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MCD New-Approach-Methodology (NAM) paper map

Curator reference for the malformation-of-cortical-development (MCD) curation cluster seeded from:

Romero DM, Bahi-Buisson N, Francis F. Genetics and mechanisms leading to human cortical malformations. Seminars in Cell & Developmental Biology 76:33–75 (2018). DOI: 10.1016/j.semcdb.2017.09.031

Tracks issue #4100 ("capture iPSC/organoid NAM evidence and modeling patterns for cortical malformation curation"), itself part of the cortical-malformation epic #4098.

Purpose

The review's Section 4 treats stem-cell / organoid systems (NAMs) as the primary tools for studying human-specific cortical-development mechanisms that rodent models miss (most notably basal/outer radial glia, bRG/oRG). This file maps the NAM papers behind that section to verified PubMed identifiers, so that every MCD entry and module in the cluster cites the same, identity-checked references with consistent evidence_source handling.

How this map was built (anti-hallucination methodology)

Per the CLAUDE.md deep-research SOP, review-summary reference numbers and recalled PMIDs are treated as leads, not ground truth. Every PMID below was:

  1. fetched with just fetch-reference PMID:<id> (never hand-created), and
  2. confirmed by reading the cached title / journal / year in references_cache/PMID_<id>.md against the paper the review describes.

Where the issue thread named a specific paper for a Romero reference bracket (e.g. [298] = Bershteyn; [299] = Iefremova; [278] = KATNB1), the bracket is reproduced from the issue. For clusters the issue gives only as a range (e.g. ZIKV [294–297, 309–311]), papers are assigned to the cluster, not to a single bracket number, because the Romero reference list was not independently available to pin exact numbers — the paper identity is verified from the cache, the exact bracket index is not. Unverified bracket→PMID guesses are listed under "Not yet mapped" rather than asserted.

Core curation rule (from #4100)

NAM data — patient-derived iPSC, hESC-derived neural rosettes, neurospheres, cerebral/forebrain organoids, organotypic fetal slices, single-cell profiling of organoids — is mechanistic evidence, classified IN_VITRO, not HUMAN_CLINICAL. Always name the model system in the evidence explanation (e.g. "patient-derived iPSC forebrain organoid", "human cerebral organoid single-cell profiling"). Use the HUMAN_MODEL_MISMATCH discussion kind (not a generic KNOWLEDGE_GAP) when the point is that human organoid/fetal data reveal biology absent or weak in rodent models — the canonical example being bRG/oRG findings.


NAM platform / foundational methods (Romero cluster [282–284, 293] and [288–291])

These establish the platforms themselves; cite them when a node's evidence rests on the validity of the model system rather than a disease-specific finding.

PMID Verified title (from cache) First author / journal / year NAM platform Cited by (MCD cluster)
16904174 Induction of pluripotent stem cells from mouse embryonic and adult fibroblast cultures by defined factors. Takahashi K et al., Cell, 2006 iPSC reprogramming (foundational) foundational platform reference (fetch-reference-verified; not yet cited in the cluster)
18035408 Induction of pluripotent stem cells from adult human fibroblasts by defined factors. Takahashi K et al., Cell, 2007 human iPSC reprogramming (foundational) foundational platform reference (fetch-reference-verified; not yet cited in the cluster)
23995685 Cerebral organoids model human brain development and microcephaly. Lancaster MA et al., Nature, 2013 cerebral organoid corticogenesis already in repo cache; cited by microtubule_dependent_neuronal_migration_failure, apical_neuroependyma_integrity_failure, pial_basement_membrane_radial_glial_endfoot_failure, reelin_terminal_translocation_lamination_failure
26644564 Human cerebral organoids recapitulate gene expression programs of fetal neocortex development. Camp JG et al., Proc Natl Acad Sci U S A, 2015 cerebral organoid single-cell corticogenesis validation already in repo cache (IN_VITRO); cited by microtubule_dependent_neuronal_migration_failure

Lissencephaly / Miller-Dieker (MDS) organoid models (Romero [298], [299])

