Literature DB >> 29799801

The Genetics of Primary Microcephaly.

Divya Jayaraman1,2,3, Byoung-Il Bae4, Christopher A Walsh1,5,6.   

Abstract

Primary microcephaly (MCPH, for "microcephaly primary hereditary") is a disorder of brain development that results in a head circumference more than 3 standard deviations below the mean for age and gender. It has a wide variety of causes, including toxic exposures, in utero infections, and metabolic conditions. While the genetic microcephaly syndromes are relatively rare, studying these syndromes can reveal molecular mechanisms that are critical in the regulation of neural progenitor cells, brain size, and human brain evolution. Many of the causative genes for MCPH encode centrosomal proteins involved in centriole biogenesis. However, other MCPH genes fall under different mechanistic categories, notably DNA replication and repair. Recent gene discoveries and functional studies have implicated novel cellular processes, such as cytokinesis, centromere and kinetochore function, transmembrane or intracellular transport, Wnt signaling, and autophagy, as well as the apical polarity complex. Thus, MCPH genes implicate a wide variety of molecular and cellular mechanisms in the regulation of cerebral cortical size during development.

Entities:  

Keywords:  DNA repair; centrosome; microcephaly; radial glial cells

Mesh:

Substances:

Year:  2018        PMID: 29799801     DOI: 10.1146/annurev-genom-083117-021441

Source DB:  PubMed          Journal:  Annu Rev Genomics Hum Genet        ISSN: 1527-8204            Impact factor:   8.929


  81 in total

1.  A missense variant in NUF2, a component of the kinetochore NDC80 complex, causes impaired chromosome segregation and aneuploidy associated with microcephaly and short stature.

Authors:  Daniela Tiaki Uehara; Hiroshi Mitsubuchi; Johji Inazawa
Journal:  Hum Genet       Date:  2021-03-15       Impact factor: 4.132

2.  The C7orf43/TRAPPC14 component links the TRAPPII complex to Rabin8 for preciliary vesicle tethering at the mother centriole during ciliogenesis.

Authors:  Adrian Cuenca; Christine Insinna; Huijie Zhao; Peter John; Matthew A Weiss; Quanlong Lu; Vijay Walia; Suzanne Specht; Selvambigai Manivannan; Jimmy Stauffer; Andrew A Peden; Christopher J Westlake
Journal:  J Biol Chem       Date:  2019-08-29       Impact factor: 5.157

Review 3.  Mendelian neurodegenerative disease genes involved in autophagy.

Authors:  Lidia Wróbel; Sandra Malmgren Hill; Claudia Puri; Sung Min Son; Motoki Fujimaki; Ye Zhu; Eleanna Stamatakou; Farah Siddiqi; Marian Fernandez-Estevez; Marco M Manni; So Jung Park; Julien Villeneuve; David Chaim Rubinsztein
Journal:  Cell Discov       Date:  2020-05-05       Impact factor: 10.849

Review 4.  Spindle positioning and its impact on vertebrate tissue architecture and cell fate.

Authors:  Terry Lechler; Marina Mapelli
Journal:  Nat Rev Mol Cell Biol       Date:  2021-06-22       Impact factor: 94.444

5.  Bi-allelic Pathogenic Variants in TUBGCP2 Cause Microcephaly and Lissencephaly Spectrum Disorders.

Authors:  Tadahiro Mitani; Jaya Punetha; Ibrahim Akalin; Davut Pehlivan; Mateusz Dawidziuk; Zeynep Coban Akdemir; Sarenur Yilmaz; Ezgi Aslan; Jill V Hunter; Hadia Hijazi; Christopher M Grochowski; Shalini N Jhangiani; Ender Karaca; Jawid M Fatih; Piotr Iwanowski; Tomasz Gambin; Pawel Wlasienko; Alicja Goszczanska-Ciuchta; Monika Bekiesinska-Figatowska; Masoumeh Hosseini; Sanaz Arzhangi; Hossein Najmabadi; Jill A Rosenfeld; Haowei Du; Dana Marafi; Susan Blaser; Ronni Teitelbaum; Rachel Silver; Jennifer E Posey; Hans-Hilger Ropers; Richard A Gibbs; Wojciech Wiszniewski; James R Lupski; David Chitayat; Kimia Kahrizi; Pawel Gawlinski
Journal:  Am J Hum Genet       Date:  2019-10-17       Impact factor: 11.025

Review 6.  Mechanism and Regulation of Centriole and Cilium Biogenesis.

Authors:  David K Breslow; Andrew J Holland
Journal:  Annu Rev Biochem       Date:  2019-01-11       Impact factor: 23.643

Review 7.  Using Drosophila to drive the diagnosis and understand the mechanisms of rare human diseases.

Authors:  Nichole Link; Hugo J Bellen
Journal:  Development       Date:  2020-09-28       Impact factor: 6.868

Review 8.  Comprehensive review on the molecular genetics of autosomal recessive primary microcephaly (MCPH).

Authors:  Muhammad Naveed; Syeda Khushbakht Kazmi; Mariyam Amin; Zainab Asif; Ushna Islam; Kinza Shahid; Sana Tehreem
Journal:  Genet Res (Camb)       Date:  2018-08-08       Impact factor: 1.588

9.  Acute Lengthening of Progenitor Mitosis Influences Progeny Fate during Cortical Development in vivo.

Authors:  Aaron Mitchell-Dick; Andrea Chalem; Louis-Jan Pilaz; Debra L Silver
Journal:  Dev Neurosci       Date:  2020-06-15       Impact factor: 2.984

10.  Mutations in ANKLE2, a ZIKA Virus Target, Disrupt an Asymmetric Cell Division Pathway in Drosophila Neuroblasts to Cause Microcephaly.

Authors:  Nichole Link; Hyunglok Chung; Angad Jolly; Marjorie Withers; Burak Tepe; Benjamin R Arenkiel; Priya S Shah; Nevan J Krogan; Hatip Aydin; Bilgen B Geckinli; Tulay Tos; Sedat Isikay; Beyhan Tuysuz; Ganesh H Mochida; Ajay X Thomas; Robin D Clark; Ghayda M Mirzaa; James R Lupski; Hugo J Bellen
Journal:  Dev Cell       Date:  2019-11-14       Impact factor: 12.270

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