1. Choi JW, Oh J, Bennett DH, Kannan K, Tancredi DJ, Miller M, Schmidt RJ, Shin HM. Gestational exposure to organophosphate esters and autism spectrum disorder and other non-typical development in a cohort with elevated familial likelihood. Environ Res. 2024; 263(Pt 2): 120141.

BACKGROUND: Gestational exposure to organophosphate esters (OPEs) is known to affect offspring neurodevelopment in animal studies. However, epidemiological evidence is inconsistent. METHODS: Participants were 277 mother-child pairs from MARBLES (Markers of Autism Risk in Babies – Learning Early Signs), a cohort with elevated familial likelihood of autism spectrum disorder (ASD). Nine OPE biomarker concentrations were quantified in maternal urine collected during the 2nd or 3rd trimesters of pregnancy. At age 3 years, children underwent clinical assessment for ASD and were classified into ASD, other non-typical development (non-TD), or typical development (TD). Multinomial logistic regression was used to estimate associations between each OPE biomarker and relative risk ratios for ASD and non-TD compared to TD. We examined effect modification by child sex and socioeconomic status. We also conducted a secondary analysis by using a continuous measure of ASD symptom severity as an outcome. Quantile-based g-computation was performed to examine the associations for an OPE mixture. RESULTS: Overall, no significant association was observed between the concentrations of each OPE biomarker or their mixture and relative risk for either ASD or non-TD. Effect modifications by child sex and maternal education were not observed. When the analysis was stratified by homeownership, among non-homeowners, ASD likelihood was increased with increased levels of bis(1-chloro-2-propyl) phosphate, bis(butoxyethyl) phosphate, and sum of di-n-butyl phosphate and di-iso-butyl phosphate (DBUP/DIBP) (p(int) < 0.10). Higher DBUP/DIBP were associated with increased ASD symptom severity scores. CONCLUSION: There was no clear evidence of gestational OPE exposure in association with relative risk for ASD; however, potential effect modification by homeownership was observed. Although our cohort includes children with elevated familial likelihood of ASD, this is the first study investigating the association between gestational OPE exposure and clinically-diagnosed ASD. Further research is needed to confirm our findings in the general population.

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2. Clair MS, Massoudi K, Tarbox J, Najdowski A, Simchoni L, Jackson M, Persicke A. Correction: Making Deception Fun: Teaching Autistic Individuals How to Play Friendly Tricks. Behav Anal Pract. 2024; 17(3): 947.

[This corrects the article DOI: 10.1007/s40617-024-00935-z.].

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3. d’Oleire Uquillas F, Sefik E, Li B, Trotter MA, Steele KA, Seidlitz J, Gesue R, Latif M, Fasulo T, Zhang V, Kislin M, Verpeut JL, Cohen JD, Sepulcre J, Wang SS, Gomez J. Multimodal evidence for cerebellar influence on cortical development in autism: structural growth amidst functional disruption. Mol Psychiatry. 2024.

Despite perinatal damage to the cerebellum being one of the highest risk factors for later being diagnosed with autism spectrum disorder (ASD), it is not yet clear how the cerebellum might influence the development of cerebral cortex and whether this co-developmental process is distinct between neurotypical and ASD children. Leveraging a large structural brain MRI dataset of neurotypical children and those diagnosed with ASD, we examined whether structural variation in cerebellar tissue across individuals was correlated with neocortical variation during development, including the thalamus as a coupling factor. We found that the thalamus plays a distinct role in moderating cerebro-cerebellar structural coordination in ASD. Notably, structural coupling between cerebellum, thalamus, and neocortex was strongest in younger childhood and waned by early adolescence, mirroring a previously undescribed trajectory of behavioral development between ASD and neurotypical children. Complementary functional connectivity analyses likewise revealed atypical connectivity between cerebellum and neocortex in ASD. This relationship was particularly prominent in a model of cerebellar structure predicting functional connectivity, where ASD and neurotypical children showed divergent patterns. Interestingly, these functional-structural relationships became more prominent with age, while structural effects were most prominent earlier in childhood, and showed significant lateralization. This pattern may suggest a developmental sequence where early uncoordinated structural growth amongst regions is followed by increasingly atypical functional synchronization. These findings provide multimodal evidence in the living brain for a cerebellar diaschisis model of autism, where both increased cerebellar-cerebral structural coupling and altered functional connectivity in cerebro-cerebellar pathways contribute to the ontogeny of this neurodevelopmental disorder.

