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Ulk4 Is Essential for Ciliogenesis and CSF Flow

Overview of attention for article published in Journal of Neuroscience, July 2016
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Title
Ulk4 Is Essential for Ciliogenesis and CSF Flow
Published in
Journal of Neuroscience, July 2016
DOI 10.1523/jneurosci.0621-16.2016
Pubmed ID
Authors

Min Liu, Zhenlong Guan, Qin Shen, Pierce Lalor, Una Fitzgerald, Timothy O'Brien, Peter Dockery, Sanbing Shen, Liu, Min, Guan, Zhenlong, Shen, Qin, Lalor, Pierce, Fitzgerald, Una, O'Brien, Timothy, Dockery, Peter, Shen, Sanbing

Abstract

Ciliopathies are an emerging class of devastating disorders with pleiotropic symptoms affecting both the central and peripheral systems and commonly associated with hydrocephalus. Even though ciliary components and three master transcriptional regulators have been identified, little is known about the signaling molecules involved. We previously identified a novel gene, Unc51-like-kinase 4 (ULK4), as a risk factor of neurodevelopmental disorders. Here we took multidisciplinary approaches and uncovered essential roles of Ulk4 in ciliogenesis. We show that Ulk4 is predominantly expressed in the ventricular system, and Ulk4(tm1a/tm1a) ependymal cells display reduced/disorganized cilia with abnormal axonemes. Ulk4(tm1a/tm1a) mice exhibit dysfunctional subcommissural organs, obstructive aqueducts, and impaired CSF flow. Mechanistically, we performed whole-genome RNA sequencing and discovered that Ulk4 regulates the Foxj1 pathway specifically and an array of other ciliogenesis molecules. This is the first evidence demonstrating that ULK4 plays a vital role in ciliogenesis and that deficiency of ULK4 can cause hydrocephalus and ciliopathy-related disorders. Ciliopathies are an emerging class of devastating disorders with pleiotropic symptoms affecting both the central and peripheral systems. Ciliopathies are commonly associated with hydrocephalus, and Unc51-like-kinase 4 (Ulk4) has been identified as one of 12 genes causing hydrocephalus in mutants. Here we uncover an essential role of Ulk4 in ciliogenesis. Ulk4 is predominantly expressed in the ventricles, and mutant ependymal cells display reduced/disorganized/nonfunctional motile cilia with abnormal axonemes and impaired CSF flow. Ulk4 modulates expression of the master regulator of ciliogenesis, Foxj1, and other ciliogenesis molecules. This is the first report demonstrating a vital role of Ulk4 in ciliogenesis. ULK4 deficiency may be implicated in human hydrocephalus and other ciliopathy-related disorders.

Twitter Demographics

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Mendeley readers

The data shown below were compiled from readership statistics for 28 Mendeley readers of this research output. Click here to see the associated Mendeley record.

Geographical breakdown

Country Count As %
Spain 1 4%
Unknown 27 96%

Demographic breakdown

Readers by professional status Count As %
Student > Ph. D. Student 9 32%
Researcher 6 21%
Student > Doctoral Student 5 18%
Student > Bachelor 3 11%
Student > Master 1 4%
Other 3 11%
Unknown 1 4%
Readers by discipline Count As %
Agricultural and Biological Sciences 13 46%
Biochemistry, Genetics and Molecular Biology 8 29%
Medicine and Dentistry 3 11%
Engineering 2 7%
Neuroscience 1 4%
Other 0 0%
Unknown 1 4%

Attention Score in Context

This research output has an Altmetric Attention Score of 1. This is our high-level measure of the quality and quantity of online attention that it has received. This Attention Score, as well as the ranking and number of research outputs shown below, was calculated when the research output was last mentioned on 25 July 2016.
All research outputs
#8,791,471
of 11,420,953 outputs
Outputs from Journal of Neuroscience
#14,867
of 16,862 outputs
Outputs of similar age
#171,564
of 265,509 outputs
Outputs of similar age from Journal of Neuroscience
#203
of 259 outputs
Altmetric has tracked 11,420,953 research outputs across all sources so far. This one is in the 19th percentile – i.e., 19% of other outputs scored the same or lower than it.
So far Altmetric has tracked 16,862 research outputs from this source. They typically receive more attention than average, with a mean Attention Score of 9.7. This one is in the 10th percentile – i.e., 10% of its peers scored the same or lower than it.
Older research outputs will score higher simply because they've had more time to accumulate mentions. To account for age we can compare this Altmetric Attention Score to the 265,509 tracked outputs that were published within six weeks on either side of this one in any source. This one is in the 29th percentile – i.e., 29% of its contemporaries scored the same or lower than it.
We're also able to compare this research output to 259 others from the same source and published within six weeks on either side of this one. This one is in the 15th percentile – i.e., 15% of its contemporaries scored the same or lower than it.