Catatonia associated with pediatric postoperative cerebellar mutism syndrome.

OBJECTIVE: To ascertain the presence of catatonia in cases of pediatric postoperative cerebellar mutism syndrome (PPCMS). METHOD: A systematic review of PPCMS case reports of patients aged 0-17 years with sufficient clinical information to extract catatonic phenomena was undertaken following PRISMA guidelines. Standardized catatonia rating scales were applied to selected cases retrospectively to ascertain whether diagnostic criteria for catatonia were met. A case known to the authors is also presented. RESULTS: Two hundred twenty-one suitable full-text articles were identified. Following screening and application of inclusion criteria, 51 articles were selected plus seven more from their references, reporting on 119 subjects. All cases met Bush and Francis (BF) diagnostic criteria for catatonia, 92.5% Pediatric Catatonia Rating Scale (PCRS), 52.9% ICD-11, and 44.5% DSM-5. All patients presented with mutism. The next most frequent signs were immobility/stupor (77.3%), withdrawal (35.3%), mannerisms (23.5%), and excitement/agitation (18.5%). Most cases presented with stuporous catatonia (75.6%). Catatonia most frequently occurred following resection of medulloblastoma (64.7%). Preoperative hydrocephalus occurred in 89 patients (74.8%). CONCLUSION: Catatonia was frequent in this PPCMS sample, with a predominant stuporous variant; it should be considered in patients with PPCMS and assessed with reliable and validated instruments for prompt diagnosis and management.

Absence of Long-Lasting Morning Stiffness at the Time of Diagnosis as Paraneoplastic Warning in Patients with Polymyalgia Rheumatica (PMR): Data from Italian Single-Center Study.

BACKGROUND: There is little literature on the paraneoplastic value of the absence of long-lasting morning stiffness (MS) at the time of diagnosis of polymyalgia rheumatica (PMR). We investigated whether and to what extent this finding was related to the probability of diagnosing a neoplasia. PATIENTS AND METHODS: This was an observational, retrospective, single-center cohort study. We enrolled all patients consecutively referred to our rheumatologic outpatient clinic between January 2015 and December 2020, who could be classified as PMR according to 2012 EULAR/ACR criteria. In particular, we assessed all patients scoring a minimum of five points with a combination of clinical and ultrasound (US) criteria. The exclusion criteria were as follows: (a) follow-up duration

Polymyalgia rheumatica and polymyalgia-like syndromes as adverse events following COVID-19 vaccines: working notes from a narrative review of published literature.

Since the 1990s, polymyalgia rheumatica (PMR) has been reported as a possible adverse event following immunization (AEFI). The aim of this narrative review is to provide an overview of PMR (and PMR-like syndromes) following the most common types of COVID-19 vaccines, namely mRNA (tozinameran and mRNA-1273) and adenovirus-vectored (ChAdOx1-S) vaccines. To date, published literature reports few cases of PMR as vaccine-linked AEFI. Yet Vigibase, the WHO pharmacovigilance database, reports a few hundred cases. Based on these data, we address the question whether PMR/PMR-like syndromes following COVID-19 vaccines can be a true adverse or a coincidental event, and discuss its possible pathogenetic mechanisms.

Catatonia in older adults: A systematic review.