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4. Fischer I, Shohat S, Leichtmann-Bardoogo Y, Nayak R, Wiener G, Rosh I, Shemen A, Tripathi U, Rokach M, Bar E, Hussein Y, Castro AC, Chen G, Soffer A, Schokoroy-Trangle S, Elad-Sfadia G, Assaf Y, Schroeder A, Monteiro P, Stern S, Maoz BM, Barak B. Shank3 mutation impairs glutamate signaling and myelination in ASD mouse model and human iPSC-derived OPCs. Sci Adv. 2024; 10(41): eadl4573.

Autism spectrum disorder (ASD) is characterized by social and neurocognitive impairments, with mutations of the SHANK3 gene being prominent in patients with monogenic ASD. Using the InsG3680 mouse model with a Shank3 mutation seen in humans, we revealed an unknown role for Shank3 in postsynaptic oligodendrocyte (OL) features, similar to its role in neurons. This was shown by impaired molecular and physiological glutamatergic traits of InsG3680-derived primary OL cultures. In vivo, InsG3680 mice exhibit significant reductions in the expression of key myelination-related transcripts and proteins, along with deficits in myelin ultrastructure, white matter, axonal conductivity, and motor skills. Last, we observed significant impairments, with clinical relevance, in induced pluripotent stem cell-derived OLs from a patient with the InsG3680 mutation. Together, our study provides insight into Shank3’s role in OLs and reveals a mechanism of the crucial connection of myelination to ASD pathology.

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5. Ge W, Zhang C, Yang G, Zhang B. Prevalence and trends of autism spectrum disorder and other developmental disabilities among children and adolescents in the United States from 2019 to 2021. Front Psychiatry. 2024; 15: 1471969.

BACKGROUND: The National Health Interview Survey (NHIS) is a comprehensive health survey conducted by the National Center for Health Statistics (NCHS) in the U.S., providing valuable insights into the health status of the population. This study focuses on the NHIS child survey between 2019 and 2021, exploring developmental disabilities in U.S. children, including autism spectrum disorder (ASD), attention deficit/hyperactivity disorder or attention-deficit disorder (ADHD/ADD), intellectual disability (ID), other developmental delay (other DD), and learning disability (LD). OBJECTIVE: Leveraging NHIS data, our primary objective is to investigate the latest trends and disparities in the prevalence of developmental disabilities among various racial-ethnic groups. METHODS: Employing a repeated cross-sectional design, we analyzed NHIS data from 2019 to 2021, focusing on children aged 3-17. The survey employed a meticulous stratified multi-stage sampling design. We utilized SAS version 9.4 for data analysis, calculating race-ethnicity-specific prevalence rates and employing weighted linear regression and the Rao-Scott chi-square test for trend analysis. RESULTS: Among 19,490 children, prevalence rates varied: ASD (3.11%), ADHD/ADD (9.50%), ID (1.85%), other DD (5.66%), and LD (7.49%). Non-Hispanic black children exhibited higher rates of ID and LD, while non-Hispanic white children had the highest ADHD/ADD prevalence. Disparities persisted across sociodemographic subgroups, with variations in prevalence rates. CONCLUSION: Our study reveals an increase in ASD prevalence and persistent disparities among racial-ethnic groups. Non-Hispanic black children face elevated risks of ID and LD, while non-Hispanic white children exhibit higher rates of ADHD/ADD.

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6. Gong M, Li J, Qin Z, Machado Bressan Wilke MV, Liu Y, Li Q, Liu H, Liang C, Morales-Rosado JA, Cohen ASA, Hughes SS, Sullivan BR, Waddell V, van den Boogaard MH, van Jaarsveld RH, van Binsbergen E, van Gassen KL, Wang T, Hiatt SM, Amaral MD, Kelley WV, Zhao J, Feng W, Ren C, Yu Y, Boczek NJ, Ferber MJ, Lahner C, Elliott S, Ruan Y, Mignot C, Keren B, Xie H, Wang X, Popp B, Zweier C, Piard J, Coubes C, Mau-Them FT, Safraou H, Innes AM, Gauthier J, Michaud JL, Koboldt DC, Sylvie O, Willems M, Tan WH, Cogne B, Rieubland C, Braun D, McLean SD, Platzer K, Zacher P, Oppermann H, Evenepoel L, Blanc P, El Khattabi L, Haque N, Dsouza NR, Zimmermann MT, Urrutia R, Klee EW, Shen Y, Du H, Rappaport L, Liu CM, Chen X. MARK2 variants cause autism spectrum disorder via the downregulation of WNT/β-catenin signaling pathway. Am J Hum Genet. 2024.

