Evaluation of Catatonia with Different Assessment Scales in Psychiatry and Neurology. / Psikiyatri ve Nörolojide Katatoninin Farkli Ölçeklerle Degerlendirilmesi.

OBJECTIVE: Catatonia is a syndrome that can be missed in clinical settings. Diagnosis of catatonia is important because the condition can be reversible and is associated with severe complications. This study aims to screen patients with catatonia admitted to a university hospital's psychiatry and neurology services, examine their characteristics, and compare the coverage of different catatonia scales. METHOD: During a consecutive 20 months study period, the Turkish adaptations of the Bush-Francis Catatonia Rating and the KANNER scales were administered in psychiatry and neurology inpatient units and patients on the waiting list for psychiatric hospitalization. The participants were also evaluated with DSM-5 criteria. In addition, the sociodemographic and clinical characteristics of the patients in the psychiatric group were compared. RESULTS: A total of 214 patients were evaluated. Twenty-eight (13.1%) screened positive for catatonia, and 23 (82.1%) were diagnosed with catatonia according to DSM-5 criteria. KANNER and Bush- Francis identified the same patients as having catatonia. In addition to schizophrenia and mood disorders; neurodevelopmental disorder, encephalitis, postpartum psychosis, obsessive-compulsive disorder, delirium, cerebrovascular disease, functional neurological symptom disorder have also been found to be associated with catatonia. The most common complication was urinary tract infection. Life-threatening complications were also observed. CONCLUSION: Overlooking catatonia may have dire consequences. Adhering solely to the DSM-5 criteria may miss some patients with catatonia. Widely and efficiently using standardized catatonia scales can improve detection capacity and enhance the management of morbidity and mortality.

JAK inhibition in Down Syndrome Regression Disorder.

Down Syndrome Regression Disorder (DRSD) is an uncommon but devastating condition affecting primarily adolescents and young adults with Down syndrome (DS). Individuals with DS display a dysregulated immune system associated with hyperactive interferon signaling, which is associated with a high incidence of autoimmune conditions. While the cause of DSRD is unknown, increasing evidence indicates that it may have an immune basis, and some individuals with DSRD have responded to intravenous immunoglobulin therapy. This case series describes three individuals with probable DSRD who received the JAK inhibitor tofacitinib and saw improvement in DSRD symptoms across multiple domains of neurological function.

Catatonia related to tacrolimus: a real world pharmacovigilance study of FDA adverse event reporting system (FAERS) database.

BACKGROUND: Emerging case reports have highlighted that catatonia may be a complication that is easily misdiagnosed. Our study aimed to summarize the clinical characteristics of patients with tacrolimus-induced catatonia and assess the association between tacrolimus and catatonia through disproportionality analysis. RESEARCH DESIGN AND METHODS: We conducted a retrospective pharmacovigilance study using the FAERS database, analyzing data up to the third quarter of 2023. The clinical characteristics of the reported cases were summarized using descriptive statistics. Information component (IC) and reporting odds ratio (ROR) were used to evaluate the association between tacrolimus and catatonia. RESULTS: There were 66 reports of tacrolimus-related catatonia, with the majority of cases occurring in the United States (78.79%). The risk signal for tacrolimus-related catatonia was significantly higher compared to all other drugs (ROR 3.222 [2.524, 4.111], IC 1.632 [1.273, 1.991]). A significant association was detected in both male and female, while the risk signal of tacrolimus-associated catatonia was only found in the subgroups aged over 40 years. CONCLUSIONS: Our study identified a safety signal for the association between tacrolimus and catatonia compared to all other drugs in FAERS database, particularly in patients aged 40 and above.

A Rare Case of Arnold Chiari Malformation Type 1 Presenting With Features of Catatonia.

