{"doi":"10.1101/2020.02.18.951756","title":"Altered patterning of trisomy 21 interneuron progenitors","abstract":"SUMMARY Individuals with Down syndrome (DS, Ts21), the most common genetic cause of intellectual disability, have smaller brains that reflect fewer neurons at pre- and post-natal stages, implicating impaired neurogenesis during development. Our stereological analysis of adult DS cortex indicates a reduction of calretinin expressing interneurons. Using Ts21 human induced pluripotent stem cells (iPSCs) and isogenic controls, we find Ts21 progenitors generate fewer COUP-TFII+ progenitors with reduced proliferation. Single cell RNA-sequencing of Ts21 progenitors confirms the altered specification of progenitor subpopulations and identifies reduced WNT signaling. Activation of WNT signaling partially restores the COUP-TFII+ progenitor population in Ts21, suggesting that altered WNT signaling contributes to the defective development of cortical interneurons in DS.","journal":"bioRxiv (Cold Spring Harbor Laboratory)","year":2020,"id":122583,"datarank":0.0,"base_score":0.0,"endowment":0.0,"self_citation_contribution":0.0,"citation_network_contribution":0.0,"self_endowment_contribution":0.0,"citer_contribution":0.0,"corpus_percentile":null,"corpus_rank":null,"citation_count":4,"citer_count":0,"citers_with_citation_signal":0,"citers_with_endowment":0,"datacite_reuse_total":0,"is_dataset":false,"is_dataset_confidence":0.9526,"is_data_producer":false,"deposit_databanks":null,"is_oa":true,"file_count":0,"downloads":0,"has_version_chain":false,"published_date":"2020-01-01","fair_score":null,"fair_percentile":null,"algorithm_id":"datarank_citation_only_1hop_v6","ranking_scope":"data_only","authors":[{"id":104312,"name":"Jie Sheng","orcid":"0000-0001-8196-8600","position":1,"is_corresponding":false},{"id":565189,"name":"Bennett Strand","orcid":null,"position":2,"is_corresponding":false},{"id":432093,"name":"Ke Xu","orcid":"0000-0002-0621-4507","position":3,"is_corresponding":false},{"id":564258,"name":"Leslie Huang","orcid":"0000-0001-9778-8102","position":4,"is_corresponding":false},{"id":565190,"name":"Margaret Medo","orcid":null,"position":5,"is_corresponding":false},{"id":564259,"name":"Kirstin A. Risgaard","orcid":"0009-0006-8623-1820","position":6,"is_corresponding":false},{"id":565191,"name":"Samuel Dantinne","orcid":null,"position":7,"is_corresponding":false},{"id":565192,"name":"Sruti Mohan","orcid":null,"position":8,"is_corresponding":false},{"id":565193,"name":"Aratrika Keshan","orcid":null,"position":9,"is_corresponding":false},{"id":565194,"name":"Roger A. Daley","orcid":null,"position":10,"is_corresponding":false},{"id":565195,"name":"Bradley Levesque","orcid":null,"position":11,"is_corresponding":false},{"id":565196,"name":"Lindsey Amundson","orcid":null,"position":12,"is_corresponding":false},{"id":298889,"name":"Rebecca Reese","orcid":"0000-0002-4352-1119","position":13,"is_corresponding":false},{"id":499328,"name":"André M. M. Sousa","orcid":"0000-0003-1740-5066","position":14,"is_corresponding":false},{"id":432091,"name":"Yunlong Tao","orcid":"0000-0003-0377-7133","position":15,"is_corresponding":false},{"id":249243,"name":"Daifeng Wang","orcid":"0000-0001-9190-3704","position":16,"is_corresponding":false},{"id":368694,"name":"Su‐Chun Zhang","orcid":"0000-0003-0563-5147","position":17,"is_corresponding":false},{"id":368693,"name":"Anita Bhattacharyya","orcid":"0000-0002-0360-7145","position":18,"is_corresponding":false},{"id":564257,"name":"Yathindar Giffin-Rao","orcid":"0000-0001-7494-0183","position":0,"is_corresponding":true}],"reference_count":84,"raw_metadata":null,"created_at":"2026-07-18T23:14:55.385653Z","pmid":null,"pmcid":null,"fwci":null,"citation_percentile":null,"influential_citations":0,"oa_status":null,"license":null,"views":0,"total_file_size_bytes":0,"version_count":0,"fair_f":null,"fair_a":null,"fair_i":null,"fair_r":null,"fair_zscore":null,"fair_rationale":null,"fair_model":null,"fair_agent_version":null,"fair_fulltext_source":null,"fair_has_llm":null,"fair_computed_at":null,"clinical_trials":[],"software_tools":[],"db_accessions":[],"linked_datasets":[],"topics":[]}