Evidence map›Paper›PMID 40607425›Full record

ArticleFrontiers in immunology2025

Complete recovery after complement factor I deficiency associated fulminant acute hemorrhagic leukoencephalitis: a case report.

Fanni Szumutku, Léna Szabó, Zoltán Liptai, Edit Varga, Tímea Seszták, Péter Barsi, Ádám Goschler, Gábor Szarvas, Klára Horváth, Simon Péter Nagy and 3 more

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In one paragraph

Article in Frontiers in immunology, 2025. 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
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

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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.

  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

13 authors.

Fanni SzumutkuTűzoltó Street Department, Pediatric Center, Semmelweis University, Budapest, Hungary.
Léna SzabóTűzoltó Street Department, Pediatric Center, Semmelweis University, Budapest, Hungary.
Zoltán LiptaiTűzoltó Street Department, Pediatric Center, Semmelweis University, Budapest, Hungary.
Edit VargaDepartment of Neuroradiology, Medical Imaging Center, Semmelweis University, Budapest, Hungary.
Tímea SesztákTűzoltó Street Department, Pediatric Center, Semmelweis University, Budapest, Hungary.
Péter BarsiDepartment of Neuroradiology, Medical Imaging Center, Semmelweis University, Budapest, Hungary.
Ádám GoschlerTűzoltó Street Department, Pediatric Center, Semmelweis University, Budapest, Hungary.
Gábor SzarvasTűzoltó Street Department, Pediatric Center, Semmelweis University, Budapest, Hungary.
Klára HorváthTűzoltó Street Department, Pediatric Center, Semmelweis University, Budapest, Hungary.
Simon Péter NagyResearch Laboratory, Department of Internal Medicine and Hematology, Semmelweis University, Budapest, Hungary.
Zoltán ProhászkaResearch Laboratory, Department of Internal Medicine and Hematology, Semmelweis University, Budapest, Hungary.
Ágnes SzilágyiResearch Laboratory, Department of Internal Medicine and Hematology, Semmelweis University, Budapest, Hungary.
Sarolta DobnerTűzoltó Street Department, Pediatric Center, Semmelweis University, Budapest, Hungary.

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

Introduction: Acute hemorrhagic leukoencephalitis (AHLE) is a rare, fulminant neuroinflammatory disease with high mortality rate. It most often occurs after infections; however, the exact etiology of the disease remains unclear. We highlight that complement factor I (FI) deficiency may be a possible cause of AHLE. Case report: We describe a 9-year-old patient presenting with fever, headache, dizziness, ataxia, and diplopia, who developed rapid neurologic decline and refractory intracranial pressure elevation. Based on clinical, laboratory, and MRI findings, AHLE was diagnosed. Successful treatment included therapeutic plasma exchange (PEX) and early decompressive craniectomy. At one year of follow-up, the patient showed complete recovery. Complement testing of the patient revealed complete FI deficiency. Genetic workup uncovered a germline pathogenic variant in the Discussion: As AHLE is an emerging phenotype of complement FI deficiency, with only a few previously reported cases in the literature, high clinical suspicion and awareness among clinicians are needed. To control the complement system, prompt blockade with complement FI substitution via PEX and early decompressive craniectomy may be life-saving. In neuroinflammatory diseases with unknown etiology, complement testing is recommended.

Indexed as

Complement Factor IHereditary Complement Deficiency DiseasesLeukoencephalitis, Acute HemorrhagicChildComplement C3Decompressive CraniectomyHumansPlasma ExchangeTreatment OutcomeCFI protein, humanComplement C3Complement Factor Iacute hemorrhagic leukoencephalitiscase reportcomplement factor I deficiencyimmune-mediated neurological disorderneuroinflammatory disease

Identifiers

PMID40607425
PMCPMC12213424

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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.