Evidence map›Paper›PMID 38668906›Full record

ArticleChild's nervous system : ChNS : official journal of the International Society for Pediatric Neurosurgery2024

Moyamoya syndrome in a patient with D-2-hydroxyglutaric aciduria type II: a rare association.

Tobias Kühnl, Elke Januschek, Sana Klinikum Offenbach

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In one paragraph

Article in Child's nervous system : ChNS : official journal of the International Society for Pediatric Neurosurgery, 2024. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 1 paper.

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0cells of the map it votes in
1citing papers in PubMed
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1 · What the graph read from it

What it found

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2 · The registry

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3 · Its place in the literature

Who cites it

1 citing paper in PubMed.

  1. Review
4 · The record

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5 · Who and what money

Authors and funding

3 authors.

Tobias KühnlDepartment of Neurosurgery, Offenbach, Hessen, Germany. tobias.kuehnl@sana.de.
Elke JanuschekDepartment of Neurosurgery, Offenbach, Hessen, Germany.
Sana Klinikum OffenbachDepartment of Neurosurgery, Offenbach, Hessen, Germany.

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

purposeSeveral underlying conditions of moyamoya syndrome (MMS) are well established, but so far, D-2-hydroxyglutaric aciduria (D-2-HGA) has not been mentioned. We are the first to describe a case of a patient suffering from D-2-HGA developing MMS.

methodsThe co-occurrence of D-2-HGA and MMS in a patient is reported. Furthermore, we describe the neurosurgical revascularization procedure performed and report on the follow-up.

resultsA 7-year-old girl suffering from D-2-HGA developed two transient ischemic attacks (TIAs). Using MRI/MRA and invasive angiography MMS was diagnosed. We performed an encephalo-duro-arterio-myo-synangiosis (EDAMS) as an indirect revascularization procedure first on the right and 2 months later on the left hemisphere. We have followed her up until the age of 10. Since the second surgery, she has not suffered further TIAs and is in a better general medical condition.

conclusionEven though children with D-2-HGA often suffer epileptic attacks, every new (transient) neurological deficit should be followed up by an MRI/MRA so as not to oversee a possible underlying MMS. After diagnosis, EDAMS in combination with acetylsalicylic acid (ASA) is recommended to prevent further ischemic events.

Indexed as

Moyamoya DiseaseBrain Diseases, Metabolic, InbornCerebral RevascularizationChildFemaleHumansMagnetic Resonance ImagingD-2-Hydroxyglutaric aciduria type IIEDAMSMoyamoya syndrome

Identifiers

PMID38668906

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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.