Spinal hemorrhages are associated with early neonatal motor function loss in human spina bifida aperta

Sival, Deborah A; Verbeek, R.J.; Brouwer, Oebele F.; Sollie, Krystyna M.; Bos, Arie; Dunnen, den Wilfred

doi:10.1016/j.earlhumdev.2007.11.003

Cited by 24 publications

(35 citation statements)

References 42 publications

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“…These rates of activity are lower than the 68.8% recorded from muscles in infants with typical development during treadmill stepping (Teulier, Sansom, Muraszko, & Ulrich, in press). Lesioning of the nervous system early in gestation, which reduces transmission of neural impulses to the muscle fibres and impacts abnormal myogenesis may account for some of the differences between the two population groups (Sival et al, 2008). These factors may also account for the lack of any activity recorded in a muscle, averaged across all trials, in 24.4% of stride cycles.…”

Section: Discussionmentioning

confidence: 99%

Changes in muscle activation patterns in response to enhanced sensory input during treadmill stepping in infants born with myelomeningocele

Pantall

Ulrich

2012

Human Movement Science

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Infants with myelomeningocele (MMC) increase step frequency in response to modifications to the treadmill surface. The aim was to investigate how these modifications impacted the electromyographic (EMG) patterns. We analyzed EMG from 19 infants aged 2–10 months, with MMC at the lumbosacral level. We supported infants upright on the treadmill for 12 trials, each 30 seconds long. Modifications included visual flow, unloading, weights, Velcro and lcriction. Surface electrodes recorded EMG from tibialis anterior, lateral gastrocnemius, rectus femoris and biceps femoris. We determined muscle bursts for each stride cycle and from these calculated various parameters. Results indicated that each of the five sensory conditions generated different motor patterns. Visual flow and friction which we previously reported increased step frequency impacted lateral gastrocnemius most. Weights, which significantly decreased step frequency increased burst duration and co-activity of the proximal muscles. We also observed an age effect, with all conditions increasing muscle activity in younger infants whereas in older infants visual flow and unloading stimulated most activity. In conclusion, we have demonstrated that infants with myelomeningocele at levels which impact the myotomes of major locomotor muscles find ways to respond and adapt their motor output to changes in sensory input.

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Section: Discussionmentioning

confidence: 99%

Changes in muscle activation patterns in response to enhanced sensory input during treadmill stepping in infants born with myelomeningocele

Pantall

Ulrich

2012

Human Movement Science

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“…Indirect support for this hypothesis is provided by Thevenet and Sengel [40] , who evaluated the impact of oligohydramnios on wound healing in an avian model and found that with reduction of the amniotic fluid volume the chick embryos develop pressure necrosis, skin erosion and abrasion in prominent areas of the body. Of note, erosions, abrasions and pressure necrosis of the exposed neural elements in human MMC fetuses have been reported [24,25,31] .…”

Section: Embryology and Pathophysiologymentioning

confidence: 99%

“…More recently, it has been postulated that ependymal denudation found in human MMC fetuses at as early as 16 weeks of gestation may precede or even trigger the de-velopment of secondary acquired neurological injury to the exposed spinal cord [29][30][31] . Ependymal differentiation occurs in a fixed temporal and spatial pattern starting at about 4 weeks of gestation and is completed during the second trimester [32] .…”

Section: Embryology and Pathophysiologymentioning

confidence: 99%

“…ependymal denudation has occurred), progenitor cells are lost and functional restoration may be impossible. Ependymal denudation is followed by macrophage invasion into the denuded areas, which subsequently leads to increased inflammation, gliosis and fibrosis in the denuded area [31] . Consequently, worsening of the pathological neurodevelopmental processes associated with MMC may result.…”

Section: Embryology and Pathophysiologymentioning

confidence: 99%

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Fetal Surgery for Myelomeningocele: Patient Selection, Perioperative Management and Outcomes

Danzer

Adzick²

2011

Fetal Diagn Ther

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Myelomeningocele (MMC), one of the most common congenital malformations, can result in severe lifelong disabilities, including paraplegia, hydrocephalus, Chiari II malformation (CM-II), incontinence, sexual dysfunction, skeletal deformations and mental impairment. MMC was the first nonlethal anomaly to be treated by fetal surgery. Experimental and clinical evidence suggests that the primary cause of the neurologic deficit associated with MMC is not simply incomplete neurulation but rather chronic mechanical injury and amniotic fluid-induced chemical trauma that progressively damages the exposed neural tissue during gestation. Case series and a prospective, randomized study show that fetal surgery for MMC before 26 weeks’ gestation may preserve neurologic function, reverse the hindbrain herniation of the CM-II and obviate the need for postnatal placement of a ventriculoperitoneal shunt. However, these studies also demonstrate that fetal surgery is associated with significant maternal and fetal risks. Consequently, further research is warranted to further expand our understanding of the pathophysiology of MMC, to evaluate the long-term impact of in utero intervention and to refine the timing and technique of fetal MMC surgery.

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“…This is referred to as the ‘two‐hit hypothesis’ . Many histopathological and clinical observations support this premise . There is a consensus that the prolonged intrauterine exposure of spinal cord tissue leads to progressive deterioration.…”

Section: Experimental and Clinical Support For Fetal MMC Surgerymentioning

confidence: 99%

Fetal surgical intervention for myelomeningocele: lessons learned, outcomes, and future implications

Danzer

Joyeux

Flake

et al. 2019

Develop Med Child Neuro

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Fetal myelomeningocele (fMMC) closure (spina bifida aperta) has become a care option for patients that meet inclusion criteria, but it is clear that fetal intervention, while improving outcomes, is not a cure. This review will: (1) focus on the rationale for fMMC surgery based on preclinical studies and observations that laid the foundation for human pilot studies and a randomized controlled trial; (2) summarize important clinical outcomes; (3) discuss the feasibility, efficacy, and safety of recent developments in fetal surgical techniques and approaches; and (4) highlight future research directions. Given the increased risk of maternal and fetal morbidity associated with prenatal intervention, accompanied by the increasing number of centres performing interventions worldwide, teams involved in the care of these patients need to proceed with caution to maintain technical expertise, competency, and patient safety. Ongoing assessment of durability of the benefits of fMMC surgery, as well as additional refinement of patient selection criteria and counselling, is needed to further improve outcomes and reduce the risks to the mother and fetus. What this paper adds High‐quality prospective studies are needed to broaden the indication for fetal surgery in the general myelomeningocele population. Innovative minimally invasive approaches have had promising results, yet lack comprehensive and robust experimental or clinical evaluation. Important information to help families make informed decisions regarding fetal surgery for myelomeningocele is provided.

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Spinal hemorrhages are associated with early neonatal motor function loss in human spina bifida aperta

Cited by 24 publications

References 42 publications

Changes in muscle activation patterns in response to enhanced sensory input during treadmill stepping in infants born with myelomeningocele

Changes in muscle activation patterns in response to enhanced sensory input during treadmill stepping in infants born with myelomeningocele

Fetal Surgery for Myelomeningocele: Patient Selection, Perioperative Management and Outcomes

Fetal surgical intervention for myelomeningocele: lessons learned, outcomes, and future implications

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