Novel Antineuronal Autoantibodies With Somatodendritic Staining Pattern in a Patient With Autoimmune Psychosis

Endres, Dominique; Rauer, Sebastian; Pschibul, Alexander; Süß, Patrick; Venhoff, Nils; Runge, Kimon; Feige, Bernd; Denzel, Dominik; Nickel, Kathrin; Schweizer, Tina; Maier, Simon; Egger, Karl; Domschke, Katharina; Meyer, Philipp T.; Prüß, Harald; Elst, Ludger Tebartz van

doi:10.3389/fpsyt.2020.00627

Cited by 8 publications

(4 citation statements)

References 26 publications

(27 reference statements)

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“…In contrast, GABAR antibodies are more probably involved in the pathogenesis of catatonia, as they are antibodies against extracellular targets. In addition to GABAAR and NMDAR antibodies, novel neural autoantibodies (against an unknown epitope in granule cells in cerebellar and hippocampal interneurons) were identified in a patient suffering from psychosis and severe chronic catatonia ( 19 ). That patient responded to immunotherapy, but not to standard treatment with antipsychotics and benzodiazepines, supporting the relevance of taking CSF and blood samples to look for both known and novel autoantibodies in order to treat these patients adequately.…”

Section: Discussionmentioning

confidence: 99%

Catatonic Schizophrenia Associated With Cerebrospinal GAD65 Autoantibodies: Case Report and Literature Review

Hansen¹,

Bartels²,

Teegen

et al. 2022

Front. Immunol.

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BackgroundGAD65 autoimmunity is reported to be associated with schizophrenia and bipolar disorder. However, there has been no evidence that glutamic acid decarboxylase 65 (GAD65) autoantibodies in cerebrospinal fluid (CSF) are associated with akinetic catatonia in schizophrenia patients.MethodsWe report the case of a 28-year-old man who underwent diagnostics including brain MRI, neuropsychological testing, and electroencephalography (EEG) as well as a tumor search via CT of the abdomen and thorax, as well as colonoscopy and gastroscopy. For clinical characterization, his patient files were retrospectively examined.ResultsOur patient presented catatonia that responded somewhat to benzodiazepines in combination with previously taken antipsychotics such as risperidone for prediagnosed paranoid schizophrenia. Diagnostics revealed GAD65 autoantibodies in his serum and CSF. MRI revealed no brain lesion, and the tumor search had no malignancy. We diagnosed catatonic schizophrenia. Furthermore, as he had not fully recovered, he was given immunotherapy entailing two cycles of intravenous immunoglobulins. Subsequent neuropsychological testing due to subjective cognitive complaints after immunotherapy revealed no objective cognitive deficits.ConclusionsWe present the novel finding of an association between GAD65 autoantibodies in the serum and CSF with catatonia in a patient suffering from prediagnosed chronic schizophrenia. Due to the presence of CSF GAD65 antibodies and the catatonia factor in prediagnosed schizophrenia, we suspect that his catatonia has an autoimmune origin. Immunotherapy stabilized the catatonia that had initially responded to lorazepam treatment. Further research should be done to characterize patients’ responses to immunotherapy and standard treatment in a large cohort of patients with GAD65 antibody-associated catatonia and schizophrenia.

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Section: Discussionmentioning

confidence: 99%

Catatonic Schizophrenia Associated With Cerebrospinal GAD65 Autoantibodies: Case Report and Literature Review

Hansen¹,

Bartels²,

Teegen

et al. 2022

Front. Immunol.

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“…Controls had a mean age of 22 years (range ; the male:female ratio was 13:38. Controls where CSF was available had a mean age of 29 years (range [27][28][29][30][31][32][33]; the male:female ratio was 1:2.…”

Section: Controlsmentioning

confidence: 99%

“…Indirect support for antibody-mediated pathology may be provided by increased intrathecal immunoglobulin (Ig) G and oligoclonal bands (OCBs), or pathologic brain immunohistochemistry (IHC) findings 31 . A growing hypothesis is that the presence of red flags may predict response to immunotherapy, even when distinct MRI abnormalities and known autoantibodies in CSF or serum are absent 32,33 . This study provides longitudinal clinical data characterizing three severely ill, previously treatment-resistant cases with signs of autoimmune OCD where rituximab treatment was followed by clear gains regarding symptom burden and function.…”

