Literature DB >> 24719385

Macrocerebellum, epilepsy, intellectual disability, and gut malrotation in a child with a 16q24.1-q24.2 contiguous gene deletion.

Andrea H Seeley1, Mark A Durham, Mark A Micale, Jeffrey Wesolowski, Bradley R Foerster, Donna M Martin.   

Abstract

Macrocerebellum is a rare condition characterized by enlargement of the cerebellum with conservation of the overall shape and cytoarchitecture. Here, we report on a child with a distinctive constellation of clinical features including macrocerebellum, epilepsy, apparent intellectual disability, dysautonomia, gut malrotation, and poor gut motility. Oligonucleotide chromosome microarray analysis identified a 16q24.1-q24.2 deletion that included four OMIM genes (FBXO31, MAP1LC3B, JPH3, and SLC7A5). Review of prior studies describing individuals with similar or overlapping16q24.1-q24.2 deletions identified no other reports of macrocerebellum. These observations highlight a potential genetic cause of this rare disorder and raise the possibility that one or more gene(s) in the 16q24.1-q24.2 interval regulate cerebellar development.
© 2014 Wiley Periodicals, Inc.

Entities:  

Keywords:  FBXO31; JPH3; MAP1LC3B; SLC7A5; deletion syndrome; macrocerebellum

Mesh:

Year:  2014        PMID: 24719385      PMCID: PMC4266592          DOI: 10.1002/ajmg.a.36569

Source DB:  PubMed          Journal:  Am J Med Genet A        ISSN: 1552-4825            Impact factor:   2.802


  34 in total

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Journal:  Ann Neurol       Date:  2005-03       Impact factor: 10.422

2.  Cerebellar abnormalities in infants and toddlers with Williams syndrome.

Authors:  Wendy Jones; John Hesselink; Eric Courchesne; Tim Duncan; Kevin Matsuda; Ursula Bellugi
Journal:  Dev Med Child Neurol       Date:  2002-10       Impact factor: 5.449

3.  Macrocerebellum: neuroimaging and clinical features of a newly recognized condition.

Authors:  J B Bodensteiner; G B Schaefer; G M Keller; J N Thompson; M K Bowen
Journal:  J Child Neurol       Date:  1997-09       Impact factor: 1.987

4.  FBXO31 is the chromosome 16q24.3 senescence gene, a candidate breast tumor suppressor, and a component of an SCF complex.

Authors:  Raman Kumar; Paul M Neilsen; Joanne Crawford; Ross McKirdy; Jaclyn Lee; Jason A Powell; Zarqa Saif; Julie M Martin; Marc Lombaerts; Cees J Cornelisse; Anne-Marie Cleton-Jansen; David F Callen
Journal:  Cancer Res       Date:  2005-12-15       Impact factor: 12.701

5.  The neuroimaging findings in Sotos syndrome.

Authors:  G B Schaefer; J B Bodensteiner; B A Buehler; A Lin; T R Cole
Journal:  Am J Med Genet       Date:  1997-02-11

6.  Coexpression of junctophilin type 3 and type 4 in brain.

Authors:  Miyuki Nishi; Hiroyuki Sakagami; Shinji Komazaki; Hisatake Kondo; Hiroshi Takeshima
Journal:  Brain Res Mol Brain Res       Date:  2003-10-21

Review 7.  Lhermitte-Duclos disease and Cowden disease: clinical and genetic study in five patients with Lhermitte-Duclos disease and literature review.

Authors:  A Pérez-Núñez; A Lagares; J Benítez; M Urioste; R D Lobato; J R Ricoy; A Ramos; P González
Journal:  Acta Neurochir (Wien)       Date:  2004-05-21       Impact factor: 2.216

8.  A case of macrocephaly, hydrocephalus, megacerebellum, white matter abnormalities and Rosenthal fibres.

Authors:  M Torreman; L M Smit; P van der Valk; J Valk; P Scheltens
Journal:  Dev Med Child Neurol       Date:  1993-08       Impact factor: 5.449

Review 9.  Clinical and radiological aspects of dysplastic gangliocytoma (Lhermitte-Duclos disease): a report of two cases with review of the literature.

Authors:  G Milbouw; J D Born; D Martin; J Collignon; P Hans; M Reznik; J Bonnal
Journal:  Neurosurgery       Date:  1988-01       Impact factor: 4.654

10.  Junctophilin-mediated channel crosstalk essential for cerebellar synaptic plasticity.

Authors:  Sho Kakizawa; Yasushi Kishimoto; Kouichi Hashimoto; Taisuke Miyazaki; Kazuharu Furutani; Hidemi Shimizu; Masahiro Fukaya; Miyuki Nishi; Hiroyuki Sakagami; Atsushi Ikeda; Hisatake Kondo; Masanobu Kano; Masahiko Watanabe; Masamitsu Iino; Hiroshi Takeshima
Journal:  EMBO J       Date:  2007-03-08       Impact factor: 11.598

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  5 in total

Review 1.  SCF ubiquitin ligase-targeted therapies.

Authors:  Jeffrey R Skaar; Julia K Pagan; Michele Pagano
Journal:  Nat Rev Drug Discov       Date:  2014-11-14       Impact factor: 84.694

2.  Isolated macrocerebellum: description of six cases and literature review.

Authors:  Felice D'Arco; Lorenzo Ugga; Ferdinando Caranci; Maria Pia Riccio; Chiara Figliuolo; Kshitij Mankad; Alessandra D'Amico
Journal:  Quant Imaging Med Surg       Date:  2016-10

3.  The amino acid transporter Slc7a5 regulates the mTOR pathway and is required for granule cell development.

Authors:  Aidan M Sokolov; Jennie C Holmberg; David M Feliciano
Journal:  Hum Mol Genet       Date:  2020-11-04       Impact factor: 6.150

4.  Compound genetic etiology in a patient with a syndrome including diabetes, intellectual deficiency and distichiasis.

Authors:  Amélie Bonnefond; Martine Doco Fenzy; Lauriane Le Collen; Brigitte Delemer; Marta Spodenkiewicz; Pascale Cornillet Lefebvre; Emmanuelle Durand; Emmanuel Vaillant; Alaa Badreddine; Mehdi Derhourhi; Tarik Ait Mouhoub; Guillaume Jouret; Pauline Juttet; Pierre François Souchon; Martine Vaxillaire; Philippe Froguel
Journal:  Orphanet J Rare Dis       Date:  2022-02-28       Impact factor: 4.123

5.  Microdeletion of 16q24.1-q24.2-A unique etiology of Lymphedema-Distichiasis syndrome and neurodevelopmental disorder.

Authors:  Marina Michelson; Gabriel Lidzbarsky; Daniella Nishri; Ifat Israel-Elgali; Rachel Berger; Michal Gafner; Noam Shomron; Dorit Lev; Yael Goldberg
Journal:  Am J Med Genet A       Date:  2022-03-21       Impact factor: 2.578

  5 in total

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