Articles
Authors: A Noon ( ) , RJ Hunter ( ) , MH Witte ( ) , B Kriederman ( ) , MJ Bernas ( ) , M Rennels ( ) , D Percy ( ) , S Enerback ( ) , RP Erickson ( )
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).