Evidence map›Paper›PMID 36621064›Full record

ArticleEuropean journal of paediatric neurology : EJPN : official journal of the European Paediatric Neurology Society2023

Pediatric recurrent acute necrotizing encephalomyelitis, RANBP2 genotype and Sars-CoV-2 infection: Diagnosis, pathogenesis and targeted treatments from a case study.

Cristina Forest, Michele Laudisi, Cristina Malaventura, Valeria Tugnoli, Giuditta Pellino, Elisabetta Marangoni, Eleonora Baldi, Luca Borgatti, Maura Pugliatti, Agnese Suppiej

Open access · bronzeAbstract readCase Reports
PubMed Publisher
In one paragraph

Article in European journal of paediatric neurology : EJPN : official journal of the European Paediatric Neurology Society, 2023. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 11 papers, 2 of them syntheses that pooled it.

0numbers the graph read from it
0cells of the map it votes in
11citing papers in PubMed, 2 pooled it
2.9field-weighted citation impact, top 9% 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

11 citing papers in PubMed, 2 syntheses or guidelines pooled it, 15 citations in OpenAlex.

  1. Pooled it
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  8. Letter to the Editor in response to Dr. Josef Finsterer et al. "Intense work-up is required for pediatric COVID-related acute necrotizing encephalopathy with RANBP2 variants".European journal of paediatric neurology : EJPN : official journal of the European Paediatric Neurology Society · 2023
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  9. Intense work-up is required for pediatric COVID-related acute necrotizing encephalopathy with RANBP2 variants.European journal of paediatric neurology : EJPN : official journal of the European Paediatric Neurology Society · 2023
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4 · The record

Corrections and comments

5 · Who and what money

Authors and funding

10 authors at 1 institution in 1 country.

Cristina ForestDepartment of Medical Sciences-Pediatric Section, University of Ferrara, Ferrara, Italy.
Michele LaudisiDepartment of Neuroscience and Rehabilitation, University of Ferrara, Italy.
Cristina MalaventuraDepartment of Medical Sciences-Pediatric Section, University of Ferrara, Ferrara, Italy.
Valeria TugnoliS. Anna University Hospital, Department of Neuroscience and Rehabilitation, Ferrara, Italy.
Giuditta PellinoDepartment of Medical Sciences-Pediatric Section, University of Ferrara, Ferrara, Italy.
Elisabetta MarangoniDepartment of Anesthesia and Intensive Care, University of Ferrara, Ferrara, Italy.
Eleonora BaldiS. Anna University Hospital, Department of Neuroscience and Rehabilitation, Ferrara, Italy.
Luca BorgattiPediatric Neuroradiology, S. Anna University Hospital, Ferrara, Italy.
Maura PugliattiDepartment of Neuroscience and Rehabilitation, University of Ferrara, Italy; S. Anna University Hospital, Department of Neuroscience and Rehabilitation, Ferrara, Italy.
Agnese SuppiejDepartment of Medical Sciences-Pediatric Section, University of Ferrara, Ferrara, Italy. Electronic address: sppgns@unife.it.
University of Ferrara · IT

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

Acute necrotizing encephalopathy (ANE) is a rare disease not yet described in children with Covid-19. RANBP2 gene variations are implicated in recurrences in the genetic form of ANE, the so called ANE1. We report the first case of pediatric ANE1 following Sars-CoV-2 infection. She had a first episode at 2 years of age following influenza type A with full recovery, many other respiratory and non-respiratory febrile viral infections without recurrences and a severe recurrence following Sars-CoV-2 infection, suggesting a potentiation effect on cytokine cascade. Her MRI showed the typical pattern of injury resembling that of mitochondrial disorders, and supported the role of RANBP2 in mitochondrial homeostasis. This case rises attention on diagnostic challenges and offers several interesting tips for discussion about new perspectives in pathogenesis and targeted treatments.

Indexed as

Brain DiseasesCOVID-19EncephalomyelitisLeukoencephalitis, Acute HemorrhagicChildCOVID-19 TestingFemaleGenotypeHumansMolecular ChaperonesNuclear Pore Complex ProteinsSARS-CoV-2Molecular ChaperonesNuclear Pore Complex Proteinsran-binding protein 2ANE1Mitochondrial homeostasisPediatricRANBP2Sars-CoV-2

Identifiers

PMID36621064
OpenAlexW4313530029

What OpenQuestion holds

Textmetadata
Read underepoch 390

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.