Zebrafish Class 1 Phosphatidylinositol Transfer Proteins: PITPβ and Double Cone Cell Outer Segment Integrity in Retina

Ile, Kristina E.; Kassen, Sean C.; Cao, Canhong; Thomas, Vihtehlic,; Shah, Sweety D.; Mousley, Carl J.; Alb, James G.; Huijbregts, Richard P. H.; Stearns, George; Brockerhoff, Susan E.; Hyde, David R.; Bankaitis, Vytas A.

doi:10.1111/j.1600-0854.2010.01085.x

Cited by 54 publications

(51 citation statements)

References 52 publications

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“…The monoclonal antibody zpr1 recognizes Arrestin-3 on the surface of the red-green double cones (Ile et al, 2010; Larison and Bremiller, 1990). In both wild type and myo7aa −/− retinas, zpr1 labeled the entire cell bodies of the red-green double cones, and robust staining was observed across the entire ONL (Fig.…”

Section: Resultsmentioning

confidence: 99%

myosin 7aa−/− mutant zebrafish show mild photoreceptor degeneration and reduced electroretinographic responses

Wasfy

Matsui

Miller

et al. 2014

Experimental Eye Research

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Mutations in myosin VIIa (MYO7A) cause Usher syndrome 1B (USH1B), a disease characterized by the combination of sensorineural hearing loss and visual impairment termed retinitis pigmentosa (RP). Although the shaker-1 mouse model of USH1B exists, only minor defects in the retina have been observed during its lifespan. Previous studies of the zebrafish mariner mutant, which also carries a mutation in myo7aa, revealed balance and hearing defects in the mutants but the retinal phenotype has not been described. We found elevated cell death in the outer nuclear layer (ONL) of myo7aa−/− mutants. While myo7aa−/− mutants retained visual behaviors in the optokinetic reflex (OKR) assay, electroretinogram (ERG) recordings revealed a significant decrease in both a- and b-wave amplitudes in mutant animals, but not a change in ERG threshold sensitivity. Immunohistochemistry showed mislocalization of rod and blue cone opsins and reduced expression of rod-specific markers in the myo7aa−/− ONL, providing further evidence that the photoreceptor degeneration observed represents the initial stages of the RP. Further, constant light exposure resulted in widespread photoreceptor degeneration and the appearance of large holes in the retinal pigment epithelium (RPE). No differences were observed in the retinomotor movements of the photoreceptors or in melanosome migration within the RPE, suggesting that myo7aa−/− does not function in these processes in teleosts. These results indicate that the zebrafish myo7aa−/− mutant is a useful animal model for the RP seen in humans with USH1B.

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Section: Resultsmentioning

confidence: 99%

myosin 7aa−/− mutant zebrafish show mild photoreceptor degeneration and reduced electroretinographic responses

Wasfy

Matsui

Miller

et al. 2014

Experimental Eye Research

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“…This defect purportedly comes without compromise of anterograde ER to Golgi trafficking -- a curious result given retrograde transport is essential for recycling of v-SNARES for anterograde ER to Golgi transport. With regard to vertebrate PITPβ, zebrafish with strongly reduced PITPβ levels develop normally, although these exhibit defects in outer segment biogenesis and/or maintenance in double cone photoreceptor cells [70]. These data argue against housekeeping roles for PITPβ in retrograde Golgi to ER trafficking, although such functions might be important in specialized contexts that require high-capacity membrane flux.…”

Section: Metazoan Lipid Transfer Proteins and The Golgi Systemmentioning

confidence: 99%

Golgi Membrane Dynamics and Lipid Metabolism

2012

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Summary The striking morphology of the Golgi complex has fascinated cell biologists since its discovery over 100 years ago. Yet, despite intense efforts to understand how membrane flow relates to Golgi form and function, this organelle continues to baffle cell biologists and biochemists alike. Fundamental questions regarding Golgi function, while hotly debated, remain unresolved. While Golgi function is historically described from a protein-centric point of view, we now appreciate that conceptual frameworks for how lipid metabolism is integrated with Golgi biogenesis and function are essential for a mechanistic understanding of this fascinating organelle. It is from a lipid-centric perspective that we discuss the larger question of Golgi dynamics and membrane trafficking. We review the growing body of evidence for how lipid metabolism is integrally written into the engineering of the Golgi system, and highlight questions for future study.

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“…At the cellular level, PITP␣ was shown to associate with the netrin receptor and to play a role in neurite outgrowth (1747). Morpholino-mediated knockdown of PITP␣ in zebrafish causes early developmental arrest (688). In Drosophila, mutation of the single class I PITP Giotto causes defects in male germline cytokenesis (500, 508), with a phenotype, similar to the defect in the PI4KB homolog fwd (182).…”

Section: A Class I Pitpsmentioning

confidence: 99%

“…The zebrafish homolog PITP␤ is required for proper development of double cone cell outer segments in the retina (688). Knockdown of PITP␤ in HeLa cells causes altered Golgi morphology and distorted nuclei and a retrograde transport defect between the Golgi and the ER (219).…”

Section: A Class I Pitpsmentioning

confidence: 99%

Phosphoinositides: Tiny Lipids With Giant Impact on Cell Regulation

Balla

2013

Physiological Reviews

1,365

1,655

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Phosphoinositides (PIs) make up only a small fraction of cellular phospholipids, yet they control almost all aspects of a cell's life and death. These lipids gained tremendous research interest as plasma membrane signaling molecules when discovered in the 1970s and 1980s. Research in the last 15 years has added a wide range of biological processes regulated by PIs, turning these lipids into one of the most universal signaling entities in eukaryotic cells. PIs control organelle biology by regulating vesicular trafficking, but they also modulate lipid distribution and metabolism via their close relationship with lipid transfer proteins. PIs regulate ion channels, pumps, and transporters and control both endocytic and exocytic processes. The nuclear phosphoinositides have grown from being an epiphenomenon to a research area of its own. As expected from such pleiotropic regulators, derangements of phosphoinositide metabolism are responsible for a number of human diseases ranging from rare genetic disorders to the most common ones such as cancer, obesity, and diabetes. Moreover, it is increasingly evident that a number of infectious agents hijack the PI regulatory systems of host cells for their intracellular movements, replication, and assembly. As a result, PI converting enzymes began to be noticed by pharmaceutical companies as potential therapeutic targets. This review is an attempt to give an overview of this enormous research field focusing on major developments in diverse areas of basic science linked to cellular physiology and disease.

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Zebrafish Class 1 Phosphatidylinositol Transfer Proteins: PITPβ and Double Cone Cell Outer Segment Integrity in Retina

Cited by 54 publications

References 52 publications

myosin 7aa−/− mutant zebrafish show mild photoreceptor degeneration and reduced electroretinographic responses

myosin 7aa−/− mutant zebrafish show mild photoreceptor degeneration and reduced electroretinographic responses

Golgi Membrane Dynamics and Lipid Metabolism

Phosphoinositides: Tiny Lipids With Giant Impact on Cell Regulation

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