DOI: 10.1242/dev.119.1.113. Interaction between splotch (Sp) and curly tail (ct) mouse mutants in the embryonic development of neural tube defects

Interaction between splotch (Sp) and curly tail (ct) mouse mutants in the embryonic development of neural tube defects

10.1242/dev.119.1.113

Crossref journal-article

The Company of Biologists

Development (237)

Abstract

ABSTRACT The mouse mutations splotch (Sp) and curly tail (ct) both produce spinal neural tube defects with closely similar morphology, but achieve this by different embryonic mechanisms. To determine whether the mutants may interact during development, we constructed mice carrying both mutations. Double heterozygotes exhibited tail defects in 10% of cases, although the single het-erozygotes do not express this phenotype. Backcrosses of double heterozygotes to ct/ct produced offspring with an elevated incidence of neural tube defects, both spina bifida and tail defects, compared with a control backcross in which Sp was not involved. Use of the deletion allele Sp2H permitted embryos carrying a splotch mutation to be recognised by polymerase chain reaction assay. This experiment showed that only embryos carrying Sp2H develop spina bifida in the backcross with ct/ct, suggesting that the genotype Sp2H/+, ct/ct is usually lethal around the time of birth as a result of severe disturbance of neurulation. The interaction between Sp and ct was investigated further by examining embryos in the backcross for developmental markers of the Sp/Sp and ct/ct genotypes. Sp/Sp embryos characteristically lack neural crest derivatives, such as dorsal root ganglia, and die on day 13 of gestation. Double mutant embryos from the backcross did not exhibit either of these characteristics suggesting that homozygosity for ct does not cause Sp/+ embryos to develop as if they were of genotype Sp/Sp. The angle of ventral curvature of the posterior neuropore region is enhanced in affected ct/ct embryos whereas it was found to be reduced in Sp/Sp embryos compared with their normal littermates. Double mutant embryos from the backcross had an angle of curvature that resembled the ct/ct pattern but was less exaggerated. We conclude that the non-allelic mutations Sp and ct interact to promote the development of neural tube defects in a manner that does not involve exacerbation of the specific developmental effects of either gene. The presence of enhanced curvature of the caudal region, which is responsible for neurulation disturbance in ct/ct embryos, summates with the reduced neurulation potential of the neuroepithelium in the Sp/+ genotype leading to the development of severe spina bifida. This study demonstrates that the finding of a significant interaction between genes in double mutant mice cannot be assumed to indicate that the genes operate in the same genetic pathway.

Authors 3

J. Peter Estibeiro (first)
Frances A. Brook (additional)
Andrew J. Copp (additional)

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{'key': '2023120222351361700_DEVELOP_119_1_113C5', 'first-page': '151', 'article-title': 'Neural tube development in mutant (curly tail) and normal mouse embryos: the timing of posterior neuropore closure in vivo and in vitro', 'volume': '69', 'author': 'Copp', 'year': '1982', 'journal-title': 'J. Embryol. exp. Morphol'} / J. Embryol. exp. Morphol / Neural tube development in mutant (curly tail) and normal mouse embryos: the timing of posterior neuropore closure in vivo and in vitro by Copp (1982)
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Dates

Type	When
Created	4 years, 4 months ago (April 25, 2021, 9:18 p.m.)
Deposited	1 year, 8 months ago (Dec. 2, 2023, 5:35 p.m.)
Indexed	1 year, 4 months ago (April 1, 2024, 1:55 p.m.)
Issued	31 years, 11 months ago (Sept. 1, 1993)
Published	31 years, 11 months ago (Sept. 1, 1993)
Published Online	31 years, 11 months ago (Sept. 1, 1993)
Published Print	31 years, 11 months ago (Sept. 1, 1993)

Funders 0

None

BibTeX

@article{Estibeiro_1993, title={Interaction between splotch (Sp) and curly tail (ct) mouse mutants in the embryonic development of neural tube defects}, volume={119}, ISSN={1477-9129}, url={http://dx.doi.org/10.1242/dev.119.1.113}, DOI={10.1242/dev.119.1.113}, number={1}, journal={Development}, publisher={The Company of Biologists}, author={Estibeiro, J. Peter and Brook, Frances A. and Copp, Andrew J.}, year={1993}, month=sep, pages={113–121} }

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  "abstract": "<jats:title>ABSTRACT</jats:title>\n               <jats:p>The mouse mutations splotch (Sp) and curly tail (ct) both produce spinal neural tube defects with closely similar morphology, but achieve this by different embryonic mechanisms. To determine whether the mutants may interact during development, we constructed mice carrying both mutations. Double heterozygotes exhibited tail defects in 10% of cases, although the single het-erozygotes do not express this phenotype. Backcrosses of double heterozygotes to ct/ct produced offspring with an elevated incidence of neural tube defects, both spina bifida and tail defects, compared with a control backcross in which Sp was not involved. Use of the deletion allele Sp2H permitted embryos carrying a splotch mutation to be recognised by polymerase chain reaction assay. This experiment showed that only embryos carrying Sp2H develop spina bifida in the backcross with ct/ct, suggesting that the genotype Sp2H/+, ct/ct is usually lethal around the time of birth as a result of severe disturbance of neurulation. The interaction between Sp and ct was investigated further by examining embryos in the backcross for developmental markers of the Sp/Sp and ct/ct genotypes. Sp/Sp embryos characteristically lack neural crest derivatives, such as dorsal root ganglia, and die on day 13 of gestation. Double mutant embryos from the backcross did not exhibit either of these characteristics suggesting that homozygosity for ct does not cause Sp/+ embryos to develop as if they were of genotype Sp/Sp. The angle of ventral curvature of the posterior neuropore region is enhanced in affected ct/ct embryos whereas it was found to be reduced in Sp/Sp embryos compared with their normal littermates. Double mutant embryos from the backcross had an angle of curvature that resembled the ct/ct pattern but was less exaggerated. We conclude that the non-allelic mutations Sp and ct interact to promote the development of neural tube defects in a manner that does not involve exacerbation of the specific developmental effects of either gene. The presence of enhanced curvature of the caudal region, which is responsible for neurulation disturbance in ct/ct embryos, summates with the reduced neurulation potential of the neuroepithelium in the Sp/+ genotype leading to the development of severe spina bifida. This study demonstrates that the finding of a significant interaction between genes in double mutant mice cannot be assumed to indicate that the genes operate in the same genetic pathway.</jats:p>",
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