Literature DB >> 31760646

Pelizaeus-Merzbacher Disease: Molecular and Cellular Pathologies and Associated Phenotypes.

Ken Inoue1.   

Abstract

Pelizaeus-Merzbacher disease (PMD) represents a group of disorders known as hypomyelinating leukodystrophies, which are characterized by abnormal development and maintenance of myelin in the central nervous system. PMD is caused by different types of mutations in the proteolipid protein 1 (PLP1) gene, which encodes a major myelin membrane lipoprotein. These mutations in the PLP1 gene result in distinct cellular and molecular pathologies and a spectrum of clinical phenotypes. In this chapter, I discuss the historical aspects and current understanding of the mechanisms underlying how different PLP1 mutations disrupt the normal process of myelination and result in PMD and other disorders.

Entities:  

Keywords:  Genotype–phenotype correlation; Hypomyelinating leukodystrophies; Molecular mechanism; PLP1 mutations; Pelizaeus-Merzbacher disease

Mesh:

Substances:

Year:  2019        PMID: 31760646     DOI: 10.1007/978-981-32-9636-7_13

Source DB:  PubMed          Journal:  Adv Exp Med Biol        ISSN: 0065-2598            Impact factor:   2.622


  14 in total

Review 1.  [Research advances in the clinical genetics of leukodystrophy in children].

Authors:  Zhe-Lan Huang; Wen-Hao Zhou
Journal:  Zhongguo Dang Dai Er Ke Za Zhi       Date:  2022-06-15

Review 2.  Molecular Biomarkers in Multiple Sclerosis and Its Related Disorders: A Critical Review.

Authors:  Maryam Gul; Amirhossein Azari Jafari; Muffaqam Shah; Seyyedmohammadsadeq Mirmoeeni; Safee Ullah Haider; Sadia Moinuddin; Ammar Chaudhry
Journal:  Int J Mol Sci       Date:  2020-08-21       Impact factor: 5.923

3.  EIF2AK2-related Neurodevelopmental Disorder With Leukoencephalopathy, Developmental Delay, and Episodic Neurologic Regression Mimics Pelizaeus-Merzbacher Disease.

Authors:  Daniel G Calame; Meagan Hainlen; Danielle Takacs; Leah Ferrante; Kayla Pence; Lisa T Emrick; Hsiao-Tuan Chao
Journal:  Neurol Genet       Date:  2020-12-17

4.  Suppression of proteolipid protein rescues Pelizaeus-Merzbacher disease.

Authors:  Matthew S Elitt; Lilianne Barbar; H Elizabeth Shick; Berit E Powers; Yuka Maeno-Hikichi; Mayur Madhavan; Kevin C Allan; Baraa S Nawash; Artur S Gevorgyan; Stevephen Hung; Zachary S Nevin; Hannah E Olsen; Midori Hitomi; Daniela M Schlatzer; Hien T Zhao; Adam Swayze; David F LePage; Weihong Jiang; Ronald A Conlon; Frank Rigo; Paul J Tesar
Journal:  Nature       Date:  2020-07-01       Impact factor: 49.962

5.  Novel Mutations in NPC1 are Associated with Pelizaeus-Merzbacher-Like Disease: A Case Report.

Authors:  Hongling Fu; Qiu Wang; Hanmin Liu
Journal:  Int J Gen Med       Date:  2021-03-09

6.  The Infantile Leukoencephalopathy-Associated Mutation of C11ORF73/HIKESHI Proteins Generates de novo Interactive Activity with Filamin A, Inhibiting Oligodendroglial Cell Morphological Differentiation.

Authors:  Kohei Hattori; Kenji Tago; Shiori Memezawa; Arisa Ochiai; Sui Sawaguchi; Yukino Kato; Takanari Sato; Kazuma Tomizuka; Hiroaki Ooizumi; Katsuya Ohbuchi; Kazushige Mizoguchi; Yuki Miyamoto; Junji Yamauchi
Journal:  Medicines (Basel)       Date:  2021-02-01

7.  Hypomyelinating Leukodystrophy 7 (HLD7)-Associated Mutation of POLR3A Is Related to Defective Oligodendroglial Cell Differentiation, Which Is Ameliorated by Ibuprofen.

Authors:  Sui Sawaguchi; Kenji Tago; Hiroaki Oizumi; Katsuya Ohbuchi; Masahiro Yamamoto; Kazushige Mizoguchi; Yuki Miyamoto; Junji Yamauchi
Journal:  Neurol Int       Date:  2021-12-22

8.  Hypomyelinating Leukodystrophy 8 (HLD8)-Associated Mutation of POLR3B Leads to Defective Oligodendroglial Morphological Differentiation Whose Effect Is Reversed by Ibuprofen.

Authors:  Sui Sawaguchi; Rimi Suzuki; Hiroaki Oizumi; Katsuya Ohbuchi; Kazushige Mizoguchi; Masahiro Yamamoto; Yuki Miyamoto; Junji Yamauchi
Journal:  Neurol Int       Date:  2022-02-16

9.  A novel non-human primate model of Pelizaeus-Merzbacher disease.

Authors:  Larry S Sherman; Weiping Su; Amanda L Johnson; Samuel M Peterson; Cassandra Cullin; Tiffany Lavinder; Betsy Ferguson; Anne D Lewis
Journal:  Neurobiol Dis       Date:  2021-08-05       Impact factor: 7.046

10.  Hypomyelinating Leukodystrophy 15 (HLD15)-Associated Mutation of EPRS1 Leads to Its Polymeric Aggregation in Rab7-Positive Vesicle Structures, Inhibiting Oligodendroglial Cell Morphological Differentiation.

Authors:  Sui Sawaguchi; Mizuki Goto; Yukino Kato; Marina Tanaka; Kenji Tago; Hiroaki Oizumi; Katsuya Ohbuchi; Kazushige Mizoguchi; Yuki Miyamoto; Junji Yamauchi
Journal:  Polymers (Basel)       Date:  2021-03-29       Impact factor: 4.329

View more

北京卡尤迪生物科技股份有限公司 © 2022-2023.