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Heritable spina bifida in sheep: A potential model for fetal repair of myelomeningocele.

Publication ,  Journal Article
Steele, JW; Bayliss, S; Bayliss, J; Lin, YL; Wlodarczyk, BJ; Cabrera, RM; Asfaw, YG; Cummings, TJ; Finnell, RH; George, TM
Published in: J Pediatr Surg
March 2020

BACKGROUND/PURPOSE: In 2004, a heritable occurrence of spina bifida was reported in sheep on a farm in the United States. We maintained and characterized the spina bifida phenotype in this flock to assess its potential as an alternative surgical model. METHODS: A breeding strategy was developed in which the sheep were crossed to maintain or increase the occurrence of spina bifida. Measurements and observations were recorded regarding lesion size, birthweight, ambulatory capacity, or urological function, and necropsies were performed on spina bifida afflicted lambs in conjunction with magnetic resonance imaging to determine the character of the spina bifida defects and assess the presence of Chiari-like malformations or hydrocephalus. RESULTS: The defects were observed to be more prevalent in ram lambs, and the rate of spina bifida per litter could be increased through backcrossing or by selection of a productive ewe breed. The lambs displayed a range of ambulatory and urological deficits which could be used to evaluate new fetal repair methodologies. Finally, affected lambs were shown to demonstrate severe Chiari malformations and hydrocephalus. CONCLUSIONS: We have determined that use of these sheep as a natural source for spina bifida fetuses is feasible and could supplement the deficits of current sheep models for myelomeningocele repair. LEVEL OF EVIDENCE: Level IV.

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Published In

J Pediatr Surg

DOI

EISSN

1531-5037

Publication Date

March 2020

Volume

55

Issue

3

Start / End Page

475 / 481

Location

United States

Related Subject Headings

  • Spinal Dysraphism
  • Sheep
  • Pregnancy
  • Pediatrics
  • Meningomyelocele
  • Fetoscopy
  • Female
  • Disease Models, Animal
  • Animals
  • 3213 Paediatrics
 

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Steele, J. W., Bayliss, S., Bayliss, J., Lin, Y. L., Wlodarczyk, B. J., Cabrera, R. M., … George, T. M. (2020). Heritable spina bifida in sheep: A potential model for fetal repair of myelomeningocele. J Pediatr Surg, 55(3), 475–481. https://doi.org/10.1016/j.jpedsurg.2019.06.019
Steele, John W., Sharon Bayliss, John Bayliss, Ying Linda Lin, Bogdan J. Wlodarczyk, Robert M. Cabrera, Yohannes G. Asfaw, Thomas J. Cummings, Richard H. Finnell, and Timothy M. George. “Heritable spina bifida in sheep: A potential model for fetal repair of myelomeningocele.J Pediatr Surg 55, no. 3 (March 2020): 475–81. https://doi.org/10.1016/j.jpedsurg.2019.06.019.
Steele JW, Bayliss S, Bayliss J, Lin YL, Wlodarczyk BJ, Cabrera RM, et al. Heritable spina bifida in sheep: A potential model for fetal repair of myelomeningocele. J Pediatr Surg. 2020 Mar;55(3):475–81.
Steele, John W., et al. “Heritable spina bifida in sheep: A potential model for fetal repair of myelomeningocele.J Pediatr Surg, vol. 55, no. 3, Mar. 2020, pp. 475–81. Pubmed, doi:10.1016/j.jpedsurg.2019.06.019.
Steele JW, Bayliss S, Bayliss J, Lin YL, Wlodarczyk BJ, Cabrera RM, Asfaw YG, Cummings TJ, Finnell RH, George TM. Heritable spina bifida in sheep: A potential model for fetal repair of myelomeningocele. J Pediatr Surg. 2020 Mar;55(3):475–481.
Journal cover image

Published In

J Pediatr Surg

DOI

EISSN

1531-5037

Publication Date

March 2020

Volume

55

Issue

3

Start / End Page

475 / 481

Location

United States

Related Subject Headings

  • Spinal Dysraphism
  • Sheep
  • Pregnancy
  • Pediatrics
  • Meningomyelocele
  • Fetoscopy
  • Female
  • Disease Models, Animal
  • Animals
  • 3213 Paediatrics