The Arnold Chiari malformation is a congenital neurological condition. It occurs due to a defect in the cerebellum. Our patient is a 19-year-old postpartum female who presented to our ER with headaches, crying spells, reduced interaction, poor self-care, and neglect of her newborn for the past five days. Before the onset of her presenting symptoms, the patient had complained of a severe headache in the back of the head and dizziness. Her baseline investigations were normal. On examination, the patient was noticed to have a fixed gaze, ambiguity, mutism, and rigidity. So, she was diagnosed with catatonia, a differential diagnosis of mental and behavioral disorders associated with pregnancy, childbirth, and puerperium, and was treated with medications appropriately. As her headache showed minimal relief with adequate analgesic measures, neuroimaging was done, which showed Arnold Chiari malformation type I with hydrocephalus. A liaison was made with the neurology team, who confirmed the diagnosis and advised her on the decompression procedure. Her family refused to undergo the procedure. Once she showed minimal improvement in her symptoms, her family members requested her discharge. In our case, the Arnold Chiari malformation type I presented with features of catatonia, unlike the usual reported presentation of depression and anxiety. This case highlights the need for a thorough evaluation of any patient presenting with catatonia.

Diagnostic and demographic factors of pediatric and adult catatonia hospitalizations: A 2016-2020 National Inpatient Sample Study.

OBJECTIVE: Catatonia is a neuropsychiatric disorder that can occur in patients of any age, but it is uncertain whether patient demographics or underlying diagnoses differ between pediatric and adult patients. This study investigates patients of all ages diagnosed with catatonia during acute care hospitalizations in the United States over a 5-year period. METHOD: The National Inpatient Sample, an all-payors database of acute care hospital discharges, was queried for patients with a discharge diagnosis of catatonia between 2016 and 2020 with patients stratified by age as pediatric (≤18 years) or adult (>18 years). RESULTS: Among 174,776,205 hospitalizations recorded in the NIS from 2016 to 2020, 61,990 (95% CI: 60,257 to 63,723; 0.035%) involved a diagnosis of catatonia. Of these, 3255 were for pediatric patients and 58,735 were for adult patients. Compared with adult patients, pediatric catatonia patients were more likely to be male and non-White. Diagnostically, psychotic disorders, encephalitis, and neurodevelopmental disorders were more common primary discharge diagnoses in pediatric patients, while adult patients more frequently were diagnosed with mood disorders. Length of stay was not significantly different between pediatric and adult catatonia hospitalizations. Physical restraints were commonly applied for patients with catatonia. CONCLUSION: Pediatric and adult catatonia patients differed in sex, race, and diagnosis, although hospital length of stay was not different between pediatric and adult catatonia hospitalizations. These results may inform catatonia diagnosis in the hospital setting and point to disparities that could be targets of quality improvement efforts.

Catatonia in Dementia: A Systematic Review of Case Reports and Case Series.

BACKGROUND: Catatonia is a neuropsychiatric syndrome characterized by motor, behavioral, and autonomic abnormalities. It is often underdiagnosed in geriatric patients with dementia despite established diagnostic criteria and treatment options. OBJECTIVE: This systematic review investigates catatonia in the elderly, particularly those with dementia, to examine their clinical presentation, treatment response, and prognosis compared to elderly patients without dementia. METHODS: We comprehensively searched MEDLINE and EMBASE, including case reports and series on catatonia in elderly patients. Reviewers independently performed data extraction and quality assessments. Statistical significance was set at a p value ≤0.05, and a multivariate logistic regression model was used to analyze differences between patients with and without dementia. RESULTS: Our review included 182 articles with 225 cases. We found no significant differences in the clinical presentation of catatonia between patients with and without dementia, with both groups commonly exhibiting the hypokinetic variant. However, patients with dementia were more frequently treated with NMDA receptor antagonists (OR: 3.27; CI: 1.05-10.11; p = 0.040) and had a lower complete response rate to treatment (OR: 0.37; CI: 0.19-0.75; p = 0.006). Patients with dementia also exhibited fewer acute medical conditions (OR: 0.17; CI: 0.05-0.65; p = 0.009). CONCLUSIONS: Catatonia in dementia does not have a different syndromic presentation. However, the diagnosis of dementia leads to varying preferences regarding the choice of symptomatic therapy and seems to be a predictor of a poorer therapeutic response. Actively treating catatonia, particularly in patients with dementia, addressing the characteristics of these patients is of paramount importance.

Catatonia and epilepsy: An underappreciated relationship.

