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@ARTICLE{Buchner:258922,
      author       = {Buchner, Felix and Dokuzluoglu, Zeynep and Grass, Tobias
                      and Rodriguez-Muela, Natalia},
      title        = {{S}pinal {C}ord {O}rganoids to {S}tudy {M}otor {N}euron
                      {D}evelopment and {D}isease.},
      journal      = {Life},
      volume       = {13},
      number       = {6},
      issn         = {2075-1729},
      address      = {Basel},
      publisher    = {MDPI},
      reportid     = {DZNE-2023-00694},
      pages        = {1254},
      year         = {2023},
      abstract     = {Motor 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.},
      subtyp        = {Review Article},
      keywords     = {development (Other) / in vitro disease modeling (Other) /
                      induced pluripotent stem cells (iPSCs) (Other) / motor
                      neuron (MN) (Other) / motor neuron diseases (MNDs) (Other) /
                      organoids (Other) / spinal cord (SpC) (Other) / spinal cord
                      organoids (SCOs) (Other)},
      cin          = {AG Rodriguez-Muela},
      ddc          = {570},
      cid          = {I:(DE-2719)1713001},
      pnm          = {352 - Disease Mechanisms (POF4-352)},
      pid          = {G:(DE-HGF)POF4-352},
      typ          = {PUB:(DE-HGF)16},
      pubmed       = {pmid:37374039},
      pmc          = {pmc:PMC10303776},
      doi          = {10.3390/life13061254},
      url          = {https://pub.dzne.de/record/258922},
}