Microtubule affinity-regulating kinase 2 (MARK2) contributes to establishing neuronal polarity and developing dendritic spines. Although large-scale sequencing studies have associated MARK2 variants with autism spectrum disorder (ASD), the clinical features and variant spectrum in affected individuals with MARK2 variants, early developmental phenotypes in mutant human neurons, and the pathogenic mechanism underlying effects on neuronal development have remained unclear. Here, we report 31 individuals with MARK2 variants and presenting with ASD, other neurodevelopmental disorders, and distinctive facial features. Loss-of-function (LoF) variants predominate (81%) in affected individuals, while computational analysis and in vitro expression assay of missense variants supported the effect of MARK2 loss. Using proband-derived and CRISPR-engineered isogenic induced pluripotent stem cells (iPSCs), we show that MARK2 loss leads to early neuronal developmental and functional deficits, including anomalous polarity and dis-organization in neural rosettes, as well as imbalanced proliferation and differentiation in neural progenitor cells (NPCs). Mark2(+/-) mice showed abnormal cortical formation and partition and ASD-like behavior. Through the use of RNA sequencing (RNA-seq) and lithium treatment, we link MARK2 loss to downregulation of the WNT/β-catenin signaling pathway and identify lithium as a potential drug for treating MARK2-associated ASD.

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7. Hakizimana O, Hitayezu J, Uyisenga JP, Onohuean H, Palmeira L, Bours V, Alagbonsi AI, Uwineza A. Genetic etiology of autism spectrum disorder in the African population: a scoping review. Front Genet. 2024; 15: 1431093.

BACKGROUND: Autism spectrum disorder (ASD) is a neurodevelopmental disorder (NDD) characterized by significant impairments in social, communicative, and behavioral abilities. However, only a limited number of studies address the genetic basis of ASD in the African population. This study aims to document the genes associated with ASD in Africa and the techniques used to identify them. Additionally, genes identified elsewhere but not yet in Africa are also noted. METHODS: Online databases such as Wiley Online Library, PubMed, and Africa Journal Online were used. The review was conducted using the keyword related to genetic and genomic ASD study in the African population. RESULT: In this scoping review, 40 genetic studies on ASD in Africa were reviewed. The Egyptian and South African populations were the most studied, with 25 and 5 studies, respectively. Countries with fewer studies included Tunisia (4), East African countries (3), Libya (1), Nigeria (1), and Morocco (1). Some 61 genes responsible for ASD were identified in the African population: 26 were identified using a polymerase chain reaction (PCR)-based method, 22 were identified using sequencing technologies, and 12 genes and one de novo chromosomal aberration were identified through other techniques. No African study identified any ASD gene with genome-wide association studies (GWAS). Notably, at least 20 ASD risk genes reported in non-African countries were yet to be confirmed in Africa’s population. CONCLUSION: There are insufficient genetic studies on ASD in the African population, with sample size being a major limitation in most genetic association studies, leading to inconclusive results. Thus, there is a need to conduct more studies with large sample sizes to identify other genes associated with ASD in Africa’s population using high-throughput sequencing technology.

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8. Janz P, Bainier M, Marashli S, Gross S, Redondo RL. Clinically-probed mechanisms of action in Fragile-X Syndrome fail to normalize translational EEG phenotypes in Fmr1 knockout mice. Neuropharmacology. 2024: 110182.

Fragile X syndrome (FXS) is a neurodevelopmental disorder caused by Fragile X Messenger Ribonucleoprotein (FMRP) deficiency. Electroencephalogram (EEG) changes in FXS include alterations of oscillatory activity and responses to sensory stimuli, some of which have been back-translated into rodent models by knocking-out the Fragile X messenger ribonucleoprotein 1 gene (Fmr1-KO). However, the validity of these EEG phenotypes as objective biomarkers requires further investigation. Potential pharmacotherapies such as mGluR5 inhibitors (e.g. CTEP; 2-chloro-4-((2,5-dimethyl-1-(4-(trifluoromethoxy)phenyl)-1H-imidazol-4-yl)ethynyl)pyridine), GABA(B)R agonists (e.g. arbaclofen) and δ-containing GABA(A)R agonists (e.g. gaboxadol) have not translated into clinical success despite rescuing many phenotypes in the Fmr1-KO model. Yet none of these treatments have been assessed on EEG phenotypes in the Fmr1-KO model. Therefore, we set out to discover new EEG phenotypes in Fmr1-KO mice, using « task-free » and auditory-evoked (AEPs) and visually-evoked potential (VEP) paradigms, and probe their modulation by CTEP, arbaclofen and gaboxadol, using within-subjects designs. First, we report Fmr1-KO-associated EEG abnormalities that closely resemble those observed in FXS, including elevated gamma-band power, reduced alpha/beta-band coherence, increased AEPs and delayed VEPs. Secondly, we found that pharmacological treatment, at best, only partially normalized EEG phenotypes. CTEP restored alpha/beta-band coherence and AEP amplitudes but failed to normalize gamma power and VEP latencies. Conversely, arbaclofen reduced gamma power but did not restore coherence or AEP amplitudes and further delayed VEPs. Gaboxadol did not normalize any EEG phenotypes. We conclude that these compounds have limited ability to normalize these EEG phenotypes.

