 publications

(14 citation statements)

References 5 publications

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“…SPECT assessment should be considered, as it revealed a hypoperfusion in parietal, temporal, and frontal lobes in more than 50% of patients having normal MRI brain when cognitive dysfunction was present. This SPECT pattern was present only in 17% of pSS patients without cognitive dysfunction [20,21,22,23,24,25,26,27,28,29,30,31,32,33,34,35,36,37,38,39,53].…”

Section: Discussionmentioning

confidence: 99%

Cognitive Function in Primary Sjögren’s Syndrome: A Systematic Review

et al. 2019

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Background: Cognitive disorders are reported to be common in patients with primary Sjogren’s syndrome (pSS). In some cases, they are the first clinical manifestation, preceding the diagnosis of pSS by two years on average. Aim: A systematic review was conducted to explore cognitive impairment in pSS, with reference to diagnostic methods and their relationship with laboratory data and clinical manifestations. Materials and Methods: According to the PRISMA 2009 checklist, we carried out a comprehensive literature search in the three main bibliographic databases: MEDLINE, EMBASE, and PsycINFO (NICE HDAS interface). The following main search terms were used: primary Sjogren syndrome, neurological manifestations, fatigue, cognitive functions, psychiatric manifestations, mild cognitive impairment, dementia, and neurocognitive disorder. The search was made on 14 September, 2018. References from all selected studies were also examined. Inclusion criteria were: all studies and case-reports published in any language from 2002 that assessed the association of pSS (according to classification criteria proposed by the 2002 American/European collaborative group (AECG)) with all types of cognitive impairment (including dementia). Exclusion criteria were: reviews, abstracts, secondary Sjögren’s syndrome (SS), and all articles in which other classification criteria were used. Results: The initial search yielded 352 articles, of which 253 were excluded based on the title and abstract review. A total of 54 articles underwent a full-length review, and 32 articles were excluded. Data were extracted from 18 studies and three case-reports involving a total of 6196 participants. In most cases, cognitive dysfunction was a brain fog or a mild cognitive impairment (MCI). Occasionally, an autoimmune dementia was present. The relationship between pSS and degenerative dementias, such as Alzheimer’s disease (AD), was a controversial issue, even if some investigators hypothesized that pSS could be a risk factor. Several unmet needs were highlighted. First, some of the included studies had not reported the severity of pSS; hence, few correlations between disease severity and cognitive function were possible. Secondly, the evaluation of the pathogenetic role of comorbid diseases was often absent. The lack of information on the type of dementia represented a third critical point in the majority of the included studies. Conclusions: This systematic review confirmed that adequate studies on cognitive function in pSS are scarce, mostly performed on small-sized samples, and often conflicting. The routine assessment of cognitive function in patients with pSS seems advisable and it will help to elucidate some of the unmet needs highlighted by this review in future appropriately designed studies.

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Section: Discussionmentioning

confidence: 99%

Cognitive Function in Primary Sjögren’s Syndrome: A Systematic Review

et al. 2019

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“…Psychiatric abnormalities have also been described including depression, anxiety, and cognitive deficits [3]. Psychosis has been reported in adult patients [3,[7][8][9][10][11][12] with pSS but has never been previously reported in the pediatric literature, although other psychiatric manifestations including major depressive disorder and obsessive compulsive disorder (OCD) have been described in pediatric patients [13].…”

Section: Introductionmentioning

confidence: 99%

Adolescent Sjogren’s syndrome presenting as psychosis: a case series

et al. 2020

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Background: Neurological involvement has been reported in up to 80% of adults with Primary Sjogren's syndrome (pSS) with psychiatric abnormalities including anxiety, depression, and cognitive dysfunction being common. Psychosis due to pSS has been reported in adult patients but has never been previously reported in the adolescent/pediatric literature. Here we describe for the first time four cases of adolescent Sjogren's syndrome that presented with psychotic symptoms. Rituximab treatment was followed by improvement of psychiatric symptoms in all patients. Case presentation: 1: 16 year old female without significant past medical history presented to the emergency department with 4 days of abnormal behavior, tremors, insomnia, polyphagia, polyuria, and suicidal ideation. 2: 16 year old female with a 4 year history of severe anxiety, OCD, and tic disorder treated with fluoxetine with partial benefit presented with an abrupt and severe worsening of anxiety, OCD and new auditory hallucinations. 3: 19 year old female without significant past medical history presented with a 3 day history of progressively altered behavior, incoherent speech, insomnia, headache, and tangential thoughts. 4: 17 year old female without significant past medical history presented with new onset suicidal ideation, paranoia, confusion, and emotional lability. Conclusion: Psychosis is more common in autoimmune disease than previously known. To our knowledge, the four teenage women described above are the first reported patients with adolescent pSS manifesting as psychosis. pSS should be considered in the differential diagnosis of young patients with new psychiatric disorders, even in the absence of sicca symptoms. Psychiatric symptoms improved with rituximab infusions in all 4 of our patients, which suggests rituximab may be an effective treatment option that should be considered early after the diagnosis of pSS-associated psychiatric disturbance.

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“…The therapeutic course suggested that catatonia with depressive episodes was a consequence of SS pathology. Although psychiatric symptoms have been commonly reported in SS [ 3 ], to the best of our knowledge, only one case of catatonia comorbid with SS has been previously reported [ 9 ].…”

Section: Discussionmentioning

confidence: 99%

A Case of Prolonged Catatonia Caused by Sjögren’s Syndrome

Inagaki

Kudo²,

Kurimoto

et al. 2020

Case Reports in Immunology

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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.

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Catatonia and Psychosis as Manifestations of Primary Sjögren’s Syndrome

Cited by 9 publications

References 5 publications

Cognitive Function in Primary Sjögren’s Syndrome: A Systematic Review

Cognitive Function in Primary Sjögren’s Syndrome: A Systematic Review

Adolescent Sjogren’s syndrome presenting as psychosis: a case series

A Case of Prolonged Catatonia Caused by Sjögren’s Syndrome

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