BACKGROUND: Catatonia is a complex psychomotor syndrome that often goes unrecognized and untreated, even though its classification has evolved in recent years. Prompt and correct identification of catatonia allows for highly effective treatment and prevention of possible complications. The underrecognition of catatonia in older patients is also frequent, and research in this population is scarce. AIM: To conduct a systematic review of the literature on catatonia in older people to ascertain its clinical characteristics across settings. METHODS: Following the PRISMA guidelines, MEDLINE, EMBASE, and PsycINFO databases were searched from inception to December 2021, with a strategy aimed at identifying all articles published on catatonia in older adults. Titles and abstracts were scanned and selected independently by two authors. Papers investigating issues related to catatonia and/or catatonic symptoms in older people, with English abstracts available, were included. References of selected articles were revised to identify other relevant studies. RESULTS: In total, 1355 articles were retrieved. After removing duplicates, 879 remained. Of the 879 identified abstracts, 669 were excluded because they did not meet the inclusion criteria. A total of 210 articles underwent full text review, and 51 were eliminated for various reasons. Fourteen more articles were selected from the references. Overall, 173 articles were reviewed: 108 case reports, 35 case series, 11 prospective cohort studies, 6 case-control studies, 3 retrospective cohort studies and 10 reviews. We found several particular aspects of catatonia in this population. Catatonia in older patients is highly prevalent and tends to have a multifactorial etiology. Older patients, compared to younger patients, have a higher risk of developing catatonia with benzodiazepine (BZD) withdrawal, in bipolar disorder, and in the general hospital. Age, together with other risk factors, was significantly associated with the incidence of deep venous thrombosis, neuroleptic malignant syndrome poor outcome, other complications and mortality. Treatment with BZDs and electroconvulsive therapy is safe and effective. Prompt treatment of its cause is essential to ensure a good prognosis. CONCLUSION: Catatonia in older patients is highly prevalent and tends to have a multifactorial etiology. The risk of developing catatonia in some settings and conditions, as well as of developing complications, is high in this population. Symptomatic treatment is safe and effective, and timely etiologic treatment is fundamental.

The Role of Tumor Necrosis Factor Alpha Antagonists (Anti TNF-α) in Personalized Treatment of Patients with Isolated Polymyalgia Rheumatica (PMR): Past and Possible Future Scenarios.

BACKGROUND: Glucocorticoids (GCs) are the cornerstone of polymyalgia rheumatica (PMR) therapy, but their long-term use (as is usually necessary in PMR patients) can induce many adverse events. Alternatives have long been sought. The primary aim of our narrative review is to provide an overview about the use of anti-tumor necrosis factor alpha (TNF-α) drugs in patients with PMR, and discuss advantages and disadvantages. MATERIALS AND METHODS: we performed a non-systematic literature search (PRISMA protocol not followed) on PubMed and Medline (OVID interface). RESULTS AND CONCLUSIONS: only two anti TNF-α drugs have been prescribed to PMR patients: infliximab in 62 patients and etanercept in 28 patients. These drugs were normally used in addition to GCs when significant comorbidities and/or relapsing PMR were present; less commonly, they were used as first-line therapy. In general, they have been scarcely successful in patients with PMR. Indeed, randomized controlled trials did not confirm the positive results reported in case reports and/or case series. However, an administration schedule and study design different from those proposed in the past could favour new scenarios in the interest of PMR patients.

Catatonia in pediatric obsessive-compulsive disorder: report of two cases.

Catatonia is a psychomotor syndrome which may occur in a wide variety of medical, neurological, and psychiatric conditions. In pediatrics, this condition is rare and is associated with high morbidity and mortality if not correctly diagnosed and treated. Catatonia in obsessive-compulsive disorder is an infrequent association that has been understudied and underdiagnosed. To add to the knowledge on this unusual clinical presentation, two pediatric patients are reported and discussed together with the other two cases described in the literature. These four cases in total of catatonia associated with OCD confirm that it is a relationship that is infrequently reported, possibly because of lack of awareness in clinicians that catatonia can also be caused by OCD, and because the similarity between some catatonic signs and some compulsive phenomena may compound the identification of the former. Most cases of catatonia in this small series seemed to have responded to the optimization of the treatment for OCD. This highlights the clinical importance of an accurate diagnosis of catatonia when associated with OCD.

Back to the future: identification and classification of polymyalgia rheumatica and polymyalgia rheumatica-like syndromes following cancer immunotherapy with checkpoint inhibitors.

Polymyalgia rheumatica (PMR) and PMR-like syndromes are among the most frequent rheumatologic immuno-related adverse events (IRAEs) induced by cancer immunotherapy with "checkpoint inhibitors" (ICIs). Our short communication addresses two key methodological issues laid bare by published literature : 1) how to diagnose PMR and PMR-like syndromes following ICI therapy, 2) how PMR/PMR-like syndromes following ICI therapy are described as adverse drug reactions (ADRs).

Identification and Classification of Polymyalgia Rheumatica (PMR) and PMR-Like Syndromes Following Immune Checkpoint Inhibitors (ICIs) Therapy: Discussion Points and Grey Areas Emerging from a Systematic Review of Published Literature.

