IPEX Syndrome, FOXP3 and Cancer - PubMed
IPEX Syndrome, FOXP3 and Cancer
Runhua Liu et al. J Syndr. 2013 Jun.
Abstract
In this review, we introduce the IPEX syndrome and its relationship with germline mutations of the FOXP3 gene. We then describe the multiple functional roles of FOXP3 in regulatory T cells and epithelial cells as well as in IPEX syndrome and tumor progression. Potential mechanisms of FOXP3 inactivation and transcriptional regulation are discussed with recent advances. Finally, we point out current issues and a potential FOXP3-mediated therapeutic strategy as well as the reactivation of FOXP3 in patients with IPEX syndrome and cancer.
Keywords: Cancer; FOXP3; IPEX; Syndrome; Tumor suppressors.
Figures
![Figure 1](https://cdn.ncbi.nlm.nih.gov/pmc/blobs/868f/4383293/ac1c31515764/nihms-578696-f0001.gif)
Functional domains and mutations in coding region of the human FOXP3 gene. FOXP3 transcript consists of 12 exons and the open reading frame is from exons 2 to 12. There are 4 potential functional domains (hollow boxes). Black starts indicate germline mutations identified from IPEX patients. Black arrows indicate somatic mutations from breast cancer patients, and solid triangles indicate somatic mutations from prostate cancer patients.
![Figure 2](https://cdn.ncbi.nlm.nih.gov/pmc/blobs/868f/4383293/71e800c91a0d/nihms-578696-f0002.gif)
Functional signaling pathways in the context of FOXP3. FOXP3 is highly expressed in Treg cells, but also detected in both epithelial and non-epithelial cells, including many cancer cells. Several hundred target genes of the FOXP3 have been identified in both Treg cells and epithelial cells. TGFβ: transforming growth factor β; TCR: T cell receptor; CTLA4: cytotoxic T-lymphocyte-associated protein 4; IL2: interleukin 2; IL2Ra: IL2 receptor a; IL7Ra: IL7 receptor a; LATS2: large tumor suppressor kinase 2; ATF2: ATF2 activating transcription factor 2; HER2: v-erb-b2 erythroblastic leukemia viral oncogene homolog 2; SKP2: S-phase kinase-associated protein 2; CDKs: cyclin-dependent kinases; mTOR: mammalian target of rapamycin; NFκB: nuclear factor κB; MMP2: matrix metalloproteinase 2; uPA: urokinase-type plasminogen activator; BRCA1: breast cancer 1, early onset; CXCR4: chemokine (C-XC motif) receptor 4.
Similar articles
-
Li B, Samanta A, Song X, Iacono KT, Brennan P, Chatila TA, Roncador G, Banham AH, Riley JL, Wang Q, Shen Y, Saouaf SJ, Greene MI. Li B, et al. Int Immunol. 2007 Jul;19(7):825-35. doi: 10.1093/intimm/dxm043. Epub 2007 Jun 22. Int Immunol. 2007. PMID: 17586580
-
Al Maawali A, Derfalvi B, Van Limbergen J, Issekutz A, Issekutz T, Ghandourah H, Rashid M. Al Maawali A, et al. Case Reports Immunol. 2020 Sep 9;2020:9860863. doi: 10.1155/2020/9860863. eCollection 2020. Case Reports Immunol. 2020. PMID: 32963853 Free PMC article.
-
Park JH, Lee KH, Jeon B, Ochs HD, Lee JS, Gee HY, Seo S, Geum D, Piccirillo CA, Eisenhut M, van der Vliet HJ, Lee JM, Kronbichler A, Ko Y, Shin JI. Park JH, et al. Autoimmun Rev. 2020 Jun;19(6):102526. doi: 10.1016/j.autrev.2020.102526. Epub 2020 Mar 29. Autoimmun Rev. 2020. PMID: 32234571
-
IPEX and FOXP3: clinical and research perspectives.
Wildin RS, Freitas A. Wildin RS, et al. J Autoimmun. 2005;25 Suppl:56-62. doi: 10.1016/j.jaut.2005.04.008. Epub 2005 Oct 21. J Autoimmun. 2005. PMID: 16243487 Review.
Cited by
-
Expanding the spectrum of IPEX: from new clinical findings to novel treatments.
Voarino M, Consonni F, Gambineri E. Voarino M, et al. Curr Opin Allergy Clin Immunol. 2024 Dec 1;24(6):457-463. doi: 10.1097/ACI.0000000000001033. Epub 2024 Oct 11. Curr Opin Allergy Clin Immunol. 2024. PMID: 39475830 Free PMC article. Review.
-
Gastric Adenocarcinoma in the Setting of IPEX Syndrome.
Steffin D, Bhar S, Fishman DS, Rider NL, Naik-Mathuria B, Martinez C, Venkatramani R. Steffin D, et al. Case Reports Immunol. 2021 Jun 25;2021:9967198. doi: 10.1155/2021/9967198. eCollection 2021. Case Reports Immunol. 2021. PMID: 34258086 Free PMC article.
-
Abele M, Grabner L, Blessing T, Block A, Agaimy A, Kratz C, Simon T, Calaminus G, Heine S, Corbacioglu S, Christiansen H, Schneider DT, Brecht IB. Abele M, et al. Cancers (Basel). 2023 Jan 3;15(1):317. doi: 10.3390/cancers15010317. Cancers (Basel). 2023. PMID: 36612313 Free PMC article.
-
FOXP3 expression is modulated by TGF‑β1/NOTCH1 pathway in human melanoma.
Skarmoutsou E, Bevelacqua V, D' Amico F, Russo A, Spandidos DA, Scalisi A, Malaponte G, Guarneri C. Skarmoutsou E, et al. Int J Mol Med. 2018 Jul;42(1):392-404. doi: 10.3892/ijmm.2018.3618. Epub 2018 Apr 4. Int J Mol Med. 2018. PMID: 29620159 Free PMC article.
References
-
- Powell BR, Buist NR, Stenzel P. An X-linked syndrome of diarrhea, polyendocrinopathy, and fatal infection in infancy. The Journal of pediatrics. 1982;100:731–737. - PubMed
-
- d'Hennezel E, Bin Dhuban K, Torgerson T, Piccirillo CA. The immunogenetics of immune dysregulation, polyendocrinopathy, enteropathy, X linked (IPEX) syndrome. Journal of medical genetics. 2012;49:291–302. - PubMed
-
- Hannibal MC, Torgerson T. In: IPEX Syndrome. GeneReviews. Pagon RA, Bird TD, Dolan CR, Stephens K, Adam MP, editors. Vol. 27. Seattle (WA): Jan, 2011. p. 2011. Last Update.
-
- Heltzer ML, Choi JK, Ochs HD, Sullivan KE, Torgerson TR, Ernst LM. A potential screening tool for IPEX syndrome. Pediatric and developmental pathology : the official journal of the Society for Pediatric Pathology and the Paediatric Pathology Society. 2007;10:98–105. - PubMed
-
- Bennett CL, Christie J, Ramsdell F, Brunkow ME, Ferguson PJ, Whitesell L, Kelly TE, Saulsbury FT, Chance PF, Ochs HD. The immune dysregulation, polyendocrinopathy, enteropathy, X-linked syndrome (IPEX) is caused by mutations of FOXP3. Nat Genet. 2001;27:20–21. - PubMed
LinkOut - more resources
Full Text Sources