Section: Introductionmentioning

confidence: 99%

Three cases with chronic obsessive compulsive disorder report gains in wellbeing and function following rituximab treatment

Cunningham,

Gallwitz,

Lindqvist

et al. 2024

Preprint

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Obsessive compulsive disorder (OCD) is a debilitating condition affecting about 2% of the population where half of patients do not respond to standard treatment. The emerging concept of an autoimmune OCD subgroup is grounded on findings that suggest immunological aetiology. We here present the longitudinal clinical course for three severely ill, functionally impaired complex cases with chronic treatment-resistant OCD. In all three cases, off-label treatment with rituximab was followed by clear and sustained gains regarding symptom burden and function over several years. The clinical phenotypes and clinical red flags are described in detail. Antibodies targeting mouse brain tissue were detected in cerebral spinal fluid for all three cases. We report exploratory analyses of immunological markers and proteomics and findings include elevated levels of sCD27 and markers indicative for complement pathway activation in CSF when compared to controls. Multidisciplinary collaboration, advanced clinical investigations and rituximab treatment are feasible in a psychiatric setting. The case histories provide a proof of principle for the newly proposed criteria for autoimmune OCD. The findings suggest that clinical red flags and biological measures may predict rituximab response in chronic treatment-resistant OCD. The report provides orientation that may inform the hypotheses and design of future treatment trials.

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“…The residual symptoms list included reduced muscle strength, recurring visual/acoustic hallucinations, dysarthria, retrograde amnesia, anterograde amnesia, mutism, seizures, hearing/speech/motor deficits, hyper/hyporeflexia, myoclonus, absent deep tendon reflexes (DTRs), varying oculomotor deficits (most commonly reporting limited abduction of R eye), and spastic paraparesis. Additional therapies that proved to be effective in treating patients diagnosed with AE conditions include alemtuzumab [9], azathioprine [10], bortezomib [11], cyclophosphamide [12][13][14][15][16], and rituximab [3,8,[17][18][19][20][21][22][23][24][25][26][27][28][29][30].…”

Section: Figure 2: Percentage Breakdown Of Successful Therapy With Di...mentioning

confidence: 99%

A Rare Presentation of a Complex Mixed Autoimmune Encephalitis Diagnosis: A Case Report and Literature Review

Khunkhun¹,

Aggarwal²,

Iqbal³

et al. 2022

Cureus

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This case report presents a unique case of a difficult differential diagnosis of autoimmune encephalitis (AE) in the setting of Mycoplasma pneumoniae. A 40-year-old female with a history of Hashimoto thyroiditis, polycystic ovarian syndrome, and a lower respiratory infection presented to the emergency department with new-onset progressive neurological symptoms. These included generalized tonic-clonic seizure and worsening respiratory status that required intubation and tracheostomy. Blood cultures returned positive for M. pneumoniae. We concluded this to be a mixed diagnosis case of anti-glutamic acid decarboxylase 65 (anti-GAD65), Bickerstaff's brainstem encephalitis (BBE), Hashimoto's encephalopathy (HE), and Miller Fisher Syndrome (MFS) concurrently in the setting of M. pneumoniae. Initial treatment with intravenous immunoglobulin showed minimal improvement; however, subsequent treatment with plasmapheresis proved to be beneficial for the patient. Over the course of the plasma exchange therapy (PLEX), the patient slowly became more alert, attentive, and verbal. She was able to answer simple questions and follow commands. Common trends of age, gender, presenting symptoms, associated antibodies, and sessions of PLEX in different AE diseases were identified through a literature review. Only 69.7% of the cases implemented PLEX or plasmapheresis. Currently, there is no standard protocol for the treatment of AE. Our case report aims to present a clinically complicated example of AE and to provide further evidence to support PLEX as an important therapeutic option.

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Novel Antineuronal Autoantibodies With Somatodendritic Staining Pattern in a Patient With Autoimmune Psychosis

Cited by 8 publications

References 26 publications

Catatonic Schizophrenia Associated With Cerebrospinal GAD65 Autoantibodies: Case Report and Literature Review

Catatonic Schizophrenia Associated With Cerebrospinal GAD65 Autoantibodies: Case Report and Literature Review

Three cases with chronic obsessive compulsive disorder report gains in wellbeing and function following rituximab treatment

A Rare Presentation of a Complex Mixed Autoimmune Encephalitis Diagnosis: A Case Report and Literature Review

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