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Woodling, N. S.

Publications and source records attributed to Woodling, N. S..

2 recordsLinked to original sources

Aβ toxicity rescued by protein retention in the ER

Accumulation of A{beta} in the brain is one of the hallmarks of Alzheimers disease (AD). In the adult Drosophila brain, human A{beta} over-expression is toxic and leads to deterioration of climbing ability and shortened lifespan. However, it remains unknown if A{beta} is inherently toxic or if it triggers toxic downstream pathways that lead to neurodegeneration. Here, we describe a novel, and previously unidentified, protective role of intracellular laminin chain accumulation. Despite high A{beta} levels, over-expression of the extracellular matrix protein subunit Laminin B1 (LanB1) resulted in a robust rescue of toxicity, highlighting a potential protective mechanism of resistance to A{beta}. Over-expression of other Laminin subunits and a Collagen IV subunit also significantly rescued A{beta} toxicity, while combining LanB1 with these subunits led to an even larger rescue. Imaging revealed that LanB1 was retained in the ER but had no effect on the secretion of A{beta} into the extracellular milieu. LanB1 rescued toxicity independently of the IRE1/XBP1-mediated branch of the ER stress response. Interestingly, over-expression of ER-targeted GFP also rescued A{beta} toxicity, indicating a potentially broader benefit of ER protein retention. Finally, in proof-of-principle lentiviral transduction experiments using murine organotypic hippocampal slice cultures, over-expression of mouse Lamb1 resulted in ER-retention in transduced cells, highlighting a conserved mechanism. Typically, retention of proteins in the ER is detrimental to cellular health, but in the context of neuronal A{beta} toxicity it may prove to be beneficial and a new therapeutic avenue for AD.

neuroscience↗

Plum is a novel regulator of synaptic function and muscle size in D. melanogaster

Alterations in the neuromuscular system underlie several neuromuscular diseases and play critical roles in the development of sarcopenia, the age-related loss of muscle mass and function. Mammalian Myostatin (MST) and GDF11, members of the TGF-{beta} superfamily of growth factors, are powerful regulators of muscle size in both model organisms and humans. Myoglianin (MYO), the Drosophila homolog of MST and GDF11, is a strong inhibitor of synaptic function and structure at the neuromuscular junction (NMJ), and a negative regulator of body weight and muscle size and function in flies. Here, we identified Plum, a cell surface immunoglobulin homologous to mammalian developmental regulators Protogenin and Nope, as a modulator of MYO function in the larval neuromuscular system. Reduction of Plum specifically in the larval body-wall muscles abolishes the previously demonstrated positive effect of attenuated MYO signalling on both muscle size and neuromuscular junction structure and function, likely by de-sequestrating the remaining MYO. In addition, downregulation of Plum on its own results in decreased synaptic strength and body weight, classifying Plum as a (novel) regulator of neuromuscular function and body (muscle) size. These findings offer new insights into possible regulatory mechanisms behind ageing- and disease-related neuromuscular dysfunctions in humans and identify potential targets for therapeutic interventions.

neuroscience↗