Morphogen-guided neocortical organoids with anteroposterior areal identity

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Authors: Yuan‐Chen Tsai, Hajime Ozaki, Axel A. Almet, Isabel Fleming, Caihao Nie, Songhao Luo, Xinyi Wang, Kaori Shiraiwa, Matthew Jung Min Noh, Sunnyana Trejo, Bret Kiyoshi Sugita, Jiya Dalal
+7 more Ruben Alberto Gonzalez, Briana De Jesus, Nate Branco, Gregory Li-Min Chen, Michael J. Gandal, Qing Nie, Momoko Watanabe
Cited by: 1
References: 66

Abstract

The human neocortex exhibits characteristic regional patterning (arealization) critical for higher-order cognitive function. Disrupted arealization is implicated in neurodevelopmental disorders (NDDs), but current neocortical organoid models largely fail to recapitulate this patterning, limiting mechanistic understanding. We establish a straightforward method for generating arealized organoids through short-term early exposure to anterior or posterior morphogens. These treatments created distinct anteroposterior (AP) signaling centers, supporting long-lasting polarization validated by spatial and single-cell RNA sequencing, which revealed area-specific molecular signatures matching the prenatal human cortex. To demonstrate utility, we modeled fragile X syndrome (FXS) in organoids with distinct AP identities. FXS organoids showed disrupted expression gradients along the AP axis, consistent with alterations in autism spectrum disorder, demonstrating how regional patterning defects may contribute to NDD pathology. Together, our study provides a robust platform for generating neocortical organoids with AP molecular signatures and highlights the importance of modeling NDDs using experimental platforms with neuroanatomic specificity.

Fields of Study & Subjects

Developmental Biology and Gene Regulation Teratomas and Epidermoid Cysts Chromatin Remodeling and Cancer

Publication details

Journal / source
Cell stem cell
Publisher
Elsevier BV
Volume
33
Issue
9
Pages
1540–1552.e9
Publication date
20 August 2026
Language
EN