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Record W4413906780 · doi:10.1186/s12969-025-01137-9

Neurological involvement in children with familial Mediterranean fever: a systematic review

2025· review· en· W4413906780 on OpenAlexaff
Saverio La Bella, Antonio Corsello, Deniz Bayraktar, Armando Di Ludovico, Giovanna Scorrano, M. Rinaldi, Yağmur Bayındır, Seza Özen, Gregorio P. Milani, Marco Gattorno, Roberta Caorsi

Bibliographic record

VenuePediatric Rheumatology · 2025
Typereview
Languageen
FieldBiochemistry, Genetics and Molecular Biology
TopicInflammasome and immune disorders
Canadian institutionsResearch Canada
Fundersnot available
KeywordsFamilial Mediterranean feverMedicinePediatricsMEFVAbdominal painDiseaseInternal medicine

Abstract

fetched live from OpenAlex

BACKGROUND: Although typical findings of familial Mediterranean fever (FMF), such as brief fever episodes and abdominal or chest pain, have been largely described, little is known about the neurological manifestations of the disease in childhood. METHODS: A systematic search of the literature was conducted in PubMed/Medline, Cochrane, and Web of Science databases in accordance with the PRISMA guidelines, using MeSH terms related to FMF and neurological manifestations. Studies involving patients under 18 years of age diagnosed with FMF with neurological manifestations were included. RESULTS: Sixty-four studies, comprising 4753 children with FMF, were included. Approximately 33.9% of them had some degree of neurological involvement. Headache was the most common neurological symptom and was often associated with FMF flares, with frequencies ranging from 4.8 to 58.8%. Febrile seizures were also relevant manifestations, as expected in children with FMF since they have more and more high fever during childhood, with frequencies ranging from 1 to 15.2%. Demyelinating disorders, such as multiple sclerosis, were rarely reported, mostly in female adolescents with the homozygous M694V MEFV genotype. A few studies have shown that cochlear and retinal involvement due to chronic and recurrent inflammation may contribute to sensorineural hearing loss and retinal abnormalities. CONCLUSION: Although causality has not been shown and reporting bias cannot be excluded, neurological involvement appears relatively common in children with FMF and may lead to long-term disability and reduced quality of life. These findings support the need for a comprehensive neurological assessment to enable early detection, appropriate management, and better long-term outcomes.

Fetched live from OpenAlex and de-inverted. Abstracts are not stored in this database: the inverted indexes are 8.6 GB of the frame’s 9.3 GB of text, and the host has 13 GB free.

How this classification was reachedexpand

Full frame distilled prediction

Teacher imitation

Not calibrated prevalence, not ground truth. Human validation pending. Learned from the 10,348 direct Codex labels and 10,348 direct Gemma labels. Candidate is the union of thresholded teacher heads; consensus is their intersection. These outputs are machine_predicted_unvalidated and are not human labels or direct frontier model labels.

metaresearch head score (Codex)0.001
metaresearch head score (Gemma)0.001
Version: codex-gemma-dda1882f352aValidation status: machine_predicted_unvalidated
Candidate categoriesMeta-epidemiology (narrow)
Consensus categoriesnone
DomainCandidate signal: none · Consensus signal: none
Study designCandidate signal: Systematic review · Consensus signal: Systematic review
GenreCandidate signal: Review · Consensus signal: Review
Teacher disagreement score0.249
Threshold uncertainty score1.000

Codex and Gemma teacher scores by category

CategoryCodexGemma
Metaresearch0.0010.001
Meta-epidemiology (narrow)0.0010.000
Meta-epidemiology (broad)0.0040.000
Bibliometrics0.0000.001
Science and technology studies0.0000.000
Scholarly communication0.0000.000
Open science0.0010.000
Research integrity0.0010.000
Insufficient payload (model declined to judge)0.0000.000

Machine scores (provisional)

The two teacher heads of the student model, read on this work. A score orders the frame for review; it never asserts a category, and the validation status ships verbatim with every row.

Baseline scores from an immature model (maturity gate not passed, 7 training rounds). Scores rank; they never assert a category.

Opus teacher head0.010
GPT teacher head0.258
Teacher spread0.248 · how far apart the two teachers sit on this one work
Validation statusscore_only:v0-immature-baseline · verbatim from the scoring run: score_only means the number may rank works, and no category label ships from it

Classification

machine, unvalidated

Machine predicted; a candidate call from one teacher head, not a consensus.

Study designSystematic review
Domainnot available
GenreReview

How this classification was reached, model by model and score by score, is at the end of the page under "How this classification was reached".

Quick stats

Citations0
Published2025
Admission routes1
Has abstractyes

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