Title

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

Authors

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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