Literature DB >> 34227036

Clinical and Molecular Aspects of the Neurodevelopmental Disorder Associated with PAK3 Perturbation.

Giulia Pascolini1, Federica Gaudioso2, Chiara Passarelli3, Antonio Novelli3, Niccolò Di Giosaffatte2, Silvia Majore2, Paola Grammatico2.   

Abstract

X-linked intellectual disability can be diagnosed in about 10-12% of intellectually disabled males. In the past, mutations affecting the PAK3 gene (p21 protein-activated kinase 3, MIM#300142) have been associated with a non-syndromic form of X-linked intellectual disability, which has to date been identified in a limited number of families.Since this neurodevelopmental disorder mostly afflicts males, descriptions of symptomatic female carriers are quite rare.We describe a female patient with neurodevelopmental delay and a novel PAK3 variant. Interestingly, she manifests craniofacial anomalies, including microcephaly, representing the second reported microcephalic female but the first for whom a detailed clinical description is available. She also displays other uncommon clinical findings, which we illustrate.Moreover, a comprehensive clinical and molecular review of all to date published patients has been made. This study contributes to further delineate the PAK3-related phenotype, which can be considered a non-syndromic X-linked intellectual disability, with seemingly recurrent craniofacial abnormalities.
© 2021. The Author(s), under exclusive licence to Springer Science+Business Media, LLC, part of Springer Nature.

Entities:  

Keywords:  Craniofacial dysmorphisms; Microcephaly; Neurodevelopmental delay; PAK3

Mesh:

Substances:

Year:  2021        PMID: 34227036     DOI: 10.1007/s12031-021-01868-w

Source DB:  PubMed          Journal:  J Mol Neurosci        ISSN: 0895-8696            Impact factor:   3.444


  15 in total

1.  Whole-exome sequencing points to considerable genetic heterogeneity of cerebral palsy.

Authors:  G McMichael; M N Bainbridge; E Haan; M Corbett; A Gardner; S Thompson; B W M van Bon; C L van Eyk; J Broadbent; C Reynolds; M E O'Callaghan; L S Nguyen; D L Adelson; R Russo; S Jhangiani; H Doddapaneni; D M Muzny; R A Gibbs; J Gecz; A H MacLennan
Journal:  Mol Psychiatry       Date:  2015-02-10       Impact factor: 15.992

2.  Short case report: Xq23 deletion involving PAK3 as a novel cause of developmental delay in a 6-year-old boy.

Authors:  Ashley Cartwright; Kath Smith; Meena Balasubramanian
Journal:  Clin Dysmorphol       Date:  2017-01       Impact factor: 0.816

3.  PAK3 mutations responsible for severe intellectual disability and callosal agenesis inhibit cell migration.

Authors:  Kévin Duarte; Solveig Heide; Sandrine Poëa-Guyon; Véronique Rousseau; Christel Depienne; Agnès Rastetter; Caroline Nava; Tania Attié-Bitach; Ferechté Razavi; Jelena Martinovic; Marie Laure Moutard; Jacqueline Cherfils; Cyril Mignot; Delphine Héron; Jean-Vianney Barnier
Journal:  Neurobiol Dis       Date:  2019-12-14       Impact factor: 5.996

Review 4.  Regulation of phosphorylation pathways by p21 GTPases. The p21 Ras-related Rho subfamily and its role in phosphorylation signalling pathways.

Authors:  L Lim; E Manser; T Leung; C Hall
Journal:  Eur J Biochem       Date:  1996-12-01

5.  Missense mutation in PAK3, R67C, causes X-linked nonspecific mental retardation.

Authors:  T Bienvenu; V des Portes; N McDonell; A Carrié; R Zemni; P Couvert; H H Ropers; C Moraine; H van Bokhoven; J P Fryns; K Allen; C A Walsh; J Boué; A Kahn; J Chelly; C Beldjord
Journal:  Am J Med Genet       Date:  2000-08-14

6.  Regional localisation of two non-specific X-linked mental retardation genes (MRX30 and MRX31).

Authors:  A J Donnelly; M W Partington; A K Ryan; J C Mulley
Journal:  Am J Med Genet       Date:  1996-07-12

7.  Improvement of Self-Injury With Dopamine and Serotonin Replacement Therapy in a Patient With a Hemizygous PAK3 Mutation: A New Therapeutic Strategy for Neuropsychiatric Features of an Intellectual Disability Syndrome.

Authors:  Gabriella A Horvath; Maja Tarailo-Graovac; Tanja Bartel; Simone Race; Margot I Van Allen; Ingrid Blydt-Hansen; Colin J Ross; Wyeth W Wasserman; Mary B Connolly; Clara D M van Karnebeek
Journal:  J Child Neurol       Date:  2018-01       Impact factor: 1.987

8.  Whole exome sequencing diagnosis of inborn errors of metabolism and other disorders in United Arab Emirates.

Authors:  Aisha Al-Shamsi; Jozef L Hertecant; Abdul-Kader Souid; Fatma A Al-Jasmi
Journal:  Orphanet J Rare Dis       Date:  2016-07-08       Impact factor: 4.123

Review 9.  A novel PAK3 pathogenic variant identified in two siblings from a Japanese family with X-linked intellectual disability: case report and review of the literature.

Authors:  Aritoshi Iida; Kyoko Takano; Eri Takeshita; Chihiro Abe-Hatano; Shinichi Hirabayashi; Yuji Inaba; Shunichi Kosugi; Yoichiro Kamatani; Yukihide Momozawa; Michiaki Kubo; Eiji Nakagawa; Ken Inoue; Yu-Ichi Goto
Journal:  Cold Spring Harb Mol Case Stud       Date:  2019-12-13

10.  A mutation in PAK3 with a dual molecular effect deregulates the RAS/MAPK pathway and drives an X-linked syndromic phenotype.

Authors:  Pamela Magini; Tommaso Pippucci; I-Chun Tsai; Simona Coppola; Emilia Stellacci; Anna Bartoletti-Stella; Daniela Turchetti; Claudio Graziano; Giovanna Cenacchi; Iria Neri; Duccio Maria Cordelli; Valentina Marchiani; Rosalba Bergamaschi; Giuseppe Gasparre; Giovanni Neri; Laura Mazzanti; Annalisa Patrizi; Emilio Franzoni; Giovanni Romeo; Domenico Bordo; Marco Tartaglia; Nicholas Katsanis; Marco Seri
Journal:  Hum Mol Genet       Date:  2014-02-19       Impact factor: 6.150

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  1 in total

Review 1.  Pathophysiological Mechanisms in Neurodevelopmental Disorders Caused by Rac GTPases Dysregulation: What's behind Neuro-RACopathies.

Authors:  Marcello Scala; Masashi Nishikawa; Koh-Ichi Nagata; Pasquale Striano
Journal:  Cells       Date:  2021-12-02       Impact factor: 6.600

  1 in total

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