Trial reportPloS one2012
Human FOXN1-deficiency is associated with αβ double-negative and FoxP3+ T-cell expansions that are distinctly modulated upon thymic transplantation.
Trial report in PloS one, 2012. The graph could read no effect estimate from its abstract, so it casts no vote on the map. It is linked to 4 registered trials, which are not on this map. Cited by 22 papers.
What it found
Each row is one number read from the abstract, on the scale the paper reported it, with its interval. Left of the dashed line favours the treatment, right favours the comparator. Under each row is the sentence it came from. New to these charts? A ten-minute tutorial.
The abstract states no effect estimate the extractor could read, or names no intervention and outcome on the map, so this paper lights no cell and moves no belief. It is still indexed, cited and linked below.
The trial behind it
Trials whose registry record cites this paper, or whose number appears in the abstract. A trial that started after this paper was published is citing it as background, not reporting it.
Thymus Transplantation With Immunosuppression, #884
Safety and Efficacy of Thymus Transplantation in Complete DiGeorge Anomaly, IND#9836
Expanded Access Protocol Thymus Transplantation for Immunodeficiency, Hematologic Malignancies, and Autoimmune Disease Related to Poor Thymic Function
A First-in-Humans Supermicrosurgical Technique for the Neonatal Thymus Transplantation
Who cites it
22 citing papers in PubMed, 39 citations in OpenAlex.
- Congenital Athymia: Unmet Needs and Practical Guidance.Therapeutics and clinical risk management · 2023Review
- Congenital Athymia: Genetic Etiologies, Clinical Manifestations, Diagnosis, and Treatment.Journal of clinical immunology · 2021Review
- Current and Future Therapeutic Approaches for Thymic Stromal Cell Defects.Frontiers in immunology · 2021Review
- T-Cell Immunodeficiencies With Congenital Alterations of Thymic Development: Genes Implicated and Differential Immunological and Clinical Features.Frontiers in immunology · 2020Review
- Heterozygous FOXN1 Variants Cause Low TRECs and Severe T Cell Lymphopenia, Revealing a Crucial Role of FOXN1 in Supporting Early Thymopoiesis.American journal of human genetics · 2019Article
- Epstein-Barr virus associated with high-grade B-cell lymphoma in nude severe combined immunodeficiency.BMJ case reports · 2019Article
- Athymic mice reveal a requirement for T-cell-microglia interactions in establishing a microenvironment supportive ofGenes & development · 2018Article
- FOXN1 Deficiency: from the Discovery to Novel Therapeutic Approaches.Journal of clinical immunology · 2017Review
- FOXN1 deficient nude severe combined immunodeficiency.Orphanet journal of rare diseases · 2017Review
- IL-7-Induced Proliferation of Human Naive CD4 T-Cells Relies on Continued Thymic Activity.Frontiers in immunology · 2017Article
- Restoration of Thymus Function with Bioengineered Thymus Organoids.Current stem cell reports · 2016Article
- Human naïve regulatory T-cells feature high steady-state turnover and are maintained by IL-7.Oncotarget · 2016Article
- Establishment and Maintenance of the Human Naïve CD4Frontiers in pediatrics · 2016Review
- Isolation and transplantation of different aged murine thymic grafts.Journal of visualized experiments : JoVE · 2015Article
- Programmed cell death 1 and Helios distinguish TCR-αβ+ double-negative (CD4-CD8-) T cells that derive from self-reactive CD8 T cells.Journal of immunology (Baltimore, Md. : 1950) · 2015Article
- Regulatory T-Cell Development in the Human Thymus.Frontiers in immunology · 2015Review
- Reduced BAFF-R and increased TACI expression in common variable immunodeficiency.Journal of clinical immunology · 2014Article
- Thymus involution and regeneration: two sides of the same coin?Nature reviews. Immunology · 2013Review
- Thymus transplantation restores the repertoires of forkhead box protein 3 (FoxP3)+ and FoxP3- T cells in complete DiGeorge anomaly.Clinical and experimental immunology · 2013Article
- Molecular evidence for a thymus-independent partial T cell development in a FOXN1-/- athymic human fetus.PloS one · 2013Article
Corrections and comments
PubMed lists nothing against this paper. Absence here is not a guarantee, only a check that was made.
Authors and funding
11 authors at 7 institutions in 4 countries.
Funding
No grant is acknowledged in the PubMed record.
Abstract
Forkhead box N1 (FOXN1) is a transcription factor crucial for thymic epithelium development and prevention of its involution. Investigation of a patient with a rare homozygous FOXN1 mutation (R255X), leading to alopecia universalis and thymus aplasia, unexpectedly revealed non-maternal circulating T-cells, and, strikingly, large numbers of aberrant double-negative αβ T-cells (CD4negCD8neg, DN) and regulatory-like T-cells. These data raise the possibility that a thymic rudiment persisted, allowing T-cell development, albeit with disturbances in positive/negative selection, as suggested by DN and FoxP3+ cell expansions. Although regulatory-like T-cell numbers normalized following HLA-mismatched thymic transplantation, the αβDN subset persisted 5 years post-transplantation. Involution of thymus allograft likely occurred 3 years post-transplantation based on sj/βTREC ratio, which estimates intrathymic precursor T-cell divisions and, consequently, thymic explant output. Nevertheless, functional immune-competence was sustained, providing new insights for the design of immunological reconstitution strategies based on thymic transplantation, with potential applications in other clinical settings.
Indexed as
Identifiers
What OpenQuestion holds
Registered trials
Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the OpenQuestion graph.