Evidence map›Paper›PMID 32197817›Full record

ArticlePediatric neurology2020

Spontaneous Third Ventriculostomy in Krabbe Disease.

Giulio Zuccoli, Aram Kim, Michele Poe, Maria L Escolar

Open access · hybridAbstract read
In one paragraph

Article in Pediatric neurology, 2020. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 1 paper.

0numbers the graph read from it
0cells of the map it votes in
1citing papers in PubMed
–field-weighted citation impact, top 85% of its field
1 · What the graph read from it

What it found

Each row is one number read from the abstract, on the scale the paper reported it, with its interval. Left of the dashed line favours the treatment, right favours the comparator. Under each row is the sentence it came from. New to these charts? A ten-minute tutorial.

The abstract states no effect estimate the extractor could read, or names no intervention and outcome on the map, so this paper lights no cell and moves no belief. It is still indexed, cited and linked below.

2 · The registry

The trial behind it

Trials whose registry record cites this paper, or whose number appears in the abstract. A trial that started after this paper was published is citing it as background, not reporting it.

Neither the registry nor the abstract names a trial number. If this is a trial report, that itself is worth knowing.

3 · Its place in the literature

Who cites it

1 citing paper in PubMed, 4 citations in OpenAlex.

  1. Article
4 · The record

Corrections and comments

PubMed lists nothing against this paper. Absence here is not a guarantee, only a check that was made.

5 · Who and what money

Authors and funding

4 authors at 2 institutions in 1 country.

Giulio ZuccoliDepartment of Radiology, UPMC Children's Hospital of Pittsburgh, University of Pittsburgh, Pittsburgh, Pennsylvania.
Aram KimDivision of Child Neurology, Department of Pediatrics, UPMC Children's Hospital of Pittsburgh, Pittsburgh, Pennsylvania.
Michele PoeProgram for the Study of Neurodevelopment in Rare Disorders, Department of Pediatrics, UPMC Children's Hospital of Pittsburgh, Pittsburgh, Pennsylvania.
Maria L EscolarProgram for the Study of Neurodevelopment in Rare Disorders, Department of Pediatrics, UPMC Children's Hospital of Pittsburgh, Pittsburgh, Pennsylvania. Electronic address: maria.escolar@chp.edu.
Children's Hospital of Pittsburgh · USUniversity of Pittsburgh · US

Funding

DTI as a tool to identify infants with Krabbe Disease in need of urgent treatmentR01NS061965 · NINDS · UNIV OF NORTH CAROLINA CHAPEL HILL · PI ESCOLAR, MARIA L · 2008 to 2012
$2.5M
NINDS NIH HHS R01 NS061965
6 · The paper itself

Abstract

introductionSpontaneous third ventriculostomies have been reported in relation to obstructive hydrocephalus and increased intracranial pressure and are most commonly seen as disruption of the floor of the third ventricle. Hydrocephalus has been reported in patients with Krabbe disease; however, it is clinically difficult to monitor for hydrocephalus in patients with Krabbe disease as symptoms of increased intracranial pressure may overlap with symptoms of Krabbe disease. We describe a case series of spontaneous third ventriculostomy and hydrocephalus, likely in response to increased intracranial pressure, in patients with infantile Krabbe disease.

methodsBrain magnetic resonance images of patients with infantile Krabbe disease were retrospectively analyzed to assess for ventricular size and presence of spontaneous third ventriculostomies. A brain atlas was used to standardize the calculation of ventricular size. Mid-sagittal, T2-weighted images around the third ventricle were assessed for spontaneous third ventriculostomies. Developmental outcomes were measured with a series of standardized and validated tests.

resultsSeventy-five patients with infantile Krabbe disease were evaluated. Twelve cases of spontaneous third ventriculostomies were identified. Head circumference (SE = 8.07; P < 0.001) and average ventricular volume were greater (left: SE = 1.47, P < 0.001) in patients with spontaneous third ventriculostomies when compared with patients without spontaneous third ventriculostomies. Patients with spontaneous third ventriculostomies also had more delayed development in adaptive (difference = 0.2, P < 0.01), gross motor (difference = 0.0, P < 0.01), and fine motor (difference = 0.1, P < 0.001) function.

conclusionsSpontaneous third ventriculostomies, likely in the context of increased intracranial pressure, were identified in patients with Krabbe disease. Although difficult to assess, our study highlights the importance of monitoring for increased intracranial pressure, which can result in spontaneous third ventriculostomies, in patients with infantile Krabbe disease.

Indexed as

AdolescentChildChild, PreschoolFemaleHumansHydrocephalusInfantIntracranial HypertensionLeukodystrophy, Globoid CellMagnetic Resonance ImagingMaleThird VentricleGloboid cell leukodystrophyIntracranial pressureKrabbe diseaseSpontaneous third ventriculostomy

Identifiers

PMID32197817
PMCPMC7263959
OpenAlexW2990534079

What OpenQuestion holds

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Registered trials

None linked

Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the OpenQuestion graph.