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| S-EPMC8096321 | biostudies-literature
Project description:IntroductionPaediatric acute-onset neuropsychiatric syndrome (PANS) is a neuroimmune condition characterised by episodic exacerbations and remissions. This study investigates the impact of PANS on children's occupational performance during these phases, using the Vineland Adaptive Behaviour Scale (VABS). Understanding these variations is crucial for developing tailored interventions and managing the condition effectively.MethodsA two-period bidirectional case-crossover design was employed to assess occupational performance in children with PANS at exacerbation (T-E) and remission (T-R) phases. Parents of children aged 4.6-13.1 years with PANS were recruited globally, with data collected via online surveys and monthly updates. The VABS evaluated occupational performance, with analysis using linear mixed models and Least Squares Means (LSMeans) for accurate representation.ResultsData from 27 participants showed significantly lower scores in the Adaptive Behaviour Composite and socialisation domain of the VABS at T-E compared with T-R, indicating a decline in occupational performance and social skills during exacerbations. During remission, participants demonstrated typical performance across all domains.ConclusionThe study highlights a marked decline in occupational performance and socialisation during PANS exacerbations, with improvements to typical levels during remission. These findings emphasise the need for targeted interventions to address occupational and social challenges in children with PANS during exacerbations, underscoring the episodic nature of the condition and the risk of misinterpreting behaviours if assessed during exacerbations. Multidisciplinary allied health support may benefit children with PANS.Consumer and community involvementAlthough there was no direct consumer and community involvement, two researchers on our team have family members with PANS. Their personal experiences provided vital insights into the challenges faced by children with PANS, deeply influencing our study's design, focus, and interpretation, ensuring it reflects the realities of those affected.Plain language summaryOur study looked at how PANS affects the things children do in their daily lives. We asked parents of children ages 4.6-13.1 years old to complete surveys measuring this at two times: once during a symptom flare-up and once during a period of wellness. Our results showed that during symptom flare-ups, children's social skills and general ability to do daily tasks were much lower compared with periods of wellness. When children were well, their abilities were similar to those of typically developing children of the same age. This highlights the need for occupational therapy during flare-ups to help improve the lives of children with PANS.
| S-EPMC11650011 | biostudies-literature
Project description:Background: Individuals with generalised joint hypermobility (GJH, present in 10-20% of the general population) are at increased risk of being diagnosed with a range of psychiatric and rheumatological conditions. It is unknown whether Paediatric acute-onset neuropsychiatric syndrome (PANS), characterised by childhood onset obsessive-compulsive disorder or restricted eating and typically associated with several comorbid neuropsychiatric symptoms, is associated with GJH. It is also unknown whether extensive psychiatric comorbidity is associated with GJH. Method: This is a case-control study including 105 participants. We compared three groups: Individuals with PANS, individuals with other mental disorders and healthy controls. Joint mobility was assessed with the Beighton scoring system, psychiatric comorbidity with the M.I.N.I. or MINI-KID interview and symptoms of PANS with the PsychoNeuroInflammatory related Signs and Symptoms Inventory (PNISSI). Results: Hypermobility was similar across groups, and high rates of psychiatric comorbidity was not associated with higher Beighton scores. Conclusion: Although GJH is associated with several psychiatric conditions, such as ADHD and anxiety, this does not seem to be the case for PANS according to this preliminary study.
| S-EPMC8678126 | biostudies-literature
Project description:BackgroundPaediatric acute-onset neuropsychiatric syndrome (PANS), an umbrella term that includes PANDAS (paediatric autoimmune neuropsychiatric disorders associated with streptococcal infections) is suggested to be a psychiatric disorder of autoimmune aetiology. PANS is characterised by an acute onset of obsessive-compulsive disorder or restricted eating with multiple comorbid symptoms. The specificity of the PANS criteria is not fully understood.AimsTo describe a cohort of patients with PANS and to determine if PANS features relating to symptoms, onset and course are more common in PANS than in other psychiatric conditions.MethodA case-control study comparing patients with interview-confirmed PANS with patients with suspected PANS and patients with a psychiatric condition but with no suspicion of PANS. Validated and non-validated measures of symptoms, onset and episodic course were used.ResultsIllness in patients with interview-confirmed PANS featured an episodic course and multiple symptoms present at onset compared with the psychiatric controls. However, individuals with interview-confirmed PANS did not present a specific symptom profile.ConclusionsPANS may be a distinct clinical entity featuring an acute onset, an episodic course and multiple symptoms at onset.Declaration of interestNone.
