COMPARATIVE LYMPHATIC, OCULAR, AND METABOLIC PHENOTYPES OF FOXC2 HAPLOINSUFFICIENT AND AP2-FOXC2 TRANSGENIC MICE
- A Noon
- RJ Hunter
- MH Witte
- B Kriederman
- MJ Bernas
- M Rennels
- D Percy
- S Enerback
- RP Erickson
Abstract
FOXC2 mutations cause the lymphatic/ocular disorder Lymphedema-Distichiasis (LD), and Foxc2 haploinsufficient mice mimic this disorder. To determine ifFOXC2 overexpression might also cause lymphatic and/or ocular abnormalities, we performed dynamic lymphatic imaging (Evansblue dye), ocular tissue examination, and metabolic profiles in mice: transgenic forFOXC2 with an adipocyte (aP2) promoter (aP2-FOXC2 Tg), heterozygous for targeted disruption of Foxc2 (Foxc2+/-), or compound heterozygous and transgenic (Foxc2+/-, Tg) compared to wild-type controls (WT). Foxc2+/-; aP2-FOXC2 Tg; and Foxc2+/-, Tg, exhibited LD's distinctive hyperplastic lymphatic phenotype characterized by increased number of lymphatic channels and lymph nodes as well as retrograde lymph reflux. Foxc2+/-, andFoxc2+/-, Tg but not a P2-FOXC2 Tg or WT showed an abnormal ocular phenotype. Previously described alterations in brown/white fat distribution and lean phenotype in aP2-FOXC2 transgenics were confirmed. AP2-FOXC2 Tg immunohistochemistry disclosed aberrant FOXC2 expression in ectopic sites,especially embryonic heart. Lymphatic system links with fat metabolism are discussed.
Keywords: FOXC2, Foxc2, lymphatic hyperplasia, haploinsufficient, transgenic, Lymphedema-Distichiasis, ocular, adipocyte
How to Cite:
Noon, A., Hunter, R., Witte, M., Kriederman, B., Bernas, M., Rennels, M., Percy, D., Enerback, S. & Erickson, R., (2006) “COMPARATIVE LYMPHATIC, OCULAR, AND METABOLIC PHENOTYPES OF FOXC2 HAPLOINSUFFICIENT AND AP2-FOXC2 TRANSGENIC MICE”, Lymphology 39(2), 84-94.
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