Evidence map›Paper›PMID 40880844›Full record

ArticleAnnals of the Child Neurology Society2025

The Use of Synaptic Extracellular Myo-Inositol to Treat Developmental and Epileptic Encephalopathy.

E Naomi Vos, Didem Demirbas, Lance Rodan, Edward Yang, Sanjay P Prabhu, Jie Chen, Xiaoping Huang, Wanshu Qi, Robin L Haynes, Maria K Lehtinen and 6 more

Abstract read
In one paragraph

Article in Annals of the Child Neurology Society, 2025. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 2 papers.

0numbers the graph read from it
0cells of the map it votes in
2citing papers in PubMed
–field-weighted citation impact
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

2 citing papers in PubMed.

  1. Article
  2. 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

16 authors.

E Naomi VosDepartment of Pediatrics, MosaKids Children's Hospital Maastricht University Medical Centre Maastricht the Netherlands.
Didem DemirbasDivision of Genetics & Genomics Boston Children's Hospital Boston Massachusetts USA.
Lance RodanDivision of Genetics & Genomics Boston Children's Hospital Boston Massachusetts USA.
Edward YangDepartment of Radiology Boston Children's Hospital Boston Massachusetts USA.
Sanjay P PrabhuDepartment of Radiology Boston Children's Hospital Boston Massachusetts USA.
Jie ChenDivision of Laboratory Medicine The Children's Hospital of Philadelphia Philadelphia Pennsylvania USA.
Xiaoping HuangMitochondrial Biology and Neurodegeneration Unit, Neurogenetics Branch, National Institute of Neurological Disorders and Stroke National Institutes of Health Bethesda Maryland USA.
Wanshu QiDivision of Genetics & Genomics Boston Children's Hospital Boston Massachusetts USA.
Robin L HaynesDepartment of Pathology Boston Children's Hospital Boston Massachusetts USA.
Maria K LehtinenDepartment of Pathology Boston Children's Hospital Boston Massachusetts USA.
Michael J BennettDepartment of Pathology and Laboratory Medicine Children's Hospital of Philadelphia and university of Pennsylvania Perelman School of Medicine Philadelphia Pennsylvania USA.
Miao HeDepartment of Pathology and Laboratory Medicine The Children's Hospital of Philadelphia Philadelphia Pennsylvania USA.
Phillip L PearlDivision of Epilepsy, Department of Neurology Harvard Medical School Boston Massachusetts USA.ORCID https://orcid.org/0000-0002-6373-1068
M Estela Rubio-GozalboDepartment of Pediatrics, MosaKids Children's Hospital Maastricht University Medical Centre Maastricht the Netherlands.
Annapurna PoduriDivision of Epilepsy, Department of Neurology Harvard Medical School Boston Massachusetts USA.
Gerard T BerryDivision of Genetics & Genomics Boston Children's Hospital Boston Massachusetts USA.

Funding

Control of neural stem cells by the nascent cerebrospinal fluidR01NS088566 · NINDS · BOSTON CHILDREN'S HOSPITAL · PI LEHTINEN, MARIA · 2014 to 2024
$5.5M
Natural History of Succinic Semialdehyde Dehydrogenase Deficiency (SSADHD), a Heritable Disorder of GABA MetabolismR01HD091142 · NICHD · WASHINGTON STATE UNIVERSITY · PI Phillip Lawrence Pearl · 2018 to 2026
$5.3M
Molecular mechanisms of mitochondrial dysfunction in ATP13A2-associated neurodegeK08NS083739 · NINDS · UNIVERSITY OF ARIZONA · PI KRUER, MICHAEL C · 2013 to 2017
$869k
NICHD NIH HHS R01 HD091142NINDS NIH HHS K08 NS083739NINDS NIH HHS R01 NS088566
6 · The paper itself

Abstract

Objective: The developmental and epileptic encephalopathies (DEE) are associated with serious and lifelong neurological conditions and risk of early mortality. Here, we describe the chronic treatment of a boy with Methods: Myo-inositol levels were measured in plasma, urine, and cerebrospinal fluid (CSF) using stable isotope dilution and selected ion monitoring gas chromatography/mass spectrometry. Brain function and structure were monitored with magnetic resonance spectroscopy, magnetic resonance imaging, and electroencephalograms. Safety studies were performed according to Food and Drug Administration requirements. Results: Treatment was well tolerated without any adverse events. There was an improvement in seizure burden and stabilization of brain atrophy that was most evident in the first and second years of life. Myo-inositol administration to the pregnant Interpretation: High-dose enteral myo-inositol supplementation was safely used in a patient with epileptic encephalopathy due to

Indexed as

epilepsyhyperpolarizationmyo‐inositolprenatalsafetytreatment

Identifiers

PMID40880844
PMCPMC12381637

What OpenQuestion holds

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Registered trials

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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.