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000258922 1001_ $$0P:(DE-2719)9001904$$aBuchner, Felix$$b0$$eFirst author$$udzne
000258922 245__ $$aSpinal Cord Organoids to Study Motor Neuron Development and Disease.
000258922 260__ $$aBasel$$bMDPI$$c2023
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000258922 520__ $$aMotor neuron diseases (MNDs) are a heterogeneous group of disorders that affect the cranial and/or spinal motor neurons (spMNs), spinal sensory neurons and the muscular system. Although they have been investigated for decades, we still lack a comprehensive understanding of the underlying molecular mechanisms; and therefore, efficacious therapies are scarce. Model organisms and relatively simple two-dimensional cell culture systems have been instrumental in our current knowledge of neuromuscular disease pathology; however, in the recent years, human 3D in vitro models have transformed the disease-modeling landscape. While cerebral organoids have been pursued the most, interest in spinal cord organoids (SCOs) is now also increasing. Pluripotent stem cell (PSC)-based protocols to generate SpC-like structures, sometimes including the adjacent mesoderm and derived skeletal muscle, are constantly being refined and applied to study early human neuromuscular development and disease. In this review, we outline the evolution of human PSC-derived models for generating spMN and recapitulating SpC development. We also discuss how these models have been applied to exploring the basis of human neurodevelopmental and neurodegenerative diseases. Finally, we provide an overview of the main challenges to overcome in order to generate more physiologically relevant human SpC models and propose some exciting new perspectives.
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000258922 650_7 $$2Other$$adevelopment
000258922 650_7 $$2Other$$ain vitro disease modeling
000258922 650_7 $$2Other$$ainduced pluripotent stem cells (iPSCs)
000258922 650_7 $$2Other$$amotor neuron (MN)
000258922 650_7 $$2Other$$amotor neuron diseases (MNDs)
000258922 650_7 $$2Other$$aorganoids
000258922 650_7 $$2Other$$aspinal cord (SpC)
000258922 650_7 $$2Other$$aspinal cord organoids (SCOs)
000258922 7001_ $$0P:(DE-2719)9002364$$aDokuzluoglu, Zeynep$$b1$$udzne
000258922 7001_ $$0P:(DE-2719)9000564$$aGrass, Tobias$$b2$$udzne
000258922 7001_ $$0P:(DE-2719)9000726$$aRodriguez-Muela, Natalia$$b3$$eLast author$$udzne
000258922 770__ $$aMolecular and Cellular Mechanisms in Neurodegenerative Diseases
000258922 773__ $$0PERI:(DE-600)2662250-6$$a10.3390/life13061254$$gVol. 13, no. 6, p. 1254 -$$n6$$p1254$$tLife$$v13$$x2075-1729$$y2023
000258922 8564_ $$uhttps://www.mdpi.com/2075-1729/13/6/1254
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