Background: Polymyalgia Rheumatica (PMR) is one of the most frequent rheumatologic immune-related adverse effects (IRAEs) in cancer patients following therapy with immune checkpoint inhibitors (ICIs). Atypical findings in many patients often lead to diagnosing PMR-like syndromes. Materials and methods: The aim of our research was to review reported diagnoses of PMR and PMR-like syndromes following ICIs therapy, and assess whether they can be redefined as adverse drug reaction (ADR). In line with PRISMA guidelines, we carried out a systematic search on three main bibliographic databases, based on a combination of subject headings and free text. We included all studies and case-reports published after 2011 (when FDA approved the use of the first ICI) describing the association of PMR or PMR-like syndromes with all types of ICIs therapy. We excluded reviews, conference abstracts, comments, secondary articles, and non-English language studies. Results: We reviewed data from seven studies and eight case-reports, involving a total of 54 patients. Limitations included: the small size of all studies; only one retrospective study used validated criteria for PMR; most reports assessed IRAEs by clinical judgment only and did not seek validation through assessment scales. To date, it remains a conundrum whether IRAEs-PMR is identical to the idiopathic form of the disease, or whether it should be considered a subset of the disease or a new entity. Conclusions: Our review indicates that the relationship between PMR and ICIs therapy is yet to be clearly understood and defined and that future research should remedy the current limits in study design.

Catatonia in N-methyl-d-aspartate receptor antibody encephalitis: Phenomenological characteristics from a systematic review of case reports.

OBJECTIVE: To ascertain the phenomenological characterisation of catatonia in N-methyl-d-aspartate receptor antibody encephalitis (NMDAr-AbE). METHODS: A systematic review of case reports was undertaken in accordance with PRISMA guidelines. Case reports of NMDAr-AbE containing sufficient information on the cases' clinical presentation and meeting the study's inclusion criteria were selected. Cases were searched for clinical data in keeping with core catatonic signs by applying the screening instrument of the Bush-Francis Catatonia Rating Scale. When two or more core signs were ascertained catatonia was considered to be present. RESULTS: 2645 records were identified through the database search. Following screening and application of eligibility/inclusion criteria 139 articles were selected reporting on 189 individual subjects. Catatonia was present in 60% of these cases. The most prevalent signs were immobility/stupor (70%), mutism (67%), excitement (50%), posturing/catalepsy (34%), stereotypies (31%), and rigidity (30%). Immobility/stupor and excitement co-occurred in the same patient in 33% of cases. CONCLUSION: The phenomenological profile of catatonia in this sample of cases of NMDAr-AbE was characterised by a preponderance of signs in the hypokinetic spectrum. However, excitement often co-occurred in these patients suggesting that fluctuations in catatonic semiology may be frequent.

Behavioral, Psychiatric, and Cognitive Adverse Events in Older Persons Treated with Glucocorticoids.

Background: Since the introduction of glucocorticoids (GCs) in the physician's pharmacological arsenal, it has been known that they are a cause of behavioral or psychiatric adverse events (BPAE), as well as of cognitive problems. To the best of our knowledge, the relationship between these adverse events and GCs in older persons has never been evaluated, except through case-reports or series with few cases. In this paper, a review of the literature regarding BPAEs and cognitive disorders in older people treated with CSs is undertaken. Methods: A comprehensive literature search for BPAEs was carried out on the three main bibliographic databases: EMBASE, MEDLINE and PsycINFO (NICE HDAS interface). Emtree terms were: Steroid, steroid therapy, mental disease, mania, delirium, agitation, depression, behavior change, dementia, major cognitive impairment, elderly. The search was restricted to all clinical studies and case reports with focus on the aged (65+ years) published in any language since 1998. Results: Data on the prevalence of the various BPAEs in older patients treated with GCs were very scarse, consisting mainly of case reports and of series with small numbers of patients. It was hence not possible to perform any statistical evaluation of the data (including meta-analysis). Amongst BPAEs, he possibility that delirium can be induced by GCs has been recently been questioned. Co-morbidities and polypharmacy were additional risk factors for BPAEs in older persons. Conclusions: Data on BPAEs in older persons treated with GCs, have several unmet needs that need to be further evaluated with appropriately designed studies.