Hydrocephalus in the H-Tx rat: A monogenic disease?

    Authors

    X. G. Cai; G. McGraw; J. V. Pattisapu; L. von Kalm; S. Willingham; D. Socci;J. S. Gibson

    Comments

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    Abbreviated Journal Title

    Exp. Neurol.

    Keywords

    hydrocephalus; H-Tx rat; hereditary mode; genetics; cross-mating; CENTRAL-NERVOUS-SYSTEM; CONGENITAL HYDROCEPHALUS; MODELS; Neurosciences

    Abstract

    The H-Tx rat is a genetic model of hydrocephalus for which there is a poor understanding of the mode of inheritance. Previous studies suggested a polygenic mode of inheritance but the breeding data to support this hypothesis have not been reported. In an attempt to clarify the hereditary mode we have analyzed the data from eight generations of H-Tx rats and four generations of cross-matings between H-Tx rats and Sprague-Dawley (SD) rats. In the H-Tx rat colony 113 of 129 random brother-sister matings (87.60%) produced hydrocephalic offspring, with males and females being equally affected. The overall incidence varied greatly with an average of 30.35%. In matings with more than three litters, all mating pairs yielded hydrocephalic pups. In cross-matings both hydrocephalic and normal H-Tx rats were mated with normal SD rats. No hydrocephalus was observed in the first generation of 124 pups (F1). Subsequent brother-sister matings of F1 animals generated hydrocephalic pups in the F2 generation with a lower incidence (4.67% in hydrocephalic HTx/SD matings and 5.11% in normal HTx/SD matings, respectively) than in the II-Tx rat colony (30.35%). Back-cross-matings between F2 rats and normal H-TK rats yielded an incidence of hydrocephalus higher than that of the cross-matings but lower than that of the II-Tx colony. These data strongly suggest that the II-Tx rat is a homozygous carrier of an autosomal recessive hydrocephalus gene with incomplete penetrance. Furthermore, the data clearly rule out sex-linked and polygenic modes of inheritance and provide further insight with respect to genetic inheritance of hydrocephalus. (C) 2000 Academic Press.

    Journal Title

    Experimental Neurology

    Volume

    163

    Issue/Number

    1

    Publication Date

    1-1-2000

    Document Type

    Article

    Language

    English

    First Page

    131

    Last Page

    135

    WOS Identifier

    WOS:000087006500014

    ISSN

    0014-4886

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