Abstract
Background
This study aimed to assess the feasibility of single-access fetal endoscopy (SAFE) for the management of myelomeningocele (MMC) using intrauterine carbon dioxide as a distension medium in a sheep model.
Methods
This prospective experimental case-control study investigated 12 lamb fetuses that had a myelomeningocele-like defect surgically created on the 75th day of gestation. Four fetuses remained untreated (control group), and eight fetuses had MMC repair using two fetoscopic approaches with carbon dioxide used to distend the amniotic cavity. A collagen patch was placed over the defect and secured with surgical sealant. Four animals had a two-port fetoscopic procedure, and four animals had SAFE. Clinical and pathologic studies were performed after delivery.
Results
This study confirmed the validity of the animal MMC model. None of the control animals was able to stand or walk, and all had a significant defect in the lumbar area with continuous leakage of cerebrospinal fluid, ventriculomegaly, and a Chiari-II malformation. All the treated animals, independently of the number of ports used in the repair, were able to walk and had a closed defect with resolution of the Chiari malformation.
Conclusions
The SAFE patch and glue coverage of surgically created fetal MMC is feasible and effective in restoring gross neurologic function in the fetal lamb model.
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References
Au KS, Ashley-Koch A, Northrup H (2010) Epidemiologic and genetic aspects of spina bifida and other neural tube defects. Dev Disabil Res Rev 16:6–15
Amari F, Junkers W, Hartge D, Beyer DA, Axt-Fliedner R, Weichert J (2010) Prenatal course and outcome in 103 cases of fetal spina bifida: a single-center experience. Acta Obstet Gynecol Scand 89:1276–1283
Adzick NS (2010) Fetal myelomeningocele: natural history, pathophysiology, and in utero intervention. Semin Fetal Neonatal Med 15:9–14
Adzick NS, Thom EA, Spong CY, Brock JW III, Burrows PK, Johnson MP, Howell LJ, Farrell JA, Dabrowiak ME, Sutton LN, Gupta N, Tulipan NB, D’Alton ME, Farmer DL, MOMS Investigators (2011) A randomized trial of prenatal versus postnatal repair of myelomeningocele. N Engl J Med 364:993–1004
Fontecha CG, Peiro JL, Aguirre M, Soldado F, Añor S, Fresno L, Martinez-Ibañez V (2009) Inert patch with bioadhesive for gentle fetal surgery of myelomeningocele in a sheep model. Eur J Obstet Gynecol Reprod Biol 146:174–179
Fontecha CG, Peiro JL, Sevilla JJ, Aguirre M, Soldado F, Fresno L, Fonseca C, Chacaltana A, Martinez V (2011) Fetoscopic coverage of experimental myelomeningocele in sheep using a patch with surgical sealant. Eur J Obstet Gynecol Reprod Biol 156:171–176
Pedreira DA, Sanchez-Oliveira RC, Valente PR, Abou-Jamra RC, Araujo A, Saldiva PH (2007) Validation of the ovine fetus as an experimental model for the human myelomeningocele defect. Acta Cir Bras 22:168–173
Kohl T, Tchatcheva K, Merz W, Wartenberg HC, Heep A, Müller A, Franz A, Stressig R, Willinek W, Gembruch U (2009) Percutaneous fetoscopic patch closure of human spina bifida aperta: advances in fetal surgical techniques may obviate the need for early postnatal neurosurgical intervention. Surg Endosc 23:890–895
Copeland ML, Bruner JP, Richards WO, Sundell HW, Tulipan NB (1993) A model for in utero endoscopic treatment of myelomeningocele. Neurosurgery 33:542–544
Bruner JP, Richards WO, Tulipan NB, Arney TL (1999) Endoscopic coverage of fetal myelomeningocele in utero. Am J Obstet Gynecol 180(1 Pt 1):153–158
Bruner JP, Tulipan NB, Richards WO, Walsh WF, Boehm FH, Vrabcak EK (2000) In utero repair of myelomeningocele: a comparison of endoscopy and hysterotomy. Fetal Diagn Ther 15:83–88
Encinas JL, García-Cabezas MA, Barkovich J, Fontecha CG, Peiró JL, Soto GM, Borrell V, Reillo I, López-Santamaría M, Tovar JA, Farmer DL (2011) Maldevelopment of the cerebral cortex in the surgically induced model of myelomeningocele: implications for fetal neurosurgery. J Pediatr Surg 46:713–722
Peiró JL, Encinas JL (2012) Fetal surgery of myelomeningocele: from experimental results to clinical evidence. Cir Pediatr 25:1–3
Kohl T (2010) Minimally invasive fetoscopic interventions: an overview in 2010. Surg Endosc 24:2056–2067
Disclosures
Jose L. Peiro, Cesar G. Fontecha, Rodrigo Ruano, Marielle Esteves, Carla Fonseca, Mario Marotta, Sina Haeri, and Michael A. Belfort have no conflicts of interest or financial ties to disclose.
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Peiro, J.L., Fontecha, C.G., Ruano, R. et al. Single-Access Fetal Endoscopy (SAFE) for myelomeningocele in sheep model I: amniotic carbon dioxide gas approach. Surg Endosc 27, 3835–3840 (2013). https://doi.org/10.1007/s00464-013-2984-6
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DOI: https://doi.org/10.1007/s00464-013-2984-6