Abstract
Objective: The purpose of this study was to determine how closely the surgically induced sheep myelomeningocele (MMC) model resembles the central nervous system derangements seen in human disease, and identify which aspects of MMC are the result of the early neuronal developmental defect, and which are secondary to the cerebrospinal fluid (CSF) drainage. Study design: An MMC-like lesion was created surgically in 16 fetal sheep at 75 days' gestation: 5 died in utero, 7 underwent no fetal repair, 4 were repaired (2-layer closure or biological glue) at 100 days' gestation. MMC sheep were delivered at term and allowed to survive up to 17 days for analysis of behavioral status and feeding behavior. Animals not repaired in utero were repaired at birth. All lambs were sacrificed and analyzed for hindbrain herniation, hydrocephalus, and other CNS derangements. Results: Hindbrain herniation was observed in 43% of animals not repaired in utero, and in 1 lamb repaired with Bioglue. No animal developed hydrocephalus or other CNS derangements. Conclusion: Although this sheep MMC-like model reproduces the CSF leak, but not the developmental defect seen in humans, it suggests CSF leak contributes to hindbrain herniation seen in humans. This model may be useful to develop new minimally invasive techniques to halt CSF leak in utero.
| Original language | English (US) |
|---|---|
| Pages (from-to) | 1456-1462 |
| Number of pages | 7 |
| Journal | American Journal of Obstetrics and Gynecology |
| Volume | 193 |
| Issue number | 4 |
| DOIs | |
| State | Published - Oct 2005 |
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Keywords
- Arnold-Chiari malformation
- Fetal surgery
- Hindbrain herniation
- Myelomeningocele
- Sheep
- Spina bifida
ASJC Scopus subject areas
- Medicine(all)
- Obstetrics and Gynecology
Cite this
Myelomeningocele : Characterization of a surgically induced sheep model and its central nervous system similarities and differences to the human disease. / Von Koch, Cornelia S.; Compagnone, Nathalie; Hirose, Shinjiro; Yoder, Suzanne; Harrison, Michael R.; Farmer, Diana L.
In: American Journal of Obstetrics and Gynecology, Vol. 193, No. 4, 10.2005, p. 1456-1462.Research output: Contribution to journal › Article
}
TY - JOUR
T1 - Myelomeningocele
T2 - Characterization of a surgically induced sheep model and its central nervous system similarities and differences to the human disease
AU - Von Koch, Cornelia S.
AU - Compagnone, Nathalie
AU - Hirose, Shinjiro
AU - Yoder, Suzanne
AU - Harrison, Michael R.
AU - Farmer, Diana L
PY - 2005/10
Y1 - 2005/10
N2 - Objective: The purpose of this study was to determine how closely the surgically induced sheep myelomeningocele (MMC) model resembles the central nervous system derangements seen in human disease, and identify which aspects of MMC are the result of the early neuronal developmental defect, and which are secondary to the cerebrospinal fluid (CSF) drainage. Study design: An MMC-like lesion was created surgically in 16 fetal sheep at 75 days' gestation: 5 died in utero, 7 underwent no fetal repair, 4 were repaired (2-layer closure or biological glue) at 100 days' gestation. MMC sheep were delivered at term and allowed to survive up to 17 days for analysis of behavioral status and feeding behavior. Animals not repaired in utero were repaired at birth. All lambs were sacrificed and analyzed for hindbrain herniation, hydrocephalus, and other CNS derangements. Results: Hindbrain herniation was observed in 43% of animals not repaired in utero, and in 1 lamb repaired with Bioglue. No animal developed hydrocephalus or other CNS derangements. Conclusion: Although this sheep MMC-like model reproduces the CSF leak, but not the developmental defect seen in humans, it suggests CSF leak contributes to hindbrain herniation seen in humans. This model may be useful to develop new minimally invasive techniques to halt CSF leak in utero.
AB - Objective: The purpose of this study was to determine how closely the surgically induced sheep myelomeningocele (MMC) model resembles the central nervous system derangements seen in human disease, and identify which aspects of MMC are the result of the early neuronal developmental defect, and which are secondary to the cerebrospinal fluid (CSF) drainage. Study design: An MMC-like lesion was created surgically in 16 fetal sheep at 75 days' gestation: 5 died in utero, 7 underwent no fetal repair, 4 were repaired (2-layer closure or biological glue) at 100 days' gestation. MMC sheep were delivered at term and allowed to survive up to 17 days for analysis of behavioral status and feeding behavior. Animals not repaired in utero were repaired at birth. All lambs were sacrificed and analyzed for hindbrain herniation, hydrocephalus, and other CNS derangements. Results: Hindbrain herniation was observed in 43% of animals not repaired in utero, and in 1 lamb repaired with Bioglue. No animal developed hydrocephalus or other CNS derangements. Conclusion: Although this sheep MMC-like model reproduces the CSF leak, but not the developmental defect seen in humans, it suggests CSF leak contributes to hindbrain herniation seen in humans. This model may be useful to develop new minimally invasive techniques to halt CSF leak in utero.
KW - Arnold-Chiari malformation
KW - Fetal surgery
KW - Hindbrain herniation
KW - Myelomeningocele
KW - Sheep
KW - Spina bifida
UR - http://www.scopus.com/inward/record.url?scp=25844443761&partnerID=8YFLogxK
UR - http://www.scopus.com/inward/citedby.url?scp=25844443761&partnerID=8YFLogxK
U2 - 10.1016/j.ajog.2005.02.110
DO - 10.1016/j.ajog.2005.02.110
M3 - Article
C2 - 16202740
AN - SCOPUS:25844443761
VL - 193
SP - 1456
EP - 1462
JO - American Journal of Obstetrics and Gynecology
JF - American Journal of Obstetrics and Gynecology
SN - 0002-9378
IS - 4
ER -