PMID Verified title (from cache) First author / journal / year NAM finding Romero ref Cited by (MCD cluster)
28111201 Human iPSC-Derived Cerebral Organoids Model Cellular Features of Lissencephaly and Reveal Prolonged Mitosis of Outer Radial Glia. Bershteyn M et al., Cell Stem Cell, 2017 MDS patient iPSC organoids: neuroepithelial apoptosis, reduced size, altered cleavage angle, defective radial migration rescued by chromosome-17 compensation; prolonged oRG mitosis (human-specific, absent in mouse) [298] neural_progenitor_centrosome_spindle_dysfunction, microtubule_dependent_neuronal_migration_failure, apical_neuroependyma_integrity_failure, pial_basement_membrane_radial_glial_endfoot_failure, reelin_terminal_translocation_lamination_failure, interneuron_specification_tangential_migration_failure, KATNB1-related_Cortical_Malformation, TUBA1A-related_Tubulinopathy, TUBB_TUBB5-related_Microcephaly, NDE1-related_Microcephaly_Lissencephaly
28380362 An Organoid-Based Model of Cortical Development Identifies Non-Cell-Autonomous Defects in Wnt Signaling Contributing to Miller-Dieker Syndrome. Iefremova V et al., Cell Rep, 2017 MDS forebrain organoids: premature neurogenesis, symmetric→asymmetric apical RGC division switch, altered N-cadherin/β-catenin/Wnt; rescue by Wnt activation [299] neural_progenitor_centrosome_spindle_dysfunction

bRG/oRG note: 28111201's prolonged-oRG-mitosis finding is the cluster's clearest case of a NAM result that disambiguates an entry boundary — oRG biology is a LIS1/MDS-specific feature not shared by DCX or most other LIS genes, and is not reproduced in mouse. Model it with a HUMAN_MODEL_MISMATCH discussion where it bears on lump/split decisions.


Centrosome / spindle progenitor models (Romero [278])

PMID Verified title (from cache) First author / journal / year NAM finding Romero ref Cited by (MCD cluster)
25521378 Mutations in KATNB1 cause complex cerebral malformations by disrupting asymmetrically dividing neural progenitors. Mishra-Gorur K et al., Neuron, 2014 KATNB1 patient-derived cells / progenitor models: disrupted asymmetric progenitor division, defective neuronal production and migration [278] neural_progenitor_centrosome_spindle_dysfunction, KATNB1-related_Cortical_Malformation, Autosomal_Recessive_Primary_Microcephaly

ZIKV neural-progenitor / organoid models (Romero cluster [294–297, 309–311])

These are the primary mechanistic evidence for the viral progenitor-cytopathy pathway (kb/modules/viral_neural_progenitor_cytopathy.yaml, #4079) and the Congenital Zika Syndrome entry (#4088). All are IN_VITRO except where noted.

