The Frequency of Inherited Hydrocephalus Is Influenced by Intrauterine Factors in H-Tx Rats

Jones, Hazel C.; Depelteau, Jamie S.; Carter, Barbara J.; Somera, Kathleen C.

doi:10.1006/exnr.2002.7921

Cited by 20 publications

(13 citation statements)

References 34 publications

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“…There are two known mutations in rats: one line (H-Tx) with a high rate of inherited hydrocephalus was derived from one mating (Cai et al, 2000;Jones et al, 2000Jones et al, , 2001Jones et al, and 2002 and the other strain (LEW/Jms) was derived from an inbred strain of Wistar-Lewis rats (Jones et al, 2003). Both mutations seem to have a complex mode of inheritance probably involving more than one gene (Jones et al, 2003).…”

Section: Discussionmentioning

confidence: 99%

Hereditary hydrocephalus internus in a laboratory strain of golden hamsters (Mesocricetus auratus)

Gebhardt-Henrich

Edwards

Famula

et al. 2008

Animal

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Golden hamsters of one common laboratory strain had a high incidence of hydrocephalus internus. When a severity score of hydrocephalus was used, a major autosomal recessive locus could be identified. However, when a binary score (hydrocephalus, no hydrocephalus) was used, no such major locus could be detected and results of test matings were not consistent with Mendelian inheritance. Golden hamsters with severe forms of hydrocephalus had a dorsally compressed and ventrally intact hippocampus. Implications for the behavior and well-being of affected hamsters are unknown but researchers using this strain should be aware of the likely presence of hydrocephalus.

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Section: Discussionmentioning

confidence: 99%

Hereditary hydrocephalus internus in a laboratory strain of golden hamsters (Mesocricetus auratus)

Gebhardt-Henrich

Edwards

Famula

et al. 2008

Animal

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“…The H-Tx rat has inherited fetal-onset hydrocephalus with phenotypic expression controlled by complex genetic and epigenetic factors (Jones et al 2001a(Jones et al , 2002. Previous studies have reported that hydrocephalus is associated with aqueduct closure and an absence or reduction in the ependymal cells of the SCO (Jones and Bucknall 1988).…”

Section: Introductionmentioning

confidence: 97%

Reduced subcommissural organ glycoprotein immunoreactivity precedes aqueduct closure and ventricular dilatation in H-Tx rat hydrocephalus

Somera

Jones

2004

Cell and Tissue Research

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The H-Tx rat has fetal-onset hydrocephalus associated with closure of the cerebral aqueduct and a reduction in the secretory cells of the subcommissural organ (SCO), a circumventricular organ situated in the dorsal wall of the cerebral aqueduct. The objective of this study was to determine the role of the SCO in hydrocephalus pathogenesis. Serial brain sections through aqueduct regions containing the SCO from H-Tx rats, together with non-hydrocephalic Fischer F344 rats, were studied at E16, before hydrocephalus onset, at E17, the beginning of onset, and at P0 when the hydrocephalus was overt. Tissues were immunostained by AFRU, an antibody against the SCO glycoprotein, and for the intermediate filament nestin. The area of SCO cells with AFRU immunostaining and the severity of lateral ventricle dilatation were quantified by image analysis. At E16 all fetuses had distinct SCO ependymal cells, open aqueducts and normal lateral ventricles. The H-Tx fetuses fell into two groups with large areas and small areas of AFRU immunoreactivity, all with a full complement of SCO cells. By E17, fetuses with small areas of immunoreactivity had reduced numbers of tall SCO secretory cells, and most had aqueducts closed posteriorly and dilated ventricles. Three additional fetuses with small areas of immunoreactivity had narrow but patent aqueducts and normal ventricles, and another had an open aqueduct and dilated ventricles. At P0, pups previously identified as hydrocephalic had small areas of AFRU immunoreactivity, an aqueduct that was closed anteriorly but open posteriorly, ventricular dilatation, and an absence of SCO secretory cells. The aqueduct even when closed was lined by typical ependymal cells throughout. Decreased nestin immunostaining accompanied the SCO changes. It is concluded that reduced SCO glycoprotein immunoreactivity precedes both aqueduct closure and expansion of the lateral ventricles in the H-Tx rat.

