2022
DOI: 10.1038/s41598-022-16369-y
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Establishment and characterization of human pluripotent stem cells-derived brain organoids to model cerebellar diseases

Abstract: The establishment of robust human brain organoids to model cerebellar diseases is essential to study new therapeutic strategies for cerebellum-associated disorders. Machado-Joseph disease (MJD) is a cerebellar hereditary neurodegenerative disease, without therapeutic options able to prevent the disease progression. In the present work, control and MJD induced-pluripotent stem cells were used to establish human brain organoids. These organoids were characterized regarding brain development, cell type compositio… Show more

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Cited by 10 publications
(1 citation statement)
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“…Brain region-specific organoid models for neurogenetic disorders Human brain organoid models have been used extensively to study neurogenetic disorders in a human cellular context (Khakipoor et al 2020, Velasco et al 2020. In most cases, cerebral organoid models have been employed, even when the cerebellum was primarily affected by the disorder (Bras et al 2022) since cerebellar differentiation protocols have been developed only recently (Hua et al 2022, Muguruma et al 2015, Nayler et al 2021, Silva et al 2020. Therefore, to date, the use of cerebellar organoids in neurogenetic disease modeling has not been demonstrated.…”
Section: Brain Region-specific Anatomical Hallmarks Recapitulated In ...mentioning
confidence: 99%
“…Brain region-specific organoid models for neurogenetic disorders Human brain organoid models have been used extensively to study neurogenetic disorders in a human cellular context (Khakipoor et al 2020, Velasco et al 2020. In most cases, cerebral organoid models have been employed, even when the cerebellum was primarily affected by the disorder (Bras et al 2022) since cerebellar differentiation protocols have been developed only recently (Hua et al 2022, Muguruma et al 2015, Nayler et al 2021, Silva et al 2020. Therefore, to date, the use of cerebellar organoids in neurogenetic disease modeling has not been demonstrated.…”
Section: Brain Region-specific Anatomical Hallmarks Recapitulated In ...mentioning
confidence: 99%