Catatonia is currently conceived in the major diagnostic manuals as a syndrome with a range of possible psychiatric and general medical underlying conditions. It features diverse clinical signs, spanning motor, verbal and behavioural domains and including stupor, catalepsy, mutism, echolalia, negativism and withdrawal. The existing literature suggests that seizure activity may underlie catatonia in approximately 2% of cases. There are three possible temporal relationships between catatonia and seizure activity: (1) ictal catatonia, in which catatonia is a presentation of non-convulsive status epilepticus; (2) postictal catatonia, in which catatonia follows a seizure, and (3) interictal catatonia, in which catatonia and seizures occur in the same individual without any clear temporal relationship between them. Electroencephalographic (EEG) abnormalities are common in catatonia, even in those cases with a presumed primary psychiatric origin, and often consist of generalised background slowing. Paradoxically, electroconvulsive therapy is an effective treatment for catatonia. There are several converging pieces of evidence suggesting that there may be underlying seizure activity in more cases of catatonia than has hitherto been recognised, though identification of these seizures may require intracranial EEG recording.

Major Depressive Disorder with Catatonia: A Phenotype Related to Autistic Traits and High Suicidality.

Background: Major Depressive Disorder (MDD) represents a significant global health concern, often complicated by comorbidities such as catatonia and autism spectrum disorder (ASD). Recognizing the interplay among these conditions and their impact on suicidal tendencies is crucial for effective clinical management. Methods: A total sample of 147 subjects with MDD was divided into Significant Catatonia (SC) and Non-Significant Catatonia (NSC) groups based on Catatonia Spectrum (CS) scores. Participants were evaluated through the Structured Clinical Interview for DSM-5, Research Version (SCID-5-RV), the Adult Autism Subtreshold Spectrum (AdAS Spectrum), and the Mood Spectrum-Self Report questionnaires. Statistical analyses included Mann-Whitney U test, Chi-square test, logistic regression analyses, and a decision tree model. Results: The SC group exhibited higher CS, AdAS Spectrum, and MOODS-SR total and domain scores compared to the NSC group. Individuals with significant autistic traits were over-represented in the SC group, as well as participants with higher suicidality, suicidal ideation, and a history of suicide attempts. The total AdAS Spectrum and MOOD-SR score, the AdAS domain "Hyper-hypo reactivity to sensory input", and the "Cognitive depressive" MOOD-SR domain were predictive of belonging to the SC group. Suicidality levels appeared to be higher in clinically significant ASD, intermediate in subjects with autistic traits (AT), and low in the absence of AT. Conclusions: the study suggests the existence of a specific phenotype of MDD associated with catatonia, characterized by elevated autistic traits and suicide risk.

Case Report of Retarded Catatonia: Always Consider Catatonia as a Differential Diagnosis of Altered Mental Status.

BACKGROUND: Catatonia is a rare neuropsychiatric condition; it is estimated that around 10% of patients with mood disorders present signs and symptoms of catatonia. A catatonic syndrome is characterised by mutism, negativism, rigidity, and stupor. CASE REPORT: We report the case of a 59-year-old patient with a medical history of bipolar disorder who was admitted to the internal medicine service due to a seizure episode. During hospitalisation, the patient presented significant worsening of her clinical condition, showing marked symptoms of stupor and catatonia. Once the neurological and metabolic etiologies of altered mental status had been ruled out, pharmacological treatment with high doses of lorazepam was started. The patient had a complete clinical remission, and her evolution was favourable without any complications. Electroconvulsive therapy was recommended as a definitive treatment. CONCLUSIONS: The diagnosis of catatonia is a challenge for both hospitalists and psychiatrists due to the clinical presentation of catatonia. In reporting this clinical case, we want to emphasise the importance of taking into account the catatonic syndrome in our differential diagnoses in patients with altered mental status.

Impacts of exposure to and subsequent discontinuation of clozapine on tripartite synaptic transmission.

