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The Role of Apoptosis in the Manifestation of Polydactyly and Arhinencephaly in Genetic Mutant Mouse Pdn/Pdn *
Author(s) -
NARUSE Ichiro,
KEINO Hiromi,
TANIGUCHI Masahiko,
MASAKI Shigeo
Publication year - 1994
Publication title -
congenital anomalies
Language(s) - English
Resource type - Journals
eISSN - 1741-4520
pISSN - 0914-3505
DOI - 10.1111/j.1741-4520.1994.tb00801.x
Subject(s) - apoptosis , embryo , programmed cell death , notochord , microbiology and biotechnology , anatomy , medicine , biology , pathology , embryogenesis , genetics
Pdn/Pdn mouse exhibits preaxial polydactyly and arhinencephaly, including various brain abnormalities such as the absence of corpus callosum and hydrocephaly. We have investigated the mechanism of the manifestation of polydactyly. The abolishment of the deep preaxial mesodermal programmed cell death, foyer primaire préxial (fpp), has been considered to be the starting point of the manifestation of preaxial polydactyly. With this hypothesis, we electrocauterized the fpp region of Pdn/Pdn embryos, because Pdn/Pdn lacks fpp. After surgery, Pdn/Pdn embryos were developed using whole embryo culture system or exo utero method. They exhibited 5 digits in their limbs which received surgery in fpp region. Meanwhile, much more apoptotic degenerations were observed in the brains of Pdn/Pdn newborns and embryos than normals. Anti‐single‐stranded DNA antibody was used to detect the localization of apoptotic cell death in the brains of Pdn/Pdn mice. TRPM‐2 gene has been considered to be an indicator of apoptosis. The brains of Pdn/Pdn newborns and embryos showed higher TRPM‐2 gene expression in Northern blot analysis. From these results, we speculated that abnormal apoptosis in the periventricular zone induced the expansion of the ventricle followed by hydrocephaly.