Literature DB >> 31112762

Dystonia genes and their biological pathways.

H A Jinnah1, Yan V Sun2.   

Abstract

The dystonias are a group of disorders characterized by excessive contraction of muscles leading to abnormal involuntary movements. The clinical manifestations are very heterogeneous, with numerous distinct syndromes. The etiologies for dystonia are also heterogeneous with idiopathic, acquired, and inherited forms. Technological advances in genetics over the past two decades have led to a rapid growth in the number of genes associated with dystonia. These genes encode proteins with very diverse biological functions. This review focusses on genes that have contributed to understanding shared biological pathways relevant to specific subgroups of dystonia syndromes. Although many potential shared biological pathways have been proposed, the ones addressed here include defects in dopamine signaling, mitochondrial dysfunction and energy maintenance, toxic accumulation of heavy metals in the brain, and calcium channels and abnormal calcium homeostasis. Elucidation of these and other shared pathways is important for understanding the biological basis for dystonia and for designing novel experimental therapeutics that have the broadest potential for multiple types of dystonia.
Copyright © 2019 Elsevier Inc. All rights reserved.

Entities:  

Keywords:  Biological pathways; Calcium homeostasis; Dopamine; Dystonia; Gene; Genetic variant; Genetics; Mitochondrial disease; Mutation; NBIA; Neurobiology

Mesh:

Year:  2019        PMID: 31112762     DOI: 10.1016/j.nbd.2019.05.014

Source DB:  PubMed          Journal:  Neurobiol Dis        ISSN: 0969-9961            Impact factor:   5.996


  17 in total

Review 1.  Neuropathology and pathogenesis of extrapyramidal movement disorders: a critical update. II. Hyperkinetic disorders.

Authors:  Kurt A Jellinger
Journal:  J Neural Transm (Vienna)       Date:  2019-06-24       Impact factor: 3.575

Review 2.  Medical and Surgical Treatments for Dystonia.

Authors:  H A Jinnah
Journal:  Neurol Clin       Date:  2020-03-02       Impact factor: 3.806

Review 3.  The apparent paradox of phenotypic diversity and shared mechanisms across dystonia syndromes.

Authors:  Alessio Di Fonzo; Alberto Albanese; Hyder A Jinnah
Journal:  Curr Opin Neurol       Date:  2022-07-05       Impact factor: 6.283

4.  DYT-PRKRA Mutation P222L Enhances PACT's Stimulatory Activity on Type I Interferon Induction.

Authors:  Lauren S Vaughn; Kenneth Frederick; Samuel B Burnett; Nutan Sharma; D Cristopher Bragg; Sarah Camargos; Francisco Cardoso; Rekha C Patel
Journal:  Biomolecules       Date:  2022-05-17

5.  Biallelic variants in TSPOAP1, encoding the active-zone protein RIMBP1, cause autosomal recessive dystonia.

Authors:  Niccolò E Mencacci; Marisa M Brockmann; Jinye Dai; Sander Pajusalu; Burcu Atasu; Joaquin Campos; Gabriela Pino; Paulina Gonzalez-Latapi; Christopher Patzke; Michael Schwake; Arianna Tucci; Alan Pittman; Javier Simon-Sanchez; Gemma L Carvill; Bettina Balint; Sarah Wiethoff; Thomas T Warner; Apostolos Papandreou; Audrey Soo; Reet Rein; Liis Kadastik-Eerme; Sanna Puusepp; Karit Reinson; Tiiu Tomberg; Hasmet Hanagasi; Thomas Gasser; Kailash P Bhatia; Manju A Kurian; Ebba Lohmann; Katrin Õunap; Christian Rosenmund; Thomas C Südhof; Nicholas W Wood; Dimitri Krainc; Claudio Acuna
Journal:  J Clin Invest       Date:  2021-04-01       Impact factor: 14.808

6.  Naming Genes for Dystonia: DYT-z or Ditzy?

Authors:  Niccolo E Mencacci; H A Jinnah
Journal:  Tremor Other Hyperkinet Mov (N Y)       Date:  2019-08-28

7.  A Multi-center Genome-wide Association Study of Cervical Dystonia.

Authors:  Yan V Sun; Chengchen Li; Qin Hui; Yunfeng Huang; Richard Barbano; Ramon Rodriguez; Irene A Malaty; Stephen Reich; Kimberly Bambarger; Katie Holmes; Joseph Jankovic; Neepa J Patel; Emmanuel Roze; Marie Vidailhet; Brian D Berman; Mark S LeDoux; Alberto J Espay; Pinky Agarwal; Sarah Pirio-Richardson; Samuel A Frank; William G Ondo; Rachel Saunders-Pullman; Sylvain Chouinard; Stover Natividad; Alfredo Berardelli; Alexander Y Pantelyat; Allison Brashear; Susan H Fox; Meike Kasten; Ulrike M Krämer; Miriam Neis; Tobias Bäumer; Sebastian Loens; Max Borsche; Simone Zittel; Antonia Maurer; Mathias Gelderblom; Jens Volkmann; Thorsten Odorfer; Andrea A Kühn; Friederike Borngräber; Inke R König; Carlos Cruchaga; Adam C Cotton; Gamze Kilic-Berkmen; Alan Freeman; Stewart A Factor; Laura Scorr; J Douglas Bremner; Viola Vaccarino; Arshed A Quyyumi; Christine Klein; Joel S Perlmutter; Katja Lohmann; Hyder A Jinnah
Journal:  Mov Disord       Date:  2021-07-28       Impact factor: 10.338

8.  An Open-Label Phase 2a Study to Evaluate the Safety and Tolerability of Perampanel in Cervical Dystonia.

Authors:  Susan H Fox; Matthew Swan; Hyder A Jinnah; Maria E T de Freitas; Lais M de Oliveira; Duha Al-Shorafat; Hubert H Fernandez; Katie Kompoliti; Cynthia Comella
Journal:  Mov Disord Clin Pract       Date:  2021-05-13

9.  Differential expression of striatal proteins in a mouse model of DOPA-responsive dystonia reveals shared mechanisms among dystonic disorders.

Authors:  Maria A Briscione; Ashok R Dinasarapu; Pritha Bagchi; Yuping Donsante; Kaitlyn M Roman; Anthony M Downs; Xueliang Fan; Jessica Hoehner; H A Jinnah; Ellen J Hess
Journal:  Mol Genet Metab       Date:  2021-06-02       Impact factor: 4.204

10.  Gut Microbiome and Serum Metabolome Alterations Associated with Isolated Dystonia.

Authors:  Lingyan Ma; Jing Keng; Min Cheng; Hua Pan; Bo Feng; Yongfeng Hu; Tao Feng; Fan Yang
Journal:  mSphere       Date:  2021-08-04       Impact factor: 4.389

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