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9. Kurahashi H, Kunisawa K, Tanaka KF, Kubota H, Hasegawa M, Miyachi M, Moriya Y, Hasegawa Y, Nagai T, Saito K, Nabeshima T, Mouri A. Autism spectrum disorder-like behaviors induced by hyper-glutamatergic NMDA receptor signaling through hypo-serotonergic 5-HT(1A) receptor signaling in the prefrontal cortex in mice exposed to prenatal valproic acid. Neuropsychopharmacology. 2024.

Autism spectrum disorder (ASD) is a neurodevelopmental disorder characterized by repetitive behaviors, social deficits, and cognitive impairments. Maternal use of valproic acid (VPA) during pregnancy is associated with an increased risk of ASD in offspring. The prevailing pathophysiological hypothesis for ASD involves excitation/inhibition (E/I) imbalances and serotonergic dysfunction. Here, we investigated the association between glutamatergic-serotonergic neuronal interactions and ASD-like behaviors in mice exposed to prenatal VPA. Prenatal VPA exposure induced excessive repetitive self-grooming behavior and impaired social behavior and object recognition memory in young adult period. Prenatal VPA mice showed hyper-glutamatergic function (increase in basal extracellular glutamate levels and CaMKII phosphorylation) and hypo-serotonergic function (decrease in 5-hydroxyindoleacetic acid and stimulation-induced serotonin [5-HT] release, but an increase in 5-HT transporter expression) in the prefrontal cortex. Treatment with a low-affinity NMDA receptor antagonist (memantine), a selective 5-HT reuptake inhibitor (fluoxetine), and a 5-HT(1A) receptor agonist (tandospirone) attenuated both the increase in CaMKII phosphorylation and ASD-like behavior of prenatal VPA mice. Opto-genetic activation of the serotonergic neuronal system attenuated impairments in social behavior and object recognition memory in prenatal VPA mice. WAY-100635-a 5-HT(1A) receptor antagonist-antagonized the effect of fluoxetine on impaired social behavior and object recognition memory. These results suggest that E/I imbalance and ASD-like behavior are associated with hypo-serotonergic receptor signaling through 5-HT(1A) receptors in prenatal VPA mice.

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10. Langhorne S, Uglik-Marucha N, Broadhurst C, Lieven E, Pearson A, Vitoratou S, Leadbitter K. Correction: The Knowledge of Autism Questionnaire-UK: Development and Initial Psychometric Evaluation. J Autism Dev Disord. 2024.

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11. Luglio DG, Kleeman MJ, Yu X, Lin JC, Chow T, Martinez MP, Chen Z, Chen JC, Eckel SP, Schwartz J, Lurmann F, McConnell R, Xiang AH, Rahman MM. Prenatal Exposure to Source-Specific Fine Particulate Matter and Autism Spectrum Disorder. Environ Sci Technol. 2024.

In this study, associations between prenatal exposure to fine particulate matter (PM2.5) from 9 sources and development of autism spectrum disorder (ASD) were assessed in a population-based retrospective pregnancy cohort in southern California. The cohort included 318,750 mother-child singleton pairs. ASD cases (N = 4559) were identified by ICD codes. Source-specific PM2.5 concentrations were estimated from a chemical transport model with a 4 × 4 km(2) resolution and assigned to maternal pregnancy residential addresses. Cox proportional hazard models were used to estimate the hazard ratios (HR) of ASD development for each individual source. We also adjusted for total PM2.5 mass and in a separate model for all other sources simultaneously. Increased ASD risk was observed with on-road gasoline (HR [CI]: 1.18 [1.13, 1.24]), off-road gasoline (1.15 [1.12, 1.19]), off-road diesel (1.08 [1.05, 1.10]), food cooking (1.05 [1.02, 1.08]), aircraft (1.04 [1.01, 1.06]), and natural gas combustion (1.09 [1.06, 1.11]), each scaled to standard deviation increases in concentration. On-road gasoline and off-road gasoline were robust for other pollutant groups. PM2.5 emitted from different sources may have different impacts on ASD. The results also identify PM source mixtures for toxicological investigations that may provide evidence for future public health policies.

