Literature DB >> 33059813

[Autosomal dominant intellectual disability-40 caused by a de novo mutation of the CHAMP1 gene: a case report].

Ming-Mei Wang1, Deng-Na Zhu, San-Song Li, Guang-Yu Zhang, Lei Yang, Yun-Xia Zhao, Han-You Liu.   

Abstract

A boy, aged 6 months, had the manifestations of intellectual and motor developmental delay, head instability, general weakness, unawareness of grasping objects by hands, and unusual facies (slightly wide eye distance, epicanthus, esotropia, mouth-opening appearance, short philtrum, and low-set ears). Gene detection results showed a de novo heterozygous frameshift mutation of the CHAMP1 gene at the chromosomal location of chr13:115089847, and nuclear acid was changed to c.530delCinsTTT, resulting in a change in amino acid to p.S177Ffs*2. Therefore, the boy was diagnosed with autosomal dominant intellectual disability-40 caused by the mutation in the CHAMP1 gene. This case report suggests that for children with unexplained intellectual disability, especially those with generalized hypotonia and severe language disorder, the possibility of CHAMP1 gene mutation should be considered, and genetic testing should be performed as early as possible.

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Year:  2020        PMID: 33059813      PMCID: PMC7569005     

Source DB:  PubMed          Journal:  Zhongguo Dang Dai Er Ke Za Zhi        ISSN: 1008-8830


  9 in total

1.  De Novo Mutations in CHAMP1 Cause Intellectual Disability with Severe Speech Impairment.

Authors:  Maja Hempel; Kirsten Cremer; Charlotte W Ockeloen; Klaske D Lichtenbelt; Johanna C Herkert; Jonas Denecke; Tobias B Haack; Alexander M Zink; Jessica Becker; Eva Wohlleber; Jessika Johannsen; Bader Alhaddad; Rolph Pfundt; Sigrid Fuchs; Dagmar Wieczorek; Tim M Strom; Koen L I van Gassen; Tjitske Kleefstra; Christian Kubisch; Hartmut Engels; Davor Lessel
Journal:  Am J Hum Genet       Date:  2015-09-03       Impact factor: 11.025

2.  De Novo Truncating Mutations in the Kinetochore-Microtubules Attachment Gene CHAMP1 Cause Syndromic Intellectual Disability.

Authors:  Bertrand Isidor; Sébastien Küry; Jill A Rosenfeld; Thomas Besnard; Sébastien Schmitt; Shelagh Joss; Sally J Davies; Robert Roger Lebel; Alex Henderson; Christian P Schaaf; Haley E Streff; Yaping Yang; Vani Jain; Nodoka Chida; Xenia Latypova; Cédric Le Caignec; Benjamin Cogné; Sandra Mercier; Marie Vincent; Estelle Colin; Dominique Bonneau; Anne-Sophie Denommé; Philippe Parent; Brigitte Gilbert-Dussardier; Sylvie Odent; Annick Toutain; Amélie Piton; Christian Dina; Audrey Donnart; Pierre Lindenbaum; Eric Charpentier; Richard Redon; Kenji Iemura; Masanori Ikeda; Kozo Tanaka; Stéphane Bézieau
Journal:  Hum Mutat       Date:  2016-02-04       Impact factor: 4.878

3.  Genome sequencing identifies major causes of severe intellectual disability.

Authors:  Christian Gilissen; Jayne Y Hehir-Kwa; Djie Tjwan Thung; Maartje van de Vorst; Bregje W M van Bon; Marjolein H Willemsen; Michael Kwint; Irene M Janssen; Alexander Hoischen; Annette Schenck; Richard Leach; Robert Klein; Rick Tearle; Tan Bo; Rolph Pfundt; Helger G Yntema; Bert B A de Vries; Tjitske Kleefstra; Han G Brunner; Lisenka E L M Vissers; Joris A Veltman
Journal:  Nature       Date:  2014-06-04       Impact factor: 49.962

4.  CAMP (C13orf8, ZNF828) is a novel regulator of kinetochore-microtubule attachment.

Authors:  Go Itoh; Shin-ichiro Kanno; Kazuhiko S K Uchida; Shuhei Chiba; Shiro Sugino; Kana Watanabe; Kensaku Mizuno; Akira Yasui; Toru Hirota; Kozo Tanaka
Journal:  EMBO J       Date:  2010-11-09       Impact factor: 11.598

