Alms1 mutant zebrafish do not show hair cell phenotypes seen in other cilia mutants

Multiple cilia-associated genes have been shown to affect hair cells in zebrafish (Danio rerio), including the human deafness gene dcdc2, the radial spoke gene rsph9, and multiple intraflagellar transport (IFT) and transition zone genes. Recently a zebrafish alms1 mutant was generated. The ALMS1 gene is the gene mutated in the ciliopathy Alström Syndrome a disease that causes hearing loss among other symptoms. The hearing loss seen in Alström Syndrome may be due in part to hair cell defects as Alms1 mutant mice show stereocilia polarity defects and a loss of hair cells. Hair cell loss is also seen in postmortem analysis of Alström patients. The zebrafish alms1 mutant has metabolic defects similar to those seen in Alström syndrome and Alms1 mutant mice. We wished to investigate if it also had hair cell defects. We, however, failed to find any hair cell related phenotypes in alms1 mutant zebrafish. They had normal lateral line hair cell numbers as both larvae and adults and normal kinocilia formation. They also showed grossly normal swimming behavior, response to vibrational stimuli, and FM1-43 loading. Mutants also showed a normal degree of sensitivity to both short-term neomycin and long-term gentamicin treatment. These results indicate that cilia-associated genes differentially affect different hair cell types.

Title	Alms1 mutant zebrafish do not show hair cell phenotypes seen in other cilia mutants
Creator	Stawicki, Tamara M.
Creator	Parkinson, L.
Publisher	PLoS ONE
Academic Department	Neuroscience
Division	Natural Sciences
Organization	Lafayette College
Date Issued	April 1, 2021
Date Available	2021-04-05
Type	Article
Language	English
Bibliographic Citation	Parkinson, L. and T. M. Stawicki (2021 Apr 1) "Alms1 mutant zebrafish do not show hair cell phenotypes seen in other cilia mutants." PLoS ONE 16 (4): e0246844.
Standard Identifier	Handle 10385/3484zj09c
Standard Identifier	DOI 10.1371/journal.pone.0246844 A
Permalink	http://hdl.handle.net/10385/3484zj09c
Rights Statement	Creative Commons - Attribution
Rights Holders	Stawicki, Tamara M.
Rights Holders	Parkinson, L.