High-content analysis of neuronal morphology in human iPSC-derived neurons

Lickfett, Selene; Menacho, Carmen; Zink, Annika; Telugu, Narasimha; Beller, Mathias; Diecke, Sebastian; Cambridge, Sidney B.; Prigione, Alessandro

doi:10.1016/j.xpro.2022.101567

Cited by 11 publications

(11 citation statements)

References 7 publications

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“… For more information on how to use CellProfiler, please refer to publicly-available online tutorials ( https://cellprofiler.org/tutorials ) and step-by-step protocols. 3 , 4 , 5 …”

Section: Step-by-step Methods Detailsmentioning

confidence: 99%

Protocol for neurogenin-2-mediated induction of human stem cell-derived neural progenitor cells

Guss,

Sathe,

Dai

et al. 2024

STAR Protocols

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“… For more information on how to use CellProfiler, please refer to publicly-available online tutorials ( https://cellprofiler.org/tutorials ) and step-by-step protocols. 3 , 4 , 5 …”

Section: Step-by-step Methods Detailsmentioning

confidence: 99%

Protocol for neurogenin-2-mediated induction of human stem cell-derived neural progenitor cells

Guss,

Sathe,

Dai

et al. 2024

STAR Protocols

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“…The image analysis presented in this protocol is performed using Harmony Perkin Elmer, a licensed image analysis software. However, similar image analysis measuring neurite outgrowth can be performed using open source image analysis software’s such as Cell Profiler 6 and ImageJ with NeuronJ PlugIn. 7 Both Cell Profiler and ImageJ support multiple image file formats such as JPEG and TIFF.…”

Section: Limitationsmentioning

confidence: 99%

Generation of human-induced pluripotent-stem-cell-derived cortical neurons for high-throughput imaging of neurite morphology and neuron maturation

Wali

Abu-Bonsrah

et al. 2023

STAR Protocols

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“…Therefore, we focused on neuronal outgrowth capacity of NGN2 neurons using our highcontent microscopy based platform (Fig. 6h) 43 . In agreement with the known role of BDNF in neuronal branching 44 , the downregulation of BDNF in NGN2 neurons impaired the development of axonal and dendritic structures (Fig.…”

Section: Cell-autonomous Bioenergetic Impairment In Pure Neurons From...mentioning

confidence: 99%

“…The cells were centrifuged at 100 x g for 5 min and resuspended in neuronal medium. We quantified neuronal branching as we previously described 43 . Briefly, 5,000 neurons were seeded per well to the 384-well plate and cultured for 4 days.…”

Section: Sirna-based Knock-down Experiments and Branching Analysismentioning

confidence: 99%

Mutant Huntingtin impairs neurodevelopment in human brain organoids through CHCHD2-mediated neurometabolic failure

Lisowski

Lickfett

Rybak-Wolf

et al. 2023

Preprint

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Expansion of the glutamine tract (poly-Q) in the protein Huntingtin (HTT) causes the neurodegenerative disorder Huntington disease (HD). Emerging evidence suggests that mutant HTT (mHTT) disrupts brain development. To gain mechanistic insights into the neurodevelopmental impact of human mHTT, we engineered induced pluripotent stem cells to introduce a biallelic or monoallelic mutant 70Q expansion or to remove the poly-Q tract of HTT. 70Q introduction caused aberrant development of cerebral organoids with loss of neural progenitor organization. The early neurodevelopmental signature of mHTT highlighted the dysregulation of the protein coiled-coil-helix-coiled-coil-helix domain containing 2 (CHCHD2), a transcription factor involved in mitochondrial integrated stress response. CHCHD2 repression was associated with abnormal mitochondrial morpho-dynamics and elevated resting energy expenditure. Elimination of the poly-Q tract of HTT normalized CHCHD2 expression and mitochondrial defects. Hence, mHTT-mediated disruption of human neurodevelopment is paralleled by aberrant neurometabolic programming mediated by dysregulation of CHCHD2, which could then serve as an early intervention target for HD.

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High-content analysis of neuronal morphology in human iPSC-derived neurons

Cited by 11 publications

References 7 publications

Protocol for neurogenin-2-mediated induction of human stem cell-derived neural progenitor cells

Protocol for neurogenin-2-mediated induction of human stem cell-derived neural progenitor cells

Generation of human-induced pluripotent-stem-cell-derived cortical neurons for high-throughput imaging of neurite morphology and neuron maturation

Mutant Huntingtin impairs neurodevelopment in human brain organoids through CHCHD2-mediated neurometabolic failure

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