Individuals with FOXP1 syndrome present with a complex neurobehavioral profile with high rates of ADHD, anxiety, repetitive behaviors, and sensory ... View Full Text


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Article Info

DATE

2021-09-29

AUTHORS

M. Pilar Trelles, Tess Levy, Bonnie Lerman, Paige Siper, Reymundo Lozano, Danielle Halpern, Hannah Walker, Jessica Zweifach, Yitzchak Frank, Jennifer Foss-Feig, Alexander Kolevzon, Joseph Buxbaum

ABSTRACT

BackgroundFOXP1 syndrome is an autosomal dominant neurodevelopmental disorder characterized by intellectual disability, developmental delay, speech and language delays, and externalizing behaviors. We previously evaluated nine children and adolescents with FOXP1 syndrome to better characterize its phenotype. We identified specific areas of interest to be further explored, namely autism spectrum disorder (ASD) and internalizing and externalizing behaviors. MethodsHere, we assess a prospective cohort of additional 17 individuals to expand our initial analyses and focus on these areas of interest. An interdisciplinary group of clinicians evaluated neurodevelopmental, behavioral, and medical features in participants. We report results from this cohort both alone, and in combination with the previous cohort, where possible. ResultsPrevious observations of intellectual disability, motor delays, and language deficits were confirmed. In addition, 24% of the cohort met criteria for ASD. Seventy-five percent of individuals met DSM-5 criteria for attention-deficit/hyperactivity disorder and 38% for an anxiety disorder. Repetitive behaviors were almost universally present (95%) even without a diagnosis of ASD. Sensory symptoms, in particular sensory seeking, were common.LimitationsAs FOXP1 syndrome is a rare disorder, sample size is limited.ConclusionsThese findings have important implications for the treatment and care of individuals with FOXP1 syndrome. Notably, standardized testing for ASD showed high sensitivity, but low specificity, when compared to expert consensus diagnosis. Furthermore, many individuals in our cohort who received diagnoses of attention-deficit/hyperactivity disorder or anxiety disorder were not being treated for these symptoms; therefore, our findings suggest that there may be immediate areas for improvements in treatment for some individuals. More... »

PAGES

61

References to SciGraph publications

  • 2011. Vineland Adaptive Behavior Scales in ENCYCLOPEDIA OF CLINICAL NEUROPSYCHOLOGY
  • 2006-09-19. The Repetitive Behavior Scale-Revised: Independent Validation in Individuals with Autism Spectrum Disorders in JOURNAL OF AUTISM AND DEVELOPMENTAL DISORDERS
  • 1994-10. Autism Diagnostic Interview-Revised: A revised version of a diagnostic interview for caregivers of individuals with possible pervasive developmental disorders in JOURNAL OF AUTISM AND DEVELOPMENTAL DISORDERS
  • 2021-04-23. FOXP1 syndrome: a review of the literature and practice parameters for medical assessment and monitoring in JOURNAL OF NEURODEVELOPMENTAL DISORDERS
  • 2018-11-01. Characterization of a recurrent missense mutation in the forkhead DNA-binding domain of FOXP1 in SCIENTIFIC REPORTS
  • 2010-06-23. Chiari I malformation, delayed gross motor skills, severe speech delay, and epileptiform discharges in a child with FOXP1 haploinsufficiency in EUROPEAN JOURNAL OF HUMAN GENETICS
  • 2014-07-16. Transcriptional Regulation by FOXP1, FOXP2, and FOXP4 Dimerization in JOURNAL OF MOLECULAR NEUROSCIENCE
  • 2017-08-23. Vineland III in ENCYCLOPEDIA OF AUTISM SPECTRUM DISORDERS
  • 2012-11-11. Standardizing ADOS Domain Scores: Separating Severity of Social Affect and Restricted and Repetitive Behaviors in JOURNAL OF AUTISM AND DEVELOPMENTAL DISORDERS
  • 2017-06-05. Diagnoses and characteristics of autism spectrum disorders in children with Prader-Willi syndrome in JOURNAL OF NEURODEVELOPMENTAL DISORDERS
  • 2016-04-05. Development, behavior, and biomarker characterization of Smith-Lemli-Opitz syndrome: an update in JOURNAL OF NEURODEVELOPMENTAL DISORDERS
  • 2001-10-04. A forkhead-domain gene is mutated in a severe speech and language disorder in NATURE
  • 2017-10-24. Prospective investigation of FOXP1 syndrome in MOLECULAR AUTISM
  • 2016-01-19. Parent-reported and clinician-observed autism spectrum disorder (ASD) symptoms in children with attention deficit/hyperactivity disorder (ADHD): implications for practice under DSM-5 in MOLECULAR AUTISM
  • 2014-09-30. Brain-specific Foxp1 deletion impairs neuronal development and causes autistic-like behaviour in MOLECULAR PSYCHIATRY
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    http://scigraph.springernature.com/pub.10.1186/s13229-021-00469-z

    DOI

    http://dx.doi.org/10.1186/s13229-021-00469-z

    DIMENSIONS

    https://app.dimensions.ai/details/publication/pub.1141495283

    PUBMED

    https://www.ncbi.nlm.nih.gov/pubmed/34588003


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