BACKGROUND AND PURPOSE: Clozapine is an effective antipsychotic for treatment-resistant schizophrenia, but its discontinuation leads to discontinuation syndrome/catatonia complicated by benzodiazepine-resistance and rhabdomyolysis. EXPERIMENTAL APPROACH: This study determined time-dependent effects of exposure and subsequent discontinuation of clozapine on expression of connexin43, 5-HT receptors, intracellular L-ß-aminoisobutyrate (L-BAIBA) and 2nd-messengers and signalling of AMPK, PP2A and Akt in cultured astrocytes and rat frontal cortex. KEY RESULTS: Intracellular L-BAIBA levels increased during clozapine exposure but immediately recovered after discontinuation. Both exposure to clozapine and L-BAIBA increased connexin43 and signalling of AMPK/Akt time-dependently, but reduced PP2A signalling, 5-HT receptor expression and IP3 level. These changes recovered within 2 weeks after discontinuation, while 5-HT receptors and IP3 transiently increased during the recovery process. L-BAIBA activated AMPK signalling, leading to attenuated PP2A signalling. Astroglial D-serine release was increased by clozapine exposure but continued to increase within 1 week after discontinuation via activation of IP3 receptor function. CONCLUSION AND IMPLICATIONS: Clozapine discontinuation restored PP2A signalling due to decreased L-BAIBA, increased 5-HT receptor expression via probably enhanced 5-HT receptor recycling, but increased astroglial D-serine release persisted by transiently activated IP3 receptors via transiently increased IP3 level. Decreased L-BAIBA caused by clozapine discontinuation is, at least partially, involved in the transiently increased 5-HT receptor and astroglial D-serine release.

Ketamine for catatonia: A novel treatment for an old clinical challenge? A systematic review of the evidence.

INTRODUCTION: Catatonia, documented since the 19th century, remains a significant challenge in terms of recognition and treatment. Over the last two decades, ketamine has brought new perspectives to psychiatry, sparking widespread interest. Concurrently, catatonia has attracted heightened scientific attention. Preliminary evidence suggests the therapeutic potential of ketamine for catatonia. METHODS: We systematically searched Medline/PubMed, Embase, PsycINFO, Lilacs, and Cochrane Library databases, as well as Google Scholar, for studies with ketamine or its enantiomers as intervention for catatonia, with no restrictions to underlying diagnosis, date, language, or study design. RESULTS: Twenty articles were included, encompassing a total of 25 catatonic patients receiving ketamine or esketamine. Predominantly female (61.9 %), with a mean age of 44.4 years, patients mostly exhibited manifestations compatible with the retarded subtype of catatonia. Mood disorders were the most prevalent underlying diagnoses. Ketamine was primarily administered intravenously over a 40-minute period and in multiple-dosing schemes. Mean response and remission rates of catatonic manifestations for the whole sample were 80 % and 44 %, respectively, with no reports of worsening catatonic features or psychotic symptoms. Only one patient discontinued treatment due to intolerable dissociative effects. CONCLUSION: Challenging the conventional contraindication of ketamine in psychotic disorders, current evidence highlights its potential efficacy, particularly in treating catatonia. Pending further research, we advocate reevaluating this contraindication, as it may offer a promising therapeutic option, especially for challenging cases. Preliminary evidence suggests potentially greater benefits for catatonic patients with underlying mood disorders compared to primary psychotic disorders.

The effectiveness of the lorazepam challenge test in pediatric catatonia: A multisite retrospective cohort study.

OBJECTIVE: Catatonia is a neuropsychiatric disorder associated with changes in behavior and affect. In adults, catatonia can respond rapidly to treatment with benzodiazepines as part of the "lorazepam challenge test." The acute effectiveness of benzodiazepine treatment in pediatric catatonia, however, has received less study. This study reports catatonia severity as measured by the Bush Francis Catatonia Rating Scale (BFCRS) in pediatric patients before and after treatment with lorazepam. METHODS: Multicenter retrospective cohort study from 1/1/2018 to 6/1/2023 of patients aged 18 and younger with a clinical diagnosis of catatonia and assessment using the BFCRS before and after treatment with lorazepam. RESULTS: Among 54 patients, median age was 16, and 26 (48.1 %) were female. Neurodevelopmental disabilities were present in 24 (44.4 %) of patients. Prior to treatment, patients had a mean BFCRS score of 16.6 ± 6.1, which significantly reduced to 9.5 ± 5.3 following treatment with lorazepam (mean paired difference 7.1; t = 9.0, df = 53, p < 0.001), representing a large effect size (Hedges's g = 1.20; 95 % CI: 0.85 to 1.55). No significant association was found between lorazepam dose or route of administration and clinical response, nor were age, sex, study site, the presence of a neurodevelopmental disorder, the presence of hyperactive catatonic features, or the time between treatment and reassessment associated with post-treatment BFCRS. CONCLUSIONS: Lorazepam resulted in a rapid improvement in BFCRS score in pediatric patients, with a large effect size. Further research is needed into optimal dosing and route of administration of the lorazepam challenge test in pediatric patients.

