{"doi":"10.70962/jhi.20250143","title":"Minoxidil restores thymic growth in 22q11.2 deletion syndrome by limiting Sox9+ chondrocyte expansion","abstract":"Thymic hypoplasia, hypoparathyroidism, and cardiac defects are common congenital malformations caused by 22q11.2 deletion syndrome (22q11.2DS; aka DiGeorge syndrome). Thymus hypoplasia reduces peripheral T cell numbers, leading to more frequent infections. We report that embryonic hypoplastic thymuses from mouse models of 22q11.2DS (Tbx1neo2/neo2) have distinct mesenchymal cell subsets, including an expansion of Sox9+ chondrocytes. Chondrocytes produce collagens and extracellular matrix (ECM) proteins, which can affect thymus size and vascularization. Two compounds, minoxidil and PGE2, restored growth for Tbx1neo2/neo2 embryonic thymuses when administered to pregnant mice prior to formation of the thymic anlage. The dysregulation of the mesenchymal and endothelial transcriptomes was corrected with minoxidil in Tbx1neo2/neo2 thymuses. This was confirmed by the diminished expression of Sox9-driven type II, IX, and XI cartilaginous collagens and other ECM proteins. Furthermore, the location of parathyroids was corrected in Tbx1neo2/neo2 embryos. In summary, these findings reveal that targeting prenatal mesenchymal differentiation can correct multiple congenital anomalies in mouse models of 22q11.2DS.","journal":"Journal of Human Immunity","year":2025,"id":542048,"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":2,"citer_count":0,"citers_with_citation_signal":0,"citers_with_endowment":0,"datacite_reuse_total":0,"is_dataset":false,"is_dataset_confidence":0.9512,"is_data_producer":false,"deposit_databanks":null,"is_oa":true,"file_count":0,"downloads":0,"has_version_chain":false,"published_date":"2025-01-01","fair_score":null,"fair_percentile":null,"algorithm_id":"datarank_citation_only_1hop_v6","ranking_scope":"data_only","authors":[{"id":919390,"name":"Neha Ahuja","orcid":"0000-0003-0750-3383","position":1,"is_corresponding":false},{"id":297436,"name":"Ashwani Kumar","orcid":"0000-0002-6374-0976","position":2,"is_corresponding":false},{"id":240348,"name":"Chao Xing","orcid":"0000-0002-1838-0502","position":3,"is_corresponding":false},{"id":917792,"name":"Angela Moses","orcid":"0009-0009-3211-4356","position":4,"is_corresponding":false},{"id":314063,"name":"Ashutosh Shukla","orcid":"0000-0002-7851-8817","position":5,"is_corresponding":false},{"id":1083416,"name":"Katelyn Boetel","orcid":"0009-0000-9951-5554","position":6,"is_corresponding":false},{"id":285068,"name":"Bret M. Evers","orcid":"0000-0001-5686-0315","position":7,"is_corresponding":false},{"id":255299,"name":"John M. Shelton","orcid":"0000-0002-2949-1638","position":8,"is_corresponding":false},{"id":1431170,"name":"Maria Teresa de la Morena","orcid":null,"position":9,"is_corresponding":false},{"id":493927,"name":"Christian Wysocki","orcid":"0000-0001-8035-8165","position":10,"is_corresponding":false},{"id":245970,"name":"Ondine Cleaver","orcid":"0000-0003-2454-6641","position":11,"is_corresponding":false},{"id":274439,"name":"Nicolai S. C. van Oers","orcid":"0000-0002-6545-8405","position":12,"is_corresponding":false},{"id":493926,"name":"Pratibha Bhalla","orcid":"0000-0001-6015-996X","position":0,"is_corresponding":true}],"reference_count":99,"raw_metadata":null,"created_at":"2026-07-19T02:52:51.593043Z","pmid":"41607489","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":[]}