A Model for In Utero Endoscopic Treatment of Myelomeningocele

Copeland, Michael; Bruner, Joseph P.; Richards, William O.; Sundell, Håkan; Tulipan, Noel

doi:10.1097/00006123-199309000-00043

Cited by 11 publications

(13 citation statements)

References 18 publications

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“…23 Indeed, studies assessing the feasibility of fetoscopic MMC patch repair in sheep models reported lower PACI pressures 24,25 of 7 to 12 mm Hg. 24,25 In our study, the maternal abdominal wall was closed during insufflation, reflecting more likely the percutaneous fetoscopic approach.…”

Section: Discussionmentioning

confidence: 99%

The effects of partial amniotic carbon dioxide insufflation in an ovine model

et al. 2018

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What's already known about this topic? PACI enables fetoscopic myelomeningocele repair as a minimally invasive alternative to open fetal surgery. PACI causes fetal hypercapnia and acidosis in sheep. Initial clinical series of fetoscopic myelomeningocele repair using PACI was associated with high rates of PPROM and preterm birth. What does this study add? Increasing PACI pressures results in larger disturbances in fetal acid‐base and uterine blood flow. Unheated, nonhumidified PACI exposure leads to fetal membrane inflammation. Important differences in sheep and human uterine and placental structure must be considered in our study interpretation.

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Section: Discussionmentioning

confidence: 99%

The effects of partial amniotic carbon dioxide insufflation in an ovine model

et al. 2018

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“…Since then, many animal studies have described the intrauterine fetal repair of spina bifida aperta [2,3,4,5,6,7,8,9]. The theoretical basis for in utero repair of a spina bifida aperta is the ‘two-hit hypothesis’.…”

Section: Introductionmentioning

confidence: 99%

“…The first in utero spina bifida repair of a human fetus was performed in 1994 and since then more than 200 in utero spina bifida closures have been done [10,11,12]. Various materials, for example, Alloderm, split or full-thickness skin graft, allogeneic bone paste or Xenoderm, have been used for in utero coverage of the spinal cord [1, 6, 7, 9,13,14,15,16]. Intrauterine repair may result in reduction of the degree of hindbrain herniation and the need for postnatal ventricular shunting.…”

Section: Introductionmentioning

confidence: 99%

Delayed Intrauterine Repair of an Experimental Spina Bifida with a Collagen Biomatrix

Eggink¹,

Roelofs²,

Feitz³

et al. 2007

Pediatr Neurosurg

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Background/Purpose: The aim of the study was to evaluate whether a collagen biomatrix is useful for delayed intrauterine coverage of a surgically created spina bifida in a fetal lamb. Methods: In 20 fetal lambs, surgery was performed at 72 or 79 days’ gestation. In 15 lambs a spina bifida was created surgically. In 8 lambs it was covered with a collagen biomatrix 2 weeks later and in 7 lambs it was left uncovered. Five lambs served as sham operated controls. Neurological examination was performed at 1 week of age and afterwards the lambs were sacrificed for further histological evaluation. Results: None of the 5 surviving lambs with the defect covered showed loss of spinal function and the architecture of the spinal cord was preserved in 4 of the 5 lambs. In the uncovered group, 1 of the 4 surviving lambs had loss of spinal function, 5 lambs were available for histological evaluation and 4 of them showed disturbance of the architecture of the spinal cord. Conclusions: Collagen biomatrices can be used for intrauterine coverage of an experimental spina bifida and can preserve the architecture of the spinal cord. Neurological outcome is not different between fetuses with their spinal cord covered and fetuses with uncovered cords.

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“…In the early 1990s the popularization of endoscopic neurosurgery prompted the extension of the results of mechanically induced myelomeningocele to a larger model, the sheep. As in the rodent experiments done earlier, the emphasis was on prevention of the exposure of the spinal cord to amniotic fluid, to minimize putative toxic effects [Copeland et al, 1993]. Meuli also considered the sheep model, using new techniques of open fetal surgery rather than endoscopic covering of the defects [Meuli et al, 1995].…”

Section: Models Of Mechanically Induced Myelomeningocelementioning

confidence: 99%

Pathophysiology, prevention, and potential treatment of neural tube defects

Manning

Jennings

Madsen

2000

Ment. Retard. Dev. Disabil. Res. Rev.

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Neural tube defects (NTD) remain a major cause of morbidity in spite of the reduction in liveborn incidence with periconceptional folic acid. However, the etiology remains unknown. This article reviews studies that address causation and potential treatment of NTD in humans and in animal models that resemble aspects of the common human NTD. Studies of nutritional markers of vitamin B12 and folic acid support a defect in homocysteine metabolism; a thermolabile variant of methylene tetrahydrofolate reductase, an enzyme that remethylates homocysteine to methionine, correlates with a risk of NTD in some human populations. Numerous mouse mutant models of NTD exist, attesting to the ease of disruption of neurulation, and a genetic basis for this malformation. Of these models, the curly tail mouse mutant most closely resembles the common human NTD. Folic acid does not prevent NTD in this model; however inositol supplementation does result in a significant reduction in incidence. Recent advances in fetal surgery, and evidence from mechanically created myelomeningocele in large animals amenable to surgical intervention suggest that the handicaps associated with myelomeningocele and associated Chiari Type II malformation may be prevented by in utero NTD closure. Success will depend on preservation of neurological tissue until such intervention is possible. Further research in animal models at the genetic and cellular levels, together with technological surgical advances, provide hope that prevention of more NTD and the associated handicaps may be possible. MRDD Research Reviews 6:6–14, 2000. © 2000 Wiley‐Liss, Inc.

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A Model for In Utero Endoscopic Treatment of Myelomeningocele

Cited by 11 publications

References 18 publications

The effects of partial amniotic carbon dioxide insufflation in an ovine model

The effects of partial amniotic carbon dioxide insufflation in an ovine model

Delayed Intrauterine Repair of an Experimental Spina Bifida with a Collagen Biomatrix

Pathophysiology, prevention, and potential treatment of neural tube defects

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