hemingway is required for sperm flagella assembly and ciliary motility in Drosophila

Area of Science:

• Cell Biology
• Developmental Biology
• Genetics

Background:

• Cilia are vital for physiological processes and development.
• Ciliary dysfunction causes human diseases known as ciliopathies.
• Motile cilia are essential for cell and fluid propulsion, with deficiencies causing primary ciliary dyskinesia.

Purpose of the Study:

• To identify novel components required for motile cilia function.
• To investigate the role of the hemingway (hmw) gene and its protein product in cilia motility.
• To understand the evolutionary conservation and functional significance of HMW in motile cilia.

Main Methods:

• Gene identification and characterization of hemingway (hmw) in Drosophila.
• Analysis of HMW protein structure, including conserved domains.
• Generation and analysis of hmw-knockout Drosophila models.
• Examination of hmw expression patterns in ciliated tissues.
• Assessment of ciliary function in auditory neurons and sperm flagella.

Main Results:

• Identified hemingway (hmw) as a novel gene essential for motile cilia function in Drosophila.
• HMW protein is conserved across species with motile cilia and contains a conserved coiled-coil domain, with a human orthologue KIAA1430.
• hmw knockout flies exhibit hearing impairment and male sterility.
• hmw is crucial for the motility of ciliated auditory sensory neurons and for sperm flagella elongation and maintenance.
• HMW is not a structural component of mature flagella but is required for acquiring motile properties.

Conclusions:

• HMW is a novel, evolutionarily conserved protein necessary for motile cilium function.
• HMW plays a critical role in the assembly and functional acquisition of sperm flagella.
• The findings highlight HMW's importance in ciliary motility and provide insights into ciliopathies.