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12. Marshall KB, Bowman KS, Tereshko L, Suarez VD, Schreck KA, Zane T, Leaf JB. Correction: Behavior Analysts’ Use of Treatments for Individuals with Autism: Trends within the Field. Behav Anal Pract. 2024; 17(3): 945-6.

[This corrects the article DOI: 10.1007/s40617-023-00776-2.].

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13. Percy AK, Neul JL, Benke TA, Berry-Kravis EM, Glaze DG, Marsh ED, Barrett AM, An D, Bishop KM, Youakim JM. Trofinetide for the treatment of Rett syndrome: Long-term safety and efficacy results of the 32-month, open-label LILAC-2 study. Med. 2024; 5(10): 1275-81.e2.

BACKGROUND: Trofinetide was approved for the treatment of Rett syndrome (RTT) in patients aged ≥2 years based on the results of the 12-week, randomized, phase 3 LAVENDER study. In LILAC, a 40-week, open-label extension study of LAVENDER, trofinetide continued to improve the symptoms of RTT, with a similar safety profile as LAVENDER. Here, we report long-term safety and efficacy results of LILAC-2, a 32-month, open-label extension study. METHODS: Females aged 5-22 years who completed LILAC were eligible to enter LILAC-2. Safety and tolerability were assessed with the incidence of adverse events (AEs). Efficacy was assessed with Rett Syndrome Behaviour Questionnaire (RSBQ) and Clinical Global Impression-Improvement (CGI-I) scores. Caregiver interviews explored the patient’s experience with RTT and the efficacy of trofinetide during study participation. FINDINGS: In total, 77 participants were enrolled in LILAC-2. The most common AEs were diarrhea (53.2%), COVID-19 (27.3%), and vomiting (19.5%). The mean (standard error [SE]) change in RSBQ score from LAVENDER baseline to week 104 of LILAC-2 was -11.8 (2.45). The mean (SE) CGI-I score from LILAC baseline to week 12 of LILAC-2 was 3.1 (0.10). Most caregivers (96%; n = 24/25) were satisfied or very satisfied with the benefits of trofinetide. CONCLUSIONS: Long-term treatment with trofinetide continued to improve RTT symptoms, without new safety concerns. Caregivers reported satisfaction with trofinetide related to improvements that were meaningful for their child and themselves. FUNDING: The study was supported by Acadia Pharmaceuticals (San Diego, CA, USA). This study was registered at ClinicalTrials.gov: NCT04776746.

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14. Rivelis E, Valicenti-McDermott M. A Short-Term Group Telehealth Cognitive Behavioral Therapy Intervention for Youth with Autism and Anxiety: A Pilot Study. J Child Adolesc Psychopharmacol. 2024.

Background: Children with autism often present with comorbid anxiety disorders. Cognitive behavioral therapy (CBT) is an effective, evidence-based approach to treating anxiety, but information on youth with autism and anxiety is limited. Coping Cat is a 16-week CBT intervention for children with anxiety but its use in a group telehealth format in an urban, predominantly Hispanic population is limited. Objectives: (a) To examine the feasibility and preliminary effectiveness of a short-term CBT telehealth group for youth with autism and anxiety disorders in an urban, predominantly Hispanic population and (b) to examine satisfaction with the intervention. Methods: Single-arm pilot study that consisted of a 16-week telehealth CBT group therapy was based on a modified Coping Cat curriculum. Youth with autism and anxiety disorders who were on a waitlist for psychotherapy at an urban developmental center were invited to participate. Anxiety was assessed pre- and posttreatment using the Screen for Child Anxiety Related Emotional Disorders, parent and self-report. Results: Eighteen children were enrolled; 16 children completed the program. Mean age was 11 ± 2.5 years (8-15 years); 89% males, 61% Hispanic. There was a significant reduction in pre-post intervention in symptoms of overall anxiety (parent: 41.0 ± 18.5 to 31.0 ± 16.3 p ≤ 0.003, self: 25.9 ± 12.8 to 14.1 ± 7.8 p ≤ 0.001), panic disorder (parent: 8.1 ± 7.0 to 4.1 ± 4.2 p = 0.013, self: 5.1 ± 4.8 to 0.8 ± 0.9 p = 0.004), and separation anxiety disorder (parent: 7.5 ± 4.8 to 5.7 ± 4.4 p = 0.041, self: 5.8 ± 3.3 to 3.8 ± 2.4 p = 0.018) as per parent and self-reports. Self-report data also revealed a significant reduction in symptoms of social anxiety disorder (6.5 ± 3.5 to 3.9 ± 2.7 p ≤ 0.001). Parents and children reported satisfaction with the group. Conclusion: In this small, predominantly Hispanic population of youth with autism and anxiety disorder, 89% of families were compliant with a group telehealth CBT intervention. Parents and youth reported a significant reduction in anxiety symptoms and program satisfaction. A modified group CBT program via telehealth represents a feasible intervention for youth with autism and anxiety disorders.