| S-EPMC6401534 | biostudies-literature
Project description:This SuperSeries is composed of the SubSeries listed below. Refer to individual Series
2014-04-08 | E-GEOD-55426 | biostudies-arrayexpress
Project description:BackgroundTreatment with intravenous immunoglobulin (IVIG) in children with Paediatric Acute-onset Neuropsychiatric Syndrome (PANS) has for many years been used on clinical indications, but the research evidence for its efficacy is insufficient.MethodsOpen-label prospective in-depth trial including ten children (median age 10.3 years) with PANS, who received IVIG treatment 2 g/kg monthly for three months. Primary outcomes were changes in symptom severity and impairment from baseline to first and second follow-up visits one month after first and one month after third treatment, using three investigator-rated scales: Paediatric Acute Neuropsychiatric Symptom (PANS) scale, Clinical Global Impression - Severity and Improvement (CGI-S and CGI-I) scales. Secondary outcomes reported here were changes in Children's Yale-Brown Obsessive Compulsive Scale (CY-BOCS) scores, and side effects.ResultsAll ten children received three treatments at one-month intervals according to study plan. From baseline to second follow-up marked reductions were seen in mean total PANS scale scores (p = .005), and CGI-S scores (p = .004). CGI-I ratings showed much to very much global improvement (mean CGI-I 1.8). Nine children had clinical response defined as > 30% reduction in PANS Scale scores. Improvements were also noted for CY-BOCS scores (p = .005), and in school attendance. Three children suffered moderate to severe temporary side effects after the first treatment, and the remaining seven had mild to moderate side effects. Side effects were much less severe after second and third treatments.ConclusionsConsiderable and pervasive improvements in symptoms and clinical impairments were seen in these ten children after three monthly IVIG treatments. Moderate to severe transient side effects occurred in three cases.Trial registrationEudraCT no. 2019-004758-27, Clinicaltrials.gov no. NCT04609761, 05/10/2020.
| S-EPMC9357317 | biostudies-literature
Project description:Background: The prevalence and impact of allergic and immune-mediated food disorders in pediatric acute-onset neuropsychiatric syndrome (PANS) are mostly unknown. Objective: We sought to explore the prevalence of atopic dermatitis (AD), asthma, allergic rhinitis (AR), IgE-mediated food allergies (FAs), and other immune-mediated food disorders requiring food avoidance in patients with PANS. In addition, to further understand the extent of food restriction in this population, we investigated the empiric use of dietary measures to improve PANS symptoms. Methods: Pediatric patients in a PANS Clinic and Research Program were given surveys regarding their caregiver burdens, allergic and food-related medical history, and whether food elimination resulted in perception of improvement of PANS symptoms. A review of health records was conducted to confirm that all responses in the survey were concordant with documentation of each patient's medical chart. Results: Sixty-nine (ages 4-20 years) of 80 subjects who fulfilled PANS criteria completed the surveys. Thirteen (18.8%) had AD, 11 (15.9%) asthma, 33 (47.8%) AR, 11 (15.9%) FA, 1 (1.4%) eosinophilic gastrointestinal disorders, 1 (1.4%) food protein-induced enterocolitis syndrome, 3 (4.3%) milk protein-induced proctocolitis syndrome, and 3 (4.3%) celiac disease. Thirty subjects (43.5%) avoided foods due to PANS; elimination of gluten and dairy was most common and was associated with perceived improvement of PANS symptoms (by parents). This perceived improvement was not confirmed with objective data. Conclusions: The prevalence of allergic and immune-mediated food disorders in PANS is similar to the general population as reported in the literature, with the exception of AR that appears to be more prevalent in our PANS cohort. More research will be required to establish whether diet or allergies influence PANS symptoms.
| S-EPMC6154445 | biostudies-literature
Project description:This SuperSeries is composed of the SubSeries listed below. Refer to individual Series
2014-05-14 | E-GEOD-57633 | biostudies-arrayexpress
Project description:OXTR modulates a variety of behaviors in mammals, including social memory and recognition. Genetic and epigenetic dysregulation of OXTR has been suggested to be implicated in neuropsychiatric disorders, including autism spectrum disorder (ASD). While the involvement of DNA methylation is suggested, the mechanism underlying epigenetic regulation of OXTR is largely unknown. This has hampered the experimental design and interpretation of the results of epigenetic studies of OXTR in neuropsychiatric disorders. From the generation and characterization of a new line of Tet1 mutant mice - by deleting the largest coding exon 4 (Tet1Δe4) - we discovered for the first time to our knowledge that Oxtr has an array of mRNA isoforms and a complex transcriptional regulation. Select isoforms of Oxtr are significantly reduced in the brain of Tet1Δe4-/- mice. Accordingly, CpG islands of Oxtr are hypermethylated during early development and persist into adulthood. Consistent with the reduced express of OXTR, Tet1Δe4-/- mice display impaired maternal care, social behavior, and synaptic responses to oxytocin stimulation. Our findings elucidate a mechanism mediated by TET1 protein in regulating Oxtr expression by preventing DNA hypermethylation of Oxtr. The discovery of epigenetic dysregulation of Oxtr in TET1-deficient mouse brain supports the necessity of a reassessment of existing findings and a value of future studies of OXTR in neuropsychiatric disorders.
| S-EPMC6328031 | biostudies-literature
Project description:This SuperSeries is composed of the SubSeries listed below. Refer to individual Series
2012-12-28 | E-GEOD-39600 | biostudies-arrayexpress