Abstract
Purpose: The purpose of this study is to describe the malformations of cortical development detected in a model of cerebrospinal fluid (CSF) leakage and the influence of surgical closure technique on developmental outcome. Methods: Using a surgically induced model of myelomeningocele (MMC) in sheep, we studied the effects of different repair methods upon the development of hydrocephalus, the presence of the Arnold-Chiari II (AC-II) hindbrain malformation, and cerebral cortex developmental anomalies using gross and histologic (hematoxylin and eosin and Nissl staining) study techniques. Results: A malformed cerebral cortex, including 2 anomalous cortical folding patterns, and lower brain weights were observed in the untreated animals. Hydrocephalus and AC-II malformations were also found in this group. These malformations were mostly prevented with prenatal 2-layer closure. Conclusions: Cerebral cortical malformations and hydrocephalus, in addition to the AC-II hindbrain malformation, are disorders caused by fetal CSF leakage. These malformations were prevented with the technique of MMC closure currently used in humans. Both observations magnify the importance of the second hit associated with chronic CSF leakage, in addition to the primary defect causing the MMC, in the development of the malformation complex.
| Original language | English (US) |
|---|---|
| Pages (from-to) | 713-722 |
| Number of pages | 10 |
| Journal | Journal of Pediatric Surgery |
| Volume | 46 |
| Issue number | 4 |
| DOIs | |
| State | Published - Apr 2011 |
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Keywords
- Brain
- Cortex
- Fetal surgery
- Folding
- Myelomeningocele
- Sheep
ASJC Scopus subject areas
- Surgery
- Pediatrics, Perinatology, and Child Health
Cite this
Maldevelopment of the cerebral cortex in the surgically induced model of myelomeningocele : Implications for fetal neurosurgery. / Encinas, Jose L.; García-Cabezas, Miguel Ángel; Barkovich, James; Fontecha, César G.; Peiró, Jose L.; Soto, Garriboli M Carmen; Borrell, Victor; Reillo, Isabel; López-Santamaría, Manuel; Tovar, Juan A.; Farmer, Diana L.
In: Journal of Pediatric Surgery, Vol. 46, No. 4, 04.2011, p. 713-722.Research output: Contribution to journal › Article
}
TY - JOUR
T1 - Maldevelopment of the cerebral cortex in the surgically induced model of myelomeningocele
T2 - Implications for fetal neurosurgery
AU - Encinas, Jose L.
AU - García-Cabezas, Miguel Ángel
AU - Barkovich, James
AU - Fontecha, César G.
AU - Peiró, Jose L.
AU - Soto, Garriboli M Carmen
AU - Borrell, Victor
AU - Reillo, Isabel
AU - López-Santamaría, Manuel
AU - Tovar, Juan A.
AU - Farmer, Diana L
PY - 2011/4
Y1 - 2011/4
N2 - Purpose: The purpose of this study is to describe the malformations of cortical development detected in a model of cerebrospinal fluid (CSF) leakage and the influence of surgical closure technique on developmental outcome. Methods: Using a surgically induced model of myelomeningocele (MMC) in sheep, we studied the effects of different repair methods upon the development of hydrocephalus, the presence of the Arnold-Chiari II (AC-II) hindbrain malformation, and cerebral cortex developmental anomalies using gross and histologic (hematoxylin and eosin and Nissl staining) study techniques. Results: A malformed cerebral cortex, including 2 anomalous cortical folding patterns, and lower brain weights were observed in the untreated animals. Hydrocephalus and AC-II malformations were also found in this group. These malformations were mostly prevented with prenatal 2-layer closure. Conclusions: Cerebral cortical malformations and hydrocephalus, in addition to the AC-II hindbrain malformation, are disorders caused by fetal CSF leakage. These malformations were prevented with the technique of MMC closure currently used in humans. Both observations magnify the importance of the second hit associated with chronic CSF leakage, in addition to the primary defect causing the MMC, in the development of the malformation complex.
AB - Purpose: The purpose of this study is to describe the malformations of cortical development detected in a model of cerebrospinal fluid (CSF) leakage and the influence of surgical closure technique on developmental outcome. Methods: Using a surgically induced model of myelomeningocele (MMC) in sheep, we studied the effects of different repair methods upon the development of hydrocephalus, the presence of the Arnold-Chiari II (AC-II) hindbrain malformation, and cerebral cortex developmental anomalies using gross and histologic (hematoxylin and eosin and Nissl staining) study techniques. Results: A malformed cerebral cortex, including 2 anomalous cortical folding patterns, and lower brain weights were observed in the untreated animals. Hydrocephalus and AC-II malformations were also found in this group. These malformations were mostly prevented with prenatal 2-layer closure. Conclusions: Cerebral cortical malformations and hydrocephalus, in addition to the AC-II hindbrain malformation, are disorders caused by fetal CSF leakage. These malformations were prevented with the technique of MMC closure currently used in humans. Both observations magnify the importance of the second hit associated with chronic CSF leakage, in addition to the primary defect causing the MMC, in the development of the malformation complex.
KW - Brain
KW - Cortex
KW - Fetal surgery
KW - Folding
KW - Myelomeningocele
KW - Sheep
UR - http://www.scopus.com/inward/record.url?scp=79954493103&partnerID=8YFLogxK
UR - http://www.scopus.com/inward/citedby.url?scp=79954493103&partnerID=8YFLogxK
U2 - 10.1016/j.jpedsurg.2010.11.028
DO - 10.1016/j.jpedsurg.2010.11.028
M3 - Article
C2 - 21496543
AN - SCOPUS:79954493103
VL - 46
SP - 713
EP - 722
JO - Journal of Pediatric Surgery
JF - Journal of Pediatric Surgery
SN - 0022-3468
IS - 4
ER -