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“…Previously, we have shown that phenotypic expression of congenital hydrocephalus in H-Tx rats is controlled by a combination of genetic and epigenetic factors (Jones et al 2000b(Jones et al , 2001a(Jones et al , 2001b(Jones et al , 2002. QTL mapping from a backcross to Fisher F344 rats identified possible hydrocephalus loci on 6 different chromosomes (Jones et al 2001a).…”

Section: Discussionmentioning

confidence: 99%

“…There is evidence that epigenetic factors are needed for phenotypic expression in that the frequency of hydrocephalus in the H-Tx strain is influenced by whether or not the female is suckling a previous litter during pregnancy (Jones et al 2002). The onset of ventricular dilatation occurs in late gestation and the number of affected pups increased if the dam was pregnant from a postpartum mating.…”

Section: Expression Of Severe Hydrocephalusmentioning

confidence: 99%

“…Hydrocephalus loci on Chromosomes 10 and 11 were most frequently associated with the heterozygous genotype (one allele from each strain), whereas the hydrocephalus locus on Chromosome 17 was predominantly associated with the H-Tx homozygous genotype. In addition to genetic loci on several different chromosomes, there is evidence that epigenetic factors affect the frequency of hydrocephalus (Jones et al 2002). A disadvantage for QTL mapping is that the minimum chromosomal interval containing the locus is relatively large, up to 30 cM, and contains hundreds of genes.…”

Section: Introductionmentioning

confidence: 99%

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Single and multiple congenic strains for hydrocephalus in the H-Tx rat

et al. 2005

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The H-Tx rat has fetal-onset hydrocephalus with a complex mode of inheritance. Previously, quantitative trait locus mapping using a backcross with Fischer F344 rats demonstrated genetic loci significantly linked to hydrocephalus on Chromosomes 10, 11, and 17. Hydrocephalus was preferentially associated with heterozygous alleles on Chrs 10 and 11 and with homozygous alleles on Chr 17. This study aimed to determine the phenotypic contribution of each locus by constructing single and multiple congenic strains. Single congenic rats were constructed using Fischer F344 as the recipient strain and a marker-assisted protocol. The homozygous strains were maintained for eight generations and the brains examined for dilated ventricles indicative for hydrocephalus. No congenic rats had severe (overt) hydrocephalus. A few pups and a significant number of adults had mild disease. The incidence was significantly higher in the C10 and C17 congenic strains than in the nonhydrocephalic F344 strain. Breeding to F344 to make F.H-Tx C10 or C11 rats heterozygous for the hydrocephalus locus failed to produce progeny with severe disease. Both bicongenic and tricongenic rats of different genotype combinations were constructed by crossing congenic rats. None had severe disease but the frequency of mild hydrocephalus in adults was similar to congenic rats and significantly higher than in the F344 strain. Rats with severe hydrocephalus were recovered in low numbers when single congenic or bicongenic rats were crossed with the parental H-Tx strain. It is concluded that the genetic and epigenetic factors contributing to severe hydrocephalus in the HTx strain are more complex than originally anticipated.

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The Frequency of Inherited Hydrocephalus Is Influenced by Intrauterine Factors in H-Tx Rats

Cited by 20 publications

References 34 publications

Hereditary hydrocephalus internus in a laboratory strain of golden hamsters (Mesocricetus auratus)

Hereditary hydrocephalus internus in a laboratory strain of golden hamsters (Mesocricetus auratus)

Reduced subcommissural organ glycoprotein immunoreactivity precedes aqueduct closure and ventricular dilatation in H-Tx rat hydrocephalus

Single and multiple congenic strains for hydrocephalus in the H-Tx rat

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