When Consumption Leads to Catastrophe: A Case of Recurrent Alcohol Withdrawal Catatonia in an Adult Patient.

Catatonia is a motor dysregulation syndrome and a multifaceted neuropsychiatric behavioral syndrome distinguished by abnormal movements, immobility, abnormal behavior, and withdrawal, where patients are unable to move normally despite full physical capacity. Catatonia, in the background of alcohol withdrawal, is a fairly rare phenomenon. Therefore, we are reporting a case where the patient has a history of binge alcohol consumption, with catatonia reoccurring with his withdrawal symptoms, and no other illicit drug use in his history. Its rarity, complex presentation, and potential diagnostic pitfalls necessitate heightened awareness among healthcare professionals.

Importance of Early Diagnosis and Treatment of Perioperative Catatonia: A Case Report.

Symptoms of catatonia include silence, motionlessness, and postural retention. Although it is important to detect and treat catatonia early, before it becomes severe, postoperative cases have inherent risks that hinder diagnosis and treatment. A 60-year-old man with schizophrenia underwent endoscopic/thoracoscopic esophagectomy and was extubated in the operating room. In the intensive care unit (ICU), he had stiffness in the neck, ankles, and knees, catalepsy during passive knee flexion, mild disturbance of consciousness, mild creatine kinase elevation, and respiratory depression. Intravenous diazepam was administered for diagnosis, and the patient's rapid improvement indicated catatonia. He was intubated and started on lorazepam; tapering produced no recurrence of symptoms. The patient was extubated and transferred to the general ward on postoperative Day 2. Because this patient was extubated in the operating room and was managed postoperatively in the ICU with a full-time doctor, his symptoms were easily recognized and early diagnosis was possible. Thus, we were able to administer drug therapy quickly and adequately and perform forward management that accounted for postoperative risks, thereby achieving a favorable outcome.

Catatonia responsive to corticosteroids in a patient with an SCN2A variant.

Variants in SCN2A are a known risk factor for developing autism spectrum disorder (ASD). Catatonia is a complex neuropsychiatric syndrome, which occurs at a higher rate in individuals with ASD. Catatonia has also been associated with COVID-19 infection, though the majority of these cases are associated with increased serum inflammatory markers. We present a case of a 15-year-old female with ASD and corticosteroid responsive stuporous catatonia to explore the relationship between SCN2A variants, ASD, COVID-19 exposure, and treatment refractory catatonia. Despite a lack of significantly elevated serum or CSF inflammatory markers, this patient showed significant improvement following initiation of corticosteroid therapy. This case presents a novel approach to the work-up and treatment of catatonia in individuals with SCN2A variants independent of elevated inflammatory markers.

Expanding the phenotype of NEDAMSS with a psychiatric perspective: analysis of a new case, and a systematic review of the literature.

Pathogenic variants in the IRF2BPL gene are associated with neurodevelopmental disorders with varying degrees of regression, loss of speech and epilepsy. The phenotype is also known as Neurodevelopmental Disorder with regression, Abnormal Movements, loss of Speech, and Seizures (NEDAMSS). The motor symptoms of this disorder share significant phenotypical characteristics with catatonia, a severe neuropsychiatric psychomotor syndrome. The objective of this article is to expand the knowledge on the presentation of NEDAMSS with a focus on psychiatric symptoms including catatonia. A systematic review of 32 case presentations of NEDAMSS, and a novel case report of a patient with NEDAMSS, exhibiting multiple psychiatric symptoms, including catatonia are presented. Psychiatric symptoms and disorders including affective disorders, psychotic symptoms, catatonia, and developmental disorders are reported in one third of the reviewed cases. Reported effects of pharmacological treatment on motor symptoms of NEDAMSS are very limited. Our case presents improvement in motor symptoms originally attributed to NEDAMSS, after treatment with Lorazepam following diagnosis with catatonia. Patients with NEDAMSS may present with both neurological and psychiatric symptoms. The clinical presentation of NEDAMSS motor symptoms and catatonia have similarities and thus poses significant challenges to the diagnostic process, with risk of incorrect or delayed treatment. The limited experience and the complex phenotype of NEDAMSS complicates pharmacological treatment and encourages caution, especially with the use of antipsychotic drugs in the presence of possible catatonic symptoms.