5.  The kinetochore protein, CENPF, is mutated in human ciliopathy and microcephaly phenotypes.

Authors:  Aoife M Waters; Rowan Asfahani; Paula Carroll; Louise Bicknell; Francesco Lescai; Alison Bright; Estelle Chanudet; Anthony Brooks; Sonja Christou-Savina; Guled Osman; Patrick Walsh; Chiara Bacchelli; Ariane Chapgier; Bertrand Vernay; David M Bader; Charu Deshpande; Mary O' Sullivan; Louise Ocaka; Horia Stanescu; Helen S Stewart; Friedhelm Hildebrandt; Edgar Otto; Colin A Johnson; Katarzyna Szymanska; Nicholas Katsanis; Erica Davis; Robert Kleta; Mike Hubank; Stephen Doxsey; Andrew Jackson; Elia Stupka; Mark Winey; Philip L Beales
Journal:  J Med Genet       Date:  2015-01-06       Impact factor: 6.318

6.  Disturbed chromosome segregation and multipolar spindle formation in a patient with CHAMP1 mutation.

Authors:  Nobuhiko Okamoto; Yuki Tsuchiya; Ichiro Kuki; Toshiyuki Yamamoto; Hirotomo Saitsu; Daiju Kitagawa; Naomichi Matsumoto
Journal:  Mol Genet Genomic Med       Date:  2017-07-12       Impact factor: 2.183

7.  An anatomically comprehensive atlas of the adult human brain transcriptome.

Authors:  Michael J Hawrylycz; Ed S Lein; Angela L Guillozet-Bongaarts; Elaine H Shen; Lydia Ng; Jeremy A Miller; Louie N van de Lagemaat; Kimberly A Smith; Amanda Ebbert; Zackery L Riley; Chris Abajian; Christian F Beckmann; Amy Bernard; Darren Bertagnolli; Andrew F Boe; Preston M Cartagena; M Mallar Chakravarty; Mike Chapin; Jimmy Chong; Rachel A Dalley; Barry David Daly; Chinh Dang; Suvro Datta; Nick Dee; Tim A Dolbeare; Vance Faber; David Feng; David R Fowler; Jeff Goldy; Benjamin W Gregor; Zeb Haradon; David R Haynor; John G Hohmann; Steve Horvath; Robert E Howard; Andreas Jeromin; Jayson M Jochim; Marty Kinnunen; Christopher Lau; Evan T Lazarz; Changkyu Lee; Tracy A Lemon; Ling Li; Yang Li; John A Morris; Caroline C Overly; Patrick D Parker; Sheana E Parry; Melissa Reding; Joshua J Royall; Jay Schulkin; Pedro Adolfo Sequeira; Clifford R Slaughterbeck; Simon C Smith; Andy J Sodt; Susan M Sunkin; Beryl E Swanson; Marquis P Vawter; Derric Williams; Paul Wohnoutka; H Ronald Zielke; Daniel H Geschwind; Patrick R Hof; Stephen M Smith; Christof Koch; Seth G N Grant; Allan R Jones
Journal:  Nature       Date:  2012-09-20       Impact factor: 49.962

8.  De novo pathogenic variants in CHAMP1 are associated with global developmental delay, intellectual disability, and dysmorphic facial features.

Authors:  Akemi J Tanaka; Megan T Cho; Kyle Retterer; Julie R Jones; Catherine Nowak; Jessica Douglas; Yong-Hui Jiang; Allyn McConkie-Rosell; G Bradley Schaefer; Julie Kaylor; Omar A Rahman; Aida Telegrafi; Bethany Friedman; Ganka Douglas; Kristin G Monaghan; Wendy K Chung
Journal:  Cold Spring Harb Mol Case Stud       Date:  2016-01

9.  Large-scale discovery of novel genetic causes of developmental disorders.

Authors: 
Journal:  Nature       Date:  2014-12-24       Impact factor: 69.504

  9 in total
  2 in total

1.  Neurodevelopmental phenotypes in individuals with pathogenic variants in CHAMP1.

Authors:  Madison Garrity; Haluk Kavus; Marta Rojas-Vasquez; Irene Valenzuela; Austin Larson; Sara Reed; Gary Bellus; Cyril Mignot; Arnold Munnich; Bertrand Isidor; Wendy K Chung
Journal:  Cold Spring Harb Mol Case Stud       Date:  2021-08-02

2.  CHAMP1 disorder is associated with a complex neurobehavioral phenotype including autism, ADHD, repetitive behaviors and sensory symptoms.

Authors:  Tess Levy; Bonnie Lerman; Danielle Halpern; Yitzchak Frank; Christina Layton; Jessica Zweifach; Paige M Siper; Joseph D Buxbaum; Alexander Kolevzon
Journal:  Hum Mol Genet       Date:  2022-08-17       Impact factor: 5.121

  2 in total

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