Cross‐sectional study of a United Kingdom cohort of neonatal vein of galen malformation

Lecce, Francesca; Robertson, Fergus; Rennie, Adam; Heuchan, Anne-Marie; Lister, Paula; Bhate, Sanjay; Bhattacharya, J. J.; Brew, Stefan; Kanagarajah, Lakshmi; Kuczynski, Adam M.; Peters, Mark; Ridout, Deborah; Schmitt, Anne; Toolis, Claire; Vargha‐Khadem, Faraneh; Ganesan, Vijeya

doi:10.1002/ana.25316

Cited by 41 publications

(37 citation statements)

References 14 publications

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“…In a recent review of the literature, Gopalan et al identified good outcome in 65 per cent of patients treated by embolization but did not determine average time of evaluation. A following publication from this group, however, described longer‐term evaluation (median age at testing: 5y 2mo, range: 1–11y) and noted similar results to us: that only half of the surviving patients had a good outcome …”

Section: Discussionsupporting

confidence: 78%

Long‐term outcome of vein of Galen malformation

Taffin

Maurey

Ozanne

et al. 2019

Develop Med Child Neuro

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Aim To describe the long‐term outcomes of children by the time they reached school age with vein of Galen aneurysmal malformation (VGAM). Method This was a retrospective observational study on a consecutive cohort of patients with VGAM. We included patients with at least one Francophone parent, aged between 6 and 11 years at the time of long‐term evaluation. The neurological outcome was assessed with the King’s Outcome Scale for Childhood Injury score and eight neurological and behavioural items from the Rivermead Postconcussion Symptoms questionnaire. Results All 52 patients (17 females, 32 males [data missing for n=3]) with at least one Francophone parent (5 fetuses and 47 children) were included. At the long‐term evaluation time‐point, 33 patients were alive and 19 patients had died. Risk of postnatal death was associated with severe neonatal cardiac failure (p=0.007) or isosystemic or suprasystemic pulmonary hypertension (p=0.014). Among survivors, 19 had a good outcome with normal schooling and 14 had a poor outcome. Moreover, among the good outcome patients, a large proportion had neurodevelopmental alterations. Interpretation Long‐term outcome of patients with VGAM appears to be less favourable than outcome described at the short‐ and medium‐term, even in the absence of encephalomalacia at birth. Even patients with good outcome often have neuropsychological disorders that may have repercussions on learning and requiring appropriate rehabilitation or medical management. What this paper adds Long‐term outcome appears to be less favourable than described at short‐ and medium‐term follow‐up. Even patients with good outcome at these time‐points often have minor neuropsychological disorders.

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Section: Discussionsupporting

confidence: 78%

Long‐term outcome of vein of Galen malformation

Taffin

Maurey

Ozanne

et al. 2019

Develop Med Child Neuro

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“…Thus, previous reports may underestimate the rate of overall impairments. Indeed, only two studies have reported on the neuropsychological outcomes of VGAM, one describing outcomes of neonatal VGAM14 and the other a case of a child treated during her preschool years 15. Lecce and colleagues14 document good outcomes in around half of neonatal VGAM survivors, but a closer review of their data shows 74% of their sample had at least one poor outcome.…”

Section: Discussionmentioning

confidence: 99%

“…Indeed, only two studies have reported on the neuropsychological outcomes of VGAM, one describing outcomes of neonatal VGAM14 and the other a case of a child treated during her preschool years 15. Lecce and colleagues14 document good outcomes in around half of neonatal VGAM survivors, but a closer review of their data shows 74% of their sample had at least one poor outcome. This rate is lower but not too dissimilar to ours, with our differences likely attributed to varying ages at investigation and cut-offs for determining problematic outcomes.…”

Section: Discussionmentioning

confidence: 99%

Outcome of vein of Galen malformation presenting in the neonatal period

et al. 2019

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ObjectiveVein of Galenaneurysmal malformation (VGAM) is a rare but important congenital malformation presenting to neonatal intensive care units (NICUs), and with a change from surgical to endovascular management, survival for this condition has improved. However, there is little reported about the medical management decisions of infants with this condition and the associated long-term neurodevelopmental outcomes. We aim to report a single centre experience of both acute treatment and long-term outcomes of VGAM for those infants admitted to our NICU soon after birth.DesignRetrospective cohort study over a 15-year period from 2001 to 2015 inclusive.SettingA quaternary NICU at The Royal Children’s Hospital, Melbourne, Australia.Participants24 newborn infants referred for management of VGAM. There were no eligibility criteria set for this study; all presenting infants were included.InterventionsNone.Main outcomes measuresClinical neuroimaging data were gathered. Surviving children were formally assessed with a battery of tests administered by a neuropsychologist and occupational therapist/physiotherapist at various ages across early to middle childhood.ResultsFifteen neonates with VGAM did not survive beyond their NICU admission. 10 of these were not offered endovascular intervention. Of the nine surviving infants, only one had a normal neurodevelopmental outcome.ConclusionsThe mortality of VGAM presenting in the neonatal period was high, and rates of normal neurodevelopmental outcome for survivors were low. These findings contribute to our understanding of which neonates should be treated and highlights the importance of providing clinical neurodevelopmental follow-up to survivors beyond their infant years.

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“…The observed deficits were speech delay, cognitive disabilities, and behavioural difficulties. A following publication 35 of this group described longer‐term follow‐up and noted that only half of the surviving patients had a good outcome. These results were confirmed by a recent French study, 29 in which the long‐term evaluation described was much poorer.…”

Section: Vgammentioning

confidence: 99%

Endovascular treatment of intracranial vascular malformations in children

Lubicz

Christiaens

2020

Develop Med Child Neuro

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Paediatric intracranial vascular malformations are rare and different from adult ones in vascular anatomy, pathophysiology, and symptoms. Their impact on the brain and their symptoms will differ in the antenatal period, in neonates, infants, and children. Clinical presentation includes seizures, focal neurological deficit, haemorrhage, congestive heart failure, hydrovenous disorder, and developmental delays. These malformations are thus associated with a poor prognosis if left untreated. Therefore, aggressive management is generally recommended and must be performed by a multidisciplinary team with extensive experience. Endovascular treatment is the first‐choice treatment for most paediatric intracranial vascular malformations. Indication and timing for treatment should be decided on the basis of a careful assessment of neurological symptoms, growth and development, cardiac and other systemic manifestations, and imaging of the malformation and the brain tissue. What this paper adds Paediatric intracranial vascular malformations are rare, but their prognosis is poor if left untreated. Improved clinical, anatomical, and pathophysiological understanding of these complex lesions has improved prognosis.

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Cross‐sectional study of a United Kingdom cohort of neonatal vein of galen malformation

Cited by 41 publications

References 14 publications

Long‐term outcome of vein of Galen malformation

Long‐term outcome of vein of Galen malformation

Outcome of vein of Galen malformation presenting in the neonatal period

Endovascular treatment of intracranial vascular malformations in children

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