Trans-synaptic Retrograde Degeneration Following Hemispherectomy in Childhood

Handley, Siân E.; Panteli, Vasiliki; Liasis, Alki

doi:10.1080/01658107.2016.1276935

Cited by 8 publications

(2 citation statements)

References 13 publications

(29 reference statements)

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“…To the best of our knowledge MMO has yet to be reported in cases with isolated lesions to the posterior visual pathways. Notably MMO was absent in a case with severe GCIPL atrophy following hemispherectomy for intractable childhood epilepsy (Handley et al 2017).…”

Section: Brain 2019mentioning

confidence: 86%

Patterns of retrograde axonal degeneration in the visual system

Vries-Knoppert

Baaijen

Petzold

2019

Brain

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Conclusive evidence for existence of acquired retrograde axonal degeneration that is truly trans-synaptic (RTD) has not yet been provided for the human visual system. Convincing data rely on experimental data of lesions to the posterior visual pathways. This study aimed to overcome the limitations of previous human studies, namely pathology to the anterior visual pathways and neurodegenerative co-morbidity. In this prospective, longitudinal cohort retinal optical coherence tomography scans were acquired before and after elective partial temporal lobe resection in 25 patients for intractable epilepsy. Newly developed region of interest-specific, retinotopic areas substantially improved on conventional reported early treatment diabetic retinopathy study (ETDRS) grid-based optical coherence tomography data. Significant inner retinal layer atrophy separated patients with normal visual fields from those who developed a visual field defect. Acquired RTD affected the retinal nerve fibre layer, ganglion cell and inner plexiform layer and stopped at the level of the inner nuclear layer. There were significant correlations between the resected brain tissue volume and the ganglion cell layer region of interest (R = −0.78, P < 0.0001) and ganglion cell inner plexiform layer region of interest (R = −0.65, P = 0.0007). In one patient, damage to the anterior visual pathway resulted in occurrence of microcystic macular oedema as recognized from experimental data. In the remaining 24 patients with true RTD, atrophy rates in the first 3 months were strongly correlated with time from surgery for the ganglion cell layer region of interest (R = −0.74, P < 0.0001) and the ganglion cell inner plexiform layer region of interest (R = −0.51, P < 0.0001). The different time course of atrophy rates observed relate to brain tissue volume resection and suggest that three distinct patterns of retrograde axonal degeneration exist: (i) direct retrograde axonal degeneration; (ii) rapid and self-terminating RTD; and (iii) prolonged RTD representing a ‘penumbra’, which slowly succumbs to molecularly governed spatial cellular stoichiometric relationships. We speculate that the latter could be a promising target for neuroprotection.

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Section: Brain 2019mentioning

confidence: 86%

Patterns of retrograde axonal degeneration in the visual system

Vries-Knoppert

Baaijen

Petzold

2019

Brain

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“…In retinitis pigmentosa (RP) OCT scans have shown to correlate well to preserved central visual function [ 9 ]. Damage to the visual pathway causes trans-synaptic degeneration (TSD), which can be detected in the eye using OCT [ 11 , 12 ]. However, the time course to this degeneration that has been demonstrated in adults has been shown to have an age effect [ 11 ].…”

Section: Introductionmentioning

confidence: 99%

What can visual electrophysiology tell about possible visual-field defects in paediatric patients

Handley

Šuštar

Pompe

2021

Eye

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Recognising a potential visual-field (VF) defect in paediatric patients might be challenging, especially in children before the age of 5 years and those with developmental delay or intellectual disability. Visual electrophysiological testing is an objective and non-invasive technique for evaluation of visual function in paediatric patients, which can characterise the location of dysfunction and differentiate between disorders of the retina, optic nerve and visual pathway. The recording of electroretinography (ERG) and visual-evoked potentials (VEP) is possible from early days of life and requires no subjective input from the patient. As the origins of ERG and VEP tests are known, the pattern of electrophysiological changes can provide information about the VF of a child unable to perform accurate perimetry. This review summarises previously published electrophysiological findings in several common types of VF defects that can be found in paediatric patients (generalised VF defect, peripheral VF loss, central scotoma, bi-temporal hemianopia, altitudinal VF defect, quadrantanopia and homonymous hemianopia). It also shares experience on using electrophysiological testing as additional functional evidence to other tests in the clinical challenge of diagnosing or excluding VF defects in complex paediatric patients. Each type of VF defect is illustrated with one or two clinical cases.

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