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BRAIN Publication-derived

An Organoid-Based Model of Cortical Development Identifies Non-Cell-Autonomous Defects in Wnt Signaling Contributing to Miller-Dieker Syndrome

Source Iefremova et al., 2017 · Institute of Reconstructive Neurobiology, University of Bonn · 10.1016/j.celrep.2017.03.047

👤 Vira Iefremova, George Manikakis, Olivia Krefft, Ammar Jabali, Kevin Weynans, Ruven Wilkens, Fabio Marsoner, Björn Brändl, Franz-Josef Müller, Philipp Koch, Julia Ladewig ⏱ 35 days 📋 3 phases 🧫 Patient-Derived iPSC (Miller-Dieker Syndrome)

Abstract

This protocol generates Miller-Dieker-syndrome-specific iPSC-derived forebrain-type organoids that model cortical development and disease-related defects. The organoids exhibit reduced expansion rates, premature neurogenesis, and disrupted cortical niche architecture due to alterations in the LIS1/NDEL1/14.3.3ε complex. The model reveals non-cell-autonomous defects in Wnt/β-catenin signaling that can be rescued by GSK3β inhibition.

Cell source
Patient-Derived iPSC (Miller-Dieker Syndrome)
Application
Disease modeling

Protocol overview

10 steps across 3 phases

iPSC Dissociation and Embryoid Body Formation Day 1–5
  1. 1 Dissociate iPSCs into single cells
  2. 2 Exchange medium every other day
  3. 3 Transfer embryoid bodies to low-adhesion plates
Neural Induction and Small Molecule Treatment Day 5–12
  1. 4 Maintain neural induction for 5 days with small molecules
  2. 5 Remove small molecules from medium
  3. 6 Embed organoids in Geltrex matrix
Organoid Culture and Maturation Day 12–35+
  1. 7 Culture organoids under continuous agitation
  2. 8 Change medium every 3–4 days
  3. 9 Optional: Add GSK3β inhibitor for β-catenin/Wnt activation
  4. 10 Harvest organoids for analysis

Full SOP

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Attribution

This SOP was authored by Organthis based on the published method in Iefremova et al., 2017. The originating laboratory holds no rights in this SOP and has not endorsed it unless marked Verified.

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