Comparative lymphatic, ocular, and metabolic phenotypes of FOXC2 haploinsufficient and AP2-FOXC2 transgenic mice

A. Noon, R. J. Hunter, M. H. Witte, B. Kriederman, M. Bernas, M. Rennels, D. Percy, S. Enerbäck, Robert P. Erickson

Research output: Contribution to journalArticle

6 Scopus citations

Abstract

FOXC2 mutations cause the lymphatic/ocular disorder Lymphedema-Distichiasis (LD), and Foxc2 haploinsufficient mice mimic this disorder. To determine if FOXC2 overexpression might also cause lymphatic and/or ocular abnormalities, we performed dynamic lymphatic imaging (Evans blue dye), ocular tissue examination, and metabolic profiles in mice: transgenic for FOXC2 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 +/-, and Foxc2+/-, Tg but not aP2-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.

Original languageEnglish (US)
Pages (from-to)84-94
Number of pages11
JournalLymphology
Volume39
Issue number2
StatePublished - Jun 1 2006

Keywords

  • Adipocyte
  • FOXC2
  • Foxc2
  • Haploinsufficient
  • Lymphatic hyperplasia
  • Lymphedema-Distichiasis
  • Ocular
  • Transgenic

ASJC Scopus subject areas

  • Immunology and Allergy
  • Hematology

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    Noon, A., Hunter, R. J., Witte, M. H., Kriederman, B., Bernas, M., Rennels, M., Percy, D., Enerbäck, S., & Erickson, R. P. (2006). Comparative lymphatic, ocular, and metabolic phenotypes of FOXC2 haploinsufficient and AP2-FOXC2 transgenic mice. Lymphology, 39(2), 84-94.