Inflammatory dilated cardiomyopathy in Abcg5-deficient mice.
Document Type
Article
Publication Date
8-2013
JAX Location
Reprint Collection
JAX Source
Toxicol Pathol 2013 Aug; 41(6):880-92.
Volume
41
Issue
6
First Page
880
Last Page
892
ISSN
1533-1601
PMID
23129576
Abstract
Dilated cardiomyopathy (DCM) in A/J mice homozygous for the spontaneous thrombocytopenia and cardiomyopathy (trac) mutation results from a single base pair change in the Abcg5 gene. A similar mutation in humans causes sitosterolemia with high plant sterol levels, hypercholesterolemia, and early onset atherosclerosis. Analyses of CD3+ and Mac-3+ cells and stainable collagen in hearts showed inflammation and myocyte degeneration in A/J-trac/trac mice beginning postweaning and progressed to marked dilative and fibrosing cardiomyopathy by 140 days. Transmission electron microscopy (TEM) demonstrated myocyte vacuoles consistent with swollen endoplasmic reticulum (ER). Myocytes with cytoplasmic glycogen and irregular actinomyosin filament bundles formed mature intercalated disks with normal myocytes suggesting myocyte repair. A/J-trac/trac mice fed lifelong phytosterol-free diets did not develop cardiomyopathy. BALB/cByJ-trac/trac mice had lesser inflammatory infiltrates and later onset DCM. BALB/cByJ-trac/trac mice changed from normal to phytosterol-free diets had lesser T cell infiltrates but persistent monocyte infiltrates and equivalent fibrosis to mice on normal diets. B- and T-cell-deficient BALB/cBy-Rag1(null) trac/trac mice fed normal diets did not develop inflammatory infiltrates or DCM. We conclude that the trac/trac mouse has many features of inflammatory DCM and that the reversibility of myocardial T cell infiltration provides a novel model for investigating the progression of myocardial fibrosis. Toxicol Pathol 2013 Aug; 41(6):880-92.
Recommended Citation
Wilson D,
Oslund K,
Lyons BL,
Foreman O,
Burzenski L,
Svenson KL,
Chase T,
Shultz LD.
Inflammatory dilated cardiomyopathy in Abcg5-deficient mice. Toxicol Pathol 2013 Aug; 41(6):880-92.