PMID Verified title (from cache) First author / journal / year NAM finding Cited by (MCD cluster)
26952870 Zika Virus Infects Human Cortical Neural Progenitors and Attenuates Their Growth. Tang H et al., Cell Stem Cell, 2016 human iPSC-derived NPCs (hNPCs) are direct ZIKV targets; increased cell death, cell-cycle dysregulation, attenuated growth viral_neural_progenitor_cytopathy, Congenital_Zika_Syndrome
27064148 Zika virus impairs growth in human neurospheres and brain organoids. Garcez PP et al., Science, 2016 reduced viability/growth of human neurospheres and brain organoids → abrogated neurogenesis viral_neural_progenitor_cytopathy, Congenital_Zika_Syndrome
27038591 Expression Analysis Highlights AXL as a Candidate Zika Virus Entry Receptor in Neural Stem Cells. Nowakowski TJ et al., Cell Stem Cell, 2016 single-cell expression: candidate entry receptor AXL enriched on human radial glia / astrocytes / endothelium / microglia viral_neural_progenitor_cytopathy, Congenital_Zika_Syndrome
27162029 Zika Virus Depletes Neural Progenitors in Human Cerebral Organoids through Activation of the Innate Immune Receptor TLR3. Dang J et al., Cell Stem Cell, 2016 hESC-derived cerebral organoids: TLR3 upregulation, perturbed cell fate, reduced organoid volume; TLR3 inhibition partially rescues viral_neural_progenitor_cytopathy, Congenital_Zika_Syndrome
27568284 Zika Virus Disrupts Phospho-TBK1 Localization and Mitosis in Human Neuroepithelial Stem Cells and Radial Glia. Onorati M et al., Cell Rep, 2016 human neuroepithelial stem cells / radial glia: pTBK1 mislocalization, disrupted centrosome/mitosis neural_progenitor_centrosome_spindle_dysfunction, viral_neural_progenitor_cytopathy, Congenital_Zika_Syndrome
28132835 Recent Zika Virus Isolates Induce Premature Differentiation of Neural Progenitors in Human Brain Organoids. Gabriel E et al., Cell Stem Cell, 2017 human brain organoids: centrosome perturbation, premature progenitor differentiation → progenitor depletion / cortical thinning neural_progenitor_centrosome_spindle_dysfunction, viral_neural_progenitor_cytopathy, Congenital_Zika_Syndrome
27279226 The Brazilian Zika virus strain causes birth defects in experimental models. Cugola FR et al., Nature, 2016 mixed-model: human organoids and mouse / non-human-primate models. Split evidence items so the organoid arm is IN_VITRO and the in-vivo arm is MODEL_ORGANISM. viral_neural_progenitor_cytopathy, Congenital_Zika_Syndrome

Adjacent ZIKV references that are not NAMs (boundary check)

These appear in the same ZIKV evidence base but must not be tagged IN_VITRO — they are listed here so curators don't misclassify them when reusing the cluster's citations.

PMID Verified title (from cache) First author / journal / year Correct evidence_source Why
27179424 Zika Virus Disrupts Neural Progenitor Development and Leads to Microcephaly in Mice. Li C et al., Cell Stem Cell, 2016 MODEL_ORGANISM in vivo mouse model (despite the journal), not a stem-cell/organoid system
26862926 Zika Virus Associated with Microcephaly. Mlakar J et al., N Engl J Med, 2016 HUMAN_CLINICAL human fetal autopsy case, not an in-vitro model
24388750 Microcephaly-associated protein WDR62 regulates neurogenesis through JNK1 in the developing neocortex. Xu D et al., Cell Rep, 2014 MODEL_ORGANISM in vivo developing-neocortex study; cited in MCPH/centrosome context but not a NAM

Rescue-branch modeling reminder

Where an organoid experiment reverses a phenotype, model the rescue as mechanistic evidence on the perturbed causal branch, not as a treatment, unless separate clinical evidence exists:

  • chromosome-17 compensation rescues bRG mitotic delay — Bershteyn 28111201
  • Wnt activation rescues premature neurogenesis in MDS organoids — Iefremova 28380362
  • TLR3 inhibition reduces ZIKV-induced organoid volume loss — Dang 27162029

Not yet mapped (open follow-ups for this issue)

The following Romero clusters from #4100 are not asserted here because no single PMID could be identity-verified against the cache in this pass. Resolve each with just fetch-reference + cached-title confirmation before citing:

  • Neural rosette / neural-precursor model papers [285–287].
  • ~~The remaining individual cerebral-organoid corticogenesis / single-cell validation papers in [288–291] beyond Lancaster 23995685 (e.g. organoid single-cell transcriptomic validation of corticogenesis pathways).~~ Partially resolved (2026-06-30): the canonical single-cell organoid corticogenesis-validation paper — Camp JG et al. 2015 (PMID:26644564, Human cerebral organoids recapitulate gene expression programs of fetal neocortex development) — was already cache-verified and already cited (IN_VITRO) in microtubule_dependent_neuronal_migration_failure; it is now recorded in the platform table above. Any further [288–291] organoid validation papers beyond Lancaster 23995685 and Camp 26644564 remain open.
  • Any additional ZIKV [309–311] papers (e.g. further AXL / Sofosbuvir rescue reports) beyond the seven verified above.

When these are verified, append them to the relevant table above with the same columns and the same cached-title-confirmation standard.