Regression of polyneural innervation in the human psoas muscle

Gramsbergen, Albert; Ijkema‐Paassen, Jos; Nikkels, Pgj; Hadders‐Algra, Mijna

doi:10.1016/s0378-3782(97)01876-8

Cited by 20 publications

(8 citation statements)

References 38 publications

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“…Therefore, polyneuronal innervation causes an apparent reduction in MUNE measurements due to overlapping territories of motor units 31. In contrast, the existing evidence from a single study in humans suggests that monosynaptic innervation is evident at birth 42. However, further studies are required to be absolutely certain that this is the case.…”

Section: Resultsmentioning

confidence: 99%

Electrophysiological biomarkers in spinal muscular atrophy: proof of concept

Arnold

Porensky

McGovern

et al. 2013

Ann Clin Transl Neurol

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ObjectivePreclinical therapies that restore survival motor neuron (SMN) protein levels can dramatically extend survival in spinal muscular atrophy (SMA) mouse models. Biomarkers are needed to effectively translate these promising therapies to clinical trials. Our objective was to investigate electrophysiological biomarkers of compound muscle action potential (CMAP), motor unit number estimation (MUNE) and electromyography (EMG) using an SMA mouse model.MethodsSciatic CMAP, MUNE, and EMG were obtained in SMNΔ7 mice at ages 3–13 days and at 21 days in mice with SMN selectively reduced in motor neurons (ChATCre). To investigate these measures as biomarkers of treatment response, measurements were obtained in SMNΔ7 mice treated with antisense oligonucleotide (ASO) or gene therapy.ResultsCMAP was significantly reduced in SMNΔ7 mice at days 6–13 (P < 0.01), and MUNE was reduced at days 7–13 (P < 0.01). Fibrillations were present on EMG in SMNΔ7 mice but not controls (P = 0.02). Similar findings were seen at 21 days in ChATCre mice. MUNE in ASO-treated SMNΔ7 mice were similar to controls at day 12 and 30. CMAP reduction persisted in ASO-treated SMNΔ7 mice at day 12 but was corrected at day 30. Similarly, CMAP and MUNE responses were corrected with gene therapy to restore SMN.InterpretationThese studies confirm features of preserved neuromuscular function in the early postnatal period and subsequent motor unit loss in SMNΔ7 mice. SMN restoring therapies result in preserved MUNE and gradual repair of CMAP responses. This provides preclinical evidence for the utilization of CMAP and MUNE as biomarkers in future SMA clinical trials.

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Section: Resultsmentioning

confidence: 99%

Electrophysiological biomarkers in spinal muscular atrophy: proof of concept

Arnold

Porensky

McGovern

et al. 2013

Ann Clin Transl Neurol

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“…Because multiple axons on each endplate have been noted in the developing muscles of a variety of mammals (Riley, 1976), including humans (Gramsbergen et al, 1997), transient hyperinnervation appears to be the general rule for establishing the motor innervation of the skeletal muscles. In the present study, the percentage of polyneuronal innervation was considerably higher in the alcoholexposed rats than in the controls.…”

Section: Discussionmentioning

confidence: 99%

Prenatal alcohol exposure and early postnatal changes in the developing nerve-muscle system

David

Krishnan

2005

Birth Defect Res A

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“…2 This technique was also applied in a recent study into polyneural innervation of the psoas muscle in the human. 15 With this method the acetylcholinesterase in the end-plate is stained, and the axons are impregnated with silver. Inherent to evaluation of polyneural innervation by histological investigations are a few peculiarities which should be kept in mind.…”

Section: Discussionmentioning

confidence: 99%

“…More recently, innervation patterns were studied in the psoas muscle in human fetuses from 15 weeks PMA and in babies until 45 weeks after term age as well as in 2 adults. 15 Until 25 weeks PMA, maximally five endings per end-plate were observed, and this decreased from then onwards. At term age still one out of 10 end-plates contains two axons.…”

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confidence: 98%

Polyneural innervation in the psoas muscle of the developing rat

Ijkema‐Paassen¹,

Gramsbergen²

1998

Muscle Nerve

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Polyneural innervation was studied in the psoas muscle in developing rats from P4 till P25 and at adult age, with the combined silver–acetylcholinesterase technique. Nerve endings were counted, and end‐plates were measured. These data were compared with such data in the human. The end of polyneural innervation in the rat (around P20) and in the human (around 12 weeks postterm age) in both cases coincides with a transformation in motor behavior and postural control. The rat's psoas muscle at early stages is less heavily innervated than this muscle in the human. Up to three axons per motor end‐plate were counted at P4, but in the human up to five axons at 25 weeks of post menstrual age. This difference might be related to the lower percentage of type I muscle fibers in the rat. © 1998 John Wiley & Sons, Inc. Muscle Nerve 21:1058–1063, 1998.

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Regression of polyneural innervation in the human psoas muscle

Cited by 20 publications

References 38 publications

Electrophysiological biomarkers in spinal muscular atrophy: proof of concept

Electrophysiological biomarkers in spinal muscular atrophy: proof of concept

Prenatal alcohol exposure and early postnatal changes in the developing nerve-muscle system

Polyneural innervation in the psoas muscle of the developing rat

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