Alteration of Functional Connectivity before and after Electroconvulsive Therapy in a Single Patient with Depression and Catatonia.

Catatonia, a severe neuropsychiatric condition, is distinguished by a range of prominent motor features such as immobility, mutism, negativism, rigidity, posturing, staring, stereotypy, automatic obedience, echolalia, and mannerism. A female patient of middle age was admitted to the open psychiatric ward of a hospital after exhibiting suicidal ideation, delusions, depression, insomnia, refusal to eat, difficulty in swallowing, and decreased motivation for four months prior to admission. Following 14 electroconvulsive therapy (ECT) sessions, her symptoms improved in the order of appetite, immobility, speech volume, mood, and delusions. The post-ECT functional connectivity was found to be improved compared to pre-ECT. The patient was discharged to outpatient clinics with medications that included aripiprazole, mirtazapine, quetiapine, and trazodone. This case reveals that ECT is an efficacious treatment in a depressive patient with catatonia, with movement symptoms responding to ECT more rapidly than affective symptoms. In addition to the improvement of movement and affective symptoms, functional connectivity much improved after ECT.

A network analysis of catatonia symptoms across diagnoses.

BACKGROUND: Catatonia, as a transdiagnostic construct, manifests across various psychiatric and non-psychiatric conditions. Understanding how symptom variations impact the catatonia construct and differ across primary diagnoses (schizophrenia, bipolar disorder, unipolar depression, and neurological/metabolic/immunological condition) is essential to refine diagnostic and therapeutic approaches. This study aims to compare the symptom networks and centrality measures of these diagnoses. METHODS: We conducted a network analysis using Bush-Francis Catatonia Rating Scale (BFCRS) data from 118 patients, examining centrality measures and network comparisons across the four primary diagnostic groups. RESULTS: In the general catatonia network, the three most central symptoms identified were Excitement (1.462), Perseveration (1.456), and Impulsivity (1.332). While the overall structure of the catatonia networks did not show significant differences between diagnoses in terms of symptom connections and centrality, variations in centrality measures were observed among the different networks. CONCLUSIONS: The study reinforces the notion of catatonia as an independent syndrome relatively to psychiatric or non-psychiatric diagnoses. However, the variation in centrality of symptoms across different primary diagnoses provides critical insights that could aid clinicians in tailoring diagnostic and therapeutic strategies. Future research should further explore these relationships and develop more refined approaches to managing catatonia.

Case report: Diagnostic challenges in an adolescent case of autistic catatonia.

Catatonia is a complex neuropsychiatric syndrome involving a constellation of psychomotor disturbances including catalepsy, waxy flexibility, stupor, mutism, negativism, agitation, posturing, stereotypes, mannerisms, grimacing, echolalia, and echopraxia. Catatonia occurs in several conditions including psychotic, affective and neurodevelopmental disorders such as autism spectrum disorder (ASD). ASD is a neurodevelopmental disorder characterized by persistent deficits in communication, social interaction, restricted interests, repetitive behaviours and sensory sensitivities. Catatonia can occur in response to life stressors such as extreme fear or threat, interpersonal conflict, tragic events or following significant loss. Those with ASD may be particularly vulnerable to the negative impact of stressors and the link between catatonia and ASD is being increasingly recognized. The overlapping features of catatonia and ASD make it difficult to differentiate often resulting in delayed or missed diagnosis. Catatonia in ASD remains a significant clinical challenge; it is difficult to diagnose and can pose debilitating difficulties for those affected. Catatonia is a treatable condition and prompt recognition is vital in securing the best possible outcome. We report a complex and unique case of a 15-year-old boy who presented with severe cognitive and functional decline with a background history of significant bullying and deterioration in his mental state. This case posed a diagnostic conundrum leading to a diagnosis of underlying ASD, anxiety and trauma.