A-A’: Representative histologic images of the mitral
valve at 2 months of age in wildtype (A) and Marfan syndrome (A’) mice
using Movat’s pentachrome. Abnormal valvular ECM remodeling comprised of
collagen fragmentation (arrows) and increased proteoglycan (arrowheads) is
present in Marfan syndrome mice (A’) versus controls (A). The anterior
leaflet is shown. B-B’: Increased proteoglycan deposition
(green) and collagen fragmentation (red) can also be seen by confocal
microscopy. Note the discontinuity of collagen (red) in mutant mitral valves
(B’). C-C’: By confocal microscopy, CD45+ leukocytes
are noticeably increased in MFS mitral valves (C’) versus controls (C)
and are localized to regions of abnormal ECM remodeling.
D-D”: MFS mitral valves exhibit immunogenic modification
of the proteoglycan layer comprised of increased IaI deposition (green;
D”) versus control mitral valves (D, D’).
E-F’’’: CD45+ leukocytes are primarily
comprised of two subsets of macrophages (CD206 and MHCII) that are spatially
distinct in wildtype valves (E). Note that MHCII+ macrophages remain at the
distal tips of the mitral valve (F’), whereas CD206+ macrophages run
along the length of the valve leaflets (F’). In diseased valves, spatial
preference of macrophages is lost, as MHCII+ and double-positive CD206+MCHII+
macrophages are located in the vicinity of CD206+ macrophages (F”,
F’’’). G-H: Quantification of CD45+ leukocyte
numbers (G) and IaI deposition (H). I-J”: Representative
fluorescent images of CCL2 expression in WT and MFS mitral valves at one and two
months of age. No significant differences in CCL2 expression was observed at one
month of age (I-I”). However, CCL2 was significantly increased by two
months of age in MFS mitral valves (J’-J”) versus control valves
(J). CCL2 protein was predominantly expressed in CD45- populations and also
deposited in the extracellular space (arrows, J”). Note that CD45+ cells
are observed in the vicinity of CCL2. Data are represented as mean ± SEM.
*p<0.05 and **p<0.01. n.s. =
not significant. n=6–8 mice/genotype/timepoint. Scale Bar = 100μm.
Abbreviations: HABP=Hyaluronan Binding Protein; COL=Collagen; IaI=
Inter-alpha-trypsin inhibitor; mo=month.