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Transcript
Functional amelioration of murine galactosialidosis by
genetically modified bone marrow hematopoietic
progenitor cells
by Thasia Leimig, Linda Mann, Maria del Pilar Martin, Erik Bonten, Derek Persons,
James Knowles, James A. Allay, John Cunningham, Arthur W. Nienhuis, Richard
Smeyne, and Alessandra d'Azzo
Blood
Volume 99(9):3169-3178
May 1, 2002
©2002 by American Society of Hematology
Retrovirally transduced PPCA−/− BM cells restore cathepsin A activity in transplanted PPCA−/−
recipients.(A) Schematic diagram of the retroviral bicistronic construct encoding the human
PPCA cDNA.
Thasia Leimig et al. Blood 2002;99:3169-3178
©2002 by American Society of Hematology
Histology of systemic organs from BM-transplanted GS mice.Organs from PPCA −/− mice
transplanted with total −/−BM transduced with the MSCV-PPCA (BMT-PPCA) retrovirus were
isolated at different time points after treatment.
Thasia Leimig et al. Blood 2002;99:3169-3178
©2002 by American Society of Hematology
Immunostaining of tissue sections from BM-transplanted GS mice with α-32 and α-GFP
antibodies.Numerous human PPCA-expressing cells were detected by immunostaining with α-32
antibody, monospecific for the human protein (left panels).
Thasia Leimig et al. Blood 2002;99:3169-3178
©2002 by American Society of Hematology
Histology of 4 regions of the brain from BM-transplanted GS mice.Hematoxylin and eosin
staining of the choroid plexus (CP), the lateral geniculate nucleus (LGN), the amygdala (AMG),
and the deep cerebellar nucleus (CbN) isolated from BMT-PPCA–treated GS mou...
Thasia Leimig et al. Blood 2002;99:3169-3178
©2002 by American Society of Hematology
Immunostaining of 4 brain regions from BM-transplanted GS mice with α-32 and α-GFP
antibodies.α-32 immunostaining of brain sections derived from BMT-PPCA–transplanted mice at
9 months after treatment revealed numerous PPCA+ endothelial cells, perivascular m...
Thasia Leimig et al. Blood 2002;99:3169-3178
©2002 by American Society of Hematology
PEP19 staining of cerebellar sections from BM-transplanted GS mice.Serial sections of the
cerebellum from a 9-month-old GS mouse transplanted with MSCV-PPCA–marked BM cells were
stained with anti-PEP19 antibody.
Thasia Leimig et al. Blood 2002;99:3169-3178
©2002 by American Society of Hematology
Purkinje cell counts in BM-transplanted GS mice.Purkinje cells were counted in recipient mice at
3 and 9 months after treatment and compared to 3- and 9-month-oldPPCA −/− mice and agematched controls.
Thasia Leimig et al. Blood 2002;99:3169-3178
©2002 by American Society of Hematology