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15. Sha L, Cao Z, Fu Y, Duan Y, Xia Y, Feng X, Tomson T, Xie X, Chen L. Global burden and management of women with epilepsy in pregnancy: A modeling study. Med. 2024; 5(10): 1326-33.e4.

BACKGROUND: Most pregnant women with epilepsy do not receive proper medical care, which creates a special burden worldwide. We aimed to qualify this special global burden and assess the impact of different clinical management strategies to reduce it. METHODS: The data used in this study were extracted from articles published between 2005 and 2022. We calculated the economic costs associated with major burdens experienced by pregnant women with epilepsy. We developed a microsimulation model to estimate the different effects of various interventions and their combinations as integrated strategies for pregnant women with epilepsy and related burden reduction. We also compared the regional differences in disease burden and interventions. FINDINGS: The total economic burden for pregnant women with epilepsy is estimated to reach $1.8 billion globally annually, which is more than three times the burden for epilepsy alone. Folic acid supplementation is projected to be the most effective intervention, with a 9.1% reduction in major congenital malformations, a 14.9% reduction in autism spectrum disorder, and a 10.8% reduction in offspring-related economic burden globally annually. Integrated strategies are associated with a reduced economic burden of up to $37.7 million annually globally. Folic acid supplementation is the most effective intervention in high- and upper-middle-income countries, whereas changes in antiseizure medication prescriptions are more effective in lower-middle- and low-income countries. CONCLUSION: This study highlights the huge burden for pregnant women with epilepsy and actions that must be taken to improve their quality of life. FUNDING: This work was supported by the Sichuan Science and Technology Program (2023YFS0047).

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16. Thakore A, Kelly A, Petursdottir AI, Stockdale M. Evaluation of a Treatment Package for Chronic, Stereotypic Hand Mouthing of a Child Diagnosed with Autism. Behav Anal Pract. 2024; 17(3): 915-20.

We describe the clinical treatment of repetitive, stereotypic hand mouthing in a 7-year old child with severe developmental delay, conducted in the context of center-based autism service. The client’s history included use of mechanical restraint to prevent tissue damage and infection. Prior antecedent- and reinforcement-based interventions were ineffective. Functional analysis results suggested hand mouthing was maintained by automatic reinforcement. Response interruption and redirection (RIRD) initially did not decrease hand mouthing, but the addition of contingent protective equipment, together with modified RIRD, was followed by reduction to near-zero level, which was maintained following withdrawal of the intervention. • Repetitive hand mouthing may require treatment due to tissue damage and other health consequences. • Repetitive hand mouthing is typically maintained in the absence of social contingencies and may be resistant to treatment. • When reinforcement-based interventions are not sufficient, contingent application of nonrestraining protective equipment may produce lasting reduction in hand mouthing. • Successful intervention in this case freed the client from mechanical (arm band) restraint and was followed by gains in skill acquisition. SUPPLEMENTARY INFORMATION: The online version contains supplementary material available at 10.1007/s40617-024-00956-8.

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17. Tropea D. Trofinetide treatment for Rett syndrome: Lessons to learn. Med. 2024; 5(10): 1194-6.

The US FDA approval of trofinetide as the first pharmacological treatment to improve Rett syndrome’s symptomatology marks a significant milestone with broad implications for various disorders. The LILAC trials demonstrate long-term safety and efficacy of trofinetide.(1)(,)(2) While further research is needed to fully resolve the condition, insights from trofinetide trials can inform strategies for future treatments and trials.

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