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Defective NKT cell development in mice and humans lacking the adapter SAP, the X-linked lymphoproliferative syndrome gene product
Open accessAbstract
SAP is an adaptor protein expressed in T cells and natural killer cells. It plays a critical role in immunity, as it is mutated in humans with X-linked lymphoproliferative syndrome (XLP), a fatal immunodeficiency characterized by an abnormal response to Epstein-Barr virus (EBV) infection. SAP interacts with the SLAM family receptors and promotes transduction signal events by these receptors through its capacity to recruit and activate the Src kinase FynT. Because it has been previously established that FynT is selectively required for the development of NKT cells, we examined NKT cells in SAP-deficient mice and in humans with XLP. In the absence of SAP, the development of NKT cells is severely impaired both in mice and in humans. These results imply that SAP is a potent regulator of NKT cell development. They also identify for the first time a defect in NKT cells associated with a human primary immunodeficiency, revealing a potential role of NKT cells in the immune response to EBV.
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Cited by
Cellular responses to viral infection in humans: lessons from Epstein–Barr virus
Hislop, A. D., Taylor, G. S., Sauce, D., & Rickinson, A. B. (2007) · Annual Review of Immunology · doi:10.1146/annurev.immunol.25.022106.141553 · Cited by 766
XIAP deficiency in humans causes an X-linked lymphoproliferative syndrome
Rigaud, S., Fondanèche, M.-C., Lambert, N., Pasquier, B., Mateo, V., Soulas, P., Galicier, L., Le Deist, F., Rieux-Laucat, F., Revy, P., et al. (2006) · Nature · doi:10.1038/nature05257 · Cited by 706
Self-citationGodfrey, D. I., Stankovic, S., & Baxter, A. G. (2010) · Nature Immunology · doi:10.1038/ni.1841 · Cited by 649
Stepwise Development of MAIT Cells in Mouse and Human
Martin, E., Treiner, E., Duban, L., Guerri, L., Laude, H., Toly, C., Premel, V., Devys, A., Moura, I. C., Tilloy, F., et al. (2009) · PLoS Biology · doi:10.1371/journal.pbio.1000054 · Cited by 598
Self-citationDustin, M. L. (2014) · Cancer Immunology Research · doi:10.1158/2326-6066.cir-14-0161 · Cited by 502
Metadata
- Authors
- Pasquier, B., Yin, L., Fondanèche, M.-C., Relouzat, F., Bloch-Queyrat, C., Lambert, N., Fischer, A., de Saint-Basile, G., & Latour, S.
- Venue
- The Journal of Experimental Medicine
- Publisher
- Rockefeller University Press
- Published
- 2005-02-28
- Volume
- 201
- Issue
- 5
- Pages
- 695-701
- ISSN
- 0022-1007 (ISSN-L)
- 1540-9538
- OpenAlex
- W2110045901
- Cited by
- 350
- References
- 0
- FWCI
- 10.19
- Type
- journal article
- Language
- en
- Open access
- green
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- CoChat. (2026). Citation check: Defective NKT cell development in mice and humans lacking the adapter SAP, the X-linked lymphoproliferative syndrome gene product. https://app.cochat.ai/papers/10.1084/jem.20042432
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- Pasquier, B., Yin, L., Fondanèche, M.-C., Relouzat, F., Bloch-Queyrat, C., Lambert, N., Fischer, A., de Saint-Basile, G., & Latour, S. (2005). Defective NKT cell development in mice and humans lacking the adapter SAP, the X-linked lymphoproliferative syndrome gene product. The Journal of Experimental Medicine. https://doi.org/10.1084/jem.20042432
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