Sperm tail axoneme alterations in the Wobbler mouse

Leestma, Jan E.; Sepsenwol, Sol

doi:10.1530/jrf.0.0580267

Cited by 36 publications

(22 citation statements)

References 5 publications

Supporting

Mentioning

Contrasting

Order By: Relevance

“…Among the best characterized of these are quaking (qk) (Sidman et al, 1964;Samorajski et al, 1970;Bennett et al, 1971), Purkinje cell degeneration (pcd) (Mullen et al, 1976;Handel and Dawson, 19811, and wobbler (wr) (Duchen and Strich, 1968;Leestma and Sepsenwol, 1980;Heimann et al, 1991). All of these mutations are autosomal recessive and all cause motor deficits as well as sterility in homozygous mutant mice.…”

Section: Discussionmentioning

confidence: 99%

“…Among the phenotypes exhibited by these mutant mice are failure of spermatid elongation, abnormal tail formation, and abnormal acrosome development (Bennett et al, 1971;Mullen et al, 1976;Leestma and Sepsenwol, 1980;Handel and Dawson, 1981;Heimann et al, 1991). Although weauer also affects terminal differentiation of sperm, most of the features of the testicular phenotype are dissimilar to what is seen in qk, pcd, and wr mice.…”

Section: Discussionmentioning

confidence: 99%

See 1 more Smart Citation

Male‐sterile phenotype of the neurological mouse mutant weaver

Harrison

Roffler-Tarlov

1994

Developmental Dynamics

View full text Add to dashboard Cite

The autosomal recessive murine mutation weauer (wu) affects postnatal differentiation in at least three neuronal populations in the brain: dopamine-containing neurons in the midbrain, and granule and Purkinje cells in the cerebellum. Neuronal populations vulnerable to the actions of weaver die in the midst of development. In addition, homozygous weaver males are sterile. We show by a histological analysis of epididymides and testes that the cause of male sterility in weaver is lack of sperm. The epididymides of the adult weaver mice examined were devoid of sperm, and few seminiferous tubules in the adult weaver's testes contained elongated spermatids. Most tubules were marked by moderate to severe degeneration of the seminiferous epithelium. A developmental study showed that the mutant phenotype emerged after the third postnatal week. By postnatal day 28, the development of weaver sperm lagged behind that of the wild-type and some seminiferous tubules contained degenerating germ cells. By postnatal day 35, terminal differentiation of spermatids appeared to be arrested in many tubules and degeneration of the seminiferous epithelium was widespread. The heterozygotes were unaffected at all ages sampled. We conclude that the normal allele at the weaver locus is necessary for spermiogenesis and the maintenance of spermatogenesis. 0 1994 Wiley-Liss, Inc.

show abstract

Section: Discussionmentioning

confidence: 99%

Section: Discussionmentioning

confidence: 99%

Male‐sterile phenotype of the neurological mouse mutant weaver

Harrison

Roffler-Tarlov

1994

Developmental Dynamics

View full text Add to dashboard Cite

show abstract

“…In contrast, it has been reported that the abnormal axoneme is induced by hereditary disease as in the Wobbler mouse and Kartagener's syndrome (LEESTMA and SEPSENWOL, 1980;AFZELIUS, 1981;AITKEN et al, 1983). Judging from extremely rare cases involving the abnormal axoneme, it is considered that the abnormal axoneme may be controlled by hereditary factors rather than other non-hereditary ones, while the periaxonemal components are controlled by other factors.…”

Section: Discussionmentioning

confidence: 99%

Flagellum Abnormalities of Spermatozoa in Seminiferous Tubules after a Short Term Vasectomy.

Hamasaki

Miyajima

Murakami

1991

Archives of Histology and Cytology

View full text Add to dashboard Cite

“…In certain t haplotypes and the Wobbler mutation (wr), sperm microtubules are also arranged in aberrant patterns in the vas deferens but appear to be normal in the testis (Dooher and Bennett 1977;Leestma and Sepsenwol 1980). The defective nature of these mutant sperm may become manifest in the epididymis because at this time they undergo dramatic changes in morphology and biochemistry and acquire motility (Hoskins and Vijayaraghavan 1990).…”

Section: Defective Sperm Motility and Axoneme Disruptionmentioning

confidence: 99%

“…The defective nature of these mutant sperm may become manifest in the epididymis because at this time they undergo dramatic changes in morphology and biochemistry and acquire motility (Hoskins and Vijayaraghavan 1990). Interestingly, sperm tail axonemes from both wr/ wr and sterile AE24 mice usually contain the same microtubule pattern (5 + 2) and are missing the same outer doublets (4--7) (Leestma and Sepsenwol 1980}. Mutations leading to sperm paralysis and male sterility have been described in humans as well as in mice (Ryder et al 1990}. The best described disease is immotile-cilia (Kartagener's) syndrome, in which men suffer from chronic sinobronchial infections and abnormal motility of both respiratory cilia and sperm flagella (Eliasson et al 1977).…”

Section: Defective Sperm Motility and Axoneme Disruptionmentioning

confidence: 99%

Inactivation of a sperm motility gene by insertion of an epidermal growth factor receptor transgene whose product is overexpressed and compartmentalized during spermatogenesis.

Merlino¹,

Stahle²,

Jhappan³

et al. 1991

Genes Dev.

View full text Add to dashboard Cite

Transgenic mice were generated with a human epidermal growth factor (EGF) receptor cDNA driven by the chicken 13-actin gene promoter. One line (AE24) that exhibited a unique expression pattern in which dramatically elevated levels of EGF receptor RNA were found only in the testis was established, suggesting that the 13-actin promoter was being influenced by an adjacent testis-specific enhancer. EGF receptor RNA was detected in primary spermatocytes, whereas the synthesis of receptor protein was restricted to elongate spermatids, indicating that transgene expression was under translational control. At spermiation, the EGF receptor was sequestered in residual bodies and excluded from mature sperm by a compartmentalization mechanism. About half of AE24 homozygous males were sterile because of sperm paralysis, whereas heterozygous males and females of either genotype were completely fertile. Electron microscopic analysis of sperm flagella from sterile AE24 homozygotes revealed an aberrant axonemal structure in which outer doublet microtubules were missing from the middle piece, resembling changes observed in the sperm of some infertile humans. Flagellar axonemal disassembly was observed in the vas deferens and epididymis but not in the testis, suggesting that outer doublets were assembled in a grossly normal manner but possessed a latent instability. These results demonstrate that in the AE24 mouse line the EGF receptor transgene was integrated into and inactivated an endogenous autosomal gene, causing sperm flagellar axonemal disruption and male sterility.

show abstract

Sperm tail axoneme alterations in the Wobbler mouse

Abstract: In Wobbler mice (neurological mutants affected with progressive motor neurone degenerative disease) a defect in sperm tail mobility and axonemal geometry was observed. Similar but less extensive abnormalities were seen in the ciliary axonemes of the ductuli efferentes.

Cited by 36 publications

References 5 publications

Male‐sterile phenotype of the neurological mouse mutant weaver

Male‐sterile phenotype of the neurological mouse mutant weaver

Flagellum Abnormalities of Spermatozoa in Seminiferous Tubules after a Short Term Vasectomy.

Inactivation of a sperm motility gene by insertion of an epidermal growth factor receptor transgene whose product is overexpressed and compartmentalized during spermatogenesis.